Abstract
Introduction
Hematohidrosis/hematidrosis/hemidrosis is a rare clinical condition in which human being sweats blood under condition of extreme physical or emotional stress [1. Jerajai HR, Bhagyashri Jaju, Phiske MM, and Nitin Lade. Hematohidrosis-a rare clinical phenomenon. Indian J Dermatol. 2009 Jul-Sep; 54(3): 290–292; 2. Martinez NL, Mas IB, Paz AFM, Boronat JL. Recurrent bleeding in an 18-year old girl. Arch Dermatol.2012; 148(8):960–961.] The capillary blood vessels that feed the sweat glands rupture, causing them to exude blood as sweat, under stressful conditions, occurring in religious (Jesus Christ prayer experience in Gethsemane) and non-religious (soldier sweat blood before battle) [2. Martinez NL, Mas IB, Paz AFM, Boronat JL. Recurrent bleeding in an 18-year old girl. Arch Dermatol.2012; 148(8):960–961.]. It is a condition where there is a spontaneous painless bleeding through unbroken skin in any part of the body. It is self-limiting in nature with a good prognosis [3. Patel RM, Mahajan S. Hematohidrosis: A rare clinical entity. Indian Dermatol Online J.2010 Jul; 1(1):30–2.]. The diagnosis of hematohidrosis is made on the presence of bloody discharge without any obvious cause through intact skin, witnessed and confirmed by health professional/doctor/nurse and the presence of blood components on biochemistry studies of the discharge. Hematohidrosis is a well-recognised diagnosis according to International Classification of Diseases (ICD-9-CM: 705–89) (ICD9 Data.com; 2013).
Case report
This study reports the clinical finding, radiology, audiology and histology of four cases of a rare isolated otological hematohidrosis. Our cases are of particular interest because to our knowledge these are the first documented cases of psychogenic hematohidrosis isolated otological (ear) presentation.
Discussion
A comprehensive literature review was performed on reports documenting hematohidrosis/hematidrosis cases. The clinical presentation, diagnosis, treatment, pathophysiology and epidemiology of hematohidrosis. The purpose is to report and raise awareness of hematohidrosis among ENT patients.
1
Introduction
Hematohidrosis [ ] is an excretion of blood or blood pigments in the sweat according to Stedman’s Medical dictionary, an observation by Aristotle “some sweat with a bloody sweat” (Hist. Animal 111, 19) . The reported cases of hematohidrosis appeared to be associated with a severe anxiety reaction with fear implicated as the inciting factor. Cases of hematohidrosis have been reported in the French literature by Broeg in 1907 and by Darier in 1930; in the British literature in 1918 by Scott, in the German literature by Riecke in 1923, and in the Russian literature by Lavsky in 1932 and by Gadzhiev and Listengarten in 1962 .
Hematohidrosis is a rare condition, of unknown aetiology (sweating blood). Hematohidrosis is actually a medical condition in which extremely high blood pressure causes blood to seep into sweat glands and escape through pores—illusion of sweating blood . Vasculitis has also been postulated as a cause of hematohidrosis . It is a condition where there is a spontaneous bleeding through unbroken skin in any part of the body . The diagnosis is made by observation of red blood corpuscles (erythrocytes) and other blood components, with no other blood or physical abnormalities found to account for the phenomenon. The documented by attending doctor/physician or any other reliable medical personnel of bloody ear discharges .
There were no recent literature reports noted of isolated otological hematohidrosis cases, to our knowledge we are reporting the first case series of psychogenic isolated otologic hematohidrosis.
After my oral presentation entitled “I sweat blood—bloody otorrhea” in our scientific national ENT congress 2003, no one believed my story. Since 2001 my interest has always been high on bloody otorrhea, hematohidrosis. I received 3 other cases communicated to me by another ENT specialist, with similar presentation. In this community of Gauteng province (RSA) in Africa. We present four case series since 2001–2013 confirmed by four different senior ENT specialists with similar history of blood stained otorrhea under severe stress with no obvious pathological cause, normal intact tympanic membrane and ear canal. It is possible that other specialists are seeing this phenomenon. This report is written to document and raise an awareness of hematohidrosis among ENT patients.
2
Case series
2.1
Patient 1: 2001–2003
A 30 year old married Black lady, a lawyer by profession, presented with intermittent bilateral spontaneous painless non-clotting bloody discharge from both ears. The episodes lasted for 15–20 min with 2–5 ml blood stained discharges from each ear per an attack, 2–3 attacks in a week in every 3–4 month interval a total of 8 episodes since 2001–2003. In the end of 2001 she got married, was appointed to a very demanding senior management position in a private company and was a part-time MBA student (a demanding stressful state). These episodes were usually aggravated by periods of severe mental stresses at work, studies and home. Each episode was preceded by tension like headaches and tiredness then the otorrhea starts like sweat from both ear canals. She said the ears got wet with blood-stained fluid ( Fig. 1 ). Bloody stained (cotton wools) from ears ( Fig. 2 ). These episodes were not associated with her menstruations and there was no history of physical abuse, ear picking, operation, trauma, bleeding tendencies, drug abuse (NAIDS), spices intake, or hypertension. No other abnormalities were detected in the rest of the examination and investigations, Radiological (CT Scan, MRIA), Audiological (Tympanometry) and blood tests (FBC, Clotting profile, Immunological screening, Biochemistry) all were normal. No obvious pathology was detected to explain the bloody discharge. Microscopic examination cotton bud swab from the ear canal discharge revealed presence of red blood corpuscle (erythrocytes) and other blood components on biochemistry analysis. Examination under anaesthesia, a normal hyperaemic tympanic membrane ( Fig. 3 ) and posterior exploratory tympanotomy no evidence of any abnormality, the biopsy from the canal skin no evidence of vasculitis. Psychotherapy, beta blockers and supportive therapy helped to reduce the frequencies of episodes and the discharges have stopped spontaneously since 2003 (By author).
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