Case report

XXX Blackwell Oxford, International IJD 1365-4632 0011-9059 2008 The UK Publishing International Journal Ltd of Dermatology Society of Dermatology

Hematidrosis: a case report and review of the literature

da Silva Carvalho Hematidrosis Dermatologic surgery et al.

Ana Carolina da Silva Carvalho, MD, Jackson Machado-Pinto, PhD, Gustavo Carneiro Nogueira, MD, Luiz Maurcio Costa Almeida, MD, and Maurcio Buzelin Nunes, MD, PhD, MSc

From the Dermatology and Pathology Departments, Santa Casa de Belo Horizonte, Brazil Correspondence Jackson Machado-Pinto, PhD Av. Francisco Sales 1463/1207 Santa Egnia Belo Horizonte-MG Brazil CEP: 30150-221 E-mail: jackpin@gold.com.br

Abstract
The authors report the case of a 9 year-old female who had bleeding episodes around the mouth after strenuous exercise or prolonged exposure to heat. Characteristically, bleeding occurred right after sweat drops started appearing on the surface of the skin around the mouth. The bleeding episodes ceased spontaneously.

Case Report A 13-year-old girl reported the onset, in November 2004, of bleeding surrounding the lips during physical exertion or an increase in room temperature, which ceased spontaneously. The episodes were not related to menses, ruling out cutaneous endometriosis. General clinical examination and all laboratory evaluations were normal. Three episodes where witnessed by medical staff. In one episode, 25 physicians were present and the whole procedure was registered on a film. Initially, the patient was examined and no wounds were found. She was then observed in a favorable environment (hot weather). In about 30 min, a red secretion was observed around the upper lip (Fig. 1). Medical staff were with her at all times to ensure that no previous injury had occurred. A smear of the upper lip secretion was carried out and a biopsy was performed. The secretion revealed the same components as found in peripheral blood. Histologic analysis showed a normal epidermis and a dermis with preserved pilosebaceous annexes and eccrine sweat glands (Fig. 2). There were small, congested, periglandular blood capillaries, some in close contact, without red blood cell extravasation (Fig. 3). The patient evolved with spontaneous improvement of the condition. Discussion Hematidrosis, the excretion of bloodsweat, is a rare disease, with only a few cases reported to date. The case reported here occurred spontaneously. Hematidrosis can be located in any part of the body, and there may be associated pain or local erythema. As isolated phenomena, bleeding episodes do not affect the patients general state of health.1 In a recent
International Journal of Dermatology 2008, 47, 10581059

Figure 1 The patient during a bleeding episode

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review of hematidrosis, 66 cases were evaluated and classified according to causative factors: systemic disease, vicarious menstruation, physical exertion, psychogenic, and unknown.1 Cases related to systemic diseases are caused mainly by
2008 The International Society of Dermatology

da Silva Carvalho et al.

Hematidrosis Case report

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Figure 2 Histopathology of the skin in the bleeding area

(hematoxylin and eosin, 40)

three categories: a single episode, recurrent episodes, and stigmatization. Blood sweating as a single bleeding occurrence generally takes place during an acute episode of fear of death or severe body injury. Recurrent episodes are a consequence of emotional excitement and hypnosis.1 Stigmatization historically refers to the transfer of the wounds of Jesus Christ to believers by fervent praying, and occurs as scars, pain, wounds, and bleeding through intact skin at regular intervals.1,3,4 A few cases have unknown origins. Diagnosis is made by the finding of peripheral blood cell exudate of intact skin, witnessed and collected by a physician or other reliable medical personnel.1,5 It should be differentiated from chromatidrosis and pseudochromatidrosis.1 It is important to rule out bleeding from self-injury.6 Little is known of the histopathology. Some studies have shown that the blood enters a duct next to the exit point on the skin surface, which indicates escape by rupture or diapedesis of superficial capillaries, and leakage through the epidermis surrounding ducts and sweat glands.1 Another recent study showed intradermal bleeding and emphraxised capillaries, with no abnormalities in the sweat glands, hair follicles, and sebaceous glands, suggesting that hematidrosis is a distinctive vasculitis.5 It is probable that there may be a minor communication between a small blood vessel and sweat gland. Because it is a rare disease and its histopathology is unknown, any suspected case of hematidrosis should be investigated both clinically and histologically. References
1 Holoubek JE, Holoubek AB. Blood, sweat and fear. J Med 1996; 27: 115133. 2 Jones HW, Wentz AC, Barnett LS, eds. Novaks Textbook of Gynecology, 11th edn. Baltimore, MD: Williams & Wilkins, 1988: 259. 3 Lifschutz JE. Hysterical stigmatization. Am J Psychiatry 1957; 114: 527531. 4 Klauder JV. Stigmatization. Arch Dermatol 1938; 37: 650 659. 5 Zhang FK, Zheng YL, Liu JH, et al. Clinical and laboratory study of a case of hematidrosis. Zhonghua Xue Ye Xue Za Zhi 2004; 25: 147150. 6 Allen AC. Disorders of cutaneous appendages. In: Allen AC, ed. The Skin: A Clinicopathological Treatise, 2nd edn. New York: Grune & Stratton, 1967: 747.

Figure 3 Congested capillary in close contact to an eccrine gland

(hematoxylin and eosin, 400)

scurvy, epilepsy, and malaria. Vicarious menstruation is a designation applied to cases of extragenital hemorrhage at periodic intervals, corresponding to the menstrual cycle, mostly related to endometriosis. The most frequent location is the nasal mucosa, as epistaxis.1,2 Cases related to excessive physical exertion occur with the performance of certain strenuous exercises. Psychogenic cases can be divided into

2008 The International Society of Dermatology

International Journal of Dermatology 2008, 47, 10581059