Asian Journal of Case Reports in Medicine and Health 5(3): 10-13, 2021; Article no.AJCRMH.67391 Hematohidrosis: A Rare and Mysterious Case Shrawan Kumar1*, K. K. Bhoi2 and Gajanan Yelme3 1Department of General Medicine, Shri Balaji Institute of Medical Sciences, Mowa, Raipur, 492001, India. 2Department of Neurology, Shri Balaji Institute of Medical Sciences, Mowa, Raipur, 492001, India. 3Department of Paediatrics, Shri Balaji Institute of Medical Sciences, Mowa, Raipur, 492001, India. Authors’ contributions This work was carried out in collaboration among all authors. Author SK manuscript preparation. Author KKB clinical follow up. Author GY concept and design. All authors read and approved the final manuscript. Article Information Editor(s): (1) Prof. Arun Singh, Rohilkhand Medical College and Hospital, India. Reviewers: (1) Usama Bin Zubair, Ireland. (2) Andreja Celofiga, University Medical Centre Maribor, Slovenia. Complete Peer review History: http://www.sdiarticle4.com/review-history/67391 Received 18 February 2021 Accepted 23 April 2021 Case Study Published 08 May 2021 ABSTRACT Aim: 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, Shri Balaji Institute of Medical Science, 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. Conclusion: Propranolol was used as an efficient beta blocker against the condition and stress was declared as one of the contributory factor for hematohidrosis. Keywords: Blood sweating; hematohidrosis; lorazepam;propranolol; recurrent hematohidrosis. _____________________________________________________________________________________________________ *Corresponding author: E-mail: [email protected]; Kumar et al.; AJCRMH, 5(3): 10-13, 2021; Article no.AJCRMH.67391 1. INTRODUCTION to be within normal limits.On detailed examination no physical or gynaecological Hematohidrosis or Hematidrosis is a rare abnormality was noted. The secretion from face condition with characterised recurrent episodes was collected and processed for benzidine test of spontaneous bleeding in normal skin and and microscopic examination. The bleeding was intact mucosa. There is a mention of related cleaned and the area was examined for trauma instance in Leonardo da Vinci’s description about or self inflicted wounds. Absence of injury was a soldier under extreme stress and also this noted and the skin over the face appeared phenomenon has been proposed as an normal. Absence of systemic disease and allergy explanation for Jesus Christ’s crucifixion in the towards drug or food was noted. Later, in the garden of Gethsemane and the claims hospital another episode of bleeding was associated with stigmata [1-2]. The possible observed and was witnessed by four physicians explanation for hematohidrosis can be systemic and nursing staff. The blood secretion was found disorders, bleeding disorders, vicarious to be spontaneous for eight to ten minutes.On menstruation, excess exertion, and psychological biochemical examinations, the secretion from stressors [3]. The exudation of blood is caused face was positive for the presence of blood by the rupture of capillary blood vessels that components with no other abnormalities. nourishes sweat glands as a result of stress [4]. Complete blood count, bleeding and clotting Previous literature suggests such bleeding from time, prothrombin time, active partial thrombin skin (hematohidrosis), eyes (hemolacria), and time, liver function tests, renal function tests, ears (blood otorrhea) [2,5-8]. Soliman et al on the hemogram, bleeding and clotting time, other hand reported one of its first kinds of prothrombin time, active partial thrombin time, Hematohidrosis in genital and perineal area [4]. liver and renal function tests, computed We hereby present a case of 13 years old Indian tomography scan of the head and girl presented with recurrent spontaneous ultrasonography of the abdomen and pelvis were bleeding mixed with sweat from intact skin, found to be normal. The secretion was confirmed diagnosed as Hematohidrosis, in order to better to be blood by benzidine test and microscopic understand the condition. examination also revealed blood elements in the sample. 2. CASE REPORT As informed by the patient and her family, A 13 years old female child resident of Raipur, symptoms usually appeared when she was afraid Chhattisgarh, India, was presented to the dept. of or in stress, particularly during her exams. Also, general medicine, Shri Balaji Institute of Medical in the hospital during the episode she showed Science, Raipur on 29 June 2018 with high grade anxiety and expressed fear and complaints of spontaneous bleeding from intact stress regarding academic performance. skin of face- on forehead, cheeks and near lateral parts of the face. The bleeding was noted The patient was prescribed vitamin C on daily to be spontaneous and recurring. The patient did basis and propranolol 10mg twice daily for ten not have any history of trauma or any other days and tapered for 1 month. No recurrence of bleeding sites such as gums/ oral mucosa/ sub the bleeding episode was noted until two months conjunctiva/ epistaxis/ hematoma; scurvy and during her academic exams. Possibility of other bleeding dyscrasias were ruled out. exacerbation associated with anxiety and stress Bleeding was found to be not associated with was noted. Propranolol repeat dose was advised menstruation. Also, no family history of any and the patient did not report any such bleeding bleeding tendency was noted. The patient had complaints for six months. similar complaints three months back and thrice in the previous year. She had history of recurrent 3. DISCUSSION bleeding episodes 4-5 times in three days. She was admitted in the hospital for observation and Hematidrosis is extremely rare and enigmatic to rule out any malingering. disorder characterised by recurrent episodes of spontaneous bleeding from intact skin, which is The patient was alert and no abnormality was usually self-limiting. The etiopathogenesis for the noted on general examination. She was found to condition as proposed by few authors includes be oriented, comprehending, and communicated post systemic disease, excessive exertion or well with nearby people. No psychotic symptoms stress, vicarious menstruation, psychogenic and were elicited and her intelligence was observed unknown [2,9,10,6]. In most of the cases, acute 11 Kumar et al.; AJCRMH, 5(3): 10-13, 2021; Article no.AJCRMH.67391 Fig.1. Sweating blood as observed in the case stress and anxiety were the precipitating factors. were found to decrease. This case and treatment In our case, the patient was advised vitamin C methodology used to decrease the episode is owing to its probable role in sympathetic activity similar to our case and results [13]. Certain role and anxiety.The phenomenon was found to be of sympathetic system has been observed in the isolated and was not found to be associated with recurrent episodes of hematohidrosis which is general health condition. usually common during emotional or physical stress, as in our case. The patients were found to Duan et al reported hematohidrosis associated be relieved and recurrence of hematohidrosis with primary thrombocytopenic purpura. decreased on administration of beta blockers Diagnosis was made by exclusion and clinically similar to our study. Bhattacharya et al noted findings of peripheral blood exudates of intact ineffectiveness of diazepam and have reported skin. In our study no such abnormality was found. effective results with propranolol thus, There is lack of knowledge about pathology of recommending the use of propranolol and the condition. However, some studies have supporting the treatment used in our study [14]. suggested increased vascular pressure leading to passage of blood cells through the duct of 4. CONCLUSION sweat glands; vasculitis of dermal vessels and exacerbated sympathetic activation leading to We treated our patient with propranolol 10mg periglandular vessel constriction and subsequent twice daily based on its similar uses in the expansion, allowing the passage of blood content literature and its effective observations. This led into ducts [6,11]. to a significant observation and remission was achieved till date. In conclusion, stress is noted to Hematohidrosis should be differentiated from be a contributory factor for the progression of the chromatidrosis, meaning coloured sweat due to abnormality. The phenomenon is noted as rare coloured apocrine secretion and also from other condition and similar and associated case studies haemorrhagic disorder including platelet are advised for the better understanding of the ambnormality, coagulation or vasculature disease and devising a treatment protocol. abnormality which usually is found to be associated with petechae, purpura, mucosal CONSENT bleding or hematoma. Differential diagnosis for the disorder must include chromhidrosis,