Case study Hematohidrosis: A rare and mysterious case . ABSTRACT Background: Hematohidrosis is basically sweating of blood. It is rare and possibly induced by stress. Few cases have been studied in the past but standard treatment protocols are yet to be introduced. Objective: To report a rare case of hematohidrosis 13 years old female child. Presentation of case: A 13 years old female child was presented at the department of general medicine, SBIMS, Raipur. The patient was found to be bleeding from intact skin of the face. The patient had no history of trauma or associated bleeding due to infection and was alert and had no abnormality in general examination. Biochemical and microscopic examinations suggested the presence blood and blood components. The patient was advised 10 mg of propranolol and no recurrent hematohidrosis was observed. Discussion: Previous literature suggests effective usage of propranolol and lorazepam as anxiolytic. We found significant and effective role of propranolol against the disease and remission was achieved. Keywords: [Hematohidrosis; blood sweating; propranolol; lorazepam; recurrent hematohidrosis 1. INTRODUCTION Hematohidrosis is a rare condition with characterised recurrent episodes of spontaneous bleeding normal skin and intact mucosa. There is a mention of related instance in Leonardo da Vinci’s description about a soldier under extreme stress and also this phenomenon has been proposed as an explanation for Jesus Christ’s crucifixion in the garden of Gethsemane and the claims associated with stigmata (Edwards et al, 1986; Jayaram et al, 2017). The possible explanation for Hematohidrosis can be systemic disorders, bleeding disorders, vicarious menstruation, excess exertion, and psychological stressors. (Champion, 1992) The exudation of blood is caused by the rupture of capillary blood vessels nourishing sweat glands as a result of stress. (Soliman et al, 2019) Previous literature suggests such bleeding from skin (hematohidrosis), eyes (hemolacria) (Norn, 1977), and ears (blood otorrhea) (Tshifularo, 2014). Soliman et al on the other hand reported one of its first kind of Hematohidrosis in genital and perineal area. (Soliman et al, 2019). Hematohidrosis associated with primary thrombocytopenia purpura by Duan et al. Migliorini reported a case of hematodrosis, otorrhea with otoerythrosis. Few cases in the literature have reported blood with sweat like discharge from facial skin. (Jayaraman et al, 2017; Biswas et al, 2013; Arakkal et al, 2017) We hereby present a case of 13 years old Indian girl presented with recurrent spontaneous bleeding mixed with sweat from intact skin, diagnosed clinically and ruling out all the other possibilities, as Hematohidrosis. 2. CASE REPORT A 13 years old female child resident of Raipur, Chhattisgarh, India, was presented to the dept. of general medicine, SBIMS, Raipur on 29 June 2018 with complaints of spontaneous bleeding from intact skin of face- on forehead, cheeks and near lateral parts of the face. The bleeding was noted to be spontaneous and recurring. The patient did not have any history of trauma or any other bleeding sites such as gums/ oral mucosa/ sub conjunctiva/ epistaxis/ hematoma; scurvy and other bleeding dyscrasias were ruled out. Bleeding was found to be not associated with menstruation. Also, no family history of any bleeding tendency was noted. The patient had similar complaints three months back and thrice in the previous year. She had history of recurrent bleeding episodes 4-5 times in three days. Symptoms usually appeared when she was afraid or in stress, particularly during her exams. She was admitted in the hospital for observation and to rule out any malingering. The patient was alert and no abnormality was noted on general examination. She was found to be oriented, comprehending, and communicated well with nearby people. No psychotic symptoms were elicited and her intelligence was observed to be within normal limits. She showed high grade anxiety and expressed fear and stress regarding academic performance. On detailed examination no physical or gynaecological abnormality was noted. The secretion from face was collected and processed for benzidine test and microscopic examination. The bleeding was cleaned and the area was examined for trauma or self inflicted wounds. Absence of injury was noted and the skin over the face appeared normal. Absence of systemic disease and allergy towards drug or food was noted. Later, in the hospital another episode of bleeding was observed and was witnessed by four physicians and nursing staff. The blood secretion was found to be spontaneous for eight to ten minutes. On biochemical examination, the secretion from face was positive for the presence of blood components with no other abnormalities. Complete blood count , bleeding and clotting time, prothrombin time, active partial thrombin time, liver function tests, renal function testsHemogram, bleeding and clotting time, prothrombin time, active partial thrombin time, liver and renal function tests, computed tomography scan of the head and ultrasonography of the abdomen and pelvis were found to be normal. The secretion was confirmed to be blood by benzidine test and microscopic examination also revealed blood elements in the sample. The patient was prescribed propranolol 10mg twice daily for ten days and tapered for 1 month. The patient was also advised vitamin C owing to its probable role in sympathetic activity and anxiety based on previous studies (Moritz et al, 2020; Ribeiro 2015). No recurrence of the bleeding episode was noted until two months during her academic exams. Possibility of exacerbation associated with anxiety and stress was noted. Propranolol repeat dose was advised and the patient did not report any such bleeding complaints for six months. 3. DISCUSSION Hematidrosis or hematidrosis means – blood in sweat. It is extremely rare and enigmatic disorder characterised by recurrent episodes of spontaneous bleeding from intact skin, which is usually self-limiting. The etiopathogenesis for the condition as proposed by few authors includes post systemic disease, excessive exertion or stress, vicarious menstruation, psychogenic and unknown (Jayaraman et al, 2017; Jerajani et al, 2009; Holobek JE and Holoubek AB, 1996, Arakkal et al, 2017). In most of the cases, acute stress and anxiety were the precipitating factors. The phenomenon was found to be isolated and was not found to be associated with general health condition. Duan et al reported hematohidrosis associated with primary thrombocytopenic purpura. Diagnosis was made by exclusion and clinically findings of peripheral blood exudates of intact skin. There is lack of knowledge about pathology of the condition. However, some studies have suggested increased vascular pressure leading to passage of blood cells through the duct of sweat glands; vasculitis of dermal vessels and exacerbated sympathetic activation leading to periglandular vessel constriction and subsequent expansion, allowing the passage of blood content into ducts. (Arakkal et al, 2017; Zhang et al, 2004; Wang et al, 2010) Hematohidrosis should be differentiated from chromatidrosis, meaning coloured sweat due to coloured apocrine secretion and also from other haemorrhagic disorder including platelet ambnormality, coagulation or vasculature abnormality which usually is found to be associated with petechae, purpura, mucosal bleding or hematoma. Differential diagnosis for the disorder must include chromhidrosis, factitious dermatitis, vicarious menstruation, vasculitis, and platelet and coagulation disorders. Lab investigations are of utmost importance. Till date there no specific treatment for the disease has been identified. However, Manolukul, et al used lorazepam as anxiolytic and significant results were obtained. Zhaoyue, et al. used propranolol with the hypothesis of sympathetic overactivity and was found to be effective against the disease. Certain role of sympathetic system has been observed in the recurrent episodes of hematohidrosis which is usually common during emotional or physical stress, as in our case. The patients were found to be relieved and recurrence of hematohidrosis decreased on administration of beta blockers. Bhattacharya et al noted ineffectiveness of diazepam and have reported effective results with propranolol. 4. CONCLUSION We treated our patient with propranolol 10mg twice daily based on its similar uses in the literature and its effective observations. This led to a significant observation and remission was achieved till date. In conclusion, stress is noted to be a contributory factor for the progression of the abnormality. The phenomenon is noted as rare condition and similar and associated case studies are advised for the better understanding of the disease and devising a treatment protocol. CONSENT Consent taken ETHICAL APPROVAL Ethical approval taken from Institutional ethical committee REFERENCES 1. Edwards WD, Gabel WJ, Hosmer FE. On the physical death of Jesus Christ. Jama. 1986 Mar 21;255(11):1455- 63. 2. Jayaraman AR, Kannan P, Jayanthini V. An interesting case report of hematohidrosis. Indian journal of psychological medicine. 2017 Jan;39(1):83-5. 3. Champion RH. Disorders of sweat glands. Textbook of Darmatology. 1992:1745-62. 4. Soliman M, Mowafy K, Soliman R, Soliman R. 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