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Recurrent Bleeding in an 18-Year-Old Girl

Recurrent Bleeding in an 18-Year-Old Girl Hematidrosis is an extremely rare and enigmatic disorder characterized by recurrent episodes of self-limited skin bleeding that can affect almost any part of the body. Although the condition is not life-threatening, affected individuals can be stigmatized owing to religious beliefs. Report of a Case An 18-year-old white woman presented with a year-long history of recurrent episodes of spontaneous bleeding lasting for a few minutes. Bleeding episodes occurred from previously healthy skin of her forehead, ears, eyelids, abdomen, and interdigital folds of both hands (Figure 1) and were related to emotional and physical stress and occasionally preceded by headache and abdominal pain. After these episodes, physical examination revealed absence of skin disruption or signs of self-inflicted lesions, even with dermoscopic examination (Figure 2). A fresh smear of the hematic exudate taken from her abdomen showed all blood elements in diluted form. The findings of a complete hematologic study were normal, and there was no evidence of internal bleeding or psychiatric abnormality. A biopsy specimen was taken immediately after interdigital fold bleeding and showed normal skin without any relevant histopathologic changes. Diagnosis of hematidrosis was made, and treatment with propranolol, 30 mg/d, was started. Frequency of the episodes decreased after starting treatment with propranolol. View LargeDownload Figure 1. Bleeding from the interdigital fold. View LargeDownload Figure 2. Dermoscopic image from interdigital fold bleeding showing absence of skin disruption. Comment Hematidrosis is a rare phenomenon that manifests itself more frequently in young women as bleeding episodes, often related to stress situations or acute fear, although they can also appear without any obvious precipitating factors.1-5 It is important to document that bleeding episodes occur from intact skin and that the exudate contains all peripheral blood components to rule out the most important differential diagnosis: self-injury and chromhidrosis (sweat discoloration due to bacteria or colored substances). Hematologic diseases affecting platelets, coagulation, or blood vessels should also be ruled out, although these conditions often present themselves with petechiae, purpura, or ecchymoses and not as blood oozing from intact skin. Very little is known about the pathologic mechanism of hematidrosis, although the latest publications try to offer a plausible explanation. In 1 case,3 multiple, distinct, dermal-located, blood-filled spaces that opened directly to the skin surface or into the follicular canals were found. These spaces contained no demonstrable endothelial cells and were not marked with factor VII or CD34. In another case,4 areas of loosening among connective tissue in dermis were also found but without red blood cells inside. The response to propranolol treatment described in 2 cases, as well as in our case, suggests that sympathetic nerve activation might play a role in these events.4,5 Although the bleeding episodes are self-limited and non–life-threatening, further research is needed to establish its cause and appropriate treatment to prevent social isolation of affected patients and their families. Back to top Article Information Corresponding Author: Dr Martinez, Department of Dermatology, Hospital General Universitario de Alicante, C/Pintor Baeza 12, Alicante, 03010 Spain (nuria_latorre_martinez@hotmail.com). Financial Disclosure: None reported. References 1. Holoubek JE, Holoubek AB. Blood, sweat and fear: “a classification of hematidrosis”. J Med. 1996;27(3-4):115-1338982961PubMedGoogle Scholar 2. Carvalho AC, Machado-Pinto J, Nogueira GC, Almeida LM, Nunes MB. Hematidrosis: a case report and review of the literature. Int J Dermatol. 2008;47(10):1058-105918986356PubMedGoogle ScholarCrossref 3. Manonukul J, Wisuthsarewong W, Chantorn R, Vongirad A, Omeapinyan P. Hematidrosis: a pathologic process or stigmata: a case report with comprehensive histopathologic and immunoperoxidase studies. Am J Dermatopathol. 2008;30(2):135-13918360116PubMedGoogle ScholarCrossref 4. Wang Z, Yu Z, Su J, et al. A case of hematidrosis successfully treated with propranolol. Am J Clin Dermatol. 2010;11(6):440-44320666570PubMedGoogle ScholarCrossref 5. Praveen BK, Vincent J. Hematidrosis and hemolacria: a case report. Indian J Pediatr. 2012;79(1):109-11121617906PubMedGoogle ScholarCrossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Dermatology American Medical Association

Recurrent Bleeding in an 18-Year-Old Girl

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References (5)

Publisher
American Medical Association
Copyright
Copyright © 2012 American Medical Association. All Rights Reserved.
ISSN
0003-987X
eISSN
1538-3652
DOI
10.1001/archdermatol.2012.1007
Publisher site
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Abstract

Hematidrosis is an extremely rare and enigmatic disorder characterized by recurrent episodes of self-limited skin bleeding that can affect almost any part of the body. Although the condition is not life-threatening, affected individuals can be stigmatized owing to religious beliefs. Report of a Case An 18-year-old white woman presented with a year-long history of recurrent episodes of spontaneous bleeding lasting for a few minutes. Bleeding episodes occurred from previously healthy skin of her forehead, ears, eyelids, abdomen, and interdigital folds of both hands (Figure 1) and were related to emotional and physical stress and occasionally preceded by headache and abdominal pain. After these episodes, physical examination revealed absence of skin disruption or signs of self-inflicted lesions, even with dermoscopic examination (Figure 2). A fresh smear of the hematic exudate taken from her abdomen showed all blood elements in diluted form. The findings of a complete hematologic study were normal, and there was no evidence of internal bleeding or psychiatric abnormality. A biopsy specimen was taken immediately after interdigital fold bleeding and showed normal skin without any relevant histopathologic changes. Diagnosis of hematidrosis was made, and treatment with propranolol, 30 mg/d, was started. Frequency of the episodes decreased after starting treatment with propranolol. View LargeDownload Figure 1. Bleeding from the interdigital fold. View LargeDownload Figure 2. Dermoscopic image from interdigital fold bleeding showing absence of skin disruption. Comment Hematidrosis is a rare phenomenon that manifests itself more frequently in young women as bleeding episodes, often related to stress situations or acute fear, although they can also appear without any obvious precipitating factors.1-5 It is important to document that bleeding episodes occur from intact skin and that the exudate contains all peripheral blood components to rule out the most important differential diagnosis: self-injury and chromhidrosis (sweat discoloration due to bacteria or colored substances). Hematologic diseases affecting platelets, coagulation, or blood vessels should also be ruled out, although these conditions often present themselves with petechiae, purpura, or ecchymoses and not as blood oozing from intact skin. Very little is known about the pathologic mechanism of hematidrosis, although the latest publications try to offer a plausible explanation. In 1 case,3 multiple, distinct, dermal-located, blood-filled spaces that opened directly to the skin surface or into the follicular canals were found. These spaces contained no demonstrable endothelial cells and were not marked with factor VII or CD34. In another case,4 areas of loosening among connective tissue in dermis were also found but without red blood cells inside. The response to propranolol treatment described in 2 cases, as well as in our case, suggests that sympathetic nerve activation might play a role in these events.4,5 Although the bleeding episodes are self-limited and non–life-threatening, further research is needed to establish its cause and appropriate treatment to prevent social isolation of affected patients and their families. Back to top Article Information Corresponding Author: Dr Martinez, Department of Dermatology, Hospital General Universitario de Alicante, C/Pintor Baeza 12, Alicante, 03010 Spain (nuria_latorre_martinez@hotmail.com). Financial Disclosure: None reported. References 1. Holoubek JE, Holoubek AB. Blood, sweat and fear: “a classification of hematidrosis”. J Med. 1996;27(3-4):115-1338982961PubMedGoogle Scholar 2. Carvalho AC, Machado-Pinto J, Nogueira GC, Almeida LM, Nunes MB. Hematidrosis: a case report and review of the literature. Int J Dermatol. 2008;47(10):1058-105918986356PubMedGoogle ScholarCrossref 3. Manonukul J, Wisuthsarewong W, Chantorn R, Vongirad A, Omeapinyan P. Hematidrosis: a pathologic process or stigmata: a case report with comprehensive histopathologic and immunoperoxidase studies. Am J Dermatopathol. 2008;30(2):135-13918360116PubMedGoogle ScholarCrossref 4. Wang Z, Yu Z, Su J, et al. A case of hematidrosis successfully treated with propranolol. Am J Clin Dermatol. 2010;11(6):440-44320666570PubMedGoogle ScholarCrossref 5. Praveen BK, Vincent J. Hematidrosis and hemolacria: a case report. Indian J Pediatr. 2012;79(1):109-11121617906PubMedGoogle ScholarCrossref

Journal

Archives of Dermatology – American Medical Association

Published: Aug 1, 2012

Keywords: hemorrhage

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