Available Online at http://www.recentscientific.com International Journal of CODEN: IJRSFP (USA) Recent Scientific

International Journal of Recent Scientific Research Research Vol. 12, Issue, 03 (C), pp. 41293-41296, March, 2021 ISSN: 0976-3031 DOI: 10.24327/IJRSR Research Article

DIAGNOSIS & MANAGEMENT OF HEMATIDROSIS (SWEATING )

Elgharably Noura1 and Mohammed Al Abadie*2

1Medical School, Birmingham University, UK 2Department of Dermatology Royal Wolverhampton NHS Trust Wolverhampton WV10 0QP UK

DOI: http://dx.doi.org/10.24327/ijrsr.2021.1203.5863

ARTICLE INFO ABSTRACT

Hematidrosis is a rare condition where an individual excretes blood through the pores. Article History: Bleeding is apparent on the intact skin in many parts of the body. The aetiology is unclear; however, th Received 10 December, 2020 theories suggest severe episodes of stress as well as other causative factors including systemic nd Received in revised form 2 diseases, vicarious menstruation, extreme exertion and psychogenic causes can stimulate the January, 2021 bleeding. It can also be seen in bleeding disorders. Reporting on several cases where sweat with th Accepted 26 February, 2021 human blood appeared from the forehead, face and body and patients had no underlying bleeding th Published online 28 March, 2021 orders, we will explore the unusual presentation of Hematidrosis and where atropine sulphate transdermal patch and have resulted in remission. Key Words:

Hematidrosis, children, blood sweat

Copyright © Elgharably Noura and Mohammed Al Abadie, 2021, this is an open-access article distributed under the terms

of the Creative Commons Attribution License, which permits unrestricted use, distribution and reproduction in any medium,

provided the original work is properly cited.

INTRODUCTION The secretion was collected and examined microscopically where many erythrocytes were observed. Peripheral smear of Hematidrosis is a condition where a human sweats blood(1). It (2) the secretion showed multiple red blood cells with gram is believed to be triggered by stress, strain or exertion .True positive cocci and bacilli, confirming the blood to be of human Hematidrosis occurs in bleeding disorders, however it may also (3) origin. The patient’s routine hemogram, blood counts, , occur under extreme levels of stress . Near the sweat glands bleeding time, clotting time, active partial thrombin time, are blood vessels which can constrict and rupture when under prothrombin time, liver and renal function tests were all within the pressure of extreme physical or emotional stress. Here, as normal limits. the sweat glands produce sweat, the blood exudes to the (3) surface, appearing as a red watery secretion .

REVIEWING CURRENT LITERATURE

Biswas et al report a case of Hematidrosis in a healthy 12-year- (4) old girl with no underlying disorders . She presented with a 2-year history of bleeding from the skin over the forehead, scalp, cheek, nose and trunk. The bleeding was episodic, occurring once or twice a day, and particularly in the morning. There was no history of or prior stressful events. The episodes began with a watery secretion which turned bright right within a few seconds, and in total lasted around 10-15 Fig 1 sweat admixed with blood from the right side of the face(4) minutes. There was no bleeding from any other sites, and nothing indicated ingestion of anticoagulants, dyes or other drugs. There was no history of medical or surgical illnesses, no familial pattern and menstruation cycles were normal. There were no abnormalities on both physical and systemic examinations and the skin over the forehead remained intact (4) with no tenderness .

*Corresponding author: Mohammed Al Abadie Department of Dermatology Royal Wolverhampton NHS Trust Wolverhampton WV10 0QP UK Elgharably Noura and Mohammed Al Abadie., Diagnosis & Management of Hematidrosis (Sweating Blood)

In this case, the patient was treated with propranolol (10mg twice a day), and symptoms began to reduce within a week. Propranolol was continued twice a day for the next month and then decreased to 5mg twice a day for another month. In follow up, the patient reported complete remission of symptoms and episodes, except for one when she had run a race and had a small amount of blood sweat on her legs. Hence a suggestion for effective treatment are b-adrenoceptor antagonists.

Jayaraman et al reported a case of a 10-year-old girl who had a

1-week history of bleeding from the intact skin of the scalp(9).

(4) This initially began following a fight with a student in her Fig 2 sweat admixed with blood from the nose school despite no physical trauma occurring at the bleeding The patient was treated with an atropine transdermal patch over site. Following this incident, the bleeding was once or twice the sites involved and noted steady remission in severity and daily and lasted for 3-5 minutes with no associated pain. frequency of episodes. After 1 month of treatment, the patient The patient had no bleeding disorder, systemic disease, and stopped getting episodes(4). skin biopsy was normal. She had a 2-year history of bed In this case, no causative factor was identified. Some authors wetting at night which was more frequent during exam periods have suggested that severe mental anxiety activates the and when scolded by parents. The patient’s parents often would sympathetic nervous system to invoke a stress-fight reaction scold and punish her physically for her exam performance, and effective enough to cause haemorrhage of the vessels which the child expressed regret about coming home. Stress analysis supply the sweat glands into the ducts of the sweat was conducted via the Stress Questionnaire-Child/Adolescent glands(5).The pathology of Hematidrosis has also previously version, which highlighted the child was severely stressed. The been suggested to be distinctive vasculitis(6), however this case child was diagnosed with mixed anxiety and depressive used direct immunofluorescence study which showed no disorder, and initial treatment consisted of imipramine and abnormalities. Treatment of this condition was successful with clonazepam with a 1-week follow up(9). atropine sulphate transdermal patch, as the patient showed During a follow up interview of the patient, an episode of red complete remission. However other studies have suggested the secretions from her scalp was evident upon explanation that she success of propranolol(7). had been punished for her academic performance. Biochemical Wang et al have reported a case of a 13-year-old girl who examination of the secretion confirmed the substance to be experienced frequent bleeding episodes for over 3 years(7). human peripheral blood, with no other abnormalities Spontaneous bleeding from intact skin was witnessed by reported(9). Skin remained intact under the scalp, and the hospital staff on more than 20 occasions in hospital admissions, bleeding resolved within a minute. from the subungual area, palms, feet, thighs, and trunk. These This case was management with parental training and psycho episodes never occurred when sleeping at night and appeared education for the parents regarding the role of stress. Positive more frequently during times of emotional excitement. The and negative reinforcement techniques were encouraged over patient’s blood count, blood biochemistry, aggregation, physical punishment, and the child was taught relaxation bleeding time, coagulation screening tests, coagulation factors techniques. The bleeding completely stopped after 4 months and von Willebrand factor were all in normal limits, excluding and follow up over the next year showed complete remission. any haemorrhagic disorders. She also did not have In this case, the child’s severe mental anxiety and fear about menorrhagia, and the episodes were unrelated to menses, her educational performance presented as spontaneous excluding vicarious menstruation. Blood components were bleeding, highlighting the role of stress management in treating confirmed to contain the same components as the peripheral Hematidrosis. Remission was achieved with pharmacotherapy, blood. However, histopathology showed normal sweat gland supportive psychotherapy and family education. structure without blood when biopsy was performed post bleeding from the affected skin on the arm. The blood Vigorous exercise and sustained heat exposure were reported to capillaries were not obstructed, but minor blood extravasation trigger an episode of Hematidrosis in a report by Carvalho et al was observed. They concluded that the blood was mixed with a of a 9-year-old female who bled around the lips(10). The episode sweat-like fluid rather than real sweat and inferred a was unrelated to menses, and general examinations and sympathetic nerve activation was responsible for this(8). laboratory findings were all normal. Medical staff witnessed three later episodes, and biopsy confirmed the secretion to be Recurrent bleeding episodes were often linked to extreme peripheral blood. In one episode the patient was pre-examined, emotional excitement or fear. In a separate report, Manonukul (5) then observed in a hot environment where the episode began. et al. administered lorazepam , an anxiolytic to a patient to Medical staff were with her prior, during, and after the episode reduce her anxiety. The patient’s bleeding was initially treated and ensured that no injury had occurred. The report details that with vitamin C, carbazochrome sodium sulphonate and the patient spontaneously improved in her condition(10). diazepam – a similar anxiolytic – however this did prove to be Shahgholi reported three patients who had Hematidrosis. This efficacious. Thus, a suggestion of the role of sympathetic nerve (11) activation was explored, and propranolol was given to limit included an 8-year-old boy and two girls – aged 9 and 11 . vascular resistance. The boy was referred due to spontaneous bleeding from nail beds of hands and feet. This occurred prior to exams, playing 41294 | P a g e International Journal of Recent Scientific Research Vol. 12, Issue, 03 (C), pp. 41293-41296, March, 2021 computer games and when parents did not satisfy his demands. beta blockers. In 2 cases, remission was spontaneous without There were no physical or laboratory abnormalities, and the any medical intervention. Management with psychotherapy, diagnosis was confirmed by a medical professional who anxiolytic drugs or beta blockers proved to be effective in observed the bleeding. This patient did not require any medical almost all cases(12). intervention, and complete remission occurred after 2-year Pathology of Hematidrosis follow up. Little is known of the pathology of Hematidrosis. Common The 9-year-old girl presented with spontaneous bleeding reported causes include extreme episodes of fear, anxiety or episodes from her ears and nose. Again, there were no excitement. Stress can have physical and psychological abnormalities reported, and a 5-minute bleeding episode was manifestations. In most literature reviewed the cases were observed by a medical professional.The patient was given children, thus stress can be considered an important epigenetic propranolol 10mg twice daily, which results in improvement in factor in this age group. Some studies suggest the pathogenesis both severity and frequency of the bleeding. At 6 months as blood vessels surrounding the blood vessels constrict and follow up, the episodes had completely stopped(11). rupture under stress. Stress causes the activation of a The 11-year-old girl was admitted with a history of child abuse sympathetic response which may affect local vasculature and and malingering. Under admission, she experienced 2 episodes rupture blood vessels nearby sweat glands. Authors have of bleeding where a moist, viscous red discharge was emitted described this as the blood entering a duct near the exit of the from her forearm and back. The skin remained intact, and the skin surface, following rupture or diapedesis of superficial patient experienced no pain or agitation in the episode. The capillaries, and then leaking via the epidermis nearby ducts and patient was treated with regular counselling for her anxiety as sweat glands(5). well as propranolol 10mg daily. At 6 months follow up, there Some authors have described Hematidrosis as a distinctive was significant improvement in frequency and severity of vasculitis(13). This is due to the presence of intradermal episodes, but complete remission did not occur(11). bleeding and emphraxised capillaries with no changes sweat In a recent review of Hematidrosis by Kluger, 25 cases were glands, hair follicles, and sebaceous glands. However, evaluated and discussed according to presenting features and Hematidrosis has also been reported in situations that were not treatment options(12). Twenty four cases reported country of preceded by a stressful event. Thus, due to both its rarity and origin, with 15 from Asia (10 from India or Pakistan), 6 from unclear pathology, clinical and histological investigation Africa (either Maghreb or South Africa), and one from Europe. should be carried out in any case where it is a possible Bleeding episodes were localised mainly on the face in 24 differential. cases, including the forehead in 10 cases. Other areas involved Management -Reviewing Current Treatment Options included the eyes and ear in 10 and 9 cases respectively, the palms in 5 cases and the soles in 2 cases. Mucosal bleeding was In most of the cases reviewed, treatment with beta blockers(7), reported in 8 patients. Analysis of the bloody secretion was anxiolytics(14) and antidepressants(15) have been effective. carried out in 18 cases, and healthcare members witnessed a Similarly, the use of psychotherapy and counselling have also bleeding episode in 16 of the cases. Skin biopsies were been noted to be beneficial(10). The importance of a complete performed in 9 cases, and all reported minimal or inconsistent history was evident to reduce potential stressors thus aid changes. Triggering factors were identified in 14 cases, with management. stress within families, work or school reported in 12 of the Complete remission of symptoms varied from as soon as 1- cases. A psychiatric diagnosis was connected to Hematidrosis week post treatment, to 12 months after onset. In 2 cases in 9 patients. Examples of this included anxiety, depression, discussed, relapse occurred after stopping beta blockers, conversion disorder and oppositional defiant disorder. highlighting the importance of follow up. However, in many Bleeding presented as sweating of a red liquid that was less cases there was no medical treatment given, and once potential viscous than blood. Biochemical analysis showed that stressors were identified, the episodes were self-resolving. haemoglobin is at lower concentrations in bloody sweaty than The cases reported often included children, thus it may be in venous blood. The review highlighted some fascinating beneficial to obtain a collateral history from parents or school insights to the distribution of Hematidrosis, with almost half of teachers. the cases originating from India, and reports from Europe and North America very low. Interestingly, some patients also DISCUSSION reported mucosal bleeding, epistaxis, intestinal bleeding or haematuria. Despite this, only one case identified presence of a This review includes many recent cases of Hematidrosis. It is bleeding disorder (12). an interesting, uncommon condition where the pathology, incidence and prevalence remains unclear however, it is Management was reported in 20 cases and oral treatments appearing more commonly in recent literature. Many of the included beta blockers (45%), anxiolytic drugs (30%) and cases covered showed typical presentation, affecting young antidepressants (10%). Local treatments like atropine patches children commonly females. It mainly presents on the face, the or adrenaline drops were rarely used. Psychotherapy, scalp and the upper limbs. There was variation in frequency of counselling and relaxation therapies were given to 7 patients, episodes between the cases, but clinically the bleeding either alone or in association with pharmacological treatments. appeared as a red liquid which was less viscous than blood. In 2 cases, no treatment was given. The outcomes for 5 patients was not described. In 13 cases, healing occurred from a few It was confirmed in many cases to be the same as human days to 12 months. In 2 cases, relapses occurred after stopping peripheral blood under cytology. In all the cases, there was 41295 | P a g e Elgharably Noura and Mohammed Al Abadie., Diagnosis & Management of Hematidrosis (Sweating Blood) improvement or complete remission and management included 2. Bhagwat PV, Tophakhane RS, Rathod RM, anxiolytic drugs, beta blockers and psychotherapy. Shashikumar BM, Naidu V Hematohidrosis. Indian J Dermatol Venereol Leprol. 2009 May-Jun; 75(3):317-8. Interestingly, many patients had ethnic backgrounds that were 3. Holoubek JE, Holoubek AB Blood, sweat and fear. "A predominantly Asian. There could be a possible social and classification of hematidrosis". J Med. 1996; 27(3- cultural aspect in the distribution, where Asian culture 4):115-33. encourages strong academic performance thus could be a 4. Biswas S, Surana T, De A, Nag F. A curious case of potential stressor. Extreme stress or fear, including from sweating blood. Indian J Dermatol 2013;58:478-80 parents or school exams, has been identified as a trigger for 5. Manonukul J., Wisuthsarewong W., Chantorn R., Hematidrosis. However, in some cases there was no trigger Vongirad A., Omeapinyan P. Hematidrosis: a pathologic known. process or stigmata. A case report with comprehensive There are some limitations to this review. The articles and case histopathologic and immunoperoxidase studies. The reports were predominantly from PubMed and record in American Journal of Dermatopathology. 2008; English. Only cases which identified Hematidrosis by the 30(2):135–139. author were included, thus we may have missed cases of 6. Zhang FK, Zheng YL, Liu JH, Chen HS, Liu SH, Xu misdiagnosis. Additionally, some cases included have not MQ, et al. Clinical and laboratory study of a case of provided a thorough follow up or exploration of the hematidrosis. Zhonghua Xue Ye Xue Za Zhi. 2004; presentation. 25:147–50. 7. Wang, Z., Yu, Z., Su, J. et al. A Case of Hematidrosis The pathology of Hematidrosis remains unclear and skin Successfully Treated with Propranolol. Am J Clin biopsies fail to report abnormal findings. There may be a role Dermatol 11, 440–443 (2010 of stress in the sympathetic nervous system causing vascular 8. Jacobsen TN, Converse Jr RL, Victor RG. Contrasting rupture and permeability. This rationalises the benefit of beta effects of propranolol on sympathetic nerve activity and blockers, anti-depressants, anxiolytics and psychotherapy. vascular resistance during orthostatic stress. Circulation CONCLUSION 1992; 85: 1072–6 9. Jayaraman AR, Kannan P, Jayanthini V.(2017). An In the cases explored, Hematidrosis presents with self-limiting Interesting Case Report of Hematohidrosis. Indian J bleeding episodes from many areas of the body, but frequently Psychol Med. 39(1):83-85. doi: 10.4103/0253- on the face with skin remaining intact. The exact cause remains 7176.198953. PMID: 28250564; PMCID: PMC unknown; however, authors explore two theories whereby the 5329997. increased vascular pressure initiated by a high stress event, 10. Carvalho AC, Machado-Pinto J, Nogueira GC, Almeida initiates the blood vessels around sweat glands to contract, then LM, Nunes MB. Hematidrosis: a case report and review dilate and rupture, allowing the blood to pass into the ducts of (5) of the literature. Int J Dermatol. 2008 Oct;47(10):1058- the sweat glands .This is intensified by sympathetic activation 9. doi: 10.1111/j.1365-4632.2008.03746.x. PMID: which causes periglandular vessel constriction and later 18986356. expansion, leading to blood passage into ducts, resulting in (5) 11. Shahgholi E. A case series of hematohidrosis: A blood combined with sweat . 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How to cite this article:

Elgharably Noura and Mohammed Al Abadie.2021, Diagnosis & Management of Hematidrosis (Sweating Blood). Int J Recent Sci Res. 12(03), pp. 41293-41296. DOI: http://dx.doi.org/10.24327/ijrsr.2021.1203.5863

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