Orthodontics in Obstructive Sleep Apnea Patients A Guide to Diagnosis, ­Treatment Planning, and Interventions Su-Jung Kim Ki Beom Kim Editors 123 Orthodontics in Obstructive Sleep Apnea Patients Su-Jung Kim • Ki Beom Kim Editors Orthodontics in Obstructive Sleep Apnea Patients A Guide to Diagnosis, Treatment Planning, and Interventions Editors Su-Jung Kim Orthodontic Department Kyung Hee University School of Dentistry Seoul Korea (Republic of) Ki Beom Kim Orthodontics, Saint Louis University Orthodontics Saint Louis MO USA ISBN 978-3-030-24412-5 ISBN 978-3-030-24413-2 https://doi.org/10.1007/978-3-030-24413-2 (eBook) © Springer Nature Switzerland AG 2020 This work is subject to copyright. All rights are reserved by the Publisher, whether the whole or part of the material is concerned, specifically the rights of translation, reprinting, reuse of illustrations, recitation, broadcasting, reproduction on microfilms or in any other physical way, and transmission or information storage and retrieval, electronic adaptation, computer software, or by similar or dissimilar methodology now known or hereafter developed. The use of general descriptive names, registered names, trademarks, service marks, etc. in this publication does not imply, even in the absence of a specific statement, that such names are exempt from the relevant protective laws and regulations and therefore free for general use. The publisher, the authors, and the editors are safe to assume that the advice and information in this book are believed to be true and accurate at the date of publication. Neither the publisher nor the authors or the editors give a warranty, expressed or implied, with respect to the material contained herein or for any errors or omissions that may have been made. The publisher remains neutral with regard to jurisdictional claims in published maps and institutional affiliations. This Springer imprint is published by the registered company Springer Nature Switzerland AG The registered company address is: Gewerbestrasse 11, 6330 Cham, Switzerland Preface Snoring and obstructive sleep apnea (OSA), as the common types of sleep-­disordered breathing (SDB), have increasingly caught the attention of dentists and orthodontists since they become greater public health issues than ever. Previously, SDB is mostly recognized and managed by sleep physicians since it is a chronic multifactorial medical disease and a potentially life-threatening disorder leading to serious medical complications. Currently, however, dental sleep medicine has been emerging to deal with SDB by means of providing critical roles in the diagnosis and treatment with various orthodontic tools in hands. This text is intended to be a reference handbook on the orthodontic approaches for the SDB patients. This handbook will help dental students, dentists, orthodontic residents, and clinicians to understand all the practical information on the SDB from orthodontic point of view. Moreover, this handbook will update the orthodontists by providing well-organized diagnostic and therapeutic protocols for the SDB patients based on the integration of sleep into orthodontic practice. This book comprises three parts of general understanding of SDB and medical approaches, orthodontic diagnostic workflow, and orthodontic treatment application. In particular, the treatment parts are subdivided into six chapters depending on the patient’s phenotype and age groups. The readers will come to realize how many modalities are available beyond the previously well-known options and how important orthodontic contributions are for the treatment of SDB patients. This handbook will be a valuable clinical guideline for both beginners and experts who are interested in expanding our aims and scopes towards preventing or managing the SDB simultaneously with correcting skeletal and dental malocclusion. The demand of sleep-related orthodontics is driven by the need of our societies and patients who are coming into our offices aware of OSA. Let us keep in mind that we are improving our patient’s life quality and general health through changing the bite, smile, breathing, and sleep. Seoul, Republic of Korea Su-Jung Kim v Contents 1General Understanding of OSA as Orthodontists���������������������������������� 1 Su-Jung Kim and Sung-Wan Kim 2Orthodontic Evaluation and Diagnostic Workflow for OSA Patients���������������������������������������������������������������������������������������� 15 Su-Jung Kim and Ki Beom Kim 3Therapeutic Pathway for Orthodontic Intervention������������������������������ 29 Su-Jung Kim, Patricia Pigato Schneider, and Ki Beom Kim 4Craniofacial Growth Modification for OSA Children���������������������������� 41 Su-Jung Kim 5Craniofacial Orthopedics for Postadolescent OSA Patients������������������ 59 Su-Jung Kim 6Surgical Maxillary Expansion for OSA Adults with Nasal Obstruction�������������������������������������������������������������������������������������� 65 Hyo-Won Ahn and Su-Jung Kim 7Maxillomandibular Advancement Surgery for Skeletal Class II OSA Patients������������������������������������������������������������ 81 Jin-Young Choi and Seung-Hak Baek 8Modification of Orthognathic Surgery for Skeletal Class III OSA Patients������������������������������������������������������������������������������ 95 Takashi Ono 9Mandibular Advancement Device for Elderly OSA Patients���������������������������������������������������������������������������������������������� 109 Su-Jung Kim and Young-Guk Park 10Oropharyngeal Exercise for OSA Patients���������������������������������������������� 131 Kyung-A Kim and Su-Jung Kim vii 1 General Understanding of OSA as Orthodontists Su-Jung Kim and Sung-Wan Kim Contents 1.1 A natomy of Upper Airway 1.2 P athophysiology of OSA 1.3 M edical Approach for Diagnosis and Treatment of OSA 1.3.1 Medical Diagnostic Approach for OSA 1.3.2 Medical Therapeutic Approach for OSA 1.3.3 Treatment Modalities for OSA References 1.1 1 2 3 3 8 10 12 Anatomy of Upper Airway The respiratory tract from the nose to the lung is divided into the upper tract and lower tract, also known as upper airway (UA) and lower airway (LA). The UA comprises nasal cavity, pharynx, and larynx, while the LA involves trachea, bronchi, and lung. In spite of inconsistent definition and terminology regarding the subdivision of UA, pharyngeal airway can be divided into three regions to be relevant to orthodontics: nasopharynx, oropharynx, and hypopharynx (Fig. 1.1). • Nasopharynx extends from behind the nasal turbinates to the level of hard palate. Adenoids (pharyngeal tonsils) affect the nasopharyngeal patency. S.-J. Kim (*) Department of Orthodontics, Kyung Hee University School of Dentistry, Seoul, South Korea e-mail: ksj113@khu.ac.kr S.-W. Kim Department of Otorhinolaryngology—Head and Neck Surgery, Kyung Hee University School of Medicine, Seoul, South Korea © Springer Nature Switzerland AG 2020 S.-J. Kim, K. B. Kim (eds.), Orthodontics in Obstructive Sleep Apnea Patients, https://doi.org/10.1007/978-3-030-24413-2_1 1 2 S.-J. Kim and S.-W. Kim Fig. 1.1 Anatomic definition of upper airway described in the lateral cephalometric (left) and CBCT images (right). NC Nasal cavity, NP Nasopharynx, VP Velopharynx, OP Oropharynx, HP Hypopharynx • Oropharynx lies behind the oral cavity extending from the soft palate to the upper border of epiglottis. This can be subdivided into the retropalatal airway, which lies behind the soft palate extending from the hard palate to the caudal margin of the soft palate (which is called velopharynx), and the retroglossal airway, which extends from the caudal margin of the soft palate to the base of the epiglottis behind the tongue. Uvula, soft palate, palatine tonsils, and tongue posture may affect the oropharyngeal patency. • Hypopharynx (laryngopharynx) lies inferior to the epiglottis and extends to the diverged area into the larynx and esophagus. Tongue base and hyoid position may influence the hypopharyngeal patency. Whereas nasal cavity is a bony structure, pharyngeal airway is a soft tissue structure surrounded by muscles constricting or dilating the UA lumen. In terms of orthodontists, genioglossus muscles are important when considering the control of tongue and hyoid posture, and palatoglossus muscles are known to be communicating muscles between the soft palate and the tongue, which contributes to opening the UA. More importantly, it should be noted that lateral pharyngeal walls comprising palatopharyngeus, stylopharyngeus, salpingopharyngeus, and pharyngeal constrictors are the most dynamic and responsive areas to the physiologic function and therapeutic application [1]. 1.2 Pathophysiology of OSA Physical obstruction of UA, due to the abnormal surrounding structures or to inadequate motor tone of the tongue and/or airway dilator muscles, prevents normal air passage. UA obstruction leads to hypoxia and hypercapnia. With oxygen desaturation, the heart rate and blood pressure rise with a ventilatory effort and sympathetic hyperactivity to stimulate pharyngeal dilator muscles. The airway opens and there 1 General Understanding of OSA as Orthodontists 3 is a correction of blood gases to normal, but sleep becomes fragmented as the cycle repeats. As such, main symptoms include loud snoring, witnessed apnea, and excessive daytime sleepiness (EDS). Consequently, the sequela of cardiovascular, cerebrovascular, and metabolic comorbidities, and all-cause mortality pose a significant public health problem [2]. OSA occurs as a result of anatomical and functional abnormalities of the UA that compromise airway space and increase pharyngeal collapsibility during sleep. Although alterations in neuromuscular and ventilatory control mechanisms can contribute to the reduced airway patency underlying OSA, anatomical abnormalities play a primary role in the development of OSA. Anatomical risk factors for OSA include obesity, excess regional adipose tissue, enlarged upper airway soft tissues, and craniofacial skeletal abnormalities. The interactions between these anatomical factors, together with patient’s demographics and symptoms, are the main determinants of the likelihood and severity of OSA in the majority of patients. In this context, orthodontic treatments that may reduce oral cavity and tongue space have been in the debate issue whether they are significant risk factors of OSA or not. We sometimes recognize the narrowed pharyngeal airway spaces in the X-ray images after excessive anterior retraction with premolar extraction or mandibular set-back surgery. However, recent consensus is that not all these patients complain about respiratory discomforts. Actually, four different responses to the decreased UA dimension are observed clinically (Fig. 1.2): (1) no significant influence on the respiratory function and occlusion even maintaining the narrowed dimension; (2) compensatory adaptation of the UA to restore original dimension without leading to any problem; (3) respiratory dysfunction like snoring or OSA affected by decreased UA volume and subsequent increased pharyngeal collapsibility; (4) rebound of UA size resulting in occlusal relapse in relation to unfavorable tongue response. Evidence-based conclusion is still insufficient due to the limited number of studies and big heterogeneity among them [3]. There is no scientific evidence that decrease of UA dimension by orthodontic treatment, if any, would turn the airway more collapsible. The correlation between the UA dimensional change and respiratory functional change has not been clearly demonstrated. Nonetheless, we had better be cautious when we plan huge retraction of anterior teeth or jaw set-back surgery in the patients with already constricted UA. 1.3 Medical Approach for Diagnosis and Treatment of OSA 1.3.1 Medical Diagnostic Approach for OSA 1.3.1.1 Chief Complaint The most common complaint of OSA patients is loud snoring. Listening to the patient’s complaints beyond snoring may help the clinician in building up patient’s trust, which is crucial in starting patient-centered care. 4 S.-J. Kim and S.-W. Kim Patient A; No significance influence on airway dimension and function Patient B; Pharyngeal constriction inducing snoring Patient C; Functional adaptation even with pharyngeal narrowing without relapse Patient D; No functional adaptation to pharyngeal narrowing inducing relapse Fig. 1.2 Various responses to the decreased upper airway dimension after premolar extraction treatment can be anticipated: Patient A, no significant influence on the airway dimension and function after treatment; Patient B, respiratory dysfunction like snoring after posttreatment pharyngeal narrowing; Patient C, compensatory functional adaptation to the pharyngeal narrowing without leading to any problem; Patient D, rebound of airway dimension in relation to occlusal relapse with unfavorable tongue response. There is no scientific evidence supporting that the patients treated with extraction treatment would have respiratory functional problems 1 General Understanding of OSA as Orthodontists 5 1.3.1.2 History Taking It is important to know other nighttime symptoms like witnessed apneas and daytime symptoms like excessive daytime sleepiness, morning headache, and difficult concentration. Sleep doctors firstly focus on some important factors such as average sleep time, sleep pattern, associated insomnia, and sleep habits. Asking about smoking and alcohol consumption are of a particular importance in OSA clinic. Social history and travel history may aid in understanding patients’ abilities or barriers toward some treatment lines. Past medical history (like history of depression disorder, uncontrolled hypertension, diabetes mellitus, cardiac diseases, etc.) and medications history (like antidepressants, hypnotics, etc.) are also important. 1.3.1.3 Questionnaire Some easy-to-use questionnaires have been developed as low-cost alternatives to PSG for detecting OSA: Berlin questionnaire [4], Epworth Sleep Scale (ESS) [5], and STOPBang [6]. ESS is a validated questionnaire that consists of eight items to discriminate the daytime sleepiness level of OSA patients from non-OSA patients (Table 1.1). The STOP-Bang questionnaire includes four sleep-related questions and four additional demographic queries, for a total of eight dichotomous (yes/no) questions: snoring, tiredness, observed apnea, high blood pressure, BMI, age, neck circumference, and gender) (Table 1.2). ESS and STOP-Bang scales are easily taken by dentists. 1.3.1.4 Polysomnography Polysomnography (PSG) is the gold standard for the diagnosis of OSA, but it is time-consuming and requires trained personnel. PSG is a noninvasive technique that involves overnight monitoring of several physiological variables including Table 1.1 Epworth sleepiness scales 6 S.-J. Kim and S.-W. Kim Table 1.2 STOP-Bang questionnaire electroencephalography (EEG), eye movements (EOG), heart rhythm (ECG), and skeletal muscle activity (EMG) as well as respiratory effort, airflow, and oxygen saturation. Respiratory events can be quantified by overnight PSG study. An apnea is characterized by complete cessation of airflow for at least 10 s, and a hypopnea is defined as airflow reduction by 30% or greater lasting for 10 seconds or longer, or in case of associated oxygen desaturation by 3% or over. To grade the severity of sleep apnea, the number of apnea plus hypopnea per hour is reported as the apnea– hypopnea index (AHI). An AHI of less than 5 is considered normal. An AHI of 5–15 is mild; 15–30 is moderate; and more than 30 events per hour indicate severe sleep apnea. For children, an AHI in excess of 1 is considered abnormal due to the different physiology: 1–5, mild; 5–10, moderate; and 10 and over, severe. In addition to AHI, we have to think of respiratory disturbance index (RDI) and oxygen desaturation index (ODI) together to appreciate the severity of OSA. The RDI means the average number of apnea, hypopnea, and respiratory-effort-related arousals (RERAs) per hour of sleep. The ODI is the number of times per hour of sleep that the blood’s oxygen level drop by 3% from baseline. 1.3.1.5 Home Sleep Test with Portable Monitoring Device Home sleep test (HST) using a portable monitoring device can be an alternative to PSG for the diagnosis of OSA, due to the convenience, expedited diagnosis, and no need of hospitalization. It seems attractive to the dentists; however, it should be 1 General Understanding of OSA as Orthodontists 7 Table 1.3 Classification of sleep-monitoring system Category Type I Type II Type III Type IV Definition Attended in-laboratory studies with full sleep staging, including EEG, EOG, ECG, EMG, respiratory efforts, airflow, pulse oximetry, and additional channels for CPAP levels (Full PSG). Unattended home studies without a technologist, recording the same variables as Type I PSG (Embletta ×100). Unattended home studies with a minimum of four channels: respiratory movement, airflow, cardiac variables, and oxygen saturation. RERAs and RDIs cannot be detected (Apnealink Plus™). These devices are called dual bioparameter devices. They record only airflow and arterial oxygen saturation. EEG Electroencephalography, EOG Eye movements, ECG Heart rhythm, EMG Skeletal muscle activity, CPAP Continuous positive airway pressure, PSG Polysomnography, RERAs Respiratory-­ effort-­related arousals, RDIs Respiratory disturbance index. performed only in conjunction with a comprehensive sleep evaluation by certified practitioner, since the sensor application, scoring, and interpretation of the collected data must be correctly set to assure accuracy and reliability. It is essential to recognize several inherent limitations when we use the HST [7]. First, HST does not record sleep but yields information regarding the number of disordered breathing events per hour. Second, disordered breathing events associated with arousals but without sufficient oxyhemoglobin desaturation cannot be assessed, leading to an underestimation of disease severity. Third, sensor failure or drop-out during the night can lead to suboptimal recordings. Fourth, the selection of specific sensors and the scoring methodology for portable monitoring are not as well formulated as they are for PSG. Lastly, due to the different sensor mechanisms, comparisons of the disordered breathing measures among devices are difficult to make. According to American Academy of Sleep Medicine (AASM)’s guideline in 2017 [8], HST is recommended only to the uncomplicated adult patients presenting with signs and symptoms of moderate-to-severe OSA. HST can also be used to evaluate the efficacy of oral appliance or surgical treatment. We should be aware of the type of HST device before use it (Table 1.3), considering the possibility of underestimation and misinterpretation. 1.3.1.6 Nasopharyngoscopy Nasopharyngeal endoscopy, nasopharyngoscopy, is the examination of the internal surfaces of the nose and throat by inserting a thin, flexible, usually fiber-optic instrument called nasopharyngoscope to detect and diagnose abnormalities in the nose and nasopharyngeal area. 1.3.1.7 Müller’s Maneuver This technique is designed to see the collapsed sites of upper airway during the inspiration with closed mouth and nose leading to the negative pressure in the chest and lungs. Introducing a flexible fiber-optic scope into the hypopharynx to obtain a view, the examiner may witness the collapse and identify weakened sections of the airway. However, the sites of obstruction with Müller's maneuver do not represent reliably the sites of obstruction during sleep. 8 S.-J. Kim and S.-W. Kim Nasal Valve Area 10.0 Septum Area (cm2) Inferior Turbinate Nose tip Nasal Valve Turbinates Nasal cavity Right 1.0 Left 0.1 -6.0 -2.0 2.0 6.0 10.0 Distance (cm) Nose tip Nasal Valve Turbinates Nasal cavity Fig. 1.3 Acoustic rhinometry to measure the nasal cavity geometry and nasal airflow change through acoustic reflection 1.3.1.8 Acoustic Rhinometry Acoustic rhinometry (AR) is a simple, fast, and noninvasive diagnostic tool measuring nasal cavity geometry and nasal airway change through acoustic reflection (Fig. 1.3). The size and the pattern of the reflected sound waves provide information on the structure and dimensions of anterior and middle parts of nasal cavity including nasal valve area, which shows the greatest nasal airflow resistance. 1.3.1.9 Dynamic Sleep MRI Dynamic sleep MRI has advantages of dynamic nature, the ability to evaluate the airway in a multiplane fashion, and more realistic information obtained in the sleeping state or a simulated sleep state. Although currently used in the research setting, these approaches may help further our understanding of levels of obstruction and impact of various treatments. 1.3.1.10 Drug-Induced Sleep Endoscopy (DISE) Drug-induced sleep endoscopy (DISE) has been introduced as an alternative to conventional endoscopy for more accurately representing patterns of collapse during the sleeping state. DISE brings us closer to understanding the dynamic airway during sleep. It seems that awake endoscopy and DISE detect retropalatal collapse equally well, but DISE may identify retrolingual, hypopharyngeal collapse more often, and also lateral pharyngeal wall collapse more specifically. Despite some shortcomings, more and more data are emerging about patterns of collapse on DISE that predict success with various surgical interventions. 1.3.2 Medical Therapeutic Approach for OSA 1.3.2.1 Traditional Concept 1: PAP or Non-PAP approach (Table 1.4) According to the European Respiratory Society task force report in 2011, continuous positive airway pressure (CPAP) has proven to improve OSA as the first-line of 1 General Understanding of OSA as Orthodontists 9 Table 1.4 Grade of recommendation (A>B>C>D) in case of CPAP intolerance treatment, and other treatment options (non-CPAP treatments) can be considered only as an alternative in case of CPAP intolerance. The grade of recommendation (A>B>C>D) was given to each non-CPAP therapy as compared to the CPAP efficacy (Table 1.4). Here, mandibular advancement device (MAD) and maxillomandibular advancement (MMA) surgery were included in this report with grade A and B, respectively. This traditional approach has a limitation of nonselective application of CPAP regardless of patients’ phenotypes. 1.3.2.2 Traditional Concept 2: Phased Approach (Fig. 1.4) The original phase I and phase II Stanford protocol [9] is described in Fig. 1.4. Nonsurgical conservative treatment was firstly considered, and phase I surgical procedures were applied according to the main obstruction sites. Only when phase I alone was not successful in achieving surgical success, the sleep surgeon could proceed to phase II surgical protocol encompassing MMA. This approach has been advocated to minimize morbidity while maximizing opportunity for successful outcomes. Here, MMA could not be considered as a primary option regardless of severe craniofacial abnormality. 1.3.2.3 Current Concept: Precision One-Step Approach Stanford treatment protocol has evolved to play a role in collaboration with medical therapies in place of sleep surgeons aiming at providing a range of therapeutic options to best suit the patient’s goals for treatment [10]. This revised protocol in 2019 is defined by precision in patient selection, procedural selection, and 10 S.-J. Kim and S.-W. Kim Fig. 1.4 Original Stanford sleep surgery protocol. CPAP Continuous positive airway pressure, MAD Mandibular advancement device, OP Oropharynx, HP Hypopharynx, UPPP Uvulopalatopharyngoplasty, UPF Uvuolpalatal flap, GA Genioglossus advancement, HMS Hyoid myotomy and suspension, TBR Tongue base reduction, PSG Polysomnography, MMA Maxillomandibular advancement (Adapted from Riley RW, Powell NB, Guilleminault C. 1993 J Oral Maxillofac Surg.) procedural accuracy. Here, MMA surgery can be considered as a primary option for OSA patient with definite dentofacial deformity and/or with complete concentric collapse of lateral pharyngeal wall in DISE, as well as secondary option for the patients who failed to other therapies. In addition, they introduced another orthodontic option of surgical maxillary expansion, called distraction osteogenesis maxillary osteotomy (DOME), for the patients with nasal obstruction. With increasing demand of orthodontist’s roles, we need our precision protocol not only to decide primary intervention but to participate in multiprofessional team better. This will be presented in Chap. 3. 1.3.3 Treatment Modalities for OSA 1.3.3.1 Lifestyle Modification: Weight Loss and Sleep Hygiene Sleep hygiene is the recommended behavioral and environmental practices, which are necessary to promote quality of nighttime sleep and daytime alertness. Clinicians assess the sleep hygiene of OSA patients who present with insomnia or depression, and offer recommendations based on the assessment. Sleep hygiene recommendations include establishing a regular sleep schedule, using naps with care, not exercising physically or mentally too close to bedtime, limiting worry, limiting exposure to light in the hours before sleep, getting out of bed if sleep does not come, not using bed for anything but sleep and sex, avoiding alcohol as well as nicotine, caffeine, and other stimulants in the hours before bedtime, and having a peaceful, comfortable, and dark-sleep environment. 1 General Understanding of OSA as Orthodontists 11 1.3.3.2 Positional Therapy Positional therapy uses devices like backpack, pillow, tennis balls attached to the night suit, or electrical sensors with alarm, that help patients to sleep on their side. The AASM task force recommends positional therapy as an effective secondary therapy for patients with positional OSA (defined as supine AHI>2×AHI). The European Respiratory Society task force on non-CPAP therapies in OSA states that positional therapy can yield a moderate reduction in AHI score, with a grade C recommendation (Table 1.4). 1.3.3.3 Positive Airway Pressure (PAP) Continuous positive airway pressure (CPAP) has been the first-line treatment for OSA due to the high efficacy in reducing sleep-disordered breathing events. In spite of the development of automatic positive airway pressure (APAP), however, lots of patients who try CPAP therapy are either completely intolerant or only partially adherent. Nowadays, the prescription of PAP to all OSA patients as a primary option is not necessary any more based on the novel concept of OSA phenotyping. If the patient has nonanatomical phenotypic causes in a progressive state of OSA, PAP would be an inevitable sole option or can be combined with other treatment modality allowing lower pressure. Otherwise, optimal treatment option needs to be considered based on the differential diagnosis of OSA phenotype. 1.3.3.4 Oral Appliance (OA) Patients mostly prefer oral appliance (OA) to CPAP despite less reduction of AHI, due to the portability, ease of use, and better comfort. OA includes tongue-retaining device (TRD) and mandibular advancement device (MAD). Although TRD directly pulls the tongue forward during sleep to open oropharynx, it is not currently used because of tissue irritation, discomfort, and limited effect. MAD can be prescribed by any sleep specialist, but should be adjusted and managed by qualified dentists. MAD will be discussed in detail in Chap. 9. 1.3.3.5 Surgical Interventions • Tracheostomy bypasses the upper airway and is thus nearly universally successful in managing OSA. However, the significant morbidity associated with tracheostomy limits its application in the OSA population. • Bariatric surgery is a primary surgical option in patients with morbid obesity. • Tonsillectomy with adenoidectomy is the first-line surgical therapy for children with OSA without craniofacial anomalies. • Nasal surgery may play a role in OSA management by improving nasal airflow. Although isolated nasal surgery is unlikely to lead to resolution of severe OSA, it may increase CPAP use and MAD adherence. • The most common palatal surgery is uvulopalatopharyngoplasty (UPPP), which involves removal of the tonsils, uvula, and posterior velum. Multiple variations of UPPP have been described. Due to the low success rate of 33%, UPPP is not recommended by the American Academy of Sleep Medicine (AASM) as a sole procedure for treating moderate-to-severe OSA [11]. 12 S.-J. Kim and S.-W. Kim • Tongue base reduction surgery involves partial glossectomy or various ablative techniques to volumetrically reduce the tongue. • Genioglossal advancement (GA) involves advancement of the genial tubercles, and may be accompanied by hyoid suspension. • Multilevel surgery is acknowledged as an acceptable option for patients with multisite obstruction, reported surgical success rates vary widely from 22% to 78% [12]. • Hypoglossal nerve stimulation is a relatively new addition to the array of surgical options for treatment of OSA, and is applied to the patients with poor neuromuscle responsiveness. • Maxillomandibular advancement (MMA) is the most successful surgical intervention for OSA aside from tracheostomy. MMA has been equated to CPAP in terms of outcomes. This procedure involves advancement of both jaws and addresses airway obstruction at multiple levels; airway collapsibility decreases due to advancement of its skeletal framework. Clinical Pearls in Understanding SDB and OSA • The natural development of sleep disordered breathing (SDB) goes from normal to obstructive hypoventilation syndrome (OHS), passing through persistent snoring, upper airway resistance syndrome (UARS), and obstructive sleep apnea (OSA) between them. • In this chapter, anatomic limits of upper airway and pathophysiology of OSA were explained in terms of orthodontists with orthodontic approaches in mind. • Orthodontists should be well informed of medical approaches for the diagnosis and treatment of OSA patients for intimate collobaration. References 1. Park JG, Ramar K, Olson EJ. Updates on definition, consequences, and management of obstructive sleep apnea. Mayo Clin Proc. 2011;86:549–54. 2. Epstein LJ, Kristo D, Strollo PJ, et al. Clinical guideline for the evaluation, management and long-term care of obstructive sleep apnea in adults. J Clin Sleep Med. 2009;5(03):263–76. 3. Hu Z, Yin X, Liao J, et al. The effect of teeth extraction for orthodontic treatment on the upper airway: a systematic review. Sleep Breath. 2015;19(2):441–51. 4. Netzer NC, Stoohs RA, Netzer CM, et al. Using the Berlin Questionnaire to identify patients at risk for the sleep apnea syndrome. Ann Intern Med. 1999;131(7):485–91. 5. Johns MW. A new method for measuring daytime sleepiness: the Epworth sleepiness scale. Sleep. 1991;14(6):540–5. 6. Chung F, Yegneswaran B, Liao P, et al. Stop questionnaire a tool to screen patients for obstructive sleep apnea. Anesthesiology. 2008;108(5):812–21. 7. Punjabi NM, Aurora RN, Patil SP. Home sleep testing for obstructive sleep apnea: one night is enough! Chest. 2013;143(2):291–4. 1 General Understanding of OSA as Orthodontists 13 8. Kapur VK, Auckley DH, Chowdhuri S, et al. Clinical practice guideline for diagnostic testing for adult obstructive sleep apnea: an American Academy of Sleep Medicine clinical practice guideline. J Clin Sleep Med. 2017;13(03):479–504. 9. Riley RW, Powell NB, Guilleminault C. Obstructive sleep apnea syndrome: a surgical protocol for dynamic upper airway reconstruction. J Oral Maxillofac Surg. 1993;51(7):742–7. 10. Liu SY-C, Awad M, Riley R, Capasso R. The role of the revised Stanford protocol in today’s precision medicine. Sleep Med Clin. 2019;14(1):99–107. 11. Aurora RN, Casey KR, Kristo D, et al. Practice parameters for the surgical modifications of the upper airway for obstructive sleep apnea in adults. Sleep. 2010;33(10):1408–13. 12. Kezirian EJ, Goldberg AN. Hypopharyngeal surgery in obstructive sleep apnea: an evidence-­ based medicine review. Arch Otolaryngol Head Neck Surg. 2006;132(2):206–13. 2 Orthodontic Evaluation and Diagnostic Workflow for OSA Patients Su-Jung Kim and Ki Beom Kim Contents 2.1 History Taking 2.1.1 Medical History 2.1.2 Simplified Questionnaire 2.2 Clinical Examination of Risk Factors 2.2.1 Physical Factors 2.2.2 Behavior Factors 2.2.3 Craniofacial Anatomic Factors 2.3 Lateral Cephalometric Analysis 2.3.1 Significance of Cephalometric Analysis 2.3.2 Cephalometric Definition and Measurements of Upper Airway 2.4 CBCT Volumetric Analysis 2.5 Functional Evaluation on Respiration and Sleep 2.5.1 Mouth Breathing 2.5.2 Bruxism 2.6 Diagnostic Workflow for SDB Patients in Orthodontic Clinic References 16 16 16 16 16 16 17 18 18 20 21 22 22 24 25 27 S.-J. Kim Department of Orthodontics, Kyung Hee University School of Dentistry, Seoul, South Korea e-mail: ksj113@khu.ac.kr K. B. Kim (*) Department of Orthodontics, Center for Advanced Dental Education, Saint Louis University, Saint Louis, MO, USA e-mail: kibeom.kim@health.slu.edu © Springer Nature Switzerland AG 2020 S.-J. Kim, K. B. Kim (eds.), Orthodontics in Obstructive Sleep Apnea Patients, https://doi.org/10.1007/978-3-030-24413-2_2 15 16 S.-J. Kim and K. B. Kim 2.1 History Taking 2.1.1 Medical History After apprehending patient’s chief complaint, orthodontic diagnosis of OSA begins with a sleep history and related medical history. The representative OSA symptoms, which should be initially evaluated, include severe snoring, witnessed apnea, sleep fragmentation, excessive daytime sleepiness (EDS), distraction, and morning headache. The presence of comorbidities such as cardiovascular and cerebrovascular diseases with medication history needs to be check. Social or behavioral history including alcohol, smoking, exercise, and travel frequency should be checked. 2.1.2 Simplified Questionnaire The representative questionnaires for the suspected OSA patients such as Berlin questionnaire, Epworth Sleep Scale (ESS), and STOP-Bang can be introduced in orthodontic clinics [1]. Instead, as a routine health maintenance evaluation including respiration and sleep functions, five questions can be simply applied to every patient not only to diagnose OSA but to prevent OSA after any orthodontic treatment: (1) Is the patient obese? (2) Is the patient retrognathic? (3) Does the patient complain of daytime sleepiness? (4) Does the patient snore? (5) Does the patient have hypertension? 2.2 Clinical Examination of Risk Factors 2.2.1 Physical Factors Firstly, obesity should be checked at the time when the patient comes in the clinic. According to the recent review articles suggesting clinical prediction models [2], the best indicator for the presence of OSA are body mass index (BMI) and neck circumference. BMI can be calculated as a person’s weight in kilogram divided by the square of height in meters (kg/m2). BMI is a simple measure of body fat based on weight and height; however, it is not always correct, since the weight covers muscles as well as fat. 2.2.2 Behavior Factors Habitual head and neck posture, lip posture in rest position, breathing pattern, and the presence of daytime sleepiness need to be observed during the clinical examination. 2 Orthodontic Evaluation and Diagnostic Workflow for OSA Patients 2.2.3 17 Craniofacial Anatomic Factors In order to check if craniofacial anatomic factors contribute to OSA, facial and chin profile, facial height ratio (posterior/anterior, lower/total), and midfacial width should be basically evaluated. More in details, facial and intraoral evaluating points, which are more critical to OSA patients than regular orthodontic patients, can be suggested as follows. Here are the essential roles of the orthodontists in diagnosing OSA. 2.2.3.1 Facial Examination: Facial Triads Retruded chin has been widely known to be the most common facial features of OSA patients. However, the patient with protruded chin sometimes reveals OSA symptoms especially in case with obesity. Therefore, facial characteristics related to obesity should be carefully checked as well as chin profile and facial proportion. Throat length, cervico-mental angle, and neck circumference can be facial triads to be examined together (Fig. 2.1). Short throat length and obtuse cervicomental angle are characteristic regardless of mandibular body length in OSA patients in relation to wide neck circumference over 17 inches and submental fat deposition. 2.2.3.2 Intraoral Examination: Intraoral Triads Dental Class II malocclusion with large overjet and arch constriction appears consistent to give strong consideration in the pathogenesis of OSA; however, this consensus exists in the studies on the Caucasian OSA population. When considering multifactorial causes including genetics and ethnicity, however, vertical and transverse malocclusions such as anterior openbite and posterior crossbite seem to be more associated with the high incidence of OSA than sagittal malocclusion like Class I, II, and III. Thus, intraoral triads to be specifically evaluated for OSA patients can be suggested as (1) narrow and deep palatal arch; (2) tongue position-related structures like large tongue, tongue tie, and tori; and (3) enlarged palatine tonsils with flabby uvula in the mouth (Fig. 2.2). To classify the degree of tonsillar hypertrophy and posterior oropharyngeal tissue collapse, Friedman classification and modified Mallampati test can be used, respectively (Fig. 2.3). These scorings will alert the dentists to identify the oropharyngeal soft tissue problems and further to predict the presence and the severity of OSA. The tonsil grading depicts the ratio of pharyngeal lateral dimension occupied by the hypertrophic tonsils (Fig. 2.3, left). With a high tonsil grading of III or IV, a patient with OSA has a better chance to improve by tonsillectomy with UPPP. The modified Mallampati scale shows the disproportion between soft tissue volume and the size of oral cavity (Fig. 2.3, right). 18 S.-J. Kim and K. B. Kim 3 2 1 3 2 1 1, throat lenght; 2, cervicomental angle; 3, neck circumference Fig. 2.1 Facial triads in clinical examination of OSA patients: (1) throat length, (2) cervico-­ mental angle, (3) neck circumference. Left patient showing short throat length with retruded chin, obtuse cervico-mental angle, and normal neck circumference indicates nonobese OSA patients with craniofacial phenotype of retruded mandible. In contrast, right patient showing short throat length despite protruded chin, obtuse cervico-mental angle, and long neck circumference indicates obese OSA patients with no definite craniofacial risk factors 1. Palatal vault 2. Tori & Tongue tie 3. Tonsils & Uvula Fig. 2.2 Intraoral triads in clinical examination of OSA patients: (1) palatal vault (left), (2) tori and tongue-tie (middle), (3) uvula and palatine tonsils (right). The presence of narrow and high palate, lingual tori, heavy lingual frenulum, flabby and long uvula, and hypertrophic tonsils can be risk factors of SDB 2.3 Lateral Cephalometric Analysis 2.3.1 Significance of Cephalometric Analysis Lateral cephalometric analysis has fundamental limitations to evaluate upper airway and diagnose OSA patients. First, it is a static image taken in an awake upright position, which can hardly characterize the asleep airway function. Second, it is a two-dimensional image, where lateral pharyngeal wall collapse linked to more severe OSA than retropalatal and retrolingual collapse (as assessed with dynamic MRI) would not be noted [3]. Third, the image is 2 Orthodontic Evaluation and Diagnostic Workflow for OSA Patients Friedman Tonsils classification 0 Surgically removed tonsils I Tonsils hidden with tonsil pillars II Tonsils extending to the pillars III IV Tonsils extending 3/4 Tonsils completely of the way to midline obstructed (Kissing tonsils) 19 Modified Mallampati classification II I Tonsils, pillars, soft palate Uvula, pillars, upper pole are clearly visible are visible III Only part of soft palate is visible IV Soft palate is not visible Fig. 2.3 Friedman classification to check tonsillar hypertrophy (left) and modified Mallampati scoring for visualization of the oropharynx based on soft palate-uvula-pillars-tongue base relationship in the mouth (right). (Cited from Friedman M et al. Clinical predictors of obstructive sleep apnea. Laryngoscope. 1999 Dec;109(12):1901–7.) inconsistently obtained depending on the head and tongue posture or the time of imaging. Also, airway images change throughout the respiratory cycle, so any measurement should be taken at a standardized point in the cycle. Fourth, no consensus exists on the most useful landmarks, measurements, and numeric norms to evaluate the pharyngeal airway. Nonetheless, lateral cephalometric analysis is worthy of a basic screening tool to determine the need for more rigorous ENT follow-up for the following reasons [4]. 1. It is the most hand-handled analyzing tool in a daily orthodontic clinic. 2. It is useful for screening the main site of pharyngeal narrowing or obstruction as an adjunctive airway assessment. 3. The pharyngeal dimension can be evaluated in relation to the craniofacial and para-pharyngeal soft tissue abnormalities as a whole. 4. A meta-analysis in 2019 [5] suggested that the lateral cephalogram exhibited very good diagnostic accuracy for the diagnosis of adenoid hypertrophy and posterior upper airway obstruction, although the rate of false-positive diagnosis should be considered. 5. It is useful to predict treatment response initially, to quantify the airway changes in relation to skeletal change after intervention, and to compare the airway changes among groups. Thus, lateral cephalometric airway assessment should be well informed of in order to understand most of the literatures and studies dealing with the prediction and treatment responses of upper airway. 20 2.3.2 S.-J. Kim and K. B. Kim ephalometric Definition and Measurements C of Upper Airway (Fig. 2.4) Here, we suggest a simplified clinical guideline of cephalometric analysis of UA as briefly explained in the Chap. 1 (Fig. 1.1). Despite the limited availability and wide variation of cephalometric measurements as mentioned above, the repetitive measuring procedure is recommended to the beginners in order to raise their appreciation ability to grasp the upper airway and craniofacial complex at a glance. Firstly, draw a line of palatal plane passing through ANS and PNS (line 1, upper limit of oropharynx). Then, draw two parallel lines to the line 1 passing through the tip of soft palate (line 2, lower limit of velopharynx) and tip of epiglottis (line 3, lower limit of oropharynx). And draw another line from PNS to the midpoint of Sella-Basion line (line 4, upper limit of nasopharynx). With these four lines, we can Fig. 2.4 Definition of anatomical limits of upper airway (left) and the landmarks and measurements of pharyngeal airway (right) in the lateral cephalogram. Se Sella, Ba Basion, AD1 Adenoid point 1 (adenoid tissue on the PNS-Ba line), AD2 Adenoid point 2 (adenoid tissue on the midpoint between sella and basion to PNS line), Go Gonion; ANS, anterior nasal spine, PNS Posterior nasal spine, Me Menton, Rg Retrognathion, Sp Tip of soft palate, Eb Base of epiglottic fold, Hy hyoidale (the most antero-superior point on the body of the hyoid bone), (1) PNS-AD2, upper nasopharyngeal airway space (width of airway along PNS-R line); (2) PNS-AD1, lower nasopharyngeal airway space (width of airway along PNS-Ba line); (3) SPAS, superior posterior airway space (width of airway behind soft palate along parallel line to palatal plane); (4) MAS, middle airway space (width of airway along parallel line to palatal plane through Sp); (5) IAS, inferior airway space (width of airway along parallel line to palatal plane through epiglottis tip); (6) SPL, soft palatal length (PNS-P); (7) MPT, maximum palatal thickness measured on-line perpendicular to PNS-P; (8) SPI, soft palate inclination (angle between ANS-PNS line and PNS-P line); (9) TGL, tongue length (Eb to tip of tongue); (10) TGH, tongue height (longest distance from Eb-tongue tip line to tongue dorsum); (11) MPH, the shortest distance from mandibular plane (Me-Go) to hyoidale; dashed triangle means the hyoid triangle 2 Orthodontic Evaluation and Diagnostic Workflow for OSA Patients 21 easily assort different pharyngeal areas and their adjacent structures. Figure 2.4 shows how to measure the pharyngeal airway, soft palate, tongue, and hyoid using minimum landmarks and reference lines. 2.4 CBCT Volumetric Analysis The use of cone-beam computed tomography (CBCT) for the airway analysis may be criticized for additional radiation exposure and high cost when thinking of the similar limitations to lateral cephalometric analysis mentioned above. Besides, CBCT has different anatomic boundaries of UA, which are more difficult to be defined, and little is known about the CBCT parameters, which can predict treatment success and their normative values. However, the addition of the third dimension offers an advantage over traditional plain films, as follows (Fig. 2.5): Fig. 2.5 CBCT image analysis of upper airway. (a) Lateral volumetric image; (b) lateral sectional image encoded by color-scale; (c) frontal volumetric image; (d) frontal sectional image showing relative transverse dimension of nasal cavity, maxillary base, and mandibular base 22 S.-J. Kim and K. B. Kim 1. Three-dimensional airway shape as well as total volume can be assessed. 2. Site-specific information can be obtained through manual or automatic airway segmentation. 3. The most useful thing is that the minimal cross-sectional area (cm2), which is more pathophysiologically relevant than volume, can be identified in relation to surrounding structures. 4. The presence and the origin of nasal cavity obstruction can be assessed in relation to transverse maxillary constriction. 5. Change of airway length can be noted with the displacement of hyoid bone in space. 6. Computational fluid dynamics (CFD) can be utilized to assess theoretical airway flow and resistance but still has limitation to represent real functional problem. 2.5 Functional Evaluation on Respiration and Sleep 2.5.1 Mouth Breathing Mouth breathers do not usually realize that they breathe through the mouth especially while sleeping. Instead, they complain about the symptoms like dry mouth, gingival or periodontal inflammation, halitosis, snoring, chronic fatigue, and excessive sleepiness. Mouth breathing is more often observed in children, when the parents should look for the signs of slow growth rate, irritability, crying episode at night, and behavioral and cognitive problems. Mouth breathing influences underdeveloped midface, transverse maxillary deficiency, high and deep palatal vault, and long face in children. In earlier stage of growth under the age 5–6, mouth breathing with extended head/neck postures, and lowered jaw/tongue postures, may disturb the mechanism of cranial base flexion, resulting in dolichocephalic pattern with narrow facial width and innate mandibular retrusion (Fig. 2.6), creating a vicious cycle between mouth breathing and craniofacial abnormalities. On the other hand, skeletal Class III pattern with strong potential of mandibular growth can be deteriorated by serious mouth breathing passing through the pubertal growth peak period, as seen in Fig. 2.7. The underlying causes of mouth breathing are adenotonsillar hypertrophy, nasal obstruction, and severe skeletal discrepancy to structurally prevent lip sealing, and poor habits. The dental office is in the best position to diagnose mouth breathing and to lead the therapy by addressing each of these causative areas. From orthodontic point of view, mouth breathing can be classified into three types: obstructive type, structural type, and habitual type (Fig. 2.8). For the obstructive type of mouth breathers, nasal obstruction caused from allergic rhinitis, septal deviation, or nasal polyps should be treated firstly, and hypertrophic adenoids and tonsils need to be removed if indicated. We need to refer the obstructive type of mouth breather to the ENT doctors. For the structural type of mouth breathers who have severely retruded chin, excessive lower facial height, constricted maxillary 2 Orthodontic Evaluation and Diagnostic Workflow for OSA Patients 23 Fig. 2.6 Two cephalograms taken at different head and neck postures at the same day. Extended head posture (left) was changed into extended neck posture (right) to secure the upper airway obstructed by hypertrophic adenoids and tonsils Fig. 2.7 An example of skeletal Class III patients with serious mouth breathing. Skeletal Class III with strong mandible, hyperdivergent pattern, anterior openbite, and maxillary arch constriction were deteriorated after pubertal growth, and the pharyngeal constriction was not improved even with mandibular overgrowth, creating the vicious cycle 24 S.-J. Kim and K. B. Kim Treatment priority of Mouth breathing 1. Obstructive MB → Adenotonsillectomy 2. Structural MB → Growth modification 3. Habitual MB → Myofunctional theraphy Fig. 2.8 Three types of mouth breathing requiring different treatment approach arch and nasal cavity, skeletal modification treatment should be considered to change the structure to allow natural lip sealing and nasal breathing. For the habitual or residual mouth breathers, active myofunctional therapy should be recommended after checking the presence of tongue tie or tori. 2.5.2 Bruxism Bruxism is divided into awake bruxism and sleep bruxism. Sleep bruxism is a sleep-­ related movement disorder that is associated with SDB. Sleep bruxism is defined as “repetitive jaw muscle activity characterized by clenching or grinding of the teeth and/or bracing or thrusting of the mandible.” [6] Diagnostic criteria for sleep bruxism include regular or frequent tooth grinding sounds along with the presence of either abnormal tooth wear or transient morning jaw muscle pain or fatigue, and/or jaw locking on awakening. Sleep bruxism peaks during childhood and decreases with age. The pathophysiology of sleep bruxism is not well understood and multifactorial in nature. Current literature [7] suggests that it is regulated centrally (pathophysiological and psychosocial factors) and not peripherally (morphological factors), implying that no effective treatment that cures or stops it permanently. Definitive diagnosis of sleep bruxism is only achieved using electrophysiological tools, and management is usually directed toward tooth protection using occlusal splint, reduction of muscle activity using botox (BTX-A) injection, or pain relief. 2 Orthodontic Evaluation and Diagnostic Workflow for OSA Patients 25 Mandibular advancement device (MAD) has shown better results to decrease muscle activity than occlusal splint [8, 9], and could be a promising alternative treatment for sleep bruxism with OSA. 2.6 iagnostic Workflow for SDB Patients D in Orthodontic Clinic In summary of diagnostic overview from orthodontic perspectives, we suggest a diagram of diagnostic workflow for the referred or suspected SDB (snoring and OSA) patients whom we will see in the orthodontic clinic (Fig. 2.9). Beforehand, we emphasize two points related to diagnosis and treatment planning of SDB patients. First, orthodontic professionals should be qualified for the differential diagnosis and selective application of orthodontic modalities toward active cooperation with sleep specialist for SDB patients. Second, orthodontists should be more alert to evaluate the respiratory function whenever we evaluate and treat the regular orthodontic patients. Fig. 2.9 Diagram of diagnostic workflow for OSA patients who are referred from sleep specialists or who visit orthodontic clinic 26 S.-J. Kim and K. B. Kim For the referred OSA patients from sleep specialists: • Based on the review of chief complaint, medical history, and PSG report, orthodontists should grasp the severity of OSA and medically related factors. • Clinical examination and imaging analysis can assist orthodontists in assessment of main obstruction site in relation to craniofacial contributing factors. • Functional examination on the stomatognathic system (bruxism and temporomandibular disorders) as well as sleep function can support the phenotypic causes of OSA. • Differential diagnosis on the craniofacial anatomical phenotyping of OSA will guide orthodontists to determine whether to intervene with orthodontic modalities or not. • Titration PSG is recommended to confirm the treatment efficacy after orthodontic intervention. Unless it is successful, more aggressive surgical intervention can be considered, or PAP may be highly recommended again. For the orthodontic patients who might have risks of OSA: • Medical history and sleep questionnaire need to be enquired to the orthodontic patient population for recognizing the present SDB or the risk of SDB after orthodontic treatment. History taking should not be ignored even in the patients who have no craniofacial discrepancy, keeping the possibility of nonanatomical phenotype of OSA in mind. • Clinical examination and imaging analysis allow orthodontists to identify the anatomical risk factors of OSA. • Functional examination not only on the stomatognathic system but also on sleep and respiration can be implemented in the case of suspected SDB patients, by using HSTs or by referring to the sleep laboratory for the PSG. According to the AASM, no treatment should be rendered without definite diagnosis through PSG. • Following the collaborative differential diagnosis of the SDB, orthodontists can play a primary role in the treatment of craniofacial phenotypes, or should be able to refer the patients with nonanatomical phenotype to the sleep specialists for collaboration. If the patient has no SDB at present but has some risks of SDB after orthodontic treatment with obesity, orthodontists need to change the orthodontic treatment planning to minimize airway dimensional constriction. • Titration HST or PSG is helpful for the evaluation of treatment efficacy. If the primary orthodontic intervention is not effective, referral to the sleep specialists should be suggested. 2 Orthodontic Evaluation and Diagnostic Workflow for OSA Patients 27 Clinical Pearls in Orthodontic Diagnosis of OSA • Orthodontic diagnostic workflow for the referred OSA patients includes consultation about the chief complaint, history taking, clinical examinations, radiographic image analysis, and functional analysis. • Whenever we evaluate and diagnose the regular orthodontic patients, dimensional and functional analyses on the upper airway and respiration are important to recognize the hidden SDB or to prevent SDB after orthodontic treatment. • Clinical examination comprises physical, behavioral, facial, and intraoral evaluations. • Two- or three-dimensional radiographic image analysis provides the upper airway morphometry and the main obstruction site in relation to the craniofacial skeletal pattern and para-pharyngeal soft tissues at a glance. • Respiration or sleep-related functional evaluation such as mouth breathing, tongue posture, bruxism, and home sleep test, if available, should be integrated with the morphometric evaluation. • Final diagnosis of SDB should be reserved for the sleep specialists, however dentists play a crucial role in evaluating patients with SDB for the suitability of various orthodontic modalities. References 1. Netzer NC, Stoohs RA, Netzer CM, et al. Using the Berlin Questionnaire to identify patients at risk for the sleep apnea syndrome. Ann Intern Med. 1999;131(7):485–91. 2. Sutherland K, Lee RW, Cistulli PA. Obesity and craniofacial structure as risk factors for obstructive sleep apnoea: impact of ethnicity. Respirology. 2012;17(2):213–22. 3. Hong S-N, Won T-B, Kim J-W, et al. Upper airway evaluation in patients with obstructive sleep apnea. Sleep Med Res. 2016;7(1):1–9. 4. Major MP, Flores-Mir C, Major PW. Assessment of lateral cephalometric diagnosis of adenoid hypertrophy and posterior upper airway obstruction: a systematic review. Am J Orthod Dentofacial Orthop. 2006;130(6):700–8. 5. Duan H, Xia L, He W, et al. Accuracy of lateral cephalogram for diagnosis of adenoid hypertrophy and posterior upper airway obstruction: a meta-analysis. Int J Pediatr Otorhinolaryngol. 2019;119:1–9. 6. Lobbezoo F, Ahlberg J, Raphael K, et al. International consensus on the assessment of bruxism: report of a work in progress. J Oral Rehabil. 2018;45(11):837–44. 7. Lobbezoo F, Naeije M. Bruxism is mainly regulated centrally, not peripherally. J Oral Rehabil. 2001;28(12):1085–91. 8. Landry-Schönbeck A, de Grandmont P, Rompré PH, Lavigne GJ. Effect of an adjustable mandibular advancement appliance on sleep bruxism: a crossover sleep laboratory study. Int J Prosthodont. 2009;22(3):251–9. 9. Singh PK, Alvi HA, Singh BP, et al. Evaluation of various treatment modalities in sleep bruxism. J Prosthet Dent. 2015;114(3):426–31. 3 Therapeutic Pathway for Orthodontic Int ervention Su-Jung Kim, Patricia Pigato Schneider, and Ki Beom Kim Contents 3.1 Phenotype-Based Orthodontic Intervention 3.2 Orthodontic Treatment Protocol for SDB Adults 3.3 Life-Long Craniofacial Management for OSA Patients 3.4 Case Examples of Various OSA Phenotypes References 3.1 29 31 32 33 39 Phenotype-Based Orthodontic Intervention Traditionally, treatment modalities for OSA adult patients represented a stepwise phased approach starting from conservative therapy such as behavioral or lifestyle modification, continuous positive airway pressure (CPAP), and oral appliance. According to the American Academy of Sleep Medicine, CPAP is the first-line treatment of OSA, and should be recommended as a primary option to all patients regardless of its severity. When the conservative therapy was not effective to improve S.-J. Kim Department of Orthodontics, Kyung Hee University School of Dentistry, Seoul, South Korea e-mail: ksj113@khu.ac.kr P. P. Schneider Department of Orthodontics, School of Dentistry at Araraquara, UNESP Sao Paulo State University, Araraquara, Sao Paulo, Brazil K. B. Kim (*) Department of Orthodontics, Center for Advanced Dental Education, Saint Louis University, Saint Louis, MO, USA e-mail: kibeom.kim@health.slu.edu © Springer Nature Switzerland AG 2020 S.-J. Kim, K. B. Kim (eds.), Orthodontics in Obstructive Sleep Apnea Patients, https://doi.org/10.1007/978-3-030-24413-2_3 29 30 S.-J. Kim et al. patients’ symptoms, phase I soft-tissue surgery was suggested by sleep surgeon. Phase II skeletal surgery was considered only when the preceded procedures did not work based on the postoperative PSG evaluation. Decrease in the patient’s motivation going through these stepwise procedures was the biggest problem, so nowadays one-step target approach has been pursued. In this context, defining phenotypic causes of OSA is necessary to determine whether craniofacial orthopedic or surgical intervention is required to the OSA patient as a primary option. Eckert et al. [1] suggested a novel concept of phenotypic classification aiming at development of novel therapeutic approaches that target underlying mechanisms in individual patients with OSA. Key pathophysiologic causes include four factors: (1) anatomically collapsible UA (high critical closing pressure); (2) inadequate responsiveness of UA dilator muscles; (3) low respiratory arousal threshold; and (4) oversensitive ventilatory control system (Fig. 3.1). The PALM scale can be determined from data captured during PSG identifying (P)crit, (A)rousal threshold, (L)oop gain, and (M)uscle responsiveness, each of which represents a different etiology of OSA and responds to differing therapies. The PALM scale is expected to aid both physicians and dentists in directing optimal treatment and in avoiding much of the trial-and-error approach. For personalized phenotyping, orthodontists should integrate all information of respiratory parameters, race, gender, age, and hormone status with 3D imaging morphometric data. Despite multifactorial pathophysiology, pharyngeal collapsibility and its related anatomy is fundamental determinant of orthodontic intervention. When a patient shows moderately collapsible UA, which is primarily anatomically driven without nonanatomic vulnerabilities, anatomic intervention is likely to be effective. (4) Oversensitive ventilator control system (3) Premature arousal (low threshold) (1) Anatomically collapsible UA (2) Poor pharyngeal dilator muscle responsiveness Fig. 3.1 A novel concept of phenotypic classification aiming at therapeutic approaches that target underlying mechanisms of OSA. Key pathophysiologic causes include four factors: (1) anatomically collapsible UA (high critical closing pressure); (2) inadequate responsiveness of UA dilator muscles; (3) low respiratory arousal threshold; and (4) oversensitive ventilatory control system. Orthodontic intervention will be effective primarily to the patients with craniofacial anatomical phenotype (1) 3 Therapeutic Pathway for Orthodontic Intervention 31 Especially in case of craniofacial skeletal discrepancy constricting the UA dimension, craniofacial modification treatment need to be primarily recommended to the patient. 3.2 Orthodontic Treatment Protocol for SDB Adults (Fig. 3.2) Once orthodontic intervention is selected for the OSA patient with craniofacial anatomic vulnerability, proper orthodontic treatment modality is decided depending on the craniofacial target. The most frequent anatomic issue is retruded small mandible followed by narrow nasomaxillary complex. When a patient has retruded small mandible narrowing pharyngeal airway, mandibular advancement is required for upward and forward positioning of tongue base and hyoid to open the airway [2, 3]. If the patient shows normal or protruded maxilla with normal or flat occlusal plane, mandibular advancement surgery would be a good option [4, 5]. Advancing genioplasty can be combined as needed [6, 7]. Alternatively, temporary mandibular advancement device (MAD) can be recommended especially to elderly patients who tend to refuse skeletal surgery [8, 9]. If a patient has retruded maxilla and steep occlusal plane with retruded mandible, in contrast, maxillomandibular advancement (MMA) surgery is highly required for significant enlargement of overall pharyngeal airway [10, 11]. Orthodontic protocol for SDB adults PAP Intolerance Refer Nonanatomic vulnerability Anatomic vulnerability Refer Para-pharyngeal soft tissue abnormality Craniofacial deformity Narrow nasomaxilla Retruded mandible Protruded Maxilla / Flat OP MARPE DOME Lateral pharyngeal walls MAD Mandibular surgery Retruded Maxilla / steep OP MMA Fig. 3.2 A therapeutic pathway of phenotype-based precision protocol for the adult OSA patients from orthodontic point of view. PAP Continuous positive airway pressure, MARPE Microimplant-­ assisted rapid palatal expansion, DOME Distraction osteogenesis maxillary expansion, MAD Mandibular advancement device, MMA Maxillomandibular advancement, OP Occlusal plane 32 S.-J. Kim et al. When a patient has transverse skeletal discrepancy with maxillary constriction and nasal obstruction, nasomaxillary skeletal expansion is necessary for the enlargement of the nasal cavity to decrease nasal resistance affecting the pharyngeal collapsibility, as well as the oral cavity to guide the tongue-up posture [12, 13]. According to the age, different expansion modality is considered. Conventional rapid palatal expansion (RPE) can be applied for the growing children under 15 years old [14, 15], microimplants-assisted RPE (MARPE) is preferred for the post-adolescents or young adults [16, 17], and surgical removal of tissue resistance using corticotomy can be combined with MARPE for adult patients [18]. Sagittal skeletal correction is concomitantly performed depending on the pattern of sagittal discrepancy. Although a severe OSA patient belongs to the nonanatomic phenotype with lateral pharyngeal wall dysfunction, the patient can be referred to the orthodontists for consideration of salvage MMA surgery in case of PAP intolerance or surgical failure [19, 20]. When a referred OSA patient has no definite craniofacial risk factors, or when a patient who wants orthodontic treatment has OSA symptoms, orthodontists should be able to refer to the sleep specialists for collaboration. 3.3 ife-Long Craniofacial Management for L OSA Patients (Fig. 3.3) In the past, craniofacial management could be accomplished by two-phase approach through the growth modification treatment before the peak height velocity (the first phase), and by the surgical reconstruction after the end of residual Fig. 3.3 Life-long sequence of craniofacial modification by orthodontic, orthopedic, and surgical interventions for the expansion of upper airway as well as skeletal framework 3 Therapeutic Pathway for Orthodontic Intervention 33 growth (the second phase). Nowadays, on the other hand, the craniofacial orthopedic treatment can be successfully achieved during the postadolescence up to young adult, which was the waiting period between the two phases, with the development of bone-anchored craniofacial orthopedic appliances. Recently, moreover, earlier orthopedic intervention during the preschool ages has been issued to secure nasal breathing as early as possible especially in the child with nasal obstruction [21]. Guilleminault et al. [22] found that abnormal breathing during sleep is related to intrinsic and extrinsic factors, which are present early in life and may induce progressive narrowing of the collapsible airway. Oronasal dysfunction should be recognized from infants for appropriate intervention to prevent OSA and secondary negative impacts on orofacial growth [23]. Further, surgically assisted orthodontic treatment for the osseopharyngeal reconstruction is being applied to no less than the elderly patients for functional improvement of sleep and respiration as well as esthetic improvement. In this context, life-long craniofacial management has been established to extend the roles of orthodontic specialists. More specific guideline of timely target intervention throughout the life will be explained in the following chapters. 3.4 Case Examples of Various OSA Phenotypes Case 1: Anatomical Phenotype with Maxillary Constriction, Hypertrophic Adenoid, and Obesity A 29-year-old Caucasian woman sought for orthodontic treatment with a chief complaint of anterior open bite. She was obese with heavy submental fat tissues. Patient reported a medical history of OSA and chronic mouth breathing. Major findings noted during the extraoral clinical examination were concave facial profile with protruded chin and substantial lip incompetence, which contributes to the inability to produce full lip closure. The intraoral examination showed a bilateral Class III malocclusion, anterior open bite, severe maxillary transverse insufficiency with deep and high palatal vault, bilateral posterior cross bite, and mild-to-moderate tooth size arch length discrepancy (Fig. 3.4). Radiographically, skeletal Class III with strong mandible and hyperdivergent vertical pattern was seen. Due to the adenoid hypertrophy and elongated soft palate, nasopharyngeal and velopharyngeal airway spaces were narrowed, probably contributing to the OSA (Fig. 3.5). Conclusively, this young adult patient belongs to the anatomical phenotype of OSA with maxillary transverse constriction, hypertrophic adenoid and thick soft palate, and obesity. At the same time with weight loss and adenoidectomy, orthodontic treatment is recommended to expand the maxilla and intrude the posterior teeth for at last maintaining the facial height with closing the anterior openbite. Surgical maxillary expansion using microimplant-assisted device (distraction osteogenesis maxillary expansion) can be rendered. 34 S.-J. Kim et al. Fig. 3.4 Facial and intraoral photographs of an OSA patient with anatomical risk factors Measurement Value Norm SNA (º) 77.7 82.0 SNB (º) 79.7 80.9 ANB (º) -6.7 1.6 Wits (mm) -7.2 -1.0 SN–GoGn(º) 35.3 32.9 FMA (º) 31.7 25.9 U1–NA (mm) 7.9 4.3 U1–SN (º) 110.9 103.1 L1–NB (mm) 3.2 4.0 L1–GoGn (º) 83.6 93.0 Fig. 3.5 Lateral cephalogram and the skeletal measurements of the OSA patient. In spite of prognathic mandible, naso- and velo-pharyngeal airway spaces were narrowed in relation to hypertrophic adenoid and thick soft palate Case 2: Anatomical Phenotype with Mandibular Retrusion, Bi-Arch Constriction, and Obesity A 28-year-old Caucasian male patient was presented with chief complaint of OSA. Patient’s physical characteristics noted were midrange obesity with BMI 3 Therapeutic Pathway for Orthodontic Intervention 35 Fig. 3.6 Facial and intraoral photographs of an OSA patient with craniofacial anatomical risk factors Measurement Value Norm SNA (º) 85.0 82.0 SNB (º) 78.4 80.9 ANB (º) 6.5 1.6 Wits (mm) 3.3 -1.0 SN–GoGn(º) 34.3 32.9 FMA (º) 31.2 25.9 U1–NA (mm) 3.3 4.3 U1–SN (º) 98.2 103.1 L1–NB (mm) 7.4 4.0 L1–GoGn (º) 97.1 93.0 Fig. 3.7 Lateral cephalogram and the skeletal measurements of the OSA patient. Retrognathic mandible, low hyoid position, and hypertrophic tonsils seem to be related to the constricted oropharynx and OSA score of 28.7. The patient was seen by an ENT specialist who found enlarged tonsils and categorized the patient as Class III Mallampati score. Furthermore, the patient’s overall AHI was 12.5 and the lowest oxygen saturation level was 91% from the baseline. Facial evaluation showed convex profile with retruded small chin, short throat length, and obtuse cervicomental angle, and thick neck circumference 36 S.-J. Kim et al. (Fig. 3.6). Intra-arch evaluation showed compensated Class I relationship with severe tooth size arch length discrepancy in the mandibular arch, maxillary and mandibular arch constriction, and missing upper right first premolar and lower right first molar (Fig. 3.6). The lateral cephalogram presented skeletal Class II with retrognathic mandible, inferiorly displaced hyoid, and hypertrophic palatine tonsils, which might contribute to the narrow oropharyngeal space. (Fig. 3.7) In summary, this young adult male patient belonged to the typical craniofacial anatomical phenotype of OSA; therefore, maxillomandibular advancement surgery was considered for the primary treatment option. Concurrently, mandibular midline distraction osteogenesis with three-piece maxillary expansion was planned and tonsillectomy was recommended. Case 3: Complex Phenotype with Craniofacial Deformity and Nonanatomical Risk Factors A 55-year-old Caucasian female patient was referred to the orthodontic department. She had been diagnosed with severe OSA associated with poor neuromuscular responsiveness and obesity, and tried to use CPAP as a sleep physician’s prescription. Her AHI reading using CPAP was at 0.8, which indicates good control of OSA with CPAP. However, the patient was interested in seeking surgical options to treat her OSA permanently. She had a history of osteoarthritis and reported occasional soreness of the TMJ. She showed a convex profile with retruded chin and heavy submental fat pads. Minimal gingival display (Fig. 3.8) was observed. Intraoral exam showed that she has Class I molar and Class II canine relationship, maxillary arch constriction with deep and high palatal arch, insufficient posterior buccal Fig. 3.8 Facial and intraoral photographs of an elderly OSA patient with complex phenotype 3 Therapeutic Pathway for Orthodontic Intervention 37 Fig. 3.9 Panorama and lateral cephalogram of the OSA patient. Retrognathic mandible, hyperdivergent vertical pattern, low hyoid position, long and flabby soft palate, and long and narrow oropharynx are observed Fig. 3.10 CBCT volumetric and sectional images representing transversely as well as sagittally collapsed minimal cross-sectional area in the oropharyngeal level overjet, moderate-to-severe tooth size arch length discrepancy, and anterior open bite. Analysis of cephalogram represented excessive lower facial height with high mandibular plane angle, Class II skeletal pattern with retrognathic mandible, inferiorly positioned hyoid bone, and constricted oropharyngeal airway (Fig. 3.9). CBCT volumetric images confirmed the transversely as well as sagittally collapsed pharyngeal airway. In terms of her TMJ, both the panoramic radiograph and CBCT imaging reveal “flattened-look” of both condyles and a noticeable osteophyte “beaking” on her right condyle (Fig. 3.10). For this elderly OSA patient with complex phenotype, CPAP is recommended for the first-line treatment and mandibular advancement device can be an alternative. Since she had TMD signs and symptoms and asked permanent osseopharyngeal reconstruction, MMA with advancing genioplasty was considered with extraction of the both mandibular first premolars to maximize mandibular advancement. 38 S.-J. Kim et al. Case 4: Adolescent OSA Patient with Obese Phenotype A 13-year-old Black male patient was referred from pediatric ENT department. His medical records showed a BMI of 35 indicating high obesity, attention deficit hyperactivity disorder (ADHD), ongoing nocturnal enuresis, and OSA. Tonsillectomy and adenoidectomy had been performed, but OSA symptoms persisted. The patient was using a full-face CPAP, but despite the efforts, the AHI reading was less than optimal at 1.6. Furthermore, due to his persistent loud snoring, his CPAP pressure gage was increased from 6 to 14 cm H2O pressure to 11 cm H2O. Major orthodontic findings upon facial examination were protruded lips with well-developed chin, but almost flat cervicomental angle due to the heavy submental Fig. 3.11 Facial and intraoral photographs of an adolescent OSA patient with obese phenotype Fig. 3.12 Panorama and lateral cephalogram of the OSA patient. Skeletal Class III with prognathic mandible and normal vertical divergency is found, which implies no craniofacial anatomical phenotype of OSA 3 Therapeutic Pathway for Orthodontic Intervention 39 fat pad. Intraoral examination showed bilateral Angle Class I malocclusion, generalized spacing, normal maxillary arch, and wide mandibular arches creating bilateral posterior cross-bite in relation to the thrusting habit of large tongue (Fig. 3.11). Tongue thrusting habit might be caused to secure the patency of narrow pharyngeal airway. Lateral cephalogram represented skeletal Class III tendency with strong mandible, which might not contribute to the OSA directly, but long, thick, and retroclined soft palate made the velopharyngeal airway space narrow in association with low tongue posture, low hyoid position, and heavy fat tissues (Fig. 3.12). For this patient, growth modification treatment would not be available to treat OSA. Weight loss should be strongly recommended considering the bariatric surgery to avoid the CPAP treatment. Clinical Pearls in Therapeutic Decision of Orthodontic Intervention • Phenotype-based orthodontic intervention: Not all SDB patients will respond to the orthodontic intervention successfully even though they have been referred from sleep clinic to the orthodontists. OSA phenotyping is important to decide whether to orthodontically intervene or not, and craniofacial phenotyping is necessary to decide how to treat the SDB patients orthodontically. • Orthodontic precision protocol: An updated therapeutic pathway for the SDB patients is established from orthodontic point of view on the basis of target-based selective application of various orthodontic modalities. • Life-long craniofacial management: Orthodontic, orthopedic, and surgical craniofacial managements for the SDB patients with craniofacial skeletal risk factors can be timely applied throughout the life. References 1. 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Large maxillomandibular advancements for obstructive sleep apnea: an operative technique evolved over 30 years. J Craniomaxillofac Surg. 2015;43(7):1113–8. 40 S.-J. Kim et al. 7. Singhal D, Hsu SSP, Lin CH, et al. Trapezoid mortised genioplasty: a further refinement of mortised genioplasty. Laryngoscope. 2013;123(10):2578–82. 8. Marklund M, Verbraecken J, Randerath W. Non-CPAP therapies in obstructive sleep apnoea: mandibular advancement device therapy. Eur Respir J. 2012;39(5):1241–7. 9. Canadian Agency for Drugs and Technologies in Health (CADTH). Oral appliances for treatment of snoring and obstructive sleep apnea: a review of clinical effectiveness. CADTH Technol Overv. 2010;(1):e0107. 10. Fairburn SC, Waite PD, Vilos G, et al. Three-dimensional changes in upper airways of patients with obstructive sleep apnea following maxillomandibular advancement. J Oral Maxillofac Surg. 2007;65(1):6–12. 11. Kim T, Kim H-H, Hong S, et al. Change in the upper airway of patients with obstructive sleep apnea syndrome using computational fluid dynamics analysis: conventional maxillomandibular advancement versus modified maxillomandibular advancement with anterior segmental setback osteotomy. J Craniofac Surg. 2015;26(8):e765–70. 12. Baratieri C, Alves M Jr, de Souza MMG, et al. Does rapid maxillary expansion have long-­ term effects on airway dimensions and breathing? Am J Orthod Dentofacial Orthop. 2011;140(2):146–56. 13. Gordon JM, Rosenblatt M, Witmans M, et al. Rapid palatal expansion effects on nasal airway dimensions as measured by acoustic rhinometry: a systematic review. Angle Orthod. 2009;79(5):1000–7. 14. Haas A. Long-term posttreatment evaluation of rapid palatal expansion. Angle Orthod. 1980;50(3):189–217. 15. Ballanti F, Lione R, Baccetti T, et al. Treatment and posttreatment skeletal effects of rapid maxillary expansion investigated with low-dose computed tomography in growing subjects. Am J Orthod Dentofacial Orthop. 2010;138(3):311–7. 16. Carlson C, Sung J, McComb RW, et al. Microimplant-assisted rapid palatal expansion appliance to orthopedically correct transverse maxillary deficiency in an adult. Am J Orthod Dentofacial Orthop. 2016;149(5):716–28. 17. Brunetto DP, Sant’Anna EF, Machado AW, Moon W. Non-surgical treatment of transverse deficiency in adults using Microimplant-assisted Rapid Palatal Expansion (MARPE). Dental Press J Orthod. 2017;22(1):110–25. 18. Jang H-I, Kim S-C, Chae J-M, et al. Relationship between maturation indices and morphology of the midpalatal suture obtained using cone-beam computed tomography images. Korean J Orthod. 2016;46(6):345–55. 19. Mehra P, Downie M, Pita MC, Wolford LM. Pharyngeal airway space changes after counterclockwise rotation of the maxillomandibular complex. Am J Orthod Dentofacial Orthop. 2001;120(2):154–9. 20. Liu SY, Awad M, Riley R, Capasso R. The Role of the Revised Stanford Protocol in Today’s Precision Medicine. 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Sleep Med Rev. 2018;40:203–14. 4 Craniofacial Growth Modification for OSA Children Su-Jung Kim Contents 4.1 ajor Differences in Pediatric OSA M 4.1.1 Different Diagnostic Criteria 4.1.2 Different Subjective Symptoms 4.2 Early Intervention: Why? 4.3 Early Intervention: How? 4.3.1 Phenotype-Based Patient Selection 4.3.2 Target-Based Treatment Selection 4.3.3 Timely Target Application of Appliances 4.4 Cases References 4.1 41 41 42 43 43 43 44 45 50 58 Major Differences in Pediatric OSA Pediatric OSA differs from adult OSA in both diagnosis and management. Pediatric OSA is more difficult to be correctly diagnosed than adult OSA due to less available PSG finding, less sensitive HST finding, less reliable questionnaire, limited information from lateral cephalometry, and uncommon application of CBCT to the growing patients. Different diagnostic criteria and subjective symptoms need to be contemplated. 4.1.1 Different Diagnostic Criteria Overnight PSG is the gold standard for diagnosing OSA in children as in adults. However, AHI as specific criterion of OSA severity is provided differently from that S.-J. Kim (*) Department of Orthodontics, Kyung Hee University School of Dentistry, Seoul, South Korea e-mail: ksj113@khu.ac.kr © Springer Nature Switzerland AG 2020 S.-J. Kim, K. B. Kim (eds.), Orthodontics in Obstructive Sleep Apnea Patients, https://doi.org/10.1007/978-3-030-24413-2_4 41 42 S.-J. Kim in adults (Table 4.1). Poor availability of pediatric sleep laboratories for overnight PSG has prompted a search for alternative diagnostic screening tools including a combination of history and physical examination findings. 4.1.2 Different Subjective Symptoms Pediatric OSA can be undiagnosed, since the symptoms are different from adults. The main differences between children and adults are compared in Table 4.2 [1]. The chief complaint of OSA children is snoring or labored breathing during sleep, and mouth breathing is frequently observed during the examination time. The most common cause is known to be adenotonsillar hypertrophy, followed by nasal obstruction. Obesity contributes to the pediatric OSA less than the adult OSA. Since sleep fragmentation, witnessed apnea, arousal, and excessive daytime sleepiness, which are very common in adult OSA, are not usually detected, it is hard to recognize OSA in children. Cardiovascular and neurovascular comorbidities rarely occur, instead, irreversible disturbances such as psychosocial and behavioral problems, neurocognitive deficits, learning disorder, physical and facial growth disturbances make serious concerns. The behavioral problems represent various aspects across ages: attention deficit hyperactivity disorder (ADHD)-like behavior in the early childhood, in contrast, depression in adolescence [2]. As a simple screening instrument of pediatric OSA, BEARS (B, Bedtime; E, Excessive daytime sleepiness; A, night Awakenings; R, Regularity and duration of sleep; S, Snoring) has been used in primary care settings [3]. Table 4.1 Different objective criteria of OSA severity between children and adults OSA severity Mild Moderate Severe Children 1 ≤ AHI < 5 5 ≤ AHI < 10 10 ≤ AHI Adults 5≤ AHI < 15 15 ≤ AHI < 30 30 ≤ AHI Table 4.2 Different subjective signs and symptoms of OSA between children and adults Signs and symptoms Chief complaint Snoring Mouth breathing Adenotonsillar hypertrophy Nasal obstruction Sleep architecture Clinical arousal Sequelae Obesity Children Snoring, difficulty breathing Continuous Common Common Common Preserved Uncommon Behavioral problems, Neurocognitive deficits, Growth disturbance Less common Adults Daytime sleepiness Intermittent with pause Less common Uncommon Less common Fragmented Common Cardiovascular, neurovasucular comorbidities Common 4 Craniofacial Growth Modification for OSA Children 4.2 43 Early Intervention: Why? Since the signs of OSA may be slow to show up in the PSG, OSA symptoms may be unrecognized for years, worsening the craniofacial growth. Dentists are responsible for detecting and interrupting oronasal dysfunction in the primary care to prevent abnormal craniofacial growth and progression to SDB. Although abnormal craniofacial growth may have different genetically or environmentally induced etiologies, craniofacial growth evaluation and prediction should be performed for the children with the oronasal dysfunction or SDB. Craniofacial growth modification treatment need to be actively provided aiming at restoration of nasal breathing during sleep as well as during wakefulness. The objectives of early intervention for growth modification are as follows: (1) to prevent irreversible disorders like somatic growth disturbance, neurocognitive and learning disorders, psychosocial and behavioral deficits; (2) to interrupt progression to adult OSA that has more serious symptoms and comorbidities; and (3) to prevent or correct the craniofacial deformity to break the vicious cycle. The ultimate goals of early growth modification treatment include UA development to secure nasal breathing, as well as unlocking or stimulating mandibular growth in skeletal Class II, and developing nasomaxillary complex in skeletal Class III patients (Fig. 4.1). 4.3 Early Intervention: How? 4.3.1 Phenotype-Based Patient Selection 4.3.1.1 Medical Phenotyping The main phenotypic causes responsible for the pediatric OSA are anatomical obstruction, inflammation, and neuromotor dysfunction (Fig. 4.2). Neuromotor dysfunction is an intrinsic factor of UA, which is related to the modified pharyngeal muscle tone and reflex response during sleep. This phenotype may not respond well to the physical treatment, so CPAP is firstly recommended, although there are concerns that long-term use of the CPAP can lead to midfacial flattening [4]. Fig. 4.1 Goals of early orthodontic intervention in terms of upper airway development in addition to skeletal development. Establishment of nasal breathing should be commonly included as well as unlocking the mandibular growth in skeletal Class II or developing nasomaxillary complex in skeletal Class III patients 44 S.-J. Kim Fig. 4.2 Phenotype-based treatment approach for OSA children. Three major phenotypic causes of OSA children are anatomical obstruction, inflammation, and neuromotor dysfunction. Growth modification treatment can be effective in the OSA children with craniofacial anatomic vulnerability as a main phenotpyic cause On the other hand, extrinsic factors of UA, including fat deposit, hypertrophic tissues associated with inflammation, and craniofacial deformity, can be controlled more favorably. Adenotonsillectomy is known to be the first-line treatment of pediatric OSA, since adenotonsillar hypertrophy has been regarded as the most frequent etiologic factor in children. According to current systematic review and meta-­analysis [5], however, only 59.8% of children were cured of OSA (based on AHI <1) following surgery. Persistent mouth breathing or residual OSA may exist after surgery, and SDB symptoms tend to recur in early puberty, probably when other phenotypic cause is involved. Accordingly, phenotyping should be preceded. If the patient’s main problem is nasal obstruction, nasal surgery, nasal expansion, or medication is required. If the patient is obese showing favorable craniofacial pattern and lymphoid tissues, weight loss with exercise is highly recommended, while craniofacial modification treatment should be actively applied to the patient with craniofacial skeletal risk factor. Combined approach should be considered to the patient with complicated phentoype. 4.3.1.2 Craniofacial Phenotyping Once a patient is categorized into the group with craniofacial risk factors for the pathogenesis of OSA, the main target should be differentiated for the target-based treatment selection, which is called craniofacial phenotyping. Major facial traits, which should be attentively observed in the clinical examination, are retruded chin, depressed midface, and long and narrow midface. These can be confirmed by cephalometric and CBCT analyses. 4.3.2 Target-Based Treatment Selection (Fig. 4.3) Children with craniofacial abnormalities have a high risk of residual OSA even after adenotonsillectomy [6]. Based on the craniofacial phenotyping, three major approaches for growth modification can be taken into consideration: unlocking or stimulating mandibular growth for skeletal Class II patient with retruded small chin; nasomaxillary 4 Craniofacial Growth Modification for OSA Children 45 Orthodontic protocol for SDB children 1st line treatment Adenotonsillectomy Residual OSA Soft tissue problem Skeletal deformity Orthognathic surgery Vertical Mx.excess Constricted Maxilla Vertical pull device Rapid palatal expander Growth modification Retruded Maxilla Face mask Retruded Mandible Functional habits Obesity Myofunctional therapy Weight loss Functional appliance Fig. 4.3 A protocol of target-based treatment selection for SDB children (Updated from Ahn et al. J Oral Maxillofac Surg 73:1827-1841, 2015) protraction for skeletal Class III patient with retruded midface; and nasomaxillary skeletal expansion for the patient with constriction of maxilla and nasal cavity. In addition, control of vertical maxillary excess should be carefully reflected at the same time. Also, myofunctional therapy needs to be combined with skeletal modification for ultimate functional improvement, particularly in patients having abnormal functional habits. In spite of concerns about the residual growth, orthognathic surgery or distraction osteogenesis is another option to fix cases like severe skeletal Class II hyperdivergent pattern especially with syndromic deformity or medically refractory OSA (Fig. 4.3) [7, 8] 4.3.3 Timely Target Application of Appliances 4.3.3.1 F unctional Appliance to Unlock or Stimulate Mandibular Growth Recent systematic review [9] summarized that functional appliances had positive effect to open the oropharyngeal airway by forward growth of the mandible and subsequent adaptive advancement of tongue base, hyoid, and soft palate, through the action of genioglossus and palatoglossus muscles (Fig. 4.4). On the other hand, little improvement of nasopharynx and hypopharynx was observed. Most of the studies found no significant restraint in the maxillary growth, which might cause negative effects on nasopharyngeal airway [10]. Interestingly, oropharyngeal enlargement in three dimension (mediolaterally greater than anteroposteriorly) 46 S.-J. Kim Functional appliance Mandibular advancement + hyoid, tongue, soft palate advancement Open Oropharyngeal airway (minimum cross-sectional increment) O2 saturation Recovery of suppressed Growth hormone Greater growth potential to treat OSA & Face Fig. 4.4 Mechanism of functional appliance to open the upper airway with mandibular advancement. Along with the advancement of tongue base, hyoid, and soft palate, oropharyngeal airway including minimum cross-sectional (MCS) area can be enlarged permanently in case of true mandibular forward growth. Increment of MCS may recover the suppressed growth hormone by increasing oxygen saturation in the blood, contributing to the treatment of OSA with better face includes the minimum cross-sectional increment [11], leading to the recovery of suppressed growth hormone with increasing oxygen saturation level. Increased growth hormone may release the suppressed physical and mandibular growth, improving face and airway better (Fig. 4.4). There was no significant difference in this effect among various types of functional appliances. Four clinical implications underlie this mechanism of action. First, the positive effect of functional appliance can be expected only in the patients with good growth potential (before pubertal peak), good growth pattern (parallel or counterclockwise growth pattern), and good compliance (Fig. 4.5). Not all patients respond to the appliance. Second, there is no scientific evidence to affirm that functional appliance is effective to treat the OSA even with successful growth response [12]. Further research focusing on the respiratory functional improvement is anticipated. Third, even though true mandibular growth cannot be favorably expected with functional appliance in an OSA child, the appliance is worthy of releasing daytime symptoms by holding the mandible forward to open the airway during sleep. Lastly, in spite of controversy on the effect of functional appliance stimulating the mandibular growth, unlocking the mandibular growth itself should be the target of early intervention. 4.3.3.2 Facemask for Nasomaxillary Protraction Maxillary protraction has the potential to improve respiratory efficiency in children with maxillary deficiency. Recent systematic reviews [13] demonstrated that skeletal modification effects induced by facemask include stimulation of forward maxillary growth with direct counterclockwise rotation of the palatal plane, and indirect clockwise rotation of the mandible. These orthopedic effects lead to the enlargement of nasopharyngeal airway by forward and downward displacement of PNS, 4 Craniofacial Growth Modification for OSA Children Initial (10y 7m) Bionator 47 12y 6m Fig. 4.5 Comparison of lateral cephalographic images and intraoral photographs between initial and 2-year after treatment using bionator in a skeletal Class II OSA patient. Oropharyngeal enlargement is noticed with mandibular advancement in spite of big tonsils and submental fat pads a b Facemask Maxillary advancement (PNS) + soft palate advancement Open nasopharyngeal airway Improvement of Snoring Fig. 4.6 Mechanism of face mask to open the upper airway with maxillary protraction. Stimulation of forward and downward maxillary growth can open the nasopharyngeal airway by pulling soft palate forward along with the displacement of posterior maxilla. In spite of clockwise mandibular rotation with maxillary protraction, oropharyngeal and hypopharyngeal airway spaces showed no significant decrease 48 S.-J. Kim Initial (7y 7m) After maxillary protraction (8y 2m) Fig. 4.7 Comparison of lateral cephalographic images before and after maxillary protraction using face mask. Yellow arrows indicate the nasopharyngeal space increase with improved maxillomandibular relationship advancing the soft palate concurrently to increase velopharyngeal space (Fig. 4.6). Although the growth of adenoid tissues can be progressed until the age of 10–12, stimulation of suppressed maxillary growth by active treatment no later than 9–10 years old, when circum-maxillary sutures are still responsive, may ensure nasopharyngeal airway enlargement likely in a child showing normal growth (Fig. 4.7). On the other hand, oropharyngeal airway space showed no significant change after maxillary protraction as a result of combined effects between the increased tongue space by maxillary advancement and the possible backward tongue position due to the clockwise mandibular rotation. 4.3.3.3 Rapid Palatal Expander for Nasomaxillary Expansion Nasal airflow is a critical factor for nasal breathing and pharyngeal collapsibility. Intranasal surgery or nasopharyngeal surgery can be considered to improve nasal airflow when there are hypertrophic or inflammatory tissues. However, constricted nasal cavity with narrow nasal floor and maxillary constriction requires structural expansion. Rapid maxillary expansion (RPE) is widely applied to the patients with nasal obstruction aiming at increasing narrow nasal cavity volume in sagittal and vertical as well as transverse dimensions, which is attributable to forward and downward displacement of nasomaxillary complex as well as maxillary basal expansion. Increased airflow will decrease pharyngeal collapsibility by lowering its critical closing pressure. Simultaneously, expanded oral cavity allows forward and upward repositioning of the tongue to open the oropharyngeal airway indirectly (Fig. 4.8). 4 Craniofacial Growth Modification for OSA Children 49 Rapid palatal expander (RPE) Open Nasal cavity Increase oral cavity (tongue-up) + soft palate, pharyngeal wall stretch Decrease nasal resistance Decrease pharyngeal collapsibility Nasal breathing Fig. 4.8 Mechanism of rapid palatal expansion to open the upper airway. Through the midpalatal sutural expansion, nasal cavity and nasal floor are enlarged to increase nasal airflow, and palatal vault and oral cavity are expanded to move the tongue forward and upward. In addition, nasomaxillary complex is displaced forward and downward, inducing three-dimensional skeletal expansion. As a result, decreased nasal resistance and indirectly decreased pharyngeal collapsibility will treat the OSA patients with nasal obstruction The success of nasomaxillary skeletal expansion depends on the stage of midpalatal sutural obliteration. Therefore, removable expander can be effectively applied to separate the suture in the preschool ages, while fixed type rigid expander should be used in the school ages before 15–16 years old [14]. Microimplants or adjunctive surgery is required to overcome the strong tissue resistance to sutural opening from postadolescence to adults. Nonetheless, microimplant-assisted rapid palatal expander (MARPE) is more favorable for both skeletal and airway expansion even in the growing children than RPE: greater maxillary base expansion and more parallel sutural separation from anterior to posterior palate with less dentoalveolar effect are expected in MARPE. 4.3.3.4 Headgear for interruption of Vertical Maxillary Excess Traditionally, headgear treatment is intended to restrict the forward growth of the maxilla; thus, some clinicians are concerned about negative impact on the airway. Despite controversies, there was a study reporting the nasopharyngeal enlargement without oropharyngeal and hypopharyngeal changes after cervical pull headgear treatment [15]. Combined activator-headgear therapy represented the potential to increase oropharyngeal airway dimensions in skeletal class II patient with vertical maxillary excess and mandibular deficiency [16]. Given that the aggravating impact of vertical maxillary in excess results in clockwise mandibular rotation with narrowing oropharyngeal airway, inhibition of vertical maxillary excess using vertical pull headgear may be superior to interrupting increased facial divergency and increasing oropharyngeal patency (Fig. 4.9). Not only posterior vertical maxillary excess with anterior openbite, but also severe gummy smile indicating anterior vertical maxillary excess needs to be carefully managed. Simultaneously with vertical maxillary inhibition, compensatory extrusion of lower 50 S.-J. Kim Headgear + splint + Myofunctional therapy Inhibition of vertical maxillary excess → CCW mandibular rotation Decrease of facial height Open oropharyngeal airway Nasal breathing with lip sealing Fig. 4.9 Mechanism of splint-supported vertical pull headgear to open the upper airway posterior teeth should be prevented by lingual arch or more active lower posterior intrusion using microimplants. Myofunctional therapy should be actively combined for the successful outcome. 4.3.3.5 Limitation: Rule Out the Poor Responders Early growth modification treatment does not work successfully in every patient. Differential diagnosis should be based on the growth potential, growth pattern, growth stage, and patient’s attitude. Here comes the orthodontist’s role to determine whether to and when to intervene in individual patient. • It is not easy to predict mandibular response to stimulate its growth, since it is controlled by genetics more than environments. At least, the concept of “unlocking the mandibular growth and airway development” should be a determinant of early intervention. • Hyperdivergent vertical growth pattern with diverged mandibular shape can be a sign of poor mandibular growth and poor treatment response. Note that most of early interventions tend to increase vertical dimension with the correction of sagittal malocclusion. This may have an adverse influence on the oropharyngeal airway development. • Due to the lack of untreated controls and prospective long-term data in the literatures, and due to high interindividual variability, controversy on both skeletal and airway effects still exists. 4.4 Cases Case 1: A Snoring Child with Locked Small Mandible A 10-year-old girl was visited orthodontic clinic with the chief complaint of upper incisor protrusion. She had no subjective nasal obstruction and no history of ENT treatment, but had snoring and structural mouth breathing due to the severely proclined upper incisors and trapped lip (Fig. 4.10). She showed a convex profile with retruded chin and short lower facial height. Dental Class II molar and canine 4 Craniofacial Growth Modification for OSA Children 51 Fig. 4.10 Initial facial and intraoral photographs. Convex profile with retruded chin and short lower facial height was observed and severely proclined upper incisors with trapped lip were noticeable. Maxillary arch constriction and anterior deep overbite was locking the mandibular growth relationship with large overjet and deep overbite, and constricted maxillary dental arch was observed. The lateral cephalometry indicated skeletal Class II malocclusion with retruded small mandible and hypodivergent vertical pattern with favorable mandibular shape for the pubertal growth (Fig. 4.11). Hypertrophic adenoid was seen in relation to the narrow nasopharyngeal and velopharyngeal airway. Her skeletal age was SMI 5–6, implying the imminent pubertal growth spurt (Fig. 4.11). In terms of the growth prediction and growth stage, her mandible was evaluated to have favorable growth potential to improve the face and the airway. The important thing here is that any environmental factors locking the mandibular growth should be checked and removed before the growth peak. The most frequent locking factors that we have to always assure are maxillary arch constriction and anterior deep overbite. Since she had both of these factors, immediate intervention to allow the catch-up growth of the mandible by expansion of maxillary arch and intrusion of anterior teeth was required. Although this case seems to have sagittal mandibular deficiency, transverse and vertical control of maxilla needs to be firstly considered rather than stimulating the mandibular growth only using functional appliance. As a result of timely orthopedic treatment passing through the pubertal growth peak, Class I molar and canine relationship with proper overjet and overbite could be accomplished with facial improvement (Fig. 4.12). The mandible underwent catch-up growth to attain skeletal and dental Class I relationship with increased pharyngeal dimension (Fig. 4.13). Spontaneous atrophy of adenoid tissue was observed along with the correction of structural mouth breathing. 52 S.-J. Kim 8 5 2 6 10 PHV 100 1 9 4 mm/year 7 3 10mm Onset 50 A MATURITY INDICATIONS End 11 5 10 15 age(year) 20 Fig. 4.11 Lateral cephalogram (left) and hand wrist X-ray (middle). SMI chart (right) indicated the growth stage of SMI 5–6, which means imminent pubertal growth peak Case 2: An OSA Child with Gummy Smile and Nasal Obstruction A 10-year-old girl was referred from ENT department for the evaluation of facial deformation. She was complaining about gummy smile and retruded chin and suffering from nasal obstruction with allergic rhinitis and subsequent mouth breathing. In the clinical examination (Fig. 4.14), she showed convex profile with retruded chin, and long face with excessive lower facial height. She had narrow and deviated nasal bridge with narrow nares. Severe gummy smile in spite of anterior openbite was present in the mouth. Maxillary arch was constricted with deep narrow palatal 4 Craniofacial Growth Modification for OSA Children 53 Initial 10y 2m Final 13y 10m Fig. 4.12 Comparison of facial and intraoral photographs between initial (above) and at the end of treatment (below) Initial 10y 2m Final 13y 10m Fig. 4.13 Comparison of lateral cephalograms between initial (left) and at the end of treatment (right) representing favorable mandibular catch-up growth to attain skeletal and dental Class I relationship with increased pharyngeal dimension vault. As a result of home sleep test (Apnealink Plus™), AHI was 2 indicating mild OSA in relation to the nasal obstruction. She was diagnosed as OSA accompanied by craniofacial risk factors like skeletal Class II with retrognathic mandible, hyperdivergent vertical pattern with anterior vertical maxillary excess, and transverse discrepancy. Treatment objectives for the first phase growth modification treatment included maxillary skeletal expansion and 54 S.-J. Kim Fig. 4.14 Initial facial and intraoral photographs. Convex profile with retruded chin and excessive lower facial height was observed and excessive gum exposure during smile was noticeable. Maxillary arch constriction and anterior openbite was related to nasal obstruction-oriented mouth breathing Fig. 4.15 Intraoral photographs before (left) and after (right) MARPE and occlusal topographic image showing parallel midpalatal sutural split inhibition of vertical maxillary excess to redirect the mandibular growth and ultimately to open the upper airway (particularly nasal cavity). Maxillary skeletal expansion was performed using microimplant-assisted rapid palatal expander (MARPE) in order to maximize skeletal effect involving nasal cavity and to minimize mandibular clockwise rotation as a response to the maxillary expansion (Figs. 4.15 and 4.16). Subsequently, high-pull headgear was applied with incisor intrusion arch to attain the flattening of upper occlusal plane and forward mandibular autorotation, as seen in the cephalometric superimposition (Fig. 4.17). As a result of 4 Craniofacial Growth Modification for OSA Children 55 Fig. 4.16 Application of MARPE for the transverse expansion as noted on the CBCT superimposition (left) and high-pull headgear with incisor intrusion arch for the vertical control to apply the directional force system (right) as confirmed by the lateral cephalogram Fig. 4.17 Superimposition of lateral cephalograms between initial (black) and at the end of treatment (red). With successful vertical control, forward mandibular growth can be induced to develop pharyngeal airway space 56 S.-J. Kim Acoustic Rhinometry matched with CBCT sectional images Area (Cm2) 1.0 0.1 -6.0 Right Left -2.0 2.0 6.0 10.0 Distance (Cm) Area (Cm2) 10.0 10.0 Right 1.0 0.1 -6.0 Left -2.0 2.0 6.0 10.0 Distance (Cm) Fig. 4.18 Acoustic rhinometric finding matched with CBCT sectional images in the nasal cavity area. Intranasal area was increased especially in the left side where serious nasal obstruction existed as a result of maxillary expansion Table 4.3 Comparison of home-sleep test between initial and at the end of growth modification treatment. AHI and ODI dropped to 0, and the lowest oxygen saturation increased up to 94% with decreased flow limitation Apnea link plus™ AHI LSaO2 (%) ODI Flow limitation, Sn Initial 2 92 1.2 29 After first treatment 0 94 0 17 acoustic rhinometry matched with the CBCT sectional images in the nasal cavity area (Fig. 4.18), the intranasal area was remarkably increased especially in the left side where almost complete obstruction existed as seen in the CBCT section, following the nasal cavity expansion with MARPE. Functional evaluation using a home-sleep test (ApneaLink Plus™) found the release of AHI with increased oxygen saturation after treatment (Table 4.3). She could have esthetic improvement with the correction of gummy smile and favorable facial profile in addition to respiratory improvement through the timely target intervention (Fig. 4.19). 4 Craniofacial Growth Modification for OSA Children 57 Fig. 4.19 Comparison of facial photographs between before (above) and after treatment (below). Gummy smile was remarkably corrected, and chin profile was improved by the aid of enhanced nasal breathing Clinical Pearls of Airway-Friendly Craniofacial Growth Modification • Timely Target approach: Based on the differential growth theory, intervention timing and appliance type should be decided according to the bony target and respiratory obstruction area. • Earlier intervention for nasal breathing: Early nasal expansion with MFT is important to prevent or interrupt the progression of SDB and facial deformation. • Bone-anchored appliances for greater airway expansion: Greater amount of skeletal modification and more favorable skeletal effects on the airway patency with microimplants or miniplates may extend the range of orthodontic and orthopedic application for SDB growing patients. • Interdisciplinary approach: We need to refer the patient with noncraniofacial or complicated phenotype to pediatric sleep specialists for intimate collaboration. 58 S.-J. Kim References 1. Li H-Y, Lee L-A. Sleep-disordered breathing in children. Chang Gung Med J. 2009;32(3):247–57. 2. Sinha D, Guilleminault C. Sleep disordered breathing in children. Indian J Med Res. 2010;131(2):311. 3. Owens JA, Dalzell V. Use of the ‘BEARS’sleep screening tool in a pediatric residents’ continuity clinic: a pilot study. Sleep Med. 2005;6(1):63–9. 4. Fauroux B, Lavis J-F, Nicot F, et al. Facial side effects during noninvasive positive pressure ventilation in children. Intensive Care Med. 2005;31(7):965–9. 5. Friedman M, Wilson M, Lin H-C, Chang H-W. Updated systematic review of tonsillectomy and adenoidectomy for treatment of pediatric obstructive sleep apnea/hypopnea syndrome. Otolaryngol Head Neck Surg. 2009;140(6):800–8. 6. Imanguli M, Ulualp SO. Risk factors for residual obstructive sleep apnea after adenotonsillectomy in children. Laryngoscope. 2016;126(11):2624–9. 7. Bell RB, Turvey TA. Skeletal advancement for the treatment of obstructive sleep apnea in children. Cleft Palate Craniofac J. 2001;38(2):147–54. 8. Ahn H-W, Lee B-S, Kim S-W, Kim S-J. Stability of modified maxillomandibular advancement surgery in a patient with preadolescent refractory obstructive sleep apnea. J Oral Maxillofac Surg. 2015;73(9):1827–41. 9. Xiang M, Hu B, Liu Y, Sun J, Song J. Changes in airway dimensions following functional appliances in growing patients with skeletal class II malocclusion: a systematic review and meta-analysis. Int J Pediatr Otorhinolaryngol. 2017;97:170–80. 10. Gordon JM, Rosenblatt M, Witmans M, et al. Rapid palatal expansion effects on nasal airway dimensions as measured by acoustic rhinometry: a systematic review. Angle Orthod. 2009;79(5):1000–7. 11. Li L, Liu H, Cheng H, et al. CBCT evaluation of the upper airway morphological changes in growing patients of class II division 1 malocclusion with mandibular retrusion using twin block appliance: a comparative research. PLoS One. 2014;9(4):e94378. 12. Carvalho FR, Lentini-Oliveira DA, Prado LB, et al. Oral appliances and functional orthopaedic appliances for obstructive sleep apnoea in children. Cochrane Database Syst Rev. 2016;(10) 13. Ming Y, Hu Y, Li Y, et al. Effects of maxillary protraction appliances on airway dimensions in growing class III maxillary retrognathic patients: a systematic review and meta-analysis. Int J Pediatr Otorhinolaryngol. 2018;105:138–45. 14. Baratieri C, Alves M Jr, de Souza MMG, et al. Does rapid maxillary expansion have long-­ term effects on airway dimensions and breathing? Am J Orthod Dentofacial Orthop. 2011;140(2):146–56. 15. Kirjavainen M, Kirjavainen T. Upper airway dimensions in Class II malocclusion: effects of headgear treatment. Angle Orthod. 2007;77(6):1046–53. 16. Hänggi MP, Teuscher UM, Roos M, Peltomäki TA. Long-term changes in pharyngeal airway dimensions following activator-headgear and fixed appliance treatment. Eur J Orthod. 2008;30(6):598–605. 5 Craniofacial Orthopedics for Postadolescent OSA Patients Su-Jung Kim Contents 5.1 Bone-Anchored Nasomaxillary Protraction 5.2 Bone-Anchored Nasomaxillary Expansion 5.3 Bone-Anchored Inhibition of Vertical Maxillary Excess References 5.1 59 61 62 64 Bone-Anchored Nasomaxillary Protraction Maxillary protraction using the intraoral device-supported facemask has been known to be effective less than 9 years due to the strong resistance of circum-­ maxillary sutures afterward [1–4]. However, miniplates inserted on the zygomatic bone area can serve the skeletal anchorage substituting the intraoral devices to directly deliver strong orthopedic forces to the nasomaxillary complex (NMC). Elastics can be applied from the maxillary miniplates to the facemask or to the mandibular miniplates or microimplants (Fig. 5.1). Surgical insertion of miniplates gives benefits in the permanent dentition with no tooth germs in the bone and with appropriate bone density; thus, 12 years old or elder teenagers would be good indications (Fig. 5.2). Here, some clinical points need to be noted: (1) With preceded maxillary skeletal expansion, maxillary protraction can be favorably accomplished in the late teens [5, 6]; (2) With long-­term wearing of this appliance until late teens, camouflage treatment of skeletal Class III malocclusion may be successfully accomplished even in the cleft lip and palate patients (Fig. 5.3), depending on the mandibular growth potentials [7, 8]; (3) Mandibular growth itself S.-J. Kim (*) Department of Orthodontics, Kyung Hee University School of Dentistry, Seoul, South Korea e-mail: ksj113@khu.ac.kr © Springer Nature Switzerland AG 2020 S.-J. Kim, K. B. Kim (eds.), Orthodontics in Obstructive Sleep Apnea Patients, https://doi.org/10.1007/978-3-030-24413-2_5 59 60 S.-J. Kim Fig. 5.1 Intraoral photographs showing bone-to-bone Class III elastics to protract the maxilla using the mandible as an anchorage ApneaLink Plus TM Initial After Bone-anchored expansion & protraction AHI 3 0 LSaO2 82% 91% Fig. 5.2 Infrazygomatic miniplate-anchored facemask application to the skeletal Class III OSA patient (11 years 6 months, female) with maxillary deficiency and mandibular excess; 2 years after miniplates-anchored maxillary protraction, AHI dropped from 3 to 0 and the lowest oxygen saturation increased up to 91% with the development of maxilla, nasal cavity, and nasopharynx. AHI Apnea–hypopnea index, LSaO2 Lowest oxygen saturation cannot be inhibited, while the maxillary development is attained. Thus, the need of mandibular surgery should be initially informed to the patients. Even if the patient will require two jaw-surgery afterward, the amount of maxillary surgical advancement can be minimal with no need of considering maxillary distraction osteogenesis due to the surgical limit; (4) In the meanwhile, enhanced nasal breathing and loss of snoring will improve the patient’s life quality [9]. 5 Craniofacial Orthopedics for Postadolescent OSA Patients Initial (9y 7m) SPAS T0 T1 T1-T0 MAS T0 T1 Final (15y 0m) IAS T1-T0 T0 61 T1 VAL T1-T0 T0 T1 PNS-ad1 T1-T0 T0 T1 T1-T0 T0 PNS-ad2 T1 T1-T0 8.66 17.03 8.37 11.58 14.95 3.37 11.06 15.54 4.48 60.72 72.98 12.26 19.32 29.93 10.61 19.02 27.15 8.13 Fig. 5.3 Cephalometric comparison between before and after miniplate-anchored facemask application in a skeletal Class III patient with unilateral cleft and lip palate. After long-term maxillary protraction for 5 years, facial profile and occlusion improved with nasomaxillary development, and pharyngeal airway spaces were significantly enlarged at all levels. SPAS Superior posterior airway space, MAS Middle airway space, IAS Inferior airway space, VAL Vertical airway length, PNS-ad1 Lower nasopharyngeal space, PNS-ad2 Upper nasopharyngeal space. T0 Before treatment, T1 After maxillary protraction 5.2 Bone-Anchored Nasomaxillary Expansion As mentioned earlier in Chap. 4, bone-anchored maxillary expansion is superior to the conventional RPE for the OSA patients: (1) greater amount of skeletal expansion up to the young adult age group; (2) greater ratio of nasal cavity expansion relative to the maxillary dental expansion; (3) less dentoalveolar effect inducing mandibular clockwise rotation which should be prevented in OSA patients; (4) more parallel sutural separation from anterior to posterior palate affecting both anterior nasal cavity and the posterior pharyngeal soft tissues (Fig. 5.4) [10–12]. Especially in skeletal Class III with midface deficiency, forward and downward displacement of NMC concurrently with maxillary expansion can be expected, which is favorable for the improvement of facial profile and occlusal relationship as well as three-dimensional upper airway enlargement [13, 14]. Moreover, responsive clockwise rotation of the mandible may improve sagittal facial profile unless the 62 S.-J. Kim Acoustic Rhinometry matched with CBCT sectional image Area (Cm2) 10.0 10.0 Left Right 1.0 0.1 -6.0 -2.0 2.0 6.0 Distance (cm) 10.0 Left Right 1.0 0.1 -6.0 -2.0 2.0 6.0 Distance (cm) 10.0 Fig. 5.4 Acoustic rhinometric finding (below) matched with CBCT sectional images (middle) before and after bone-anchored maxillary expansion. After MARPE, parallel midpalatal separation was obtained and nasal floor and nasal cavity was increased with maxillary base expansion (above) as supported by increased intranasal area by acoustic rhinometry patient has no hyperdivergent vertical pattern. On the other hand, this overall skeletal effect should be carefully reflected in the treatment of skeletal Class II patients with maxillary constriction. For a postadolescent OSA patient with Class II hyperdivergent pattern and maxillary constriction, MARPE can be applied, if needed, followed by active intrusion of posterior teeth or total arch intrusion using microimplants. 5.3 Bone-Anchored Inhibition of Vertical Maxillary Excess NMC undergoes differential growth in its width, length, and height. The growth of NMC ends the earliest in width, next in depth, and the latest in height. Vertical maxillary growth rates peak during adolescence at the same time as stature, and may continue throughout the postadolescent period especially in patients with chronic mouth breathing. Therefore, the inhibition of vertical maxillary growth or intrusion 5 Craniofacial Orthopedics for Postadolescent OSA Patients 63 Fig. 5.5 Intrusion of upper and lower posterior teeth using TADs for reducing vertical dimension by mandibular autorotation. Posterior vertical maxillary excess and compensatory mandibular posterior extrusion existed creating anterior openbite with two-step upper and lower occlusal plane (above). With TADs-assisted intrusion (yellow arrows indicating intrusion force vectors from microimplants), proper overbite was obtained (below) with counterclockwise mandibular rotation enlarging the hypopharyngeal airway dimension as seen on the cephalometric superimposition (right) of posterior teeth is actively required during the postadolescence to interrupt or correct the excessive facial height in OSA patients. For the targeted vertical inhibition anticipating the counterclockwise autorotation of the mandible with no concern of sagittal maxillary restriction, temporary anchorage devices (TADs) can be a useful tool substituting the headgear. At the same time, intrusion of lower posterior teeth using the TADs is required for greater effect of mandibular autorotation. TADsassisted bimaxillary total arch intrusion needs to be considered to maximize the airway opening effect (Fig. 5.5) [15]. Clinical Pearls of Craniofacial Orthopedics for Postadolescent OSA Patients • Significance: Active craniofacial orthopedic treatment using TADs-assisted appliances can contribute to improving the respiratory function targeting the NMC in postadolescence. • Nasomaxillary protraction: Miniplates-assisted facemask or bone–bone Class III elastics will protract the NMC increasing the nasopharyngeal airway patency in Class III OSA or snoring patients regardless of residual mandibular growth. • Nasomaxillary expansion: Microimplants-assisted rapid palatal expansion will enlarge the nasal cavity/floor and oral cavity to directly increase nasal airflow and indirectly decrease the pharyngeal collapsibility in postadolescent or young adult OSA patients with structural nasal obstruction. • Inhibition of vertical maxillary excess (VME): Microimplants-assisted active intrusion of maxillary and mandibular posterior teeth will interrupt the clockwise mandibular rotation at least to prevent the pharyngeal narrowing with VME, and further to improve oropharyngeal patency with mandibular closure. 64 S.-J. Kim References 1. Campbell PM. The dilemma of Class III treatment: early or late. Angle Orthod. 1983;53(3):175–91. 2. McNamara JA Jr. An orthopedic approach to the treatment of Class III malocclusion in young patients. J Clin Orthod. 1987;21(9):598–608. 3. Kircelli BH, Pektaş ZÖ, Uçkan S. Orthopedic protraction with skeletal anchorage in a patient with maxillary hypoplasia and hypodontia. Angle Orthod. 2006;76(1):156–63. 4. De C, Hugo J, et al. Orthopedic traction of the maxilla with miniplates: a new perspective for treatment of midface deficiency. J Oral Maxillofac Surg. 2009;67(10):2123–9. 5. Vaughn GA, et al. The effects of maxillary protraction therapy with or without rapid palatal expansion: a prospective, randomized clinical trial. Am J Orthod Dentofacial Orthop. 2005;128(3):299–309. 6. da Silva Filho OG, Magro AC, Capelozza Filho L. Early treatment of the Class III malocclusion with rapid maxillary expansion and maxillary protraction. Am J Orthod Dentofacial Orthop. 1998;113(2):196–203. 7. Baek S-H, Kim K-W, Choi J-Y. New treatment modality for maxillary hypoplasia in cleft patients: protraction facemask with miniplate anchorage. Angle Orthod. 2010;80(4):783–91. 8. Yatabe M, et al. Bone-anchored maxillary protraction therapy in patients with unilateral complete cleft lip and palate: 3-dimensional assessment of maxillary effects. Am J Orthod Dentofacial Orthop. 2017;152(3):327–35. 9. Nguyen T, et al. Effect of Class III bone anchor treatment on airway. Angle Orthod. 2014;85(4):591–6. 10. Lin L, et al. Tooth-borne vs bone-borne rapid maxillary expanders in late adolescence. Angle Orthod. 2014;85(2):253–62. 11. MacGinnis M, et al. The effects of micro-implant assisted rapid palatal expansion (MARPE) on the nasomaxillary complex—a finite element method (FEM) analysis. Prog Orthod. 2014;15(1):52. 12. Choi S-H, et al. Nonsurgical miniscrew-assisted rapid maxillary expansion results in acceptable stability in young adults. Angle Orthod. 2016;86(5):713–20. 13. Pangrazio-Kulbersh V, et al. Cone beam computed tomography evaluation of changes in the naso-maxillary complex associated with two types of maxillary expanders. Angle Orthod. 2011;82(3):448–57. 14. Hershey HG, Stewart BL, Warren DW. Changes in nasal airway resistance associated with rapid maxillary expansion. Am J Orthod Dentofacial Orthop. 1976;69(3):274–84. 15. Buschang PH, Carrillo R, Rossouw PE. Orthopedic correction of growing hyperdivergent, retrognathic patients with miniscrew implants. J Oral Maxillofac Surg. 2011;69(3):754–62. 6 Surgical Maxillary Expansion for OSA Adults with Nasal Obstruction Hyo-Won Ahn and Su-Jung Kim Contents 6.1 Mechanism to Open Upper Airway 6.2 Phenotype-Based Patient Selection 6.3 Differential Effects Among Three Procedures 6.4 Distraction Osteogenesis Maxillary Expansion (DOME) 6.5 Multipiece Maxillary Segmental Osteotomy (SO) 6.6 Cases References 6.1 65 66 66 67 69 70 78 Mechanism to Open Upper Airway Transverse maxillary deficiency is characterized by constricted maxilla and high palatal vault, which can lead to increased nasal airflow resistance and displacement of tongue posteriorly. When combined with nasal disease such as nasal septum deviation, or large inferior turbinate, transverse maxillary constriction would increase the prevalence of nasal obstruction. For these adult OSA patients, transverse maxillary expansion can be used to expand nasal cavity and nasal floor as well as palate to improve nasal airflow and to decrease pharyngeal collapsibility. The greatest volume increase is in the lower anterior nasal cavity in the nasal valve area, which is the region of greatest nasal airflow resistance. Many studies showed a triangular expansion pattern after surgically assisted rapid palatal expansion (SARPE), more expansion is found anteriorly than posteriorly. Deeb et al. [1] reported the expansion ratios of anterior, middle, and posterior segment were 59%, 26%, and 15% under the treatment of SARPE with bone-borne expanders using CT data. Zambon et al. [2] also supported the H.-W. Ahn · S.-J. Kim (*) Department of Orthodontics, Kyung Hee University School of Dentistry, Seoul, South Korea e-mail: hyowon@khu.ac.kr; ksj113@khu.ac.kr © Springer Nature Switzerland AG 2020 S.-J. Kim, K. B. Kim (eds.), Orthodontics in Obstructive Sleep Apnea Patients, https://doi.org/10.1007/978-3-030-24413-2_6 65 66 H.-W. Ahn and S.-J. Kim improved breathing after SARPE, which resulted from increased area of nasal cavity, increased expiratory and inspiratory nasal flow, and decreased airway resistance using the acoustic rhinometry and rhinomanometry. Surgical maxillary expansion also allows favorable tongue position for upward direction, which results in a greater posterior pharyngeal airway. In addition, expansion of the maxilla contributes tension on the muscles attached to the palate such as palatoglossus muscle and palatopharyngeus muscle, which reduce the collapsibility of the upper airway. Abdullatif et al. [3] reviewed the effectiveness of transverse maxillary expansion as treatment for OSA adult patients, and meta-analysis of 6 studies (36 patients) showed reduced AHI by 59.3% (from 24.3 to 9.9) and improved lowest oxygen saturation (from 84.3 to 86.9). Other systematic review (11 studies, 204 patients) also reported SARPE was effective for the increase in the nasal cavity volume, which persisted at least 63 months, although the effect on oropharyngeal volume is not clear [4]. Therefore, treatment modality of surgically maxillary expansion would be preferred for the OSA patients with nasal obstruction. 6.2 Phenotype-Based Patient Selection Craniofacial phenotype for treatment of surgically maxillary expansion is nasomaxillary transverse constriction with nasal obstruction. The high palatal vault with or without posterior crossbite is usually accompanied. Guilleminault et al. [5] reported a 10.9-fold increase in odds of OSA with this phenotype. For postadolescent or adult patients, skeletal maturity has occurred, increasing degree of bony interdigitation and ossification of the midpalatal and other circummaxillary sutures. The resistance area, which impedes the maxillary expansion are the piriform aperture pillars (anteriorly), the zygomatic buttress (laterally), the pterygoid junction (posteriorly), and the midpalatal suture (medially) [6]. Surgical maxillary expansion is indicated in these adult OSA patients to overcome the resistance of these ossified sutures, as an alternative to RPE or MARPE in children or adolescent stage. 6.3 Differential Effects Among Three Procedures The three surgical techniques—surgically assisted rapid palatal expansion (SARPE), distraction osteogenesis maxillary expansion (DOME) equal to the surgically assisted and microimplants-assisted RPE (SMARPE), and multipiece segmental osteotomy (SO)—can be considered for surgical maxillary expansion. The key point of surgical treatment planning is to determine the exact location and the amount of expansion where need to be widened. Briefly, when the patients require NMC expansion targeting nasal floor and nasal cavity, fan-type expansion (anterior > posterior) would be preferred using SARPE, or DOME procedure (Fig. 6.1). Both the procedures reduce the unwanted expansion of the upper level of the maxilla, such as nasal or zygomatic bone area. The DOME is performed by 1-stage surgery, which is less invasive compared with SARPE, and especially when combined with bone-borne type expander; DOME provides more parallel or convergent expansion pattern at the frontal view. 6 Surgical Maxillary Expansion for OSA Adults with Nasal Obstruction MARPE SMARPE(DOME) 67 Segmental osteotomy Fig. 6.1 Three types of maxillary skeletal expansion. According to the expander design with or without microimplants and adjunctive surgical procedure, various type of midpalatal split can be designed to serve the purpose. DOME Distraction osteogenesis maxillary expansion, SO segmental osteotomy, MARPE miniscrew assisted rapid palatal expansion, SMARPE surgically assisted MARPE, and one type of SMARPE is DOME The multipiece SO is primarily considered as part of the orthognathic surgery when MMA is planned. It is advantageous when posterior maxillary expansion is required. However, the transverse expansion with segmental surgery has a tendency of high immediate relapse due to the tension of palatal mucosa. The selection guideline of surgical maxillary expansion protocols is described in Table 6.1. 6.4 Distraction Osteogenesis Maxillary Expansion (DOME) By the advent of bone-borne type expander, which gives the stress closure to the center of maxilla and the increased preference of minimally invasive procedures, DOME was advocated recently by the sleep surgery team at Stanford university [9]. The original SARPE procedure is a two-stage surgery including bilateral osteotomy of lateral nasal wall to pterygomaxillary suture usually 5 mm above the apices of the teeth, with pterygomaxillary separation, midpalatal separation, and nasal septum release. Slight modification in surgical technique of SARPE existed mainly in the area of midpalatal and pterygomaxillary junction. The releasing of pterygomaxillary articulation allows more expansion on the posterior part; significant volumetric expansion of the nasopharynx and minimum cross-sectional area of the oropharynx [7], and better 68 H.-W. Ahn and S.-J. Kim Table 6.1 Selection guideline of surgical maxillary expansion protocols Comparators SARPE Interincisal separation >3–4 mm Expansion AnteroFan-type: Ant > Post Pattern posterior Frontal Tooth-borne device → Divergent expansion DOME Microimplant-­ assisted device → parallel/ convergent expansion 1-stage buccal approach No midpalatal osteotomy Surgical insult 2-stage surgery: Palatal → Buccal Anatomic limitation Vomer Pterygomaxillary junction Indication Adult OSA patients requiring NMC expansion targeting nasal cavity and floor Post-surgical stability Stable with distraction device Segmental osteotomy 2-pieces 3- or 4-pieces <3–4 mm 0 mm Posterior Parallel expansion expansion or Reverse maintaining fan-type: ICW Post > Ant All patterns can be designed. with Le-Fort I osteotomy Coronoid process Thickness of palatal mucosa Thickness of maxillary posterior wall When MMA surgery is planned Posterior maxillary expansion is needed Unstable, High immediate relapse SARPE Surgically assisted rapid palatal expansion, DOME Distraction osteogenesis maxillary expansion, NMC nasomaxillary complex, MMA Maxilla-mandibular advancement, ICW ­intercanine width periodontal support [8]. However, several studies reported that removing the resistance from the zygomatic buttress is sufficient for true orthopedic expansion [10, 11]. Based on this, the DOME is a one-stage procedure to release the resistance on the buccal zygomatic buttress and intermaxillary suture. The procedure of DOME is as follows: First, a bone-borne type expander is placed before surgery. The maxillary expander is custom-fabricated to fit the narrow palatal vault and fixed with 4–6 miniimplants placed along the midpalatal suture. Since the SARPE procedure involves midpalatal osteotomy, placement of the TADs in the midpalatal region would be limited. Second, two small incisions are made 1 cm above the maxillary mucogingival junction bilaterally, and maxillary osteotomy at Le Fort I level is performed toward piriform rim medially and the maxillary buttress laterally. A vertical incision is made between the maxillary incisor roots. A piezoelectric saw is used to deepen the primordial groove of the midpalatal suture. Thin straight osteotomes are used to wedge the midpalatal suture apart. A diastema is seen immediately as the suture opens. The expander is activated until a 1 mm diastema is seen in symmetric pattern with easy separation [9]. The activation procedure and postsurgical orthodontic treatment is similar with those of SARPE. Usually, the patients activate the expander 0.25–0.5 mm per day, and approximately 1 cm of expansion at the nasal floor is accomplished 6 Surgical Maxillary Expansion for OSA Adults with Nasal Obstruction 69 within a month. The expander is not removed during the consolidation period of 3 months. The separation pattern of midpalatal suture in both SARPE and DOME is triangular, greater expansion on the anterior than posterior palate. The type of expander depending on the area to be supported (bone-borne vs. tooth-borne) influences the expansion pattern. Nada et al. [12] reported that significant increase in the nasal airway volume was observed in both expander groups at 22-months retention (12.9% for bone-borne expanders, and 9.7% for tooth-borne expanders) after SARPE. Compared to SARPE using tooth-borne expanders (TB), DOME combined with bone-borne (BB) or tooth-bone-borne (TBB) type expander allows force exerted on the height of palatal vault and nasal floor, which provides more parallel or convergent expansion pattern at the frontal view. In addition, the DOME with BB has little side effects on tooth inclination (buccal tipping), alveolar bone bending, or periodontal support (root dehiscence or fenestration). Other systematic review also showed the BB devices led to greater skeletal expansion than TB appliances (standardized mean difference, 0.92 mm) although there was no significant difference in the amount of dental expansion [13]. In clinical aspects, DOME shows smaller expansion on the teeth level than SARPE under the similar amounts of skeletal expansion; thus, it would be suitable for the OSA adult patients whose main target is the nasal cavity and floor as well as palatal vault. 6.5 Multipiece Maxillary Segmental Osteotomy (SO) Generally, for transverse discrepancy less than 5 mm (typically 3–5 mm), multipiece Le Fort I osteotomy can be considered [14, 15], while the greater amount of total expansion would be appropriate for SARPE or DOME to reduce risk of avascular necrosis or relapse. The key limitation factor of bipartition during SO is how to manage the expanded width during the bone-healing period, under the tension of the mucosa of hard palate. The surgical technique consists of Le Fort I osteotomy combined with additional multipiece osteotomy. For 2-piece SO, the osteotomy is performed between central incisors, and for 3-piece or 4-piece osteotomy, between the canines and first premolars or between the lateral incisors and the canines. A parasagittal osteotomy was made along the palate behind the nasopalatine foramen. When the intercanine width is planned to be maintained, 3-piece osteotomy is appropriate to achieve the expansion only at the posterior area, however 4-piece osteotomy would be better to minimize surgical relapse on the midpalatal osteotomy area. As the number of segments increases, it becomes more difficult to move the segment precisely in three dimensions, which affects postoperative stability. A review study showed that the 2-piece SO provided greater dental stability in the anterior region of the maxilla than the 3-piece SO [16]. Yao et al. [14] compared SARPE with TB and multipiece SO using CBCT, and they showed hinge-like fashion with more expansion on anterior in SARPE, whereas greater posterior skeletal and dental expansion occurred in SO group. In addition, the multipiece SO produced more expansion skeletally than dentally compared with 70 H.-W. Ahn and S.-J. Kim the SARPE (posterior and anterior ratios of dental/skeletal expansion are 0.70 and 0.58 in SO, and 25.19 and 31.80 in SARPE, respectively). Seeberger et al. [17] also showed the expansion with SARPE was over the entire length of the maxilla from anterior to posterior, whereas the expansion of the 2-piece SO was reverse V-shaped targeting posterior part of the maxilla. Nonetheless, the stability issue should be taken into consideration when applying the multipiece SO especially in the OSA patients [18]. For greater expansion and greater skeletal stability, DOME may have the precedence over SO. 6.6 Cases Case 1: SARPE with Modified Genioplasty as an Alternative to MMA A 28-year-old male patient was referred from the ENT department with severe OSA. He had treatment history of acute sinusitis at 7-year old and had a nasal bone reduction surgery at 16-year old. He was suffering from EDS and loud snoring. The AHI score was 37.1 and the lowest SaO2 was 86%. The BMI index was 30.2, indicating obesity. Accordingly, the importance of weight control was stressed. The extraoral examination showed a convex profile with retruded chin and flat cervico-mental angle even with habitually protruded mandibular position with stubborn mouth breathing. Intraorally, constricted upper and lower arches, moderate crowding, and scissors bite on the left second molar area were observed (Fig. 6.2). The lateral cephalometry reported skeletal Class II with retruded mandible, hyperdivergent vertical pattern, labioversion of lower incisors, and narrow pharyngeal airway. CBCT analysis found that the nasal septum was deviated significantly, and narrow airway with deposition of fat tissue in the submental area (Fig. 6.3). He was diagnosed as skeletal Class II with narrow maxilla and the mandible, hyperdivergent vertical pattern, and dental Class II subdivision, with severe OSA. The maxillo-mandibular advancement (MMA) surgery was considered as the first treatment option; however, the patient strongly refused it. Instead of orthognathic surgery, surgically assisted maxillary expansion of the NMC and advancing genioplasty was planned. The fan-type expansion (greater ratio of anterior/posterior expansion) of the maxilla was required in this patient targeting the nasal cavity, SARPE procedure was preferred. Since two-step traditional surgical procedure was performed, TB type expander was used in this patient. The first surgery was done for the midpalatal suture split under the influence of local anesthesia (Fig. 6.4a). After two weeks of healing, TB type expander was delivered in the palate (Fig. 6.4b), and the second surgery including intermaxillary suture split and bilateral osteotomy from lateral nasal walls to pterygomaxillary sutures without complete pterygomaxillary separation was performed. Concurrently, advancing genioplasty surgery was combined to stretch the genioglossus muscles (Fig. 6.4c). A 1 mm of diastema was confirmed with 6-turm activation of the expander right after surgery in the operation room. The chin was advanced by 8 mm involving the 6 Surgical Maxillary Expansion for OSA Adults with Nasal Obstruction 71 Fig. 6.2 Facial and intraoral photographs of Case 1. Habitually protruded mandibular position and mouth opening was noted implying respiratory obstruction Fig. 6.3 Initial sagittal and frontal CBCT sectional images of Case 1. Narrow pharyngeal airway at levels is observed by the retruded chin, low hyoid, and accumulated fat in submental area. At frontal images, septum deviation, high palatal vault, and the narrow maxilla are seen 72 H.-W. Ahn and S.-J. Kim Fig. 6.4 Surgical procedures of Case 1. (a) The first SARPE surgery (midpalatal split) under local anesthesia; (b) delivery of a tooth-borne type expander at 2 weeks after the first surgery; (c) the second surgery and modified genioplasty; (d) lateral cephalogram taken immediately after the surgery Fig. 6.5 Final photographs of Case 1. Favorable profile was obtained with Class I occlusion with nonextraction treatment and dental arch forms were expanded. Loss of submental fats was noticed due to the weight loss genioglossus muscles with modified design for OSA patient, and profile was improved (Fig. 6.4d). The expansion protocol included the 2 turns per a day for 14 days afterward. The scissors bite was corrected using the TADs on the palatal mucosa, and crowding was resolved without labioversion of incisors due to the increased arch with and perimeter. As a result, Class I ideal occlusion with favorable facial profile was accomplished (Fig. 6.5). At the end of treatment, AHI decreased to 5.56 (by 85.3%) with increment of the lowest oxygen saturation up to 90%, total volume of upper airway increased by 96%, and minimum cross-sectional area also 6 Surgical Maxillary Expansion for OSA Adults with Nasal Obstruction 73 Fig. 6.6 Pharyngeal airway enlargement as seen on the color-mapped airway segments (left) and sleep functional improvement as confirmed from PSG records (right). MCA Minimum cross-­ sectional area (cm2), BMI Body mass index, AHI Apnea–hypopnea index, RDI Respiratory disturbance index, LSaO2 Lowest oxygen saturation enlarged by 94.6% (Fig. 6.6). The decrease of BMI might contribute to improving the patient’s respiratory and sleep function as well as esthetics, with this alternative treatment option to MMA. Case 2. Two-Piece Osteotomy for Surgical Maxillary Expansion Combined with MMA A 21-year-old male patient was referred from ENT department for the treatment of OSA. He had EDS, headache, and severe snoring. The PSG records showed AHI 40.8, RDI 43.0, and the lowest SaO2 75.0%. He had retruded chin, convex profile, narrow and constricted maxillary arch, high palatal vault, severe crowding on both arches, bilateral crossbite, and enlarged lingual torus (Fig. 6.7). Cephalometric analysis showed skeletal Class II with retrognathic mandible, hyperdivergent vertical pattern with steep occlusal plane, and labioversion of lower incisors (Fig. 6.8a). The constricted maxilla relative to the mandible and narrow retropalatal airway were confirmed by CBCT images (Figs. 6.8b and 6.9). He was diagnosed as suffering from severe OSA with craniofacial phenotype of skeletal Class II with retruded chin, hyperdivergent vertical pattern, and severely constricted maxilla. Treatment objective was the improvement of OSA symptoms by resolving skeletal discrepancy three dimensionally. Treatment plan included extraction of four first premolars and rotational maxilla-mandibular advancement (MMA) surgery combined with 2-piece segmental osteotomy on the maxilla and anterior segmental osteotomy on the mandible. During presurgical orthodontic treatment, decrowding of the upper dentition, protraction of upper molars into extraction spaces, distal 74 H.-W. Ahn and S.-J. Kim Fig. 6.7 Facial and intraoral photographs of Case 2 a b Fig. 6.8 Initial lateral cephalogram (a), and frontal and axial CBCT images (b) of Case 2 uprighting of lower molars were achieved. The basal bone width discrepancy between the maxilla and the mandible was −5.82 mm and large expansion on the posterior part of the maxilla was required; therefore, 2-piece Le Fort I osteotomy with 6 mm expansion at the first molar level was planned. To maximize the advancement of the mandible, anterior impaction of the maxilla, and intrusion and retraction of lower anterior teeth by anterior segmental osteotomy were performed simultaneously with MMA surgery (Fig. 6.9). After surgery, sufficient transverse expansion 6 Surgical Maxillary Expansion for OSA Adults with Nasal Obstruction 75 a b Fig. 6.9 The surgical procedure of Case 2. (a) The 2-piece segmental osteotomy with reverse fan type expansion and anterior impaction was performed on the maxilla, and (b) advancement and counterclockwise rotation of the mandible with anterior segmental osteotomy is performed a b c Fig. 6.10 The CBCT images at presurgery (a) and after surgery (b), and superimposition of the maxilla (c) of the maxilla and 10 mm advancement of the mandible were obtained (Fig. 6.10). The stabilizing transpalatal arch (TPA) was inserted throughout the postsurgical orthodontic treatment. At the end of treatment, his profile was improved by change of chin projection, and Class I functional occlusion was achieved (Fig. 6.11). The superimposition between initial and final cephalograms showed counterclockwise rotation of the maxillo-mandibular complex, significant advancement of the mandible, changes of soft palate inclination, anterior and upward movement of hyoid bone, and enlarged airway (Fig. 6.12). The CBCT data for airway analysis showed 61.6% increase of 76 H.-W. Ahn and S.-J. Kim Fig. 6.11 Facial and intraoral photographs of case 2 at the end of treatment Fig. 6.12 Final lateral cephalogram (a), and superimposition between initial and final using CBCT (b) a b total airway volume, 151% increase of minimum axial area, and increased nasal cavity (Fig. 6.13). He was very satisfied with the release of EDS and snoring. The AHI and RDI score reduced and the lowest SaO2 increased at the end of treatment, but the improvement was not sufficient probably in relation to the increased BMI. However, all PSG parameters showed greater improvement at 1-year retention period with decrease of BMI (Table 6.2). 6 a Surgical Maxillary Expansion for OSA Adults with Nasal Obstruction b c 77 d Fig. 6.13 Comparison of CBCT sectional images showing the airway dimensional changes between initial (a, c) and final (b, d) Table 6.2 Cephalometric analysis, airway dimension, and PSG records of Case 2 1. Cephalometric Analysis Divergency SUM (°) PFH/AFH (%) OP-FH (°) MP-FH (°) Maxillo-Mandibular ANB (°) AB-FH (°) relationship APDI (°) Maxilla SNA (°) N perp—Point A (mm) Mandible SNB (°) N perp—Point B (mm) Denture Interincisal angle (°) U1 to FH (°) IMPA (°) ANS-U1 tip (mm) U6-PP (mm) 2. Airway dimension Total airway volume (cc) Minimum axial area (mm2) 3. PSG records BMI AHI RDI Lowest SaO2 (%) Mean 395.7 65.5 7.2 26.8 2.8 86.0 81.2 81.8 1.0 79.0 −5.0 128.3 116.6 90.1 29.3 25.2 Initial 16,334.9 98.5 Initial 22.5 40.8 43.0 75.0 Initial 402.0 59.0 14.0 35.5 7.0 73.0 75.5 83.0 2.5 77.0 −7.5 114.0 113.5 97.5 34.0 24.3 Final 26,390.6 248.0 Final 27.7 24.1 33.7 89.0 Final 398.0 63.1 11.0 31.5 1.0 82.5 88.0 83.0 2.0 82.0 −2.5 130.0 108.8 88.0 29.5 23.0 Increment (%) 61.6 151.8 1Y Retention 25.0 14.6 20.7 90.5 BMI Body mass index, AHI Apnea–hypopnea index, RDI Respiratory disturbance index 78 H.-W. Ahn and S.-J. Kim Clinical pearls of Surgical Maxillary Expansion • Mechanism: Surgical maxillary expansion increases not only nasal cavity volume lowering nasal resistance and thus decreasing pharyngeal collapsibility indirectly, but also oral cavity allowing tongue-up posture followed by pharyngeal enlargement. • Indication: The OSA patients with craniofacial anatomical phenotype with nasomaxillary transverse constriction, high and deep palatal vault, and nasal obstruction are indicated. • Procedures: One-stage DOME with microimplant-assisted expander is currently preferred to the conventional two-stage SARPE for the OSA adults. Multipiece SO can be selected for the patients requiring MMA surgery due to the concomitant sagittal and vertical skeletal deformity. • Expansion pattern: Depending on the target area, anterior maxillary expansion including nasal cavity and/or posterior maxillary expansion affecting the pharyngeal walls can be selectively designed. References 1. Deeb W, Hansen L, Hotan T, et al. Changes in nasal volume after surgically assisted bone-­ borne rapid maxillary expansion. Am J Orthod Dentofacial Orthop. 2010;137(6):782–9. 2. Zambon C, Ceccheti M, Utumi E, et al. Orthodontic measurements and nasal respiratory function after surgically assisted rapid maxillary expansion: an acoustic rhinometry and rhinomanometry study. Int J Oral Maxillofac Surg. 2012;41(9):1120–6. 3. Abdullatif J, Certal V, Zaghi S, et al. Maxillary expansion and maxillomandibular expansion for adult OSA: a systematic review and meta-analysis. J Craniomaxillofac Surg. 2016;44(5):574–8. 4. Buck LM, Dalci O, Darendeliler MA, Papadopoulou AK. Effect of surgically assisted rapid maxillary expansion on upper airway volume: a systematic review. J Oral Maxillofac Surg. 2016;74(5):1025–43. 5. Guilleminault C, Partinen M, Hollman K, et al. Familial aggregates in obstructive sleep apnea syndrome. Chest. 1995;107(6):1545–51. 6. Gautam P, Valiathan A, Adhikari R. Stress and displacement patterns in the craniofacial skeleton with rapid maxillary expansion: a finite element method study. Am J Orthod Dentofacial Orthop. 2007;132(1):5. e1–5. e11 7. de Medeiros JR, Bezerra MF, Costa FG, et al. Does pterygomaxillary disjunction in surgically assisted rapid maxillary expansion influence upper airway volume? A prospective study using Dolphin Imaging 3D. Int J Oral Maxillofac Surg. 2017;46(9):1094–101. 8. Sygouros A, Motro M, Ugurlu F, Acar A. Surgically assisted rapid maxillary expansion: cone-­ beam computed tomography evaluation of different surgical techniques and their effects on the maxillary dentoskeletal complex. Am J Orthod Dentofacial Orthop. 2014;146(6):748–57. 9. Liu SY-C, Guilleminault C, Huon L-K, Yoon A. Distraction osteogenesis maxillary expansion (DOME) for adult obstructive sleep apnea patients with high arched palate. Otolaryngol Head Neck Surg. 2017;157(2):345–8. 10. Northway WM, Meade JB Jr. Surgically assisted rapid maxillary expansion: a comparison of technique, response, and stability. Angle Orthod. 1997;67(4):309–20. 6 Surgical Maxillary Expansion for OSA Adults with Nasal Obstruction 79 11. Seeberger R, Kater W, Davids R, Thiele OC. Long term effects of surgically assisted rapid maxillary expansion without performing osteotomy of the pterygoid plates. J Craniomaxillofac Surg. 2010;38(3):175–8. 12. Nada RM, van Loon B, Schols JG, et al. Volumetric changes of the nose and nasal airway 2 years after tooth-borne and bone-borne surgically assisted rapid maxillary expansion. Eur J Oral Sci. 2013;121(5):450–6. 13. Hamedi-Sangsari A, Chinipardaz Z, Carrasco L. Following surgically assisted rapid palatal expansion, do tooth-borne or bone-borne appliances provide more skeletal expansion and dental expansion? J Oral Maxillofac Surg. 2017;75(10):2211–22. 14. Yao W, Bekmezian S, Hardy D, et al. Cone-beam computed tomographic comparison of surgically assisted rapid palatal expansion and multipiece Le Fort I osteotomy. J Oral Maxillofac Surg. 2015;73(3):499–508. 15. Marchetti C, Pironi M, Bianchi A, Musci A. Surgically assisted rapid palatal expansion vs. segmental Le Fort I osteotomy: transverse stability over a 2-year period. J Craniomaxillofac Surg. 2009;37(2):74–8. 16. Junior OH, Guijarro-Martínez R, de Sousa Gil A, et al. Stability and surgical complications in segmental Le Fort I osteotomy: a systematic review. Int J Oral Maxillofac Surg. 2017;46(9):1071–87. 17. Seeberger R, Gander E, Hoffmann J, Engel M. Surgical management of cross-bites in orthognathic surgery: surgically assisted rapid maxillary expansion (SARME) versus two-piece maxilla. J Craniomaxillofac Surg. 2015;43(7):1109–12. 18. Kretschmer WB, Baciut G, Baciut M, et al. Transverse stability of 3-piece Le Fort I osteotomies. J Oral Maxillofac Surg. 2011;69(3):861–9. 7 Maxillomandibular Advancement Surgery for Skeletal Class II OSA Patients Jin-Young Choi and Seung-Hak Baek Contents 7.1 M echanism of MMA to Open Upper Airway 7.2 Patient Selection; Indication 7.3 Surgical Design of MMA 7.3.1 Four Subtypes of MMA 7.3.2 Adjunctive Surgery with MMA 7.3.3 Factors to Determine the MMA Subtype 7.4 Surgery-First Approach for OSA Patients 7.5 Pre- and Postsurgical Orthodontic Strategy 7.5.1 Presurgical Orthodontics 7.5.2 Postsurgical Orthodontics 7.6 Case: An OSA Patient with Craniofacial Phenotype Treated with Segmental MMA Surgery References 7.1 81 82 83 83 84 85 86 86 86 87 87 93 Mechanism of MMA to Open Upper Airway Maxillomandibular advancement (MMA) expands the skeletal frame to which the pharyngeal structures and tongue – are attached [1–3]. Advancement of maxilla including the posterior nasal spine (PNS) provides larger oral cavity for the tongue and induces forward movement of soft palate. Advancement of chin pulls the tongue and hyoid forward by increasing the tension of tongue–hyoid complex, J.-Y. Choi Department of Oral and Maxillofacial Surgery, Seoul National University School of Dentistry, Seoul, Korea S.-H. Baek (*) Department of Orthodontics, School of Dentistry, Seoul National University, Seoul, South Korea e-mail: drwhite@unitel.co.kr © Springer Nature Switzerland AG 2020 S.-J. Kim, K. B. Kim (eds.), Orthodontics in Obstructive Sleep Apnea Patients, https://doi.org/10.1007/978-3-030-24413-2_7 81 82 J.-Y. Choi and S.-H. Baek leading to upright of soft palate through the palatoglossus muscles. In addition, MMA showed improvement of lateral pharyngeal wall collapsibility on DISE, supported by significant correlation with surgical success [4]. Overall, MMA results in not only increasing total volume of upper airway at all levels, especially at the minimum cross-sectional area, but also decreasing critical closing pressure of pharyngeal airway for respiratory functional improvement. A CBCT study [5] showed the ratio of lateral/sagittal dimension increased up to 2.37 at the oropharyngeal level with MMA. The sagittal dimension of pharyngeal airway increased, on average, 56% of the maxillary advancement at the velopharynx, 49% at the oropharynx, and 58% at the hypopharynx. Surprisingly, lateral dimension of pharyngeal airway increased 71% of advancement at the velopharynx and 132% at the oropharynx, and 66% at the hypopharynx. Skeletal advancement can dilate the lateral dimension of the pharyngeal airway more than twice that of the sagittal expansion. 7.2 Patient Selection; Indication MMA has reported the highest success and cure rates of all procedures but must be tailored to the appropriate patient. However, not all OSA patients respond to the MMA successfully. Craniofacial anatomic phenotype would be a good candidate of MMA; however, other factors should be considered together for the decision. Indications for the MMA are provided in the Table 7.1 [6]. Controversies still exist on the correlation of preoperative AHI (initial OSA severity) or BMI with the surgical success rate or cure rate. MMA has been applied to the patients who failed to respond to the conservative treatments or phase I surgery as a stepwise approach; however, it can be recommended as a primary option in case of OSA with definite dentofacial deformity. Notably, in addition, particularly for severe OSA patients with complete lateral pharyngeal collapse on DISE, strong consideration should be given to MMA as first-line therapy even though they have no definite dentofacial deformity [7, 8]. Table 7.1 Indication criteria of MMA Guidelines History PSG Obstruction site Cephalometric area Dentofacial deformity DISE Indication • Fail to conservative treatment or phase I surgery • Medically stable condition • AHI > 15, AI > 5, RDI > 30, LSaO2 < 90% • No prevalent central/mixed apnea • Responsive to MAD-titration PSG • Overall constriction • Hypopharyngeal narrowing <9 mm • Severe mandibular retrusion or bimaxillary retrusion • Steep occlusal plane/mandibular plane • Complete lateral pharyngeal wall collapse, even in absence of dentofacial deformity PSG Polysomnography, AHI Apnea–hypopnea index, AI Apnea index, RDI Respiratory disturbance index, LSaO2 Lowest oxygen saturation, DISE Drug-induced sleep endoscopy. 7 Maxillomandibular Advancement Surgery for Skeletal Class II OSA Patients 7.3 Surgical Design of MMA 7.3.1 Four Subtypes of MMA 83 7.3.1.1 Conventional Straightforward MMA Surgical design of conventional MMA includes maxillary and mandibular osteotomies for anterior reposition of maxillomandibular complex (MMC) along the occlusal plane. A meta-analysis [9] indicated the direct relationship between the amount of MMC advancement and an increased airway volume [10, 11]. Bimaxillary advancements greater than 10 mm are considered effective to improve OSA, especially in case of bimaxillary retrusion with normal occlusal plane steepness. In Caucasian OSA patients who have a convex profile, obtuse nasolabial angle, and large nose with high dorsum, 9–12 mm of con-MMA can achieve maximum expansion of the upper airway without aggravation of facial profile [12–18]. Especially, in middle-aged OSA patients, large amount of MMA can somewhat rejuvenate their face and give a younger looking impression [6, 12, 14, 15]. 7.3.1.2 Rotational MMA Counterclockwise (CCW) rotation of MMC has been widely accepted to maximize mandibular advancement while minimizing the cosmetic impact of the surgery in the nasomaxillary region (Fig. 7.1, Left). Especially for OSA patients with hyperdivergent skeletal Class II pattern with high occlusal plane angle, surgical flattening of occlusal plane not only enhances the esthetic profile by optimizing the advancement of chin but also improves the airway volume and function. After rotational MMA, the sagittal dimension of pharyngeal airway increased by 47% and 76% of the amount of mandibular advancement at the oropharynx and hypopharynx, respectively [19]. A recent meta-analysis [20] confirmed that both conventional and rotational MMA surgeries resulted in a significant AHI reduction; however, they found no evidence that CCW rotation would enhance the surgical gain as compared to MMA alone due to the insufficient data to determine superiority of either operation. Fig. 7.1 Diagram showing the impact of posterior nasal spine (PNS) advancement on the soft palate change to open the oropharynx, through CCW rotation of maxillary occlusal plane (left) and anterior segmental osteotomy to protract posterior maxilla maintaining anterior maxilla (right). This is based on the review reporting that the highest surgical predictor of MMA success is the amount of maxillary advancement rather than that of mandibular advancement 84 J.-Y. Choi and S.-H. Baek 7.3.1.3 Segmental MMA Segmental MMA, also called modified MMA, which combines MMA with upper and/or lower anterior segmental osteotomy (ASO), has been developed to simultaneously improve sleep function and facial esthetics in Asian OSA patients who have tendency of lip protrusion, acute nasolabial angle, and low nasal bridge [21]. (Fig. 7.1, Right) The mechanisms of segmental MMA are as follows: First, minimization of advancement or small amount of setback of the anterior segment of the maxilla according to cephalometric norms can prevent excessive protrusion of the upper lip. Second, advancement of the posterior segment of the maxilla to close the maxillary premolar extraction space stretches the soft palate and protracts the velopharyngeal aponeurosis. Third, the mandibular premolar extraction space is closed by setback of the anterior segment of the mandible. Fourth, total advancement of the distal segment of the mandible can protract tongue base with pterygomandibular aponeurotic attachments [13–15]. Segmental MMA is worth allowing surgery-first approach through the surgical correction of proclined or extruded lower incisors and surgical dental arch coordination [22]. Considering that lower ASO and genioplasty could not be performed at the same time, strategic application is needed in terms of greater advancement of chin and tongue base [23]. 7.3.1.4 Segmental-Rotational MMA To attain maximum amount of maxillomandibular advancement in an already protrusive OSA patient, combined type with CCW rotation and segmental Le Fort I maxillary osteotomy can further advance the posterior nasal spine and palatine bone to open velopharynx without protrusion of anterior maxilla. By segmental-rotational MMA, the pharyngeal airway increased in sagittal dimension at the velopharynx by 60% and at the oropharynx by 70% of the mandibular advancement. 7.3.2 Adjunctive Surgery with MMA 7.3.2.1 Modified Genioplasty Genioplasty can be offered solely (as a compromised option) or with MMA in terms of increasing the tension of genioglossus and geniohyoid muscles to pull the tongue forward and to suspend the hyoid, as well as improving esthetics. To improve the labiomental fold and chin projection, a sliding genioplasty can be performed as a horizontal osteotomy over the central portion of the mandible lower than bilateral mental nerve. In patients with OSA, on the other hand, the surgical design of genioplasty should be modified to cut more superiorly at the central area below the lower central incisors and to include the genioglossus tubercle within the advancing segment (reverse T mandibular osteotomy) [24], although a recent meta-analysis showed that genioplasty in addition to MMA did not impact PSG outcomes [25]. 7 Maxillomandibular Advancement Surgery for Skeletal Class II OSA Patients 85 7.3.2.2 Piriform Rim Recontouring, Septoplasty Piriform rim recontouring should be performed to compensate the reduced nasal cavity on the piriform aperture area in case of anterior maxillary impaction for CCW rotation of MMC during MMA surgery. In addition, septoplasty can be done to adjust the septal length to the reduced space by anterior maxillary impaction, and to prevent or correct the septal deviation for better nasal clearance after MMA. 7.3.3 Factors to Determine the MMA Subtype To determine optimal surgical designs for each OSA patient with different dentofacial and pharyngeal patterns, individualized surgical treatment objectives (STOs) including airway prediction are required. Nose height, anteroposterior position of maxilla, upper lip projection, lower incisor inclination, overjet, occlusal plane, and upper incisor exposure are proposed as critical decision factors of MMA design (Fig. 7.2). Out of these, anteroposterior position of maxilla and upper lip Nose Nasolabial angle Maxilla, Upper lip Max.incisor showing Maxillary Occlusal Plane high low obtuse acute Retrusive Protrusive or normal Normal Conventional total advancement of the maxilla with one-piece LeFort I osteotomy LeFort I osteotomy and segmental osteotomy • Ant. segment : minimization of advancement or slight setback to prevent excessive protrusion of the upper lip • Post. segment : advancement to close the Max. premolar etraction space and also protract the velopharygeal aponeurosis. No or Even impaction of the maxilla (Ant = Post) excessive Normal Steep Differential impaction of the maxilla (Ant > Post) for counterclockwise rotation of the mandible Conventional total advancement of the mandible with BSSRO Mandible, Man. incisor, Lower lip, Chin DJD Retrusive Protrusive or normal with or without Genioglossus Advancement BSSRO and segmental osteotomy • Ant. segment : to close the Man. premolar extraction space • Total advancement of the mandible : to protract tongue base with pterygomandibular aponeurotic attachments Reduction of the amount of Mandibular advancement / Genioglossus Advancement Fig. 7.2 Selection criteria for conventional maxillomandibular advancement (MMA) and segmental MMA with or without CCW rotation and genioplasty in adult OSA patients 86 J.-Y. Choi and S.-H. Baek projection (relative to the nose height) deserve to be firstly considered. Unless a patient has protrusive maxilla and upper lip, straightforward MMA or rotational MMA could be taken into account according to the degree of maxillary retrusion, combined with advancing genioplasty if needed. Then, if the patient has normal range of incisor exposure and occlusal plane simultaneously, straight MMA would be satisfactory. With excessive upper incisor exposure and steep occlusal plane, however, rotational MMA should be finally chosen. On the contrary, if a patient has protrusive maxilla and upper lip, segmental MMA with maxillary ASO is needed. Concurrently with small overjet with lower incisor proclination, bimaxillary ASOs should be the first option for the maximum mandibular advancement. If the patient has excessive upper incisor exposure and steep occlusal plane with maxillary protrusion, segmental-­rotational MMA is beneficial. 7.4 Surgery-First Approach for OSA Patients It must be kept in mind that the goal of MMA is to treat a serious medical condition of OSA and to drop the related risks. For this reason, MMA surgery without orthodontics may be pursued to the patients with favorably adapted occlusion or even if this yields a less optimal occlusal result. Nonetheless, many OSA patients belonging to the craniofacial phenotype have dental malocclusion, which should be corrected concurrently with MMA. Even in case, surgery-first approach or minimum presurgical orthodontic approach needs to be considered for the OSA patients. Conventional presurgical orthodontic decompensation to increase overjet by lower incisor retraction can increase the amount of surgical mandibular advancement; however, OSA symptoms may become aggravated during the presurgical orthodontic period due to the decreased tongue space. The delay in the resolution of chief complaint caused by presurgical orthodontic decompensation also must be considered. For the surgical success and stability with surgery-first MMA for OSA patients, (1) virtual surgical planning needs to be provided for the accurate surgical and postsurgical prediction; (2) multipiece osteotomy can be considered for immediate surgical arch coordination to minimize the prematurity on the postsurgical occlusion; (3) strategic postsurgical orthodontic treatment should be followed. Concerning unstable factors of segmental osteotomy, presurgical minimum orthodontics to expand maxillary arch, and to remove the mandibular shifting factors need to be contemplated. 7.5 Pre- and Postsurgical Orthodontic Strategy 7.5.1 Presurgical Orthodontics As mentioned above, presurgical orthodontic period should be as minimal as possible. The goals of presurgical orthodontics include arch coordination, removal of prematurity leading to immediate surgical relapse, maxillary orthopedic expansion if needed. In addition, distalization of lower posterior teeth is important to ensure 7 Maxillomandibular Advancement Surgery for Skeletal Class II OSA Patients 87 sufficient mandibular advancement in a patient with compensated molar relationship into Class I in a skeletal Class II pattern. Lower anterior retraction with premolar extraction is not recommended before MMA surgery; instead, surgical anterior retraction using lower ASO would be better thinking of patient’s OSA symptoms. 7.5.2 Postsurgical Orthodontics The goals of postsurgical orthodontics in surgery-first approach or minimum presurgical orthodontic approach are to decompensate occlusion and to ensure skeletal stability. It is initiated as early as possible, or immediately after surgery in stable cases, to take advantage of unlocked occlusion and accelerated tooth movement. With active NiTi archwires inserted for aligning and leveling, bone-tobone intermaxillary elastics are more efficient to maintain or guide the new jaw position than interdental elastics. It should be cautious that postsurgical Class III elastics for sagittal decompensation of dentition may lead to early surgical relapse of mandibular position. For the case receiving maxillary segmental osteotomy, the sectional surgical wires should be replaced with continuous archwires at the first postsurgical orthodontic appointment. However, initiation timing of active tooth movement depends on the segmental stability, which should be kept with longer wafer wearing or other retentive appliance if needed. 7.6 ase: An OSA Patient with Craniofacial Phenotype C Treated with Segmental MMA Surgery Diagnosis and Treatment Planning A nonobese young adult OSA patient (21 years-old male; BMI, 21.1) was referred from Department of ENT in Seoul National University Hospital and Department of Oral and Maxillofacial Surgery, Seoul National University Dental Hospital. His chief complaint was severe snoring and OSA. The initial PSG results indicated a moderate OSA (AHI, 14.2; RDI, 29.0; LSaO2, 87%; Table 7.2). Facial examination presented convex profile with severely retruded chin and normal upper lip angle, short throat length, obtuse cervicomental angle, and hyperactive mentalis muscles (Fig. 7.3). Molar and canine relationships were compensated into Class I with normal overjet and overbite, and upper and lower dental arches were constricted, which make sufficient mandibular advancement difficult. Cephalometrically, he had skeletal Class II pattern with normal maxilla and retruded small mandible, and hyperdivergent vertical pattern. While maxillary Table 7.2 Comparison of the polysomnography data AHI RDI LSaO2 T1 (before surgery) 14.2 29.0 87% T2 (6m after surgery) 7.9 8.3 90% 88 J.-Y. Choi and S.-H. Baek Fig. 7.3 Facial and intraoral photographs taken at initial visit Fig. 7.4 Lateral and PA cephalograms and panoramic X-ray taken at initial visit incisal inclination was normal, mandibular incisors were proclined compensating the overjet, which also make surgical chin advancement insufficient. Transverse skeletal discrepancy was not present. Pharyngeal airway was severely constricted at the velopharyngeal level, behind the soft palate. (Fig. 7.4 and Table 7.2). Treatment objectives involved maximum advancement of mandible and posterior maxilla to improve the pharyngeal collapsibility and facial profile without creating upper lip protrusion, as per the patient’s request. Accordingly, the segmental MMA with maxillary and mandibular ASOs was planned to protract the posterior maxilla enough to stretch the lateral pharyngeal walls, all the while maintaining the anteroposterior position of the anterior maxilla, and to maximally advance the mandible. In addition, maxillary differential impaction was included to induce clockwise rotation of palatal plane, occlusal plane, and mandibular plan. 7 Maxillomandibular Advancement Surgery for Skeletal Class II OSA Patients 89 Treatment Progress and Results Minimum presurgical orthodontic treatment was performed for 2 months just for dental arch form modification in order to reduce the time of uncomfortable presurgical orthodontic treatment and to improve the sleep function as early as possible. In surgery, the posterior segment of the maxilla was advanced by 6.7 mm to close the extraction space of the maxillary first premolars with no remarkable change of anteroposterior position of anterior maxilla. The anterior segment of the mandible was moved backward to close the extraction space of the mandibular first premolars and the total mandible was advanced 10.5 mm. In the meanwhile, upper incisor inclination was decreased by clockwise rotation of anterior maxillary segment, and lower incisor inclination was not sufficiently decreased due to the parallel setback of anterior mandibular segment. Simultaneously, the maxilla was differentially impacted by 3.5 mm at the ANS level, and by 2.5 mm at the PNS level. Flattening of occlusal plane could not be obtained despite greater anterior maxillary impaction, while mandibular plane angle was decreased as a result of posterior maxillary impaction. During surgery, CAD/CAM-made condylar jigs (Orapix Co, Ltd, Seoul, South Korea) were used to stabilize the condyle in the centric relation position [25]. Postsurgical orthodontic treatment was started at two months after surgery concerning the stability of bony segments. Extraction spaces were almost closed by ASO; therefore, remaining space consolidation, arch coordination, and additional intrusion of maxillary molars using microimplants were performed to obtain bite closure and complete interdigitation (Fig. 7.5). Total treatment time was 12 months. As a result, Class I canine and molar relationships, normal overbite and overjet, solid buccal occlusion were established (Figs. 7.6 and 7.7). Despite still insufficient chin profile (Fig. 7.6), patient was satisfied with his facial and respiratory improvement. According to the PSG at 6 months after surgery, there was significant improvement in OSA parameters (Table 7.2) and enlargement of the velopharyngeal airway (Table 7.3). Fig. 7.5 Intraoral photographs in postsurgical orthodontic treatment 90 J.-Y. Choi and S.-H. Baek Fig. 7.6 Facial and intraoral photographs taken at the end of treatment Fig. 7.7 Superimposition of the lateral cephalograms between initial and final Retention At 2-year retention, the upper airway space was well maintained (Table 7.3) with favorable skeletal and occlusal stability. (Figs. 7.8, 7.9, 7.10, and 7.11). 7 Maxillomandibular Advancement Surgery for Skeletal Class II OSA Patients Table 7.3 Comparison of the cephalometric measurements SNA (°) SNB (°) ANB (°) FMA (°) U1 to SN (°) IMPA (°) IIA (°) UI exposure at rest (mm) nasolabial angle (°) PNS-AD2 (mm) PNS-AD1 (mm) SPAS (mm) MAS (mm) IAS (mm) Mean 81.8 79.0 2.8 26.8 109.3 90.1 128.3 3.36 75.47 25.5 23.3 12.8 13.6 12.0 Initial 78.5 73.6 4.9 32.7 106.8 101.1 109.1 3.2 78 17.5 18.0 3.5 8.2 9.5 Final 80.9 76.0 5.0 29.5 99.1 112.3 110.5 1.0 75 23.5 22.5 8.0 11.5 11.5 Fig. 7.8 Facial and intraoral photographs taken at 2-year retention 2-year retention 80.6 74.8 5.8 27.9 101.2 113.3 108.4 0.0 81 22.0 21.0 7.5 10.5 10.5 91 92 J.-Y. Choi and S.-H. Baek Fig. 7.9 Superimposition of the lateral cephalograms between final and 2-year retention. Surgical skeletal changes were well maintained a b c Fig. 7.10 Comparison of facial photographs among initial (a), final (b), and 2-year retention (c). Facial profile was improved and well maintained a b c Fig. 7.11 Comparison of lateral cephalograms among initial (a), final (b), and 2-year retention (c). Velopharyngeal airway space (yellow arrow), which was particularly constricted as the minimum cross-sectional area was enlarged after treatment and favorably maintained. (Abnormal posture of deformed soft palate in the 2-year retention cephalogram (c) was a taking error in swallowing.) 7 Maxillomandibular Advancement Surgery for Skeletal Class II OSA Patients 93 Clinical Pearls of Maxillomandibular Advancement (MMA) Surgery • Mechanism: Advancement of chin and posterior maxilla moves the tongue, hyoid, and soft palate forward with stretching the lateral pharyngeal walls opening the upper airway three dimensionally at all levels. • Indication: For severe OSA patients with craniofacial anatomical phenotype, or with concentric collapse of lateral pharyngeal walls, MMA can be the first-line therapy as a salvage treatment. • MMA Subtypes: According to the patient’s dento-skeletal pattern such as upper lip and maxillary protrusion, maxillary incisor showing, and occlusal plane angle, surgical design can be modified from straightforward MMA to rotational MMA, segmental MMA, or segmental-rotational MMA. • Surgery-first approach (SFA): SFA or minimum presurgical orthodontics is preferred for the OSA patients in terms of resolving the chief complaint firstly and preventing the aggravation of OSA symptoms during the presurgical decompensation stage. • Orthodontic strategy: Pre- and post-surgical orthodontic strategies are more important in OSA patients than in regular orthodontic patients for greater airway enlargement and long-term stability. References 1. Riley RW, Powell NB, Guilleminault C. Obstructive sleep apnea syndrome: a surgical protocol for dynamic upper airway reconstruction. J Oral Maxillofac Surg. 1993;51(7):742–7. 2. John C, Gandhi S, Sakharia A, James T. Maxillomandibular advancement is a successful treatment for obstructive sleep apnoea: a systematic review and meta-analysis. Int J Oral Maxillofac Surg. 2018;47:1561–71. 3. Zaghi S, Holty J-EC, Certal V, et al. Maxillomandibular advancement for treatment of obstructive sleep apnea: a meta-analysis. JAMA Otolaryngol Head Neck Surg. 2016;142(1):58–66. 4. Camacho M, Liu SY, Certal V, Capasso R, Powell NB, Riley RW. Large maxillomandibular advancements for obstructive sleep apnea: an operative technique evolved over 30 years. J Craniomaxillofac Surg. 2015;43(7):1113–8. 5. Fairburn SC, Waite PD, Vilos G, et al. Three-dimensional changes in upper airways of patients with obstructive sleep apnea following maxillomandibular advancement. J Oral Maxillofac Surg. 2007;65(1):6–12. 6. Holty J-EC, Guilleminault C. Maxillomandibular advancement for the treatment of obstructive sleep apnea: a systematic review and meta-analysis. Sleep Med Rev. 2010;14(5):287–97. 7. Liu SY-C, Huon L-K, Powell NB, et al. Lateral pharyngeal wall tension after maxillomandibular advancement for obstructive sleep apnea is a marker for surgical success: observations from drug-induced sleep endoscopy. J Oral Maxillofac Surg. 2015;73(8):1575–82. 8. Liu SY-C, Awad M, Riley R, Capasso R. The role of the revised stanford protocol in today’s precision medicine. Sleep Med Clin. 2019;14(1):99–107. 94 J.-Y. Choi and S.-H. Baek 9. Rosario HD, Oliveira GMS, Freires IA, de Souza Matos F, Paranhos LR. Efficiency of bimaxillary advancement surgery in increasing the volume of the upper airways: a systematic review of observational studies and meta-analysis. Eur Arch Otorhinolaryngol. 2017;274(1):35–44. 10. Butterfield KJ, Marks PL, McLean L, Newton J. Pharyngeal airway morphology in healthy individuals and in obstructive sleep apnea patients treated with maxillomandibular advancement: a comparative study. Oral Surg Oral Med Oral Pathol Oral Radiol. 2015;119(3):285–92. 11. Ahn H-W, Cho I-S, Cho K-C, Choi J-Y, Chung J-W, Baek S-H. Surgical treatment modality for facial esthetics in an obstructive sleep apnea patient with protrusive upper lip and acute nasolabial angle. Angle Orthod. 2012;83(2):355–63. 12. Kim T, Kim H-H, Hong S, et al. Change in the upper airway of patients with obstructive sleep apnea syndrome using computational fluid dynamics analysis: conventional maxillomandibular advancement versus modified maxillomandibular advancement with anterior segmental setback osteotomy. J Craniofac Surg. 2015;26(8):e765–70. 13. Baek SH, Choi JY. Treatment of obstructive sleep apnea patients (Chapter 16). In: Baik HS, editor. Clinical combined Surgico-Orthodontics. Seoul, Korea: DaehanNarae Publishing Inc.; 2017. p. 525–37. 14. Hong SO, Baek SH, Choi JY. Individualized orthodontic treatment and surgical plan for obtaining facial esthetics as well as better sleep results in adult patients with obstructive sleep apnea syndrome (Chapter 37). In: Kim KB, et al., editors. Management of obstructive sleep apnea—a multidisciplinary textbook with evidence-based guidelines: Springer International Publishing AG (In Press). 15. Holty J-EC, Guilleminault C. Surgical options for the treatment of obstructive sleep apnea. Med Clin. 2010;94(3):479–515. 16. Li KK. Maxillomandibular advancement for obstructive sleep apnea. J Oral Maxillofac Surg. 2011;69(3):687–94. 17. Li KK. Surgical therapy for adult obstructive sleep apnea. Sleep Med Rev. 2005;9(3):201–9. 18. Brunetto DP, Velasco L, Koerich L, de Souza Araújo MT. Prediction of 3-dimensional pharyngeal airway changes after orthognathic surgery: a preliminary study. Am J Orthod Dentofacial Orthop. 2014;146(3):299–309. 19. Mehra P, Downie M, Pita MC, Wolford LM. 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J Craniofac Surg. 2014;25(6):2002–7. 8 Modification of Orthognathic Surgery for Skeletal Class III OSA Patients Takashi Ono Contents 8.1 T wo-Jaw Surgery Versus Mandibular One-Jaw Surgery 8.2 Maxillary One-Jaw Advancement Surgery 8.2.1 Case 1: Non-REM-Related Non-POSA 8.2.2 Case 2: REM-Related POSA 8.2.3 Clinical Significance of the Two Cases with Different Phenotypes 8.3 Maxillary Distraction Osteogenesis 8.4 Trimming of Pyriform Aperture in Case of Superior Maxillary Impaction 8.5 Maxillary Horseshoe Osteotomy in Case of Superior Maxillary Impaction References 8.1 95 97 98 99 100 101 104 105 107 Two-Jaw Surgery Versus Mandibular One-Jaw Surgery A cephalometric and portable PSG study conducted at Tufts University [1] examined the effects of mandibular setback with or without maxillary advancement on the development of OSA in 26 patients with skeletal Class III malocclusion. They found that patients who underwent one-jaw surgery with movement more than 5 mm exhibited a higher incidence of mild-to-moderate OSA than did patients who underwent two-jaw surgery (Fig. 8.1). This finding indicated that combined two-jaw surgery may be more favorable than one-jaw surgery from the perspective of respiratory function. Another study by Tamari and colleagues [2] revealed a significant correlation between the tongue position and the width of the mandibular arch. The T. Ono (*) Department of Orthodontic Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University (TMDU), Tokyo, Japan e-mail: t.ono.orts@tmd.ac.jp © Springer Nature Switzerland AG 2020 S.-J. Kim, K. B. Kim (eds.), Orthodontics in Obstructive Sleep Apnea Patients, https://doi.org/10.1007/978-3-030-24413-2_8 95 96 T. Ono 70% 1-jaw (Md set-back) 60% n=26 Percent of patients 50% 40% 30% 2-jaw (Md set-back + Mx advancement) 20% 10% 0% negative mild moderate Severity severe Fig. 8.1 Comparisons of cephalometric and portable polysomnographic changes between the one-jaw and two-jaw groups authors consequently speculated that orthodontic treatment may affect the mandibular arch shape and influence the hyoid bone position and pharyngeal airway. In our previous study [3], we compared 2D changes in UA between patients with Class III malocclusion (n = 65) who underwent mandibular setback surgery (one-jaw group) and those who underwent maxillary/mandibular surgery (two-jaw group) for correction of their occlusion and facial profile. A set of lateral cephalograms was obtained before surgery (T0), 3 months after surgery (T1), and 2 years after surgery (T2). Parameters for evaluation of morphological changes in UA were determined and included the naso-, oro-, and hypopharyngeal airway spaces and the horizontal and vertical positions of the hyoid bone (Fig. 8.2). As a result, similar changes in the UA dimensions and hyoid position appeared in both groups (Fig. 8.2): (1) The nasopharyngeal airway dimension significantly decreased from T1 to T2; (2) Both the oro- and hypo-pharyngeal airway dimensions significantly decreased from T0 to T1; (3) The hyoid bone moved posteroinferiorly from T0 to T1 and anterosuperiorly from T1 to T2. On the other hand, we found significant negative correlations between the amount of mandibular displacement and changes in the UA dimensions from T0 to T2 in the one-jaw group, but not in the two-jaw group. Comparisons of the longitudinal decrease in the UA dimensions from T0 to T2 between the one-jaw and twojaw groups revealed that the rate of decrease in the oro- and hypo-pharyngeal airway spaces was significantly lower in the two-jaw group. Based on this, we suggest twojaw surgery to negate the risk of postoperative sleep-disordered breathing in Class III patients. In the future, it will be necessary to delineate the effects of the direction and amount of maxillary displacement on the UA morphology and function. 8 Modification of Orthognathic Surgery for Skeletal Class III OSA Patients 97 Fig. 8.2 Landmarks and reference points (upper) and comparisons of the longitudinal reduction of the UA dimensions from T0 to T2 between the one-jaw and two-jaw groups (lower) 8.2 Maxillary One-Jaw Advancement Surgery It has been reported that the amount of surgical maxillary advancement rather than mandibular advancement is the significant correlation factor with the surgical success of MMA in skeletal Class II OSA patients [4]. Then, is the surgical maxillary advancement critical factor in skeletal Class III OSA patient as well? Is the maxillary advancement effective to treat OSA in all patients with various phenotypes? In this section, we will focus on OSA phenotypes and compare the outcomes of maxillary advancement surgery between a case of non-REM-related non-POSA and a case of REM-related POSA (REM, rapid eye movement; POSA, positional obstructive sleep apnea). 98 T. Ono 8.2.1 Case 1: Non-REM-Related Non-POSA Diagnosis and Treatment Plan A 37-year-old man presented with complaints of chewing difficulties due to the anterior crossbite and openbite (Fig. 8.3) [5]. He was not worried about his facial appearance, but flat cervicomental angle and thick neck circumference with heavy submental fat deposition were concerned. He was obese, with a BMI of 34 kg/m2, and PSG revealed an AHI of 56 indicating severe OSA. He belonged to a non-REM-­ dependent, and a nonpositional OSA phenotype. He was suffering from EDS and frequent arousal. Intraoral examination showed anterior crossbite with a large and flaccid tongue with tongue thrust. Cephalometric analysis revealed skeletal openbite with hyperdivergent mandible and skeletal Class III with an ANB angle of −4.0°. Considering the respiratory complications by mandibular setback surgery for this severe OSA patient, we decided to perform only maxillary advancement surgery. Treatment Progress Following alignment with nonextraction, orthognathic surgery was performed. The maxilla was advanced by 10 mm without any soft tissue resistance. There were no immediate postoperative complications, and the patient reported that he had never slept better. Postsurgical orthodontic treatment was completed in 10 months, and the entire duration of active treatment was 2 years and 2 months. Fixed retainers were bonded on both arches, and a circumferential maxillary retainer with a tongue crib was provided for use while sleeping. Treatment Results Because only maxillary advancement surgery was performed, the mandible remained deviated to the left side. However, the facial profile was improved. Comparison of pre- and posttreatment lateral cephalograms revealed a change in the ANB angle from −3.9° to +3.4°. Moreover, superimposition of pre- and Before treatment After treatment Fig. 8.3 Facial and intraoral photographs and cephalometric radiographs obtained before treatment and after treatment along with the superimposition of lateral cephalometric tracings obtained from before (black line) and after treatment (red line) 8 Modification of Orthognathic Surgery for Skeletal Class III OSA Patients 99 posttreatment cephalograms showed an improvement in the skeletal discrepancy and tongue position. The patient no longer experienced EDS and night awakening. Posttreatment PSG found a decrease in AHI and the snoring frequency and an increase in the lowest oxygen saturation. CBCT images obtained before surgery and during retention represented the enlargement in both anteroposterior and lateral dimensions at the most constricted site in spite of persistent obesity. In particular, the increase in the anteroposterior dimensions of nasopharynx and velopharynx was quite remarkable in this case with maxillary advancement without mandibular surgery. Our findings were in accordance with the findings of Tonogi and colleagues [6], who suggested that the lateral dimension increased with forward movement of the mandible. Of note, 3D changes in UA are not necessarily correlated with the 2D changes. It is important to understand the 3D changes in UA after orthognathic surgery [5]. 8.2.2 Case 2: REM-Related POSA Diagnosis and Treatment Plan A 20-year-old man presented with a chief complaint of a concave facial profile and difficulty in chewing (Fig. 8.4). His AHI of 15.3 indicated moderate OSA, and he also exhibited a low BMI. He belonged to a REM-dependent, and a positional OSA phenotype. He showed bilateral Class III molar relationships and lower midline deviation to the right side. All first premolars were missing. Cephalometric analysis revealed skeletal Class III with maxillary retrusion and hyperdivergent pattern, and pharyngeal spaces seemed to be within normal range. He was diagnosed with skeletal Class III malocclusion with moderate OSA. We decided to perform orthognathic surgery for esthetic and respiratory improvements. In general, MMA is recommended as the first treatment option from the perspective of respiratory function. However, maxillary advancement by onejaw surgery was chosen, since mandibular advancement was not feasible from esthetic point of view. Because maxillary advancement can result in some undesirable outcomes such as widening of the alar bases, loss of the vermilion, and downward sloping of the commissures [6], we applied alar cinch suture and a mucomusculo-­periosteal V-Y closure (ACVY) for this patient. Treatment Results The patient’s AHI decreased from 15.3 to 2.8 after treatment, and satisfactory occlusion and facial profile were achieved. Despite maxillary advancement, nasal deformities were alleviated by ACVY surgery. Although the maxilla was moved forward by 6.5 mm and the SNA angle was increased by 5.0°, airway enlargement in the anteroposterior dimension could not be observed on cephalometric superimposition. The treatment outcomes were maintained during and after the retention phase, as confirmed by superimposition of cephalograms obtained before treatment and after retention. PSG data showed that the treatment improved the respiratory function, particularly in the supine position, of this young adult patient with REM-­related POSA. 100 T. Ono Before treatment After treatment Fig. 8.4 Facial and intraoral photographs and cephalometric radiographs obtained before treatment and after treatment along with the superimposition of lateral cephalometric tracings obtained from before treatment stage (black line) to after treatment stage (red line). (Cited from Ishida et al. Angle Orthod. 2019) 8.2.3 linical Significance of the Two Cases with Different C Phenotypes To our knowledge, no study has investigated the effects of the OSA phenotype on the outcomes of surgical orthodontic treatment for patients with OSA. Therefore, we focused on changes in AHI after orthognathic surgery involving only maxillary advancement for a patient with non-REM-related non-POSA (Case 1) and a patient with REM-related POSA (Case 2). The decrease in AHI was small in Case 1 and remarkable in Case 2. Tables 8.1 and 8.2 show the changes in AHI according to the sleeping position and the level of sleep, respectively. We found that the supine AHI decreased after treatment in both cases, whereas the non-supine AHI showed increase in Case 1 but little change in Case 2. These findings suggest that maxillary advancement surgery can decrease only the supine AHI, consistent with the findings of Okushi’s study [7], where maxillary advancement was found to expand the anteroposterior diameter of UA. Table 8.1 shows that both the supine AHI and the total AHI were decreased during REM sleep after treatment in both cases. On the other hand, AHI during non-­ REM sleep improved in Case 2, with little change in Case 1. During REM sleep, the muscles remain relaxed, while the brain shows activity similar to that during waking states. During non-REM sleep, the brain activity is considerably reduced, while the muscles show a certain degree of activity. Shepard and colleagues [8] reported that airway collapse at the level of the hyoid bone was not observed during non-REM sleep in obese patients with severe OSA. Moreover, the sleeping position had little effect on the extent of airway collapse during sleep, which is independent of the sleep state. Our Case 1 showed similar findings. In such cases, if airway stenosis occurs at the caudal site of the hyoid bone during non-REM sleep, the physical distance from the anteriorly moved maxilla remains large, thus leading to little improvement in the non-REM AHI. In summary, there are several differences in the influences of different OSA phenotypes on sleep apnea and the general status after orthognathic surgery. 8 Modification of Orthognathic Surgery for Skeletal Class III OSA Patients 101 Table 8.1 Changes in AHI according to the sleep position Supine AI Supine HI Supine AHI Nonsupine AI Nonsupine HI Nonsupine AHI Case 1 (Non-REM, non-POSA) Pre-Tx Post-Tx 0.6 2.7 50.7 40.1 51.3 42.8 0 4 40 41.9 40 45.9 Case 2 (REM, POSA) Pre-Tx Post-Tx 1 0.4 15.5 5.4 16.5 5.8 0 0 1.7 1.6 1.7 1.6 Table 8.2 Changes in AHI according to the level of sleep and sleep position AHI index NREM supine NREM nonsupine NREM total REM supine REM nonsupine REM total Case 1 (Non-REM, non-POSA) Pre-Tx Post-Tx 40.7 38.6 40 46.2 40.7 41.7 75.3 60 40 38.1 71 39.6 Case 2 (REM, POSA) Pre-Tx Post-Tx 9.8 4.3 1.9 0.5 9.1 1.6 37 9.9 0 5.8 35.3 7.3 Moreover, phenotypes such as POSA and REM-related OSA significantly influence the orthodontic diagnosis in terms of respiratory function. However, the influences remain almost uncertain. If a reliable and accurate pathophysiological pattern for each patient with OSA could be identified, a customized treatment plan would become feasible. Orthognathic surgery can change the UA morphology; therefore, it is necessary to assess respiratory function and formulate a personalized treatment plan for each patient with OSA. Accordingly, we suggest that full PSG data should be obtained before treatment planning for these patients in order to understand the AHI patterns and respiratory dynamics in different sleeping positions and during the different phases of sleep. 8.3 Maxillary Distraction Osteogenesis Maxillary distraction osteogenesis (DO) may be an effective method substituting the Le fort I osteotomy when a large amount of maxillary advancement is required. Combined maxillary DO with mandibular setback surgery can be effective in the treatment of skeletal Class III malocclusion with OSA. Here, we describe a case of an adult man with skeletal Class III malocclusion accompanied by OSA who was successfully managed by two-jaw surgery involving distraction osteogenesis (DO) instead of conventional Le Fort I osteotomy for the maxilla. Diagnosis and Treatment Plan A 42-year-old man presented with a chief complaint of mandibular protrusion. The patient diagnosed with mild OSA with obesity on the basis of PSG findings. BMI was 30 kg/m2 and AHI was 11.4 (Table 8.3). He showed a concave profile with deficient midface and protruded chin (Fig. 8.5). Cephalometric analysis demonstrated 102 T. Ono Table 8.3 Posttreatment changes of BMI, PSG, and pharyngeal airway spaces measured on lateral cephalograms BMI AHI (/hr) PNS-ad1 (mm) MAS (mm) IAS (mm) Initial 30.0 11.4 23.0 9.7 14.0 After maxillary DO 26.8 1.0 28.3 16.0 13.9 Final 27.4 1.0 25.2 13.7 12.7 PNS-ad1 Nasopharyngeal airway space, MAS Oropharyngeal airway space, IAS Hypopharyngeal airway space as defined in Fig. 1.1 Initial After Maxillary DO Final Fig. 8.5 Facial photographs and cephalometric radiographs obtained before treatment (left), after maxillary distraction osteogenesis (middle), and after treatment with mandibular setback surgery (right) a severe Class III skeletal relationship with ANB angle of −8.1°, and the pharyngeal airway widths looked normal. Bilateral class III molar and canine relationships, large negative overjet of −12.0 mm, and overbite of 3 mm were observed (Fig. 8.6). He was wearing a partial denture in the left maxillary premolar region. A panoramic radiograph showed that the maxillary right central incisor, maxillary left first and second premolars, mandibular left second molar, and mandibular right first molar 8 Modification of Orthognathic Surgery for Skeletal Class III OSA Patients 103 Initial Final Fig. 8.6 Intraoral photographs obtained before (above) and after treatment (below) were missing, with endodontic and restorative treatments in several teeth. He was diagnosed with skeletal Class III malocclusion with mandibular protrusion and maxillary deficiency accompanied by mild OSA. To improve the occlusion and facial esthetics, following treatment options can be considered: (1) mandibular setback surgery only (SSRO); (2) maxillary advancement surgery combined with mandibular setback surgery (conventional two-jaw surgery; Le Fort I osteotomy and SSRO); (3) maxillary distraction osteogenesis combined with mandibular setback surgery (SSRO). However, OSA symptoms should be concurrently improved by surgery for this patient. With mandibular setback surgery alone, the amount of mandibular displacement may exceed 15 mm, which can lead to worsening of OSA. Because the patient exhibited a significant freeway space and hypoplastic maxilla, two-jaw surgery was desirable. Moreover, forward movement of the maxilla by 10.0 mm would maintain satisfactory respiratory function and minimize the amount of mandibular setback. This could be achieved by distraction osteogenesis rather than Le Fort I osteotomy [9]. Therefore, option (3) was selected. We also instructed the patient to follow a weight-loss regimen before initiating the maxillofacial treatment. Treatment Progress and Results Maxillary DO was performed first. After 13 days of distraction, the maxilla moved anteroinferiorly and the mandible exhibited clockwise rotation (Fig. 8.7), improving facial profile. After the retention period of DO, the distraction device was removed and mandibular setback surgery was performed. The restorations and prosthesis were also corrected after completion of the orthodontic treatment finished. The patient’s BMI had decreased in the early phase of treatment. Superimposition of pre- and post-DO cephalograms revealed the increase of nasopharyngeal and oropharyngeal airway spaces after maxillary distraction (Fig. 8.7). Although these increased sagittal spaces turned back after SSRO, the AHI dropped from 11.4 to 1.0 after treatment, indicating surgical cure (Table 8.3). 104 T. Ono - Initial - After maxillary DO - After maxillary DO - Final Fig. 8.7 Superimposition of lateral cephalometric tracings obtained from before treatment (black line) to after maxillary distraction stage (red line) on the left and from after maxillary distraction stage (black line) to after treatment stage (red line) on the right 8.4 rimming of Pyriform Aperture in Case of Superior T Maxillary Impaction Skeletal Class III patients frequently require superior maxillary impaction along with maxillary advancement to accommodate the posteriorly repositioned mandible without stretching of pterygomasseteric slings. Moreover, if a skeletal Class III hyperdivergent patient has OSA symptoms, superior impaction of maxilla especially in the anterior part is needed to reduce the vertical dimension with surgical flattening of occlusal plane. In patients who undergo maxillary impaction, nasal cavity volume may be reduced without compensatory contouring at the inferior edge of pyriform aperture (Fig. 8.8). However, the efficacy of this approach remains unclear. We analyzed the influence of orthognathic surgery involving superior maxillary impaction on nasal respiratory function using computational fluid dynamics (CFD) and determined the efficacy of bone trimming at the inferior edge of the pyriform aperture. The sample comprised 10 patients with mandibular prognathism who underwent two-jaw surgery involving superior maxillary impaction and bone trimming at the inferior edge of the pyriform aperture [10]. 3D models of the nasal cavity were reconstructed from pre- and postoperative CT images and simulated using CFD. The pressure effort (ΔP) and cross-sectional area (CSA) after treatment were evaluated for the 8 Modification of Orthognathic Surgery for Skeletal Class III OSA Patients 105 Fig. 8.8 Photograph of bone trimming at the inferior edge of the pyriform aperture after Le Fort I downfracture in two-jaw surgery anterior (a), middle (m), and posterior (p) parts of the nasal cavity and the pharyngeal airway (PA). The pressure effort was defined as the product of the airway resistance and the volume flow rate. We found that the pressure effort decreased after surgery. One of the most important factors may have been the increase in CSA-NV (cross-sectional area at nasal valve) after bone trimming at the inferior edge of the pyriform aperture. Nasal ventilation worsened after surgery without trimming at the inferior edge of the pyriform aperture. These results suggest that pyriform trimming is useful for the maintenance of nasal respiratory function after orthognathic surgery involving superior maxillary impaction. In addition, it is suggested that changes in the pressure effort are more likely to occur in the nasal cavity than in the pharyngeal airway in these patients. 8.5 axillary Horseshoe Osteotomy in Case of Superior M Maxillary Impaction It is well known that conventional Le Fort I osteotomy for maxillary impaction seemed to be disadvantageous from the viewpoint of respiratory function. Horseshoe osteotomy was first introduced in order to impact the maxillary molar region by up to 5 mm without compromising the nasal airway and causing nasal deformities. A major difference between horseshoe osteotomy and conventional Le Fort I osteotomy is that the hard palate remains pedicled on the nasal septum and the vomer bone in the former procedure (Fig. 8.9). PNS does not move upward in horseshoe osteotomy. Horseshoe osteotomy has two additional advantages. First, the nasal cavity is not compromised despite a decrease in the facial height. Second, the maxillary incisor 106 T. Ono 5 Displacement of PNS (mm) 4 –40.0 3 2 1 –30.0 –20.0 –10.0 0 0.0 10.0 20.0 30.0 40.0 –1 Increment of upper airway volume (%) Fig. 8.9 Correlation of displacement of PNS and increment of upper airway volume. Osteotomy line indicates horseshoe-shaped osteotomy inclination can be arbitrarily adjusted. Recently, we treated 15 patients with maxillomandibular surgery involving horseshoe osteotomy. Nine of the patients exhibited an increase in the UA volume, while six exhibited a decrease (Fig. 8.9). The reason why some patients showed a large amount of increase or decrease remains unknown; however, for patients exhibiting a significant decrease, we believe that a difficult configuration between the dentoalveolar and midpalatal segments may have played a role. Because the palatal root of the molar is sometimes located on the inner side of the cutting line, the dentoalveolar segment collides with the midpalatal segment, and both of them move upward together. This theory should be confirmed in further studies. 8 Modification of Orthognathic Surgery for Skeletal Class III OSA Patients 107 Clinical Pearls of Orthognathic Surgery for Skeletal Class III OSA Patients • When orthognathic surgery is planned for skeletal Class III patients who have OSA symptoms or risks of occurrence, two-jaw surgery is preferred to mandibular one-jaw surgery to compensate the pharyngeal narrowing by mandibular setback. • In severe OSA patients with skeletal Class III patients, maxillary one-jaw surgery without mandibular setback can be considered. • In OSA patients requiring large amount of maxillary advancement, maxillary distraction osteogenesis can substitute Le Fort I surgery. • Superior maxillary impaction is usually combined with maxillary advancement and mandibular setback surgery in skeletal Class III patients with risks of OSA. In case, trimming of pyriform aperture should be performed to compensate the decreased nasal cavity and at least to maintain the nasal airflow especially on the nasal valve area. • Maxillary horseshoe osteotomy, which is the modification of Le Fort I osteotomy for superior maxillary impaction, can be applied not to compromise the nasal airway. References 1. Tan SK, Leung WK, Tang ATH, Zwahlen RA. Effects of mandibular setback with or without maxillary advancement osteotomies on pharyngeal airways: an overview of systematic reviews. PLoS One. 2017;12(10):e0185951. 2. Tamari K, Shimizu K, Ichinose M, Nakata S, Takahama Y. Relationship between tongue volume and lower dental arch sizes. Am J Orthod Dentofacial Orthop. 1991;100(5):453–8. 3. Sakai K, Shimazaki K, Kokai S, Fukuyama E, Ono T. Effects of different surgical procedures on the upper-airway dimension in subjects with mandibular prognathism. Jap J Jaw Deformities. 2012;22(4):239–43. 4. Holty J-EC, Guilleminault C. Maxillomandibular advancement for the treatment of obstructive sleep apnea: a systematic review and meta-analysis. Sleep Med Rev. 2010;14(5):287–97. 5. Ishida T, Manabe A, Yang S-S, Watakabe K, Abe Y, Ono T. An orthodontic-orthognathic patient with obstructive sleep apnea treated with Le Fort I osteotomy advancement and alar cinch suture combined with a muco-musculo-periosteal VY closure to minimize nose deformity. Angle Orthod. 2019; https://doi.org/10.2319/052818-406.1. 6. Muradin M, Rosenberg A, Van Der Bilt A, Stoelinga P, Koole R. The effect of alar cinch sutures and VY closure on soft tissue dynamics after Le Fort I intrusion osteotomies. J Craniomaxillofac Surg. 2009;37(6):334–40. 7. Okushi T, Tonogi M, Arisaka T, et al. Effect of maxillomandibular advancement on morphology of velopharyngeal space. J Oral Maxillofac Surg. 2011;69(3):877–84. 8. Shepard J, Thawley SE. Localization of upper airway collapse during sleep in patients with obstructive sleep apnea. Am Rev Respir Dis. 1990;141(5 Pt 1):1350–5. 9. Baker DL, Stoelinga PJ, Blijdorp PA, Brouns JJ. Long-term stability after inferior maxillary repositioning by miniplate fixation. Int J Oral Maxillofac Surg. 1992;21(6):320–6. 10. Bloching MB. Disorders of the nasal valve area. GMS Curr Topi Otorhinolaryngol Head Neck Surg. 2007;6:Doc07. 9 Mandibular Advancement Device for Elderly OSA Patients Su-Jung Kim and Young-Guk Park Contents 9.1 M echanism to Open Pharyngeal Airway 9.2 Patient Selection: Indication and Contraindication 9.2.1 Predictors of Success 9.2.2 Rule Out The Poor Responders 9.3 Appliance Design 9.3.1 Monoblock Versus Biblock Types 9.3.2 Titratable Versus Non-titratable Types 9.3.3 Custom-Made Versus Prefabricated Types 9.3.4 Mono-layer Versus Dual-layer Types 9.4 Bite Registration and Jaw Titration 9.4.1 Where to Establish the Mandibular Position? 9.4.2 How to do the Titration? 9.4.3 Why and How to Take Bite Registration? 9.5 Side Effects and Their Management 9.6 Instruction Manual for the Patients 9.7 Cases References 9.1 109 110 111 111 111 112 112 113 113 113 113 115 115 117 119 120 129 Mechanism to Open Pharyngeal Airway Advancement of the mandible produces three-dimensional configurational changes and improves the patency of the velopharynx and the oropharynx are attached [1–3]. Mandibular advancement stretched the soft palate stiffening the S.-J. Kim (*) Department of Orthodontics, Kyung Hee University School of Dentistry, Seoul, South Korea e-mail: ksj113@khu.ac.kr Y.-G. Park Department of Orthodontics, Kyung Hee University School of Dentistry, Seoul, South Korea e-mail: ygpark@khu.ac.kr © Springer Nature Switzerland AG 2020 S.-J. Kim, K. B. Kim (eds.), Orthodontics in Obstructive Sleep Apnea Patients, https://doi.org/10.1007/978-3-030-24413-2_9 109 110 S.-J. Kim and Y.-G. Park Fig. 9.1 Lateral cephalometric images at initial (left) and with wearing MAD. Increased pharyngeal airway widths are observed through forward and upward repositioning of soft palate, tongue base, hyoid bone as well as mandible b a Lateral pharyngeal walls 12 cm Palatoglossus Genioglossus Fig. 9.2 (a) A merged image of stomatognathic neuromuscular system on the lateral cephalogram, which describes the mechanism of opening airway by key muscles of genioglossus, palatoglossus, and lateral pharyngeal walls. (b) With wearing MAD, minimum cross-sectional area on the CBCT axial section increased transversely more than anteroposteriorly, as a result of harmonized action of the pharyngeal dilator muscles velopharynx by palatoglossus muscles as well as repositioned the tongue base and hyoid forward and upward by genioglossus muscles (Fig. 9.1). Moreover, transverse diameter of minimal cross-­sectional area in the velopharynx increased greater than the sagittal one due to the connection between lateral pharyngeal walls and the soft palate (Fig. 9.2). It is influenced by not only upper-airway dimensions but also complex neuromuscular reflex interactions. 9.2 Patient Selection: Indication and Contraindication Because as many as 50% of subjects discontinue use of an OA in the first year [4], a better understanding of what prognostic factors exist for OAT success is required. 9 Mandibular Advancement Device for Elderly OSA Patients 9.2.1 111 Predictors of Success As a general rule, MAD has been indicated for mild-to-moderate OSA patients or CPAP-intolerant severe OSA patients. However, the severity of OSA simply indicated by AHI is not enough to predict the effectiveness of MAD due to the various underlying pathophysiologies among OSA patients, who are differently affected by the mechanism of action of MAD. Currently, the reference of patient selection has been changed from the OSA severity to the patient’s phenotype. MAD can be used as a sole treatment for the patients with mild-to moderate “pharyngeal collapsibility,” without low arousal threshold, high loop gain, and/or UA muscle dysfunction [5]. Based on this, the strong predictors of success with MAD include nonobese craniofacial anatomic phenotype with retruded mandible affecting the pharyngeal collapsibility, who is represented by hypopnea-dominant type (HI≥2xAI), or supine-dependent positioner type (Supine AHI≥2xAHI) of OSA. 9.2.2 Rule Out The Poor Responders • The central sleep apnea (CSA) patient or multiple comorbid patient. • The OSA patient in severe dysfunctional state with poor dilator muscle responsiveness: The mechanism of MAD to open pharyngeal airway does not work to this patient. CPAP is inevitable. • The patient suffering from frequent arousal and/or insomnia: MAD may increase arousal or disturb falling asleep in this patient. • The obese patient with heavy submental fat pads and large neck circumference: Fat tissues may act as dominate factors affecting the pharyngeal collapsibility. • The patient with hypertrophic para-pharyngeal soft tissues: Soft tissue surgery is firstly indicated. • The patients with TMD, multiple teeth missing, poor periodontal support, or active periodontitis: Greater side effects than airway effect are expected [6]. • The patient with severe craniofacial deformity with severe hyperdivergent vertical pattern and/or nasal cavity obstruction: Maxillomandibular surgery and/ or nasomaxillary expansion is firstly required. MAD usually increases vertical dimension, which cancels the oropharyngeal opening by mandibular rotation especially in skeletal Class II hyperdivergent pattern. MAD can hardly improve the respiration in patient with nasal obstruction. 9.3 Appliance Design As far as appliance design is concerned, any uniform type is not satisfactory to all patients. The thing is that customized, adjustable, and jaw-retainable type MAD is preferred for better effectiveness, efficiency, and compliance. 112 9.3.1 S.-J. Kim and Y.-G. Park Monoblock Versus Biblock Types Clinicians tend to prefer two-piece biblock type, since it allows interarch freedom, motility of lower jaw, and possibility of gradual titration. However, it usually require larger vertical jaw opening to yield the thickness of two pieces and it hardly holds the mandible in patients with severe mouth opening during sleep. The one-piece monoblock type can be simply fabricated with low cost, and it has a virtue of jaw-­ retainable type. However, it may induce more jaw and muscle discomforts in the patients with heavy masseter activity or unstable TMJs, and cannot allow the mandibular movement at all. Many papers [7, 8] reported higher efficacy and tolerance in biblock type than in monoblock type in terms of “titration capability,” whereas a few papers [9, 10] found greater AHI reduction in monoblock type due to its “jaw-­ retainable feature.” Accordingly, individualized prescription is more important than any preference. The biblock type is favorable for the patients with hyperactive jaw muscles, bruxism, and high risk of TMD symptoms. If the biblock type is prescribed, elastic bands need to be applied to the patients with persistent mouth opening during sleep. Conversely, some patients may benefit from monoblock design if they feel discomforts from too much freedom of movement. 9.3.2 Titratable Versus Non-titratable Types Due to dose-dependent effects, a clinical titration can improve the effect of MAD without significant side effects, and increase the amount of achievable advancement through gradual patient’s adaptation. Recent consensus is that the adjustable MAD provides a greater reduction in subjective daytime sleepiness and snoring than the fixed MAD (Fig. 9.3), although there is still controversy on the objective Fig. 9.3 Various designing of MAD. (a) Monoblock type; (b) Twin-block type; (c) EMA; (d) Sleep herbst; (e) Somnomed; (f) Narval CC 9 Mandibular Advancement Device for Elderly OSA Patients 113 improvement of AHI and oxygen saturation. Considering its disadvantage requiring weekly or biweekly visits for 4–6 months, however, one-step jaw positioning or minimum titration procedures need to be contemplated depending on the patient’s condition. 9.3.3 Custom-Made Versus Prefabricated Types Lately, thermoplastic prefabricated MAD is being imprudently used under no specialist’s care, because of low cost and one-step fabrication at the day of first visit. According to the meta-analysis by AASM task force [11], noncustomized MAD decreased AHI far less than customized MAD, and could not improve oxygen saturation and ESS scores significantly. With noncustomized MAD, the amount and the direction of mandibular advancement cannot be controlled, and the amount of advancement is very limited especially in Class III OSA patients with already protruded mandible. Due to high flexibility of the material with no teeth-supported design, dentoalveolar side effects with occlusal changes and morning tooth pain inevitably occur in noncustomized MAD. Long-term health management of teeth, occlusion, periodontium, and TMJ should be attained with customized MAD under specialist’s care. Immediately adapted thermoplastic MAD may be a temporary strategy just to screen the response to MAD therapy. 9.3.4 Mono-layer Versus Dual-layer Types Regarding the materials of MAD, dual-layer type, which is composed of inner soft layer and outer hard layer, is preferred for maximum covering of teeth surfaces without discomfort during insertion and removal. Overall rigidity with inner flexibility is favorable for minimizing unwanted teeth movement or pain by dispersing the generated forces from the appliance to the teeth. Flexible monolayer type used in some prefabricated appliances is apt to induce teeth movement with poor durability. Rigid monolayer type is hard to cover teeth surfaces enough to resist dentoalveolar side effects especially in elderly patients with severe crowing and poor periodontal support. 9.4 Bite Registration and Jaw Titration 9.4.1 Where to Establish the Mandibular Position? 9.4.1.1 Determination of the Amount of Mandibular Advancement Many studies have reported dose-dependent improvement of airway dimension or AHI reduction by MAD, and recommended the range of 50–75% of maximum advancement [3, 12–14]. According to a recent systematic review and meta-analysis 114 S.-J. Kim and Y.-G. Park [15], however, the AHI improvement resulted to be not proportional to the mandibular advancement increase. Most literatures did not provide the rationale to choose the advancement amount due to a high interindividual variability. Because of different anatomic features and physiologic responses across the patients, it is important to individualize the amount of mandibular protrusion for a compromise between efficacy and side effects by starting from 50% of maximum protrusion in each patient, which is called “titration.” 9.4.1.2 Determination of the Amount of Vertical Opening Once the final level of advancement is established, the amount of vertical opening (VO) should be determined considering the patient’s compliance and the thickness of acrylic overlay. There is a concern that excessive VO may offset the airway opening mechanism of mandibular advancement. Vroegop et al. [16] suggested that VO was associated with a significant increase in total respiratory resistance leading to increased pharyngeal collapsibility. In contrast, Pitsis et al. [17] indicated that the amount of VO did not affect treatment efficacy, but that patients preferred minimal VO as they were more comfortable. Ferguson et al. [18] concluded that the effect of VO on the efficacy of MAD remains unclear. Considering that patient’s comfort is important for the compliance and continuation of MAD, minimum VO is recommended, particularly in the patient requiring large amount of mandibular advancement. 9.4.1.3 Verification of Midline Position To determine the lateral position of the advanced mandible, firstly check the lower midline to the upper midline in the maximum intercuspal position (MICP) and the presence of mandibular lateral shift from centric relation (CR) to the MICP. In case of no midline deviation without mandibular shift, bring the mandible straight forward maintaining the upper and lower midline relationship in bite registration. It should be carefully checked if the patient shows mandibular lateral deviation along with mandibular opening possibly due to unilateral disk displacement. If so, two-­piece appliance design allowing lateral jaw movement can be considered. In case that lower midline deviation exists, however, the presence of mandibular shift accompanied by unilateral condylar displacement should be firstly checked. If the lower midline is deviated due to the mandibular shifts to the same side accompanied by condylar displacement on the contralateral side, bringing the mandible straight forward may adversely affect the TMJ. The side where the condyle is more displaced at MICP needs to be less pulled to reduce the lower midline deviation in a protruded mandibular position. On the other hand, if the patient has deviated lower midline without mandibular shift and no condylar displacement on both sides, the deviated lower midline is originated from mandibular skeletal asymmetry. Thus, straight forward mandibular advancement maintaining the midline relationship would be favorable for the same amount of condylar displacement with MAD. Careful initial examination on the condylar position, disk derangement, and static and dynamic occlusion should not be ignored. 9 Mandibular Advancement Device for Elderly OSA Patients 9.4.2 115 How to do the Titration? There are three protocols of titration: subjective, objective, and multiparametric titration. Subjective titration is based on the patient’s self-reported improvement of symptoms and patient’s physical limits regardless of objective improvement of OSA severity. MAD is advanced until the maximum comfortable protrusion of the patient is reached without causing pain or discomfort; therefore, it takes long time over several months. Objective titration is based on the overnight PSG only. One-­ night titration can be carried out either with temporary appliance by awakening the patient to advance the mandible, or without awakening the patient by using a motorized advancement system like remotely controlled mandibular positioner (RCMP). This may offer more accurate titration and can be used to predict the therapeutic efficacy of MAD in each patient; however, there still exist technical difficulties and practical limitation with the device. Third, multiparametric titration, which is composed of subjective and objective criteria, may compensate the discrepancy between the two outcomes. Ideally, the titration procedure is very useful for the prediction or evaluation of MAD efficacy. Nevertheless, it is not easy to utilize PSG and RCMP for the purpose of objective or multiparametric titration to every patient who was referred to dental clinic. Moreover, there is a practical issue that most patients are not willing to accept weekly visits for a long period over 3–4 months for subjective titration. For this reason, shorter and simpler method of bite registration to decide the mandibular position needs to be considered. 9.4.3 Why and How to Take Bite Registration? Bite registration in the patient’s mouth by a dentist is very important: (1) to reduce the number of visit and the need of subjective titration of the device; (2) to reproduce the three-dimensionally reconstructed maxillomandibular relationship from the mouth to the articulator; (3) to minimize TMJ and muscle discomforts for better compliance. The recommended step-by-step procedure of bite registration using the gauge is as follows: • Ask the patient to snore in an inclined supine rest position. • Instruct the patient to protrude the mandible as far as possible, and measure the distance between the tips of upper and lower central incisors (Fig. 9.4a). • Calculate the patient’s total protrusion amount by adding the initial overjet, and get the amount of 50% protrusion. Set this value into the gauge. • Position the gauge on the patient’s upper dentition for the upper central incisors to touch the upper notch. Guide the patient’s mandible to protrude until the lower incisor tips touch the lower incisor notch (Fig. 9.4b). • Check the amount of vertical opening to be minimal and verify the initial midline relationship. 116 S.-J. Kim and Y.-G. Park • Ask the patient to snore again with gauge in the mouth. The patient can notice that the snoring noise decreases or disappears. If it is not enough, adjust the screw on the gauge for the mandible to be more protruded. Check again if the noise disappears. At the same time, ask the patient if he or she feels discomfort or pain on the TMJ and masseter muscle area. • The dentist can find a clinically optimal trial point as a balanced position between the noise release and the free of symptoms. • Inject the light body silicon material into the gap between upper and lower teeth and the gauge using a bite registration automix system. The dentist can verify the mandibular position relative to the maxillary arch by inserting the registered bite between the upper and lower stone models (Fig. 9.4c). • Additional minor adjustment of mandibular position can be done at the day of delivering MAD (Fig. 9.4d). Fig. 9.4 Procedure of bite registration. (A) Instruct the patient to protrude the mandible as far as possible, and measure the distance between the tips of upper and lower central incisors. (B) Position the gauge on the patient’s upper dentition for the upper central incisors to touch the upper notch. Guide the patient’s mandible to protrude until the lower incisor tips touch the lower incisor notch. (C) Inject the light body silicon material into the gap between upper and lower teeth and the gauge. (D) The constructed mandibular position can be confirmed at the day bite registration 9 Mandibular Advancement Device for Elderly OSA Patients 9.5 117 Side Effects and Their Management The management of side effects is essential to maximize treatment adherence and the clinical effectiveness of MAD. In the initial period of MAD use, mild transient side effects, such as morning jaw pain, masticatory muscle tenderness, tooth pain, gum irritation, and dry mouth or excessive salivation, commonly appear. However, these can be prevented or quickly disappear with well-designed and well-titrated MAD by specialists (Fig. 9.5). Otherwise, patient’s poor adherence to MAD will cause discontinuation of its use, or persistent TMD with dentofacial and periodontal changes may occur in case of long-term wearing. In 2017, the American Academy of Dental Sleep Medicine developed a consensus on the management of side effects created by MAD, classifying into categories of temporomandibular joint-related, intraoral tissue-related, occlusal changes, damage to teeth and restorations, and appliance issues. Transient jaw pain, masticatory muscle tenderness, and tooth pain occurring in the morning usually disappears spontaneously after eating breakfast or during the day. Therefore, (1) watchful waiting just confirming the original TMJ problems, (2) palliative care including hot-pack, massage, and jaw stretching, and (3) isometric contraction and passive jaw exercise are considered first-line treatments [19]. (4) Decreasing the titration rate will be helpful for fast initial adaptation. Regarding the relationship between MAD and TMD, a controlled study [20] suggested that the possible development of TMD is not a contraindication for MAD. Because the occurrence of the pain-related TMD is the highest in 2–3 months 2009.12.24; MAD delivery 2018.04.04: 8y 6m after MAD Fig. 9.5 Intraoral photos comparing the occlusion between initial and 8.5 years after daily wearing of well-fitted MAD in an OSA patient aged 62 years old. No occlusal changes are observed 118 S.-J. Kim and Y.-G. Park Fig. 9.6 Intraoral photos showing occlusal changes and periodontal side effects after 3-year wearing of poorly designed MAD. Upper dentition shifted backward and lower dentition shifted forward into Class III occlusal relationship with decreased overjet and overbite. Serious alveolar bone dehiscence on the lingual side of lower incisors due to the root-lingual and crown-labial movement of lower incisors with MAD of using MAD due to the strain in the muscles or ligaments of temporomandibular complex, however, this tends to return to the baseline during 1-year follow-up through adaptive capacity of the complex. MAD does not cause mandibular dysfunction. Accordingly, if the patient complains persistent TMD with MAD, the possible reasons involve improper patient selection having original TMJ problem, inappropriate appliance type, improper jaw titration with excessive advancement and/or vertical opening, and long wearing time. In case, following managements are considered: (1) decreasing wearing time less than 6 h/night; (2) reducing the titration rate with minimum vertical opening; (3) temporarily discontinuing use or intermittent use of MAD; (4) changing MAD design to yield proper freedom of jaw movement and posterior disclusion between the two pieces; (5) permanently discontinuing use. The most frequently concerned side effects are occlusal changes such as decreased overjet and overbite with lower incisor proclination and upper incisor retroclination, shifting into Class III molar and canine relationship (Fig. 9.6). Depending on the appliance type, posterior openbite or anterior openbite may occur. Along with progressive lower incisor proclination, gingival recession and tooth mobility can be followed in the elderly patients. Since patients are often unaware of or tolerant of these changes, periodic check-up every 6 months is highly recommended. First of all, the occlusal changes should be differentiated whether they are originated from true teeth movement or from changed habitual jaw position. When the habitual mandibular position has been being changed forwardly, morning jaw exercise and muscle stretching is firstly recommended. On the other hand, to minimize dentoalvelar side effects, following managements are considered: (1) decreasing wearing time; (2) chewing hard foods or gum right after MAD removal; (3) using 9 Mandibular Advancement Device for Elderly OSA Patients 119 morning occlusal guide to recover the occlusal relationship; (4) changing appliance design to cover all surfaces of all teeth using specified dual materials; (5) temporarily or permanently discontinuing use of MAD if the changes are of seriously concern to the patient. Sometimes, patients complain excessive salivation or dry mouth wearing MAD. Excessive salivation (drooling) only rarely precludes use because it is not very often and typically transient over the first few weeks. Therefore, (1) preceding explanation and reassurance usually suffice to manage drooling. (2) Modification to the appliance needs to be considered to decrease vertical dimension allowing for easier lip sealing and swallowing. (3) Certain medication can be utilized to decrease salivation in case that patient’s medical history does not contraindicate such use. Dry mouth is reported more often than drooling but also rarely precludes use, because it usually subsides in a few weeks. However, it may continue in patients with mouth breathing and/or nasal obstruction, or in patients having some medication. Therefore, (1) patients should be informed in advance of possible dry mouth, and watchful waiting with reassuring them mostly suffices. (2) Modification to the appliances can be considered to decrease vertical dimension to encourage lip sealing and to hold the mandible not to open. (3) Soaking the appliance in cold water prior to wearing it can be helpful for hydration. (4) When medications are responsible for dry mouth, consultation with the patient’s physician is needed. (5) Tobacco, alcohol, caffeine, sugary or salty foods, alcoholic mouth rinse should be avoided prior to bedtime. (6) When nasal airway resistance appears to lead to mouth breathing during sleep, refer the patient to an otolaryngologist. As an appliance issue, appliance breakage commonly occurs due to acute stress or accumulated fatigue from the resistance of stomatognathic system. It has been reported that some appliance designs are prone to the breakage especially due to broken components like telescopic metal bars, acrylic flanges, clasps, etc. [21, 22]. Here is dentist’s role to decide if repair of the appliance would be enough or replacement with different design is required. If the breakage occurs repeatedly, the patient’s sleep behavior and anatomic variation should be firstly checked. Botox injection on the heavy master muscles can be considered to the patient with hyperactive masseteric activity in low angle skeletal pattern. 9.6 Instruction Manual for the Patients • Wear it after tooth-brushing and device-cleaning. • Keep wearing it only during sleep (less than 6 hours). Remove it right after wake-up. • Make sure to position the tongue forward to touch the appliance. Not to evade it. • MAD-titrated PSG evaluation is highly recommended to check your objective improvement of apnea and oxygen saturation level. • This appliance has common side effects like myofascial pain, TMJ discomforts, tooth pain, different bite, or gum irritation. You don’t need to worry about them since they usually disappear in 2–3 months. 120 S.-J. Kim and Y.-G. Park • Have breakfast right after removing the MAD for the rebound of tissues. • Warm massage and jaw stretching in the morning is helpful for fast recovery of discomforts. • Sometimes, the side effects may persist despite your regulatory compliance and periodic visit. In case, reduction of jaw titration, replacement with other design, temporary discontinuation may be required for you. • If pain or discomforts worsen without subjective improvement of OSA symptoms, you may not be a good responder to MAD. Other treatment options like surgery or PAP should be considered. • Even after the completion of titration, follow-up check is very important for long-term adherence to MAD: 1 month → 3 months → 6 months thereafter. 9.7 Cases Case 1: A Good Responder to MAD with Noncraniofacial Phenotype A 63-year-old male visited the ENT department with the chief compliant of witnessed apnea, and diagnosed as severe OSA. He was referred to the orthodontic department for oral appliance, since he refused wearing the PAP. He had been taking a medicine for the control of hypertension for 3 years. He was obese with BMI of 26.2. He was not suffering from snoring and excessive daytime sleepiness subjectively, as supported by ESS scores of 5. According to PSG findings, however, he belonged to severe OSA with AHI of 50.7 and RDI of 62.9, hypopnea-dominant type (AI, 13.0 ≪ HI, 37.7), and positioner (supine-AHI 75.1 with supine sleep time of 52.8%). The lowest oxygen saturation was 81% and ODI was 25.8. He had very low sleep efficiency of 74.3% possibly associated with his chief complaint of witnessed apnea. In the clinical examination, he showed a straight facial profile with well-­ developed chin, but had typical facial triads such as short throat length, obtuse cervico-­mental angle, and thick neck circumference, in relation to obesity (Fig. 9.7). Intraorally, the palatal vault configuration was within normal limit, and neither tongue-related obstacles nor hypertrophic pharyngeal soft tissues were noted. He had acceptable occlusion, in spite of mild dento-alveolar arch constriction and deep overbite. Generalized teeth attrition was present due to the nocturnal bruxism. Lateral cephalometric image showed normal pharyngeal airway dimension in relation to normal craniofacial skeletal pattern (Fig. 9.8), which means that he had no skeletal and soft tissue anatomical contributing factors to severe OSA. He represented normal maxilla and mandible, normal vertical divergency, and normal hyoid and tongue position. When we matched with the finding of Muller’s maneuver, we found that the main obstruction occurred in the oropharyngeal area by lateral collapse of lateral pharyngeal wall rather than sagittal collapse. Taken together, he was diagnosed as severe OSA with high pharyngeal collapsibility of lateral pharyngeal walls. Although he might belong to the nonanatomical phenotype predominantly, we decided to apply MAD with positional 9 Mandibular Advancement Device for Elderly OSA Patients 121 Fig. 9.7 Clinical examination, taking the intraoral photos to check the patient’s intraoral and occlusal status and taking the facial photos to analyze the patient’s facial profiles. In this case, the patient showed a straight facial profile with well-developed chin, but had typical facial triads such as short throat length, obtuse cervico-mental angle, and thick neck circumference, in relation to obesity therapy to this patient for the following reasons: (1) He strongly refused the PAP and not indicated to soft tissue or skeletal surgeries; (2) He showed hypopnea-dominant and supine-­ dependent type with long supine sleep time, which might respond favorably to the MAD mechanism; (3) He had healthy condyles without TMD history and favorable periodontal tissue support. As a result, the patient was satisfied with the MAD therapy showing good cooperation of daily wearing. When we compared two lateral cephalograms taken without and with MAD in the mouth, oropharyngeal airway enlargement was noted (Fig. 9.9). He was happy to feel deeper sleep with MAD than before, supposedly due to improved sleep efficiency and decreased bruxism activity. He only felt mild discomfort on the masseter muscles area temporarily after wake-up, in relation to increased masseter activity with MAD as seen in the electromyographic (EMG) finding (Fig. 9.10). Interesting thing is that masseter and temporal activity was rather decreased with MAD in a clenching state, which implies the release of sleep bruxism. He did not have TMJ and teeth pain. According to the MAD-titration PSG (Table 9.1), sleep efficiency was improved from 74.3% to 86.4%. AHI was decreased by 72% from 50.7 to 14.0. Complete apnea almost disappeared (AI 0.5; HI 13.5). Supine-AHI dropped by 78% from 75.1 to 16.1. The increment of the lowest oxygen saturation was not sufficient (86%), but ODI was significantly decreased from 25.8 to 6.1. Snore time was also reduced from 51.3% into 15.5%. MAD application was successful in this patient. 122 S.-J. Kim and Y.-G. Park Fig. 9.8 The pictures of Muller’s maneuver taken at velo-, oro-, and hypopharyngeal airway to see main obstruction site, which are matched with the static pharyngeal airway morphology on the lateral cephalogram. Lateral collapse of oropharyngeal airway is noted Fig. 9.9 Lateral cephalograms taken at initial (left) and wearing MAD (right). Oropharyngeal enlargement was seen with mandibular advancement 9 Mandibular Advancement Device for Elderly OSA Patients 123 Fig. 9.10 Comparison of electromyographic (EMG) findings between without and with wearing MAD in a rest state and in a clenching state, respectively. The activities of temporal and masseter muscles increased with MAD in rest state; however, they decreased in clenching state, which implies the release of sleep bruxism. TA (red), anterior belly of temporal muscles; MM (green), masseter muscles; SCM (purple), sternocleidomastoid muscles; DA (blue), anterior belly of digastric muscles Table 9.1 Comparison of polysomnographic (PSG) summaries between initial and MAD titration. Wearing MAD, AHI was reduced by 72% indicating successful response to MAD 124 S.-J. Kim and Y.-G. Park Case 2: A Good Responder to MAD with Craniofacial Anatomical Phenotype A 48-year-old male visited the ENT department with chief complaint of severe snoring, witnessed apnea, and excessive daytime sleepiness (EDS). He was referred to the orthodontic department for oral appliance before trying PAP therapy. He had no medical history of diabetes or hypertension and no medication. He was obese with BMI of 27.2. ESS scores was 19, indicating the possibility of severe OSA. According to PSG findings, however, he belonged to moderate OSA with AHI of 19.8 and RDI of 26.9, hypopnea-dominant type (AI, 3.2 ≪ HI, 16.6), and positioner (supine-AHI 32.8). These findings indicated favorable response to MAD; however, the lowest oxygen saturation dropped to the level of 83% although ODI was 5.6. He snored during 37.1% of sleep time. In the clinical examination, he showed a convex profile with retruded chin, with short throat length and obtuse cervico-mental angle. The neck circumference was normal in spite of high BMI (Fig. 9.11). Intraorally, the palatal vault configuration was within normal limit, and neither tongue-related obstacles nor hypertrophic soft tissues were observed. He showed Class II malocclusion with large overjet and deep Fig. 9.11 Clinical examination, taking the intraoral photos to check the patient’s intraoral and occlusal status and taking the facial photos to analyze the patient’s facial profiles. In this case, he showed a convex profile with retruded chin, with short throat length and obtuse cervicomental angle. The neck circumference was normal in spite of high BMI and intraorally, the palatal vault configuration was within normal limit, and neither tongue-related obstacles nor hypertrophic soft tissues were observed. He showed Class II malocclusion with large overjet and deep overbite 9 Mandibular Advancement Device for Elderly OSA Patients 125 Fig. 9.12 Panoramic film of the patient’s initial visit. He had multiple prosthesis including five implants and generalized alveolar bone loss overbite. He had multiple prosthesis including five implants and generalized alveolar bone loss in the panoramic film (Fig. 9.12). Lateral cephalometric image showed severely constricted oropharyngeal airway in relation skeletal Class II hyperdivergent pattern with retrognathic small mandible (Fig. 9.13), which implied that he had craniofacial anatomical contributing factors to OSA. He had long soft palate and posteriorly displaced hyoid and tongue base. When we matched with the finding of Muller’s maneuver, we found that velo- and oro-pharyngeal airway was collapsed anteroposteriorly. CBCT analysis confirmed these findings in three dimensions (Fig. 9.14). Total airway volume was reduced, and minimum cross-sectional area was severely constricted in the oropharyngeal level. In addition, CBCT revealed no transverse skeletal risk factors like nasomaxillary constriction and showed favorable TMJ condition (Fig. 9.15). Taken together, he was diagnosed as moderate OSA with craniofacial contributing factors and obesity. We recommended MAD with weight control as a primary option to this patient anticipating favorable response, since he could not afford skeletal surgery and orthodontic treatment. Implant-supported prosthesis would be good anchorages to prevent occlusal side effect by resisting the generating force from MAD. The patient was satisfied with released snoring and EDS. Lateral cephalograms taken without and with MAD showed that oropharyngeal airway was clearly enlarged with forward and upward displacement of tongue base and soft palate along with mandibular advancement (Fig. 9.16). EMG analysis displayed increased activity of digastric muscles wearing MAD in a rest position (Fig. 9.17), but he did not feel any early discomfort on the muscles, TMJs, and teeth. According to the MAD-titration PSG (Table 9.2), AHI was decreased by 65.2% from 19.8 to 6.9. 126 S.-J. Kim and Y.-G. Park Fig. 9.13 The pictures of Muller’s maneuver taken at velo-, oro-, and hypo-pharyngeal airway to see main obstruction site, which are matched with the static pharyngeal airway morphology on the lateral cephalogram Fig. 9.14 CBCT images of the patient. We found that velo- and oro-pharyngeal airway was collapsed anteroposteriorly. CBCT analysis confirmed these findings in three dimensions 9 Mandibular Advancement Device for Elderly OSA Patients 127 Fig. 9.15 CBCT images of the patient’s condyle head. CBCT analysis revealed no transverse skeletal risk factors like nasomaxillary constriction and favorable TMJ condition Initial Wearing MAD Fig. 9.16 Lateral cephalograms taken at initial (left) and wearing MAD (right). Oropharyngeal enlargement was seen with mandibular advancement Complete apnea almost disappeared (AI 0.4; HI 6.5). Supine AHI dropped by 70.7% from 32.8 to 9.6. The lowest oxygen saturation improved up to 94% with ODI of 0.1. Snore time also reduced from 37.1%to 12.4%. MAD application was successful in this patient. 128 S.-J. Kim and Y.-G. Park Rest- sitting Rest- Clenching MAD- sitting MAD- Clenching Fig. 9.17 Comparison of electromyographic (EMG) findings between without and with wearing MAD in a rest state and in a clenching state, respectively. TA (red), anterior belly of temporal muscles; MM (green), masseter muscles; SCM (purple), sternocleidomastoid muscles; DA (blue), anterior belly of digastric muscles Table 9.2 Comparison of PSG parameters between initial and MAD titration states 9 Mandibular Advancement Device for Elderly OSA Patients 129 Clinical Pearls of Mandibular Advancement Device (MAD) • Mechanism: MAD opens the oropharyngeal airway three dimensionally (laterally greater than sagittally) through the actions of genioglossus, palatoglossus, and lateral pharyngeal walls. • Indication: MAD would be effective for the patients with nonobese, hypopnea-dominant, and supine-dependent OSA phenotypes, and with craniofacial anatomical phenotype of retruded mandible and nonhyperdivergent vertical pattern. MAD can be an alternative option to the PAP-­ intolerant elderly patients even though they belong to the nonanatomical phenotype. • Contraindication: Rule out the poor responders having not only severely dysfunctional muscle responsiveness, high loop gain, high obesity, but also temporomandibular disorder, multiple teeth missing, and active periodontitis. • Appliance type: Individualized MAD design allowing jaw titration will increase the patient’s cooperation and success rate. • Complication: Short-term and long-term side effects should be managed by specialized dentists. References 1. Schmidt-Nowara W, Lowe A, Wiegand L, Cartwright R, Perez-Guerra F, Menn S. Oral appliances for the treatment of snoring and obstructive sleep apnea: a review. Sleep. 1995;18(6):501–10. 2. Ramar K, Dort LC, Katz SG, et al. Clinical practice guideline for the treatment of obstructive sleep apnea and snoring with oral appliance therapy: an update for 2015. J Clin Sleep Med. 2015;11(07):773–827. 3. 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Impact of type D personality on adherence to oral appliance therapy for sleep-disordered breathing. Sleep Breath. 2013;17(3):985–91. 9. Milano F, Mutinelli S, Sutherland K, et al. Influence of vertical mouth opening on oral appliance treatment outcome in positional obstructive sleep apnea. J Dent Sleep Med. 2018;5(1):17–23. 10. Lee WH, Wee JH, Lee CH, et al. Comparison between mono-bloc and bi-bloc mandibular advancement devices for obstructive sleep apnea. Eur Arch Otorhinolaryngol. 2013;270(11):2909–13. 11. Schwartz M, Acosta L, Hung Y-L, Padilla M, Enciso R. Effects of CPAP and mandibular advancement device treatment in obstructive sleep apnea patients: a systematic review and meta-analysis. Sleep Breath. 2018;22(3):555–68. 12. Gindre L, Gagnadoux F, Meslier N, Gustin J-M, Racineux J-L. Mandibular advancement for obstructive sleep apnea: dose effect on apnea, long-term use and tolerance. Respiration. 2008;76(4):386–92. 13. Marklund M, Verbraecken J, Randerath W. Non-CPAP therapies in obstructive sleep apnoea: mandibular advancement device therapy. Eur Respir J. 2012;39(5):1241–7. 14. Aarab G, Lobbezoo F, Hamburger HL, Naeije M. Effects of an oral appliance with different mandibular protrusion positions at a constant vertical dimension on obstructive sleep apnea. Clin Oral Investig. 2010;14(3):339–45. 15. Bartolucci ML, Bortolotti F, Raffaelli E, D’Antò V, Michelotti A, Bonetti GA. The effectiveness of different mandibular advancement amounts in OSA patients: a systematic review and metaregression analysis. Sleep Breath. 2016;20(3):911–9. 16. Vroegop AV, Vanderveken OM, Van de Heyning PH, Braem MJ. Effects of vertical opening on pharyngeal dimensions in patients with obstructive sleep apnoea. Sleep Med. 2012;13(3):314–6. 17. Pitsis AJ, Darendeliler MA, Gotsopoulos H, Petocz P, Cistulli PA. Effect of vertical dimension on efficacy of oral appliance therapy in obstructive sleep apnea. Am J Respir Crit Care Med. 2002;166(6):860–4. 18. 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Oropharyngeal Exercise for OSA Patients 10 Kyung-A Kim and Su-Jung Kim Contents 10.1 W hat Is the Oropharyngeal Exercise as the Treatment Modality of OSA Patients? 10.2 Is it Effective for OSA Patients? 10.2.1 Effects of Oropharyngeal Exercises on Pediatric OSA 10.2.2 Effects of Oropharyngeal Exercises on Adult OSA 10.3 How to Do Oropharyngeal Exercises for OSA Patients? 10.3.1 Soft Palate Exercises 10.3.2 Tongue and Suprahyoid Muscle Exercises 10.3.3 Facial Muscle Exercises 10.3.4 Oropharyngeal Exercises for Stomatognathic Functions 10.4 Is There Any Limitation to do Oropharyngeal Exercises Clinically? References 10.1 131 132 132 133 133 133 134 135 138 139 140 hat Is the Oropharyngeal Exercise as the Treatment W Modality of OSA Patients? Orofacial myofunctional therapy (MFT) is defined as the treatment of subjects with orofacial myofunctional disorders to eliminate oral habits, to re-pattern and change the function of the oral and facial muscles, and to lead the normal development of orofacial structures [1, 2]. Generally, orofacial MFT is based on repetitive muscle training that enhances orofacial muscle in sensitivity, proprioception, mobility, coordination and strength as well as rehabilitates an appropriate function of respiration, mastication, deglutition, and speech. Upper airway is a soft tissue structure surrounded by muscles which constrict or dilate the upper airway lumen and contribute to the genesis of OSA [3, 4]. Among K.-A. Kim · S.-J. Kim (*) Department of Orthodontics, Kyung Hee University School of Dentistry, Seoul, South Korea e-mail: ksj113@khu.ac.kr © Springer Nature Switzerland AG 2020 S.-J. Kim, K. B. Kim (eds.), Orthodontics in Obstructive Sleep Apnea Patients, https://doi.org/10.1007/978-3-030-24413-2_10 131 132 K.-A. Kim and S.-J. Kim the oropharyngeal muscles, genioglossus muscles are important to control the tongue and hyoid position, palatoglossus muscles are known to be communicating muscles between the soft palate and the tongue and allow upper airway to open and close, and lateral pharyngeal walls involving palatopharyngeus muscles and pharyngeal constrictor muscles are related to function of respiration directly. Oropharyngeal exercises (OPE) proposed by Guimarães have been applied for reducing OSA severity and associated symptoms in adult patients since the 1990s [5]. OPE consists of isometric and isotonic exercises and target the tongue, soft palate, suprahyoid muscle, lateral pharyngeal wall, and facial muscles in order to reinforce the muscles for dilating the upper airways during sleep [6]. For reducing upper airway collapsibility during sleep, the repetitive muscle training while awake is the concept of OPE in OSA patients [7, 8]. 10.2 Is it Effective for OSA Patients? Most studies showed that OPE consisting of isometric and isotonic exercises involving the tongue, soft palate, and lateral pharyngeal wall for at least minimum 3 months improved OSA symptoms both subjectively and objectively [9]. 10.2.1 Effects of Oropharyngeal Exercises on Pediatric OSA Recent randomized studies showed the significant reduction of respiratory symptoms and the apnea–hypopnea index (AHI) and demonstrated the significant increased percentage of lowest oxygen saturation (LSaO2) after OPE in pediatric OSA [10, 11]. Regarding the snoring as the chief complaint of OSA children, most studies demonstrated that OPE ameliorated the snoring intensity and frequency [12]. Although there was a significant improvement, pediatric studies of OPE effect on snoring are lacking due to the subjective measurements of snoring. Younger children tend to sleep with their parents, while older children usually sleep far away from their parents, so the parents are more likely to find snoring of younger children [13]. But there is a risk of bias concerning snoring outcomes; most studies were consistent in their findings of decreased snoring noted after OPE. In addition to the improvement of OSA symptoms, OPE has critical role in pediatric OSA. There are various etiologic factors including craniofacial vulnerability, soft tissue vulnerability, neuromotor dysfunction, and inflammation in pediatric OSA. Except the cases of neuromotor dysfunction, adenotonsillectomy is known to be the first-line treatment option of pediatric OSA, since adenotonsillar hypertrophy has been the most frequent etiologic factor in children. The treatments including surgical (adenotonsillectomy), orthodontic, and medical (reducing lymphadenoid tissue size) correct the oropharyngeal structure, but it does not guarantee normal function such as normal tongue position or normal orofacial muscle strength during sleep. Mouth breathing and lip hypotonia as the typical symptom of pediatric OSA are persistent after surgical and medical treatment and may be the cause of residual OSA and abnormal airway development. So OPE should be applied to optimize 10 Oropharyngeal Exercise for OSA Patients 133 normal development of the airway and orofacial muscle strength as well as to maintain other treatment modalities effect. As the result, OPE leads the normal nasal breathing during sleep and normal orofacial growth. The importance of early recognition and treatment in children is paramount to maximizing resolution of symptoms and potential avoidance of OSA syndrome during adulthood. 10.2.2 Effects of Oropharyngeal Exercises on Adult OSA The recent systematic review demonstrated an improvement in snoring by approximately 50% in adult patients with mild-to-moderate OSA, and the 50% improvement in snoring seen is consistent with the improvement seen in OSA severity [12]. Snoring index, snoring frequency, and snoring time measured by Berlin questionnaires, visual analog scale, grading scale and PSG results, and snoring intensity reported by the bed partner were all decreased after OPE in patients with primary complaint of snoring or mild-to-moderate OSA [9, 14, 15]. Guimarães et al. analyzed the effect of OPE on the OSA severity for adults patients with moderate OSA to do oropharyngeal isometric and isotonic exercises involving the tongue, soft palate, and lateral pharyngeal wall for 3 months [16]. As the results, OPE reduced not only OSA severity by 39% measured by the AHI and lowest oxygen saturation but also subjective symptoms evaluated by snoring, daytime sleepiness, and sleep quality. According to the recent systematic review and meta-analysis, despite the heterogeneity in OPE, all studies demonstrated significant AHI and AI reduction, increased lowest oxygen saturation, and a reduction of arousal index compared to baseline [9, 12, 14–18]. Overall the improvement in polysomnographic outcome and daytime sleepiness were consistent. Epworth sleepiness scale was used as a measurement of result of OPE regarding daytime sleepiness in almost studies analyzed. 10.3 How to Do Oropharyngeal Exercises for OSA Patients? Oropharyngeal exercise program for OSA patients consists of isometric and isotonic exercises involving the soft palate, tongue, lips, facial muscle, and lateral pharyngeal wall designed to improve nasal breathing, swallowing, sucking, chewing, and speech functions, which are closely related and are part of stomatognathic system (Table 10.1). 10.3.1 Soft Palate Exercises OSA patients typically have elongated and floppy soft palate and uvula. Soft palate exercises consist of pronouncing an oral vowel [a, e, i, o, u] intermittently (isotonic exercise) and continuously (isometric exercise). (Fig. 10.1) The exercises targeting soft palate elevation use speech exercises that recruit several muscles including the palatopharyngeus, palatoglossus, uvula, tensor veli palatini, and levator veli palatini muscles [19–21]. These exercises have to be repeated daily for 3 min. 134 K.-A. Kim and S.-J. Kim Table 10.1 Instruction manual of oropharyngeal exercises for the OSA patients Target area Soft palate Tongue and Suprahyoid muscle Facial muscle Instructions Pronounce an oral vowel [a, e, i, o, u] intermittently and continuously Brush the superior and lateral surfaces of the tongue while the tongue is positioned in the mouth floor Place the tip of the tongue against the anterior palate and slide the tongue backward Press the entire tongue against the palatal wall Place the tongue tip in contact with the mandibular incisors and force posterior region of tongue downward Press anterior part of tongue against the palate during the mouth opening with head back Obicularis oris Pull the button with mouth closed Buccinators Stomatognathic functions Levator anguli oris Nasal breathing Swallowing and Chewing Frequency 3 min/day 5 times 3 sets/day 3 min/day 3 min/day 3 min/day 3 min/day 30 s/time 3 times/day 3 min/day Suck the cheek contracting only the buccinators with repetitions and holding position Put index fingers in the mouth and then 3 min/day stretch the buccinator muscle outward Put index fingers in the mouth corner and 10 sets/day stretch up and down of levator anguli oris Forced nasal inspiration and oral 5 times/day expiration while sitting Balloon inflation with prolonged nasal inspiration and then forced blowing Nasal inspiration on one side and then nasal expiration on the other side Alternate bilateral chewing and deglutition with tongue in the palate and mouth closed Alternate bread mastication with mouth closed Gum chewing with mouth closed Fig. 10.1 Soft palate exercises. Pronounce the vowel [a, e, i, o, u] intermittently and continuously for 3 min per day 10.3.2 Tongue and Suprahyoid Muscle Exercises Position and dimensions (area and volume) of tongue are significantly associated the upper airway collapsibility [22], so the tongue exercises focus the motility and muscle strength of tongue. Regarding the position, the tongue should be kept in a high position with its dorsal-terminal end in constant contact with the anterior 10 Oropharyngeal Exercise for OSA Patients 135 palatal striae. According to the Hirata RP et al. [22], tongue fat is the main factor of the increased tongue volume in OSA patients compared to controls and is related to the compromised ability of extrinsic muscles to tongue position and the decreased air space in the retroglossal region. Tongue exercises consist of moving the tongue in different directions with or without sticking the tongue out, pressing against bony and soft tissue structures within the oral cavity, sucking the tongue against the palate, and other tongue movements with or without resistance [16]. The suprahyoid muscles including the geniohyoid, stylohyoid, mylohyoid, anterior digastric, and posterior digastric muscles together with the styloglossus and genioglossus muscles play an important role in tongue movement [15, 18, 23]. The movements of “placing the tip of the tongue against the anterior palate and sliding the tongue backward,” “forced tongue sucking upward against the palate,” and “pressing the entire tongue against the palate” result in increased resistance and fatigue threshold of the suprahyoid muscles and reach the appropriate tongue positioning during rest, sleeping, and deglutition consequently [16, 24]. And elevation of the tongue position promoted by the suprahyoid muscles displaces the lower positioned hyoid bone in an anterior direction and upper direction [22, 25]. Tongue and suprahyoid muscle exercises are as follows (Fig. 10.2): • Tongue brushing: Brush the superior and lateral surfaces of the tongue while the tongue is positioned in the mouth floor (5 times, 3 sets/day). • Tongue sliding: Place the tip of the tongue against the anterior palate and slide the tongue backward (3 min/day). • Pressing the palatal wall: Press the entire tongue against the palatal wall (3 min/day). • Pressing the mandibular incisors: Place the tongue tip in contact with the mandibular incisors and force posterior region of tongue downward (3 min/day). • Pressing the palate during the mouth opening with head back: Press anterior part of tongue against the palate during the mouth opening with head back (3 min/day). 10.3.3 Facial Muscle Exercises The exercises of the facial musculature recruit the orbicularis oris, buccinator, major zygomaticus, minor zygomaticus, levator labii superioris, levator anguli oris, lateral pterygoid, and medial pterygoid muscles. The volume and flaccidity of cheek have possibility to affect the volume of oropharynx, so facial musculature exercises as the oropharyngeal exercises are applied [26]. The facial exercises according the targeted muscles are as follows (Fig. 10.3): 1. Orbicularis oris: • Pull the button with mouth closed to train orbicularis oris muscle (isometric exercise). • Recruited to close with pressure for 30 s, and right after, requested to realize the posterior exercise (3 times/day). 136 Fig. 10.2 Tongue and suprahyoid muscle exercises. (a) Tongue brushing; (b) tongue sliding; (c) pressing the palatal wall; (d) pressing the mandibular incisors; (e) pressing the palate during the mouth opening with head back K.-A. Kim and S.-J. Kim 10 Oropharyngeal Exercise for OSA Patients 137 Fig. 10.3 Facial muscle exercises. (a) Button pulling for orbicularis oris muscle training; (b) suction movements contracting the buccinators; (c) stretch the buccinators outward; (d) stretch up and down of levator anguli oris 138 K.-A. Kim and S.-J. Kim 2. Buccinators: • Suck the cheek contracting only the buccinators. These exercises were performed with repetitions (isotonic) and holding position (isometric) (3 min/day). • Put index fingers in the buccal mucosa of mouth and then stretch the buccinator muscle outward (3 min/day). 3. Levator anguli oris: • Put index fingers in the mouth corner and stretch up and down of levator anguli oris (isometric exercise) and after, with repetitions (isotonic exercise) (10 sets/day). 10.3.4 Oropharyngeal Exercises for Stomatognathic Functions Oropharyngeal exercises consist of habit elimination and behavior modification, jaw stabilization exercises, repatterning the oral facial muscles and changing their function for optimal nasal breathing, oral rest position, chewing, and swallowing. 1. Nasal breathing training (Fig. 10.4): • Forced nasal inspiration and oral expiration while sitting (5 times/day). Fig. 10.4 Nasal breathing training. (a) Alternate nasal inspiration and oral expiration; (b) blowing a balloon; (c) alternate nasal inspiration and expiration 10 Oropharyngeal Exercise for OSA Patients 139 • Balloon inflation with prolonged nasal inspiration and then forced blowing (5 times/day). • Nasal inspiration on one side and then nasal expiration on the other side (5 times/day). 2. Swallowing and Chewing: • Alternate bilateral chewing and deglutition, with the tongue in the palate, mouth closed and without perioral contraction. • Alternate bread mastication bilaterally aims to correct position of the tongue while eating in order to target the appropriate function and movement of the tongue and jaw. The patients were instructed to incorporate mastication pattern whenever they were eating. • Gum chewing with mouth closed for enhancing the jaw closing muscle to seal the lip during rest and sleep. 10.4 I s There Any Limitation to do Oropharyngeal Exercises Clinically? In treating pediatric OSA, there are some limitations. (1) Current forms of OPE are so difficult for younger children to do. (2) The poor compliance with daily exercises and the absence of continuous parental involvement with the OPE are major causes of failure of treatment. So the absolute need to have continuous parental involvement during the OPE for pediatric OSA is critical key for success. And compliance, how well patients adhere to OPE is the most important feature in adults as well. Another challenge is heterogeneous in exercises type, frequency and targeted muscle between studies. OPE is based on an integrative program with combined several exercises, and it is hard to determine the effects of specific exercises including the target muscle and frequency; therefore, future studies are needed to consider the effect of individual exercises protocol. Clinical Pearls of Oropharyngeal Exercise • MFT for the OSA patients represents oropharyngeal exercise (OPE) targeting the soft palate and the pharyngeal wall muscles in addition to lips, tongue, cheek, and hyoid muscles. • OPE includes the functional training of nasal breathing in addition to swallowing and chewing. • OPE alone may be effective in growing OSA patients with habitual mouth breathing and abnormal tongue posture. • OPE needs to be combined with craniofacial modification in growing OSA patients with structural mouth breathing and nasal obstruction. • OPE is necessary to ultimately improve the pharyngeal collapsibility by repositioning the soft tissue structures after skeletal enlargement in OSA adults. 140 K.-A. Kim and S.-J. Kim References 1. de Felício CM, da Silva Dias FV, Trawitzki LVV. Obstructive sleep apnea: focus on myofunctional therapy. Nat Sci Sleep. 2018;10:271. 2. Hanson M. Orofacial myofunctional therapy-historical and philosophical considerations. 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