LAUSR

Hematidrosis is a rare disease in which bloody sweat-like fluids exude through the skin. Although its etiopathogenesis is poorly understood, it is considered to be caused by emotional or physical stress. It is more common in people from Asia (especially India and Pakistan), and there is a previous report of a Japanese case. We report a case of hematidrosis in a Japanese girl with a background of dissociative disorders, who was treated with oral propranolol and psychotherapy. The treatment was approved by the ethics committee of Saitama Children’s Medical Hospital and consent for publication of the report was obtained from the patient’s parents. An 11-year-old girl presented to our hospital with a 2-year history of recurrent episodes (a few times per week) of bloody sweat-like fluid secretion, accompanied by pain in the forehead, hands, feet, and scalp (Fig. 1a-c). She had a history of dissociative disorders from the age of 7 years and had begun self-harming at the age of 9. Hematology findings showed no abnormalities in blood counts or clotting function. Self-injury as a cause of hematidrosis was excluded because the bleeding was repeatedly witnessed by our medical staff, and no wounds were found. The bloody sweat contained red blood cells and other normal blood components. Immediately after the hemorrhage, a skin biopsy was performed but no pathological abnormalities were noted, except for a slightly prominent blood vessel adjacent to the sweat gland (Fig. 1d). Repeated bleeding occurred subsequently, with no evidence of trauma at the bleeding site, resulting in a diagnosis of hematidrosis. Oral propranolol was commenced because the drug is considered effective for hematidrosis. No immediate effects were observed after propranolol administration; thus, psychotherapy was initiated because of the possible involvement of dissociative disorders in hematidrosis. The frequency of bleeding decreased remarkably and disappeared within 2 years. Propranolol treatment was, thus, terminated, while psychotherapy was continued. Although there are no established diagnostic criteria for hematidrosis, in our case, the diagnosis was confirmed by the presence of red blood cells in the bloody secretions, witnessing of the bleeding by our medical staff, and the absence of self-harm. In recent reports of hematidrosis, 64% of cases were diagnosed by direct confirmation of bleeding by a healthcare provider. We conducted follow-up hospitalization, during which we found no evidence of self-harm, and could confirm directly that the bleeding was caused by hematidrosis. The diagnosis was delayed because self-injury had been suspected for over 2 years due to the patient’s history of psychiatric disorder. It is common to misdiagnose hematidrosis as self-mutilation because it is often triggered by severe stress or psychiatric disorders. Early diagnosis may enable rapid treatment and intervention for underlying diseases and stress, making it important to understand this disease and its clinical features. The cause of hematidrosis is unknown, although some reports suggest that blood vessel rupture around sweat glands causes bleeding into sweat, while others reported the presence of microvessels adjacent to sweat glands in skin biopsies from the bleeding site and aggregation of red cells at follicular openings. In the present case, while skin biopsies obtained immediately after the bleeding showed no obvious abnormalities, microvessels were identified adjacent to the sweat glands, confirming previous reports. Although there is no established treatment for hematidrosis, many patients improve spontaneously without treatment or with the use of beta-blockers (e.g. propranolol) or psychotropic agents (e.g. lorazepam). A summary of recent reports showed that eight of nine cases improved after treatment with beta-blockers. In most cases, the symptoms disappeared within a few days to 2 months after treatment onset. In contrast, we observed a slight improvement with propranolol; however, it took time for the symptoms to disappear. Therefore, we conclude that patients with underlying psychiatric disorders may not improve with propranolol alone. Hematidrosis is an extremely rare disease and often occurs in the settings of psychiatric disorders and severe stress. It is important to be familiar with the disease and its clinical features for early diagnosis and treatment. Correspondence: Ryo Matsuoka, MD, Department of Pediatrics, Jikei University School of Medicine 3-25-8, Nishi-Shimbashi, Minato-ku, Tokyo 105-8461, Japan. Email: [email protected] Received 21 January 2020; revised 29 February 2020; accepted 4 March 2020. doi: 10.1111/ped.14223