AN INTERESTING CASE OF BLOODY SWEAT (HEMATOHIDROSIS) ABSTRACT Hematohidrosis or hematidrosis is a rare disorder characterized by one or more attacks of spontaneous, selflimited, bloody sweating from the intact skin. In this literature, a case of a 9-year-old girl was presented with recurrent self-limiting episodes of bloody sweating from the left upper arm upon exposure to extreme stress and anxiety. The patient had no history of bleeding disorders, physical trauma, or drug intake. All blood investigations of the patient were within the normal range. The outcome was favorable in this case with pharmacotherapy (beta-blocker) and psychological support. INDRODUCTION Hematidrosis or hematohidrosis is a rare clinical condition in which blood is excreted with sweat on exposure to the extreme physical or emotional stress.[1] The exact etiopathogenesis of this condition is not clear. One hypothesis proposed for etiopathogenesis of hematohidrosis[2] was that multiple blood vessels present in a net-like form around the sweat gland constrict on exposure to extreme anxiety. When the anxiety passes out, the blood vessels dilate to the point of rupture. The blood gets into the sweat glands, which will be pushed out it along with sweat to the surface, presenting as droplets of blood mixed with sweat. Anxiety activates the sympathetic nervous system to invoke stress-fight reaction to severe degree as to cause hemorrhage of the blood vessels supplying the sweat glands into the ducts of the sweat glands.[3] Etiology can be systemic disorders (bleeding disorders), vicarious menstruation, excessive physical exertion, and psychological stressors.[1] True hematohidrosis means the presence of underlying bleeding disorders.[4] Treatment of the underlying cause helps in the remission. However, in the cases with systemic etiology, psychological stressors act as a precipitating factor. There are case reports of bleeding from skin (hematohidrosis), bleeding from eyes (hemolacria)[5] and ears (blood otorrhea) [6]. CASE REPORT A 9-year-old girl was brought to the OPD with complaint of blood oozing from the left upper arm for past 1 month. First episode of bleeding happened 1 month before at the school following the incident when she fought with her co student and was punished by her teacher. The teacher noticed bleeding from her left upper arm. They gave first aid, informed to her mother and sent home. Since then, the bleeding occurred about once or twice a week, which was spontaneous, lasted for about 3–5 min from left upper arm. Not associated with pain. No history of bleeding from any other site, drug intake was present. No history of bleeding disorder or any skin infection was found. Family milieu patient was fearful of her father who was an alcoholic. He would scold her frequently, comparing her academic performances with her sibling. On examination, the child was alert, oriented and communicates relevantly. Physical examination revealed normal vital signs and average body built. On presentation, blood was oozing from the left arm, which was wiped off with sterile gauze. No signs of any physical injury. No tenderness, no sinus, no regional lymphadenopathy. BCG scar +. Written informed consent by the patient was obtained. The laboratory results were normal, regarding platelets count (3.2lacs), activated partial thromboplastin time (33.9 s), prothrombin time (12.7 s), and normal bleeding and clotting time. Microscopic examination of the discharge fluid revealed RBCs and other components of blood. Parents were counselled about the child's condition. Parents were taught about positive and negative reinforcement techniques and their advantages over punishment. Child was taught relaxation exercises and pharmacotherapy was initiated treatment with propranolol 10 mg twice daily. After 2 weeks of treatment, the bleeding episode gradually reduced in frequency and drug continued. The child is under regular follow up. DISCUSSION Hematohidrosis also known as hematidrosis, hemidrosis, and hematofolliculohidrosis is a rare clinical condition in which capillary blood vessels that feed the sweat glands rupture, causing bloody sweat; it occurs under conditions of extreme physical or emotional stress.[1] There are a various theory that explain the etiopathogenesis of hematohidrosis. The general consensus statement relates to the intensified sympathetic activation due to extreme physical or emotional stress. The sympathetic flight-or-fight response to intense stress leads to constriction of capillary vessels feeding sweat glands. When the anxiety subsides, the blood vessels dilates to the point of rupture, leading to them passage of blood through the ducts of sweat glands and presenting as the droplets of blood mixed with sweat on the intact skin surface in almost any part of the body. [7,8] The term “hematofolliculohidrosis” was proposed as it appears along with sweat and the blood pushes via follicular canals.[9] Diagnosis of hematohidrosis can be made if the following criteria are met: (i) recurrent, spontaneous, painless and self-limited oozing of bloody discharge from skin surface is witnessed and confirmed by health care professionals, (ii) usual blood components are found on biochemistry studies of the bloody discharge, and (iii) the site of bleeding is intact with no signs of abrasion, telangiectasia or purpura and after wiping the area, there is no evidence of oozing. All of these three criteria must be met in order to rule out other causes of bleeding like organic bleeding disorders, self-inflicted bleeding, factitious disorder by proxy, and chromhidrosis.[10] Histopathological examination of the affected by Zhang et al., revealed few intradermal bleeding and obstructed capillaries in the affected area. No apparent abnormality was found in sweat glands, hair follicles, and sebaceous glands.[3] Biopsy examination during asymptomatic period did not reveal any blood-filled vascular spaces, intradermal bleeding or apparent abnormality in hair follicle, sebaceous, or the sweat glands. Benzidine test can be used as important tool for diagnosis, where hemoglobin in blood reacts with H2O2-liberating oxygen, which then reacts with reagent producing a green to blue colored compound.[6] Treatment of this condition is not clear. There was one report of successful use of propranolol.[8] In this case, the patient shown improvement to pharmacotherapy (propranolol) along with psychotherapy. CONCLUSION Hematohidrosis should be considered as a miscellaneous differential diagnosis of bleeding in a patient with normal physical examination and laboratory investigations. Establishing awareness among the physicians play an important role in ensuring a non-judgmental and compassionate approach. Needed effective case reporting after diagnosing hematohidrosis, to identify etiology and risk factors of this condition. And to establish effective clinical management protocol. REFERENCES 1.Holoubek JE, Holoubek AB. Blood, sweat and fear.“A classification of hematidrosis” J Med. 1996;27:115–33. [PubMed] [Google Scholar] 2.Patel RM, Mahajan S. Hematohidrosis: A rare clinical entity. Indian Dermatol Online J 2010;1:30-2. 3.Zhang FK, Zhang YL, Liv JH, et al. Clinical and laboratory study of a case of hematidrosis. 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