A2012050803
(L201201396)
1-21 Superior tracheostomy through cricoid cartilage removal with forceps (extrabuccal)
- Chihiro Kanno 1), Tsutomu Moma 1), Hiroshi Hasegawa 1), Eiju Sato 2), Masato Kano 2)
1) Dental Surgery Department, Fukushima Medical University
2) Cervicofacial Surgery Department, Ohara General Hospital
In tracheostomy, the medial or inferior trachea is generally resected. At the present conference,
we reported on superior tracheostomy performed through cricoid cartilage removal with forceps;
particularly in terminal stage patients, as this is a highly safe technique. Since we experienced a
case of an irregular path of the common carotid artery surrounding the thyroid, in which we
performed superior tracheostomy through cricoid cartilage removal with forceps resulting in the
closure of a hole in the airway, we are reporting it here.
Case: [Patient] A 75-year-old female. [Disease name] Mandibular central cancer (T4N0M0) [Past
history] Hypertension [Treatment progress] The patient was suffering from mandibular central
cancer. Almost complete extirpation of the mandible, bilateral cervical radical dissection, and
mandibular reconstruction with a titanium plate and a free rectus abdominis flap were performed.
Since an irregular path of the right carotid artery was observed, tracheostomy was performed
through cricoid cartilage removal with forceps. Closure commenced from post-operative Day 17,
and there have been no observed abnormalities such as incomplete closure or abnormal
proliferation of granuloma; and progress is issue-free.
[Conclusion] It was considered that superior tracheostomy through cricoid cartilage removal with
forceps is a technically simple and safe technique.
G1218437
1-22 A case of bisphosphonate-related osteomyelitis of the mandible for which reconstruction
was performed using a fibula - Discussion related to the area of resection - (extrabuccal)
- Shujiro Makino 1), Masahi Takano 1), Takuya Asaka 1), Eiji Kitagawa 1), Yoshihiro Yasuhiko 2)
1) Dental Surgery, Hokuto Hospital
2) Health Science University of Hokkaido
[Introduction] In recent years, there have been reports on surgical resection and bone
reconstruction to treat BRONJ for which it is difficult to provide conservative treatment. At the
annual meeting in 2010 we reported a case of mandibular reconstruction through segmental
resection and using a fibula to treat BT-related osteomyelitis of the mandible. The present paper
reports the details of an investigation into the range of such resection.
[Case and progress] The patient was a 64-year-old female. She was already taking Fosamac for
four years in addition to steroids and methotrexate for rheumatoid arthritis. Upon a tooth
extraction in 2004, the patient started suffering from osteomyelitis of the mandible. Although the
patient was treated by her previous doctor and underwent conservative therapy and a surgical
anti-inflammatory intervention procedure at our Department over the course of six years, these
treatments were not efficacious, and the patient underwent surgery. Since there is no knowledge
and scare evidence on the range of segmental resection for BRONJ, we used as an index the Tc
accumulation area, in addition to the cortical bone change area on CT and the bone marrow lowintensity signal area on MRI, taking into account the mechanism of action of BP.
[Conclusion] In the present case, we set the resection area to be the infection area and the
inflammation reaction area which has an impact on local BP concentration, which are considered
to be related to the onset of BRONJ. It was considered that it is necessary to accumulate relevant
cases in order to set an appropriate resection area.
- 37 -
A201205083
(L201300727)
0326
182
J1300607
Mar. 2013
A case of immediate reconstruction after segmental mandiblectomy in a patient with refractory
osteomyelitis of the jaw caused by oral bisphosphonate therapy
Takuya Asaka 1, 2), Shujiroh Makino 1), Masashi Takano 1), Takahiro Abe 1,2), Yoshihiro Abiko 3),
Yoshimasa Kitagawa 2)
Introduction
In principle, the treatment for bisphosphonate (hereinafter referred to as BP)-related
osteonecrosis of the jaw (hereinafter referred to as BRONJ) is conservative therapy; however,
there are some refractory cases for which surgical treatments such as segmental resection of the
mandible are covered. In applying surgical treatment, immediate reconstruction using
reconstruction plates is recommended. Since bone transplantation has the risk of incomplete
union between the transplanted bone and the underlying bone, bone transplantation has
generally not been performed 1-4).
In recent years, there have been an increasing number of reports on immediate reconstruction
performed using blood vessel-bearing bone transplants following mandibular segmental
mandiblectomy to treat refractory progressive cases of BRONJ 5 – 10), however, there have been no
such reports in Japan. In the present paper, we report on the favorable treatment progress after
performing segmental mandiblectomy, and after immediate mandibular reconstruction using a
fibula, to treat refractory mandibular osteomyelitis.
Case
Patient: A 64-year-old female
Initial diagnosis: February 2008
Chief complaint: Pain in the right mandible
Past history: The patient was orally taking prednisolone (10 mg per day) since 2000 and
methotrexate (hereinafter referred to as MTX, 2 mg per week) since 2001 for chronic rheumatoid
arthritis (hereinafter referred to as RA).
Received on: Mar 13, 2012
Accepted on: Dec 25, 2012
36
Vol. 59 No. 3
183
A case of segmental mandiblectomy and immediate reconstruction in a patient with osteomyelitis
of the jaw while orally taking BP
The patient was also orally taking alendronate (5 mg per day) to treat steroidal osteoporosis.
Familial history: None in particular
Current history: In July 2005, an abscess in the alveolus area at tooth 5 (right) on the tongue side
was found at the dental surgery department at another hospital, and due to being diagnosed with
chronic periodontitis, the administration of antibiotics and local washing were commenced (Photo
1.) The abscess expanded to teeth 6-3 (right) at the alveolus part on the tongue side. Partial bone
exposure was observed with an appearance of sequestrum, and, in December 2005, tooth 5 (right)
was extracted and the sequestrum was removed under general anesthesia upon diagnosis with
inferior mandibular osteomyelitis.
Photo 1: Panoramic x-ray taken by previous doctor
No clear image of resorption was found in the mandible
The fistula at the same location did not disappear after the surgery, and in January 2006, the
sequestrum was removed and teeth 4, 3, and 2 (right) were extracted. Also in September 2006,
the sequestrum was removed and tooth 1 (right) was extracted.
In October 2006, in addition to the fistula in the in the oral cavity, the formation of an abscess was
observed in the inferior mentum. From around this time, the symptoms were suspected of being
related to BP, therefore, the dosage of prednisolone was reduced to 3.5 mg/day in February 2007,
and the administration of alendronate was discontinued after a consultation with the prescribing
doctor. Thereafter, swelling and pain in tooth 12 (left) in alveolar part were newly observed, and in
May 2007, the sequestrum was removed and tooth 12 (left) was extracted, however, there was
repeated remission and reflaring of the abscesses inside and outside the oral cavity, and the
patient was referred to our Department for an initial consultation.
Current symptoms:
Systemic observations: height: 156 cm, body weight: 34 kg, wearing devices on both ankle joints
due to rheumatoid arthritis, and independent in terms of ADL when using walking sticks.
Observations outside the oral cavity: The patient's face was symmetric, and fistulae were
observed in the inferior mentum. Pressure pain was found in the right mandibular bone; however,
there was no dullness of perception in the inferior alveolar nerve area (Photo 2A).
Observations inside the oral cavity: The only tooth remaining in the mandible was tooth 6 (left).
Fistula formation was observed at three sites in the alveolus of the mandibular front tooth part
(Photo 2B).
Imaging observations: An image of uneven bone resorption was observed on the alveolar crest
side and the lower edge side from the right mandibular bone to the mandibular center (Photo 2C).
Photo 2: Photos and imaging examinations at initial diagnosis
A: Appearance of face; fistula formation observed in the inferior mentum
B: Photo of inside the mouth; fistula formation was observed at three sites (arrows) in the alveolus
in the front tooth of the mandible
C: Panoramic x-ray image; uneven bone resorption image observed in alveolar crest and the lower
edge sides from the right mandible bone to the mandibular center
D: CT; sclerotic change and osteolysis images observed from the right mandible to the left mental
foramen
E: MRI (T1 enhanced); T1 low intensity signal areas were observed from the right mandible to the
left mental foramen (arrow)
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The Japanese Journal of Oral Maxillofacial Surgery
Mar. 2003
Photo 3: Preoperative imaging examination
A: Osteolysis images were observed up to the cortical bone lower edge on the tongue side of the
mandible in the same way as at initial consultation (arrow). No sequestrum separation images
were observed.
B: Preoperative 99mTc bone scintigraphy; accumulation was observed from the right mandibular
angle towards tooth 4 (left) and a cold area considered to be osteonecrotic was observed on the
lower edge side of the right mandible (arrow). Additionally, local accumulation was observed in
the part near the tooth 6 (left) extraction cavity.
Photo 4: Intraoperative photo and post-operative panoramic x-ray
A: No abnormalities were observed with the naked eye in the bone marrow of the resection edge,
and favorable bleeding was observed (arrow)
B: Panoramic x-ray image; fibula osteomized from two sites was fixed to the mandible using a
plate for mandibular reconstruction
An uneven CT bone resorption image was observed on the cortical bone on the tongue and cheek
sides from the right mandibular bone to the left mental foramen, and in the marrow cavity there
was a mixture of images of sclerotic change and osteolysis (Photo 2D). On T1-enchanced MRI,
bone marrow low-intensity signal areas were observed from the right mandibular bone to the left
mental foramen (Photo 2E). Also, a slight imaging effect was observed on an MRI image of the
same area.
Clinical diagnosis: Osteomyelitis of the mandible
Treatment and progress: An infusion of antibiotics (sulbactam sodium and sodium ampicillin, 3.0
g/day) was commenced while the patient was hospitalized. Hyperbaric oxygen therapy
(hereinafter referred to as HBO) was commenced from hospitalization Day 5, pain was observed to
have disappeared on hospitalization Day 10, and the patient was discharged. Thereafter, the oral
administration of antibiotics (400 mg/day of clarithromycin) was continued, and HBO (a total of 20
sessions: five sessions during hospitalization, and 15 sessions on an outpatient basis) was
performed. Two months after discharge, fistulae outside the oral cavity disappeared; however,
there was pain found on both sides of the mandible five months post-discharge. Thereafter,
conservative therapy was provided with antibiotics (cefuroxime axetil 750 mg/day, administered
for several days, and 400 mg/day of clarithromycin, administered for several weeks) while acute
symptoms were exacerbated. However, abscess formation was observed on the left mandibular
inferior edge in April 2009.
Since imaging found that the mixture of osteolysis images and sclerosis images extended to the
lower edge of the mandible, since the symptoms did not disappear after the four occasions of
sequestrum removal by the previous doctor, and since the administration of antibiotics together
with HBO at our department did not lead to the symptoms disappearing, we determined that a
complete cure through conservative therapy would be difficult, and planned a segmental
mandiblectomy and mandibular reconstruction using a fibula.
During presurgical preparation, perturbation was observed, and tooth 6 (left) was extracted
because there was a possibility that it may have been a source of infection during surgery.
Epithelium formed two months after extraction.
Moreover, pre-surgical HBO (a total of 20 times) was performed, fistulae in the lower part of the
mentum disappeared directly before the surgery, and those in the oral cavity were turning into
epithelium. On CT, osteolysis images reaching the mandibular tongue side cortical bone lower
edge were observed in the same way as at the initial consultation (Photo 3A). Also, no clear
changes were observed on MRI, and accumulation was observed from the right mandibular angle
to the area of tooth 4 on 99mTc bone scintigraphy, and cold areas, considered to be osteonecrotic,
were observed on the lower edge side of the right mandibular part. Meanwhile, accumulation was
observed in a part near the tooth 6 (left) extraction cavity, which was separate from these areas
(Photo 3B).
In August 2009, segmental mandiblectomy and mandibular reconstruction using a fibula were
performed. Clinical progress was referred to in order to determine the area for resection, and
sclerotic changes on CT, low intensity bone marrow signal areas on MRI, and findings on 99mTc
bone scintigraphy were used as indices for setting the area, and it was centered around tooth 6
(left) from the right mandibular angle. The surgery was performed through resection in the
mandibular part without visual crossover of the surgical field and the inside of the oral cavity.
Further, an ultrasound scalpel (Sonopet ®) was used to extirpate a nerve and vessel fascicle in the
inferior alveolus on both sides, and they were preserved. Bleeding from the bone marrow at the
resection edge in the mandibular bone was favorable during surgery, and there were no
abnormalities observed with the naked eye (Photo 4A). Additionally, osseous defects and
granuloma tissue were observed in the lower edge of resected cortical bone on the tongue side of
the mandible.
There were no abnormalities observed on the 13 cm of fibula extracted from the lower left limb,
and, with reference to a replica created preoperatively, osteotomy was performed on two sites
following the morphology of the resected mandible, and each block and mandibular bone were
fixed with plates for mandibular reconstruction (Photo 4B).
Vol. 59 No. 3
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A case of segmental mandiblectomy and immediate reconstruction in a patient with osteomyelitis
of the jaw while orally taking BP
Photo 5: 3D-CT after plate removal
Bone union was observed in the fibula resection
part and the fibula-mandible connection site
Photo 6: Histopathological image of the right
mandible
(HE staining, Bar: 100 µm)
The inside of the connective tissue in the bone
consisted of necrotic tissue, and no entrapment
of bone cells was observed in small bone
cavities. Bacterial clusters were observed in
some parts.
Cefuroxime axetil, an antibiotic agent, was administered (2 g/ day) through an infusion up to 24
hours post-operation.
One month following the surgery, blood flow in the transplanted bone was observed on 3-phase
99m
Tc bone scintigraphy. Fistula formation was neither observed inside nor outside the oral cavity,
and since the patient was able to orally consume watery rice porridge, she was discharged one
month following the operation. Six months following the operation, union of the transplanted
bone and the mandible was observed on CT, and there were no osteomyelitis findings.
Additionally, the patient started wearing a full set of artificial teeth on the mandible one year
following surgery, allowing her to consume food normally. Since the administration of BP was
expected to be resumed to treat RA, reconstruction plates were removed one year and nine
months following surgery because of risks such as those of infection due to artificial teeth ulcers,
as well as the risk of BRONJ onset. At present, although the patient has mild perception
abnormality on both sides of the inferior lip, there has been no reflaring of the osteomyelitis
condition, and progress has been good (Photo 5.)
Pathohistological findings: There were no entrapped bone cells nor osteoclasts found in the
majority of compact bone from the right mandibular molar part to the left mandibular molar part.
Also, fibrous bone considered to be sequestrum with no bone cells entrapped was observed close
to the alveolar part, around which connective tissue was found accompanied by pronounced
inflammatory cellular invasion mainly with lymphocytes and plasmocytes. In particular, bacterial
clusters were observed in certain sites near to the inferior edge of the right mandibular bone, and
fibrous connective tissue inside the bone was found to have necrotized (Photo 6). Meanwhile,
bone cells in the compact bone were observed to be entrapped on the bilateral mandibular
resection edge, and no inflammatory changes were observed in the bone marrow.
Discussion
The patient in the present case was already taking BP for approximately five years. Although bone
exposure was observed by the previous doctor, it was not observed at the time of initial
consultation at our Department, and we diagnosed the patient with refractory mandibular
osteomyelitis, which occurred when the patient was orally taking BP. According to the BRONJ
diagnostic criteria 2), the present case was categorized as Stage 0. However, the patient underwent
sequestrum removal by the previous doctor, suffered from fistulae inside and outside the oral
cavity, and an image of broad osteolysis was found on CT, and these were similar to the clinical
findings for Stage III; therefore, we referred to treatment guidelines for this Stage when treating
the patient.
At present, when treating a patient in BRONJ Stage III suffering from broad lesions, for which
conservative therapy is not efficacious, it is recommended to resect the sequestrum, including the
area peripheral thereto, or to perform segmental resection; and it is also recommended to
perform reconstruction using metal plates when carrying out segmental resection 2-4). One reason
why bone reconstruction is not recommended is that a large number of Stage III BRONJ patients
are using BP injectable agents to treat malignant tumors, which are the underlying disease, and
therefore, in many such cases doctors are hesitant to recommend surgery when considering the
invasiveness of surgery and systemic condition of the patient. Also, it is not possible to rule out the
possibility of metastasis to transplanted bone, and further, it is pointed out that there is a
possibility of osteonecrosis in the remaining mandible and transplanted bone 1, 2). However, there
have been reports 5-10) on cases of BRONJ in which favorable healing progress was found after
bone reconstruction through the transplantation of bone [six papers, 24 cases (22 malignant
tumor cases, and 2 osteoporosis cases)]. These reports are on cases in which the benefits of
surgery outweighed the risks of it.
The case we experienced did not achieve a cure even after removing sequestrum four times and
after long-term antibiotic administration together with HBO, and images of osteolysis reaching the
lower edge of the mandible were observed. Therefore, we diagnosed the patient with a condition
for which segmental resection is covered by insurance. Further, since long-term maintenance of
QOL is expected based on the patient’s age and prognosis, since the lesion broadly spread to the
bilateral mandible part, and as there was a possibility that plate reconstruction could have caused
exposure and fracture, we selected mandibular reconstruction through a vascular pedicle bone
graft.
In setting the region for segmental resection, we referred to therapeutic experience related to
osteoradionecrosis (hereinafter referred to as ORN) 11). With ORN, we judged non-contrasted
areas on contrast MRI to be necrotized, and, considering areas where the imaging effect is weak
on contrast MRI and which had low signal intensity on simple MRI to have poor blood flow, we
ultimately decided the resection area from intraoperative findings.
However, the blood flow in the mandible was decreased due to metabolism suppression by BP,
and since there was a possibility of involvement in the mechanism of onset of BRONJ 12), in the
case we experienced, all sites with poor blood flow were included in the resected region.
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The Japanese Journal of Oral Maxillofacial Surgery
Mar. 2003
Also, it was considered that 99mTc accumulation in the tooth 6 (left) extraction cavity was caused
over the course of healing of the extraction site cavity. Our policy in this case was to include it in
the resected area. This policy was based on the theory that the mechanism of BRONJ onset is a
locally-elevated BP concentration caused by inflammatory change 13).
In reports on BRONJ segmental resection 5-10), it is stated that bone union is achieved only in cases
in which normal bone tissue is found on the resection edge of the mandible. Usually, long bones
such as a fibula, do not undergo bone remodeling due to application of pressure from occlusion in
the way that jaw bones do, and long bones to not have infection foci such as with oral cavity
bacteria; therefore, it is considered that BP accumulation in such bones is low compared to in the
mandible 5, 7). Thus, it is considered that if inflammatory foci are completely resected at the time of
segmental resection it is possible to achieve bone union through BRONJ bone transplant
reconstruction. Since the most important issue is infection control of the surgical wound, in the
present case, with the aim of avoiding actual infection as much as possible in the surgical field, we
extracted tooth 6 (left) preoperatively and performed surgical resection without visual crossover
of the surgical field and the oral cavity.
The utility of HBO in treating BRONJ has not yet been established, however, there have been
reports in recent years supporting it 4). In the present case, osteomyelitis was the main issue, and
HBO was performed 20 times preoperatively in order to shrink the resection area by
preoperatively improving inflammation, and also for the purpose of promoting post-operative
wound site healing.
Also, there have been some reports in recent years of mandibular necrosis accompanying MTX 14).
The present case concerned an initial consultation prior to such reports. MTX was continued,
however, since the impact of MTX on decreased immunocompetence could not be ruled out, we
consider that it is necessary in future to carry out long-term follow-up observation of this case,
considering the fact that the re-administration of BP is being considered.
The incidence rate of BRONJ due to oral BP administration is higher in Japan than in Europe 2), and
cases of BP usage similar to that in the present case are expected to occur going forwards. The
principle of treatment is conservative therapy, however, if it cannot be avoided to perform surgical
treatment such as segmental resection, it is considered useful to carry out reconstruction through
vascular pedicle bone graft according to the area of the lesion.
Acknowledgements
As a final note to this paper, we would like to thank Dr. Takashi Konishi of Tokeikai Kyoritsu
Hospital, who kindly provided documents on the present case, as well as Dr. Eiji Kitagawa at the
Dental Surgery Department of the same hospital, who provided kind support in creating artificial
teeth.
References
3) Masahiro Urade, Noriaki Tanaka et. al, A follow-up study on 30 cases of mandibular
osteomyelitis and mandibular necrosis considered to be related to bisphosphonate administration
- The condition two years later -. Jpn. J. Oral Maxillofac. Surg.; 553-561, 2009
11) Kenichi Notani, Yu Yamazaki, et al.: Treatment of and prognosis of osteoradionecrosis with a
particular focus on surgical indication and times. Jpn. J. Oral Maxillofac. Surg.; 38: 1652 – 1658,
1992
13) Toru Ikeda, Akira Yamaguchi, et al.: Bisphosphonate related osteonecrosis of the jaw Discussion from the point of view of pathology. Jpn. J. Oral Maxillofac. Surg.; 56:352-356, 2010.
14) Daisuke Sano, Kenichiro Ishibashi, et al.: A case of methotrexate-related lymphoproliferative
disease accompanying osteonecrosis of the jaw. Jpn. J. Oral Maxillofac. Surg.; 58: 655-659, 2012
186
Mar. 2013
Also, the 99mTc accumulation in the tooth 6 (left) extraction cavity, which was outside this region,
was considered to be accumulation during healing of the extraction wound; however, we adopted
a policy of including it in the resection area in this case.