Hematidrosis, hemolacria, and trichotillomania in an adolescent patient

Abstract

Hematidrosis, also known as hematohidrosis or hemidrosis, is a condition in which a person sweats blood from intact skin without hemostatic disorders. Historically, it has captivated artists and clergymen.1 Many reports highlighted its association with stress. We report herein the case of a 13-year-old girl who experienced depression and stressful events that were subsequently associated with recurrent episodes of bloody sweat and trichotillomania. A 13-year-old girl presented with trichodynia of the frontal hairline. She reported psychological distress following the discovery of persistent left superior vena cava and a non-stenotic subaortic diaphragm. A few months later, she developed weekly episodes of bleeding from the scalp (Fig. 1). Each episode began with pain lasting 2–3 minutes, resolving spontaneously without scars. There is no history of trauma, medication intake, or hematological disease. Physical examination, coagulation profile, antinuclear antibody, and anti-neutrophil cytoplasmic antibody tests were unremarkable. Microscopic examination of the red fluid revealed red blood cells, ruling out the diagnosis of chromhidrosis. The diagnosis of hematidrosis was made. The patient received propranolol 20 mg daily. An episode of hemolacria was reported by her ophthalmologist, who treated her with timolol eye drops. Significant improvement was noted after 3 months. During follow-up, she experienced hair loss (Fig. 2a) and painful excoriations on her face and trunk. Physical examination revealed alopecic patches on the scalp with short hair and thinning eyebrows. Trichoscopy showed multiple broken hairs with different lengths, trichorrhexis, trichoptilosis, and tulip hair (Fig. 2b). The diagnosis of trichotillomania was made. She was referred to child and adolescent psychiatry for evaluation. The diagnosis of depressive disorder was made, she was hospitalized, and received fluoxetine with psychological support and close follow-up, with a good response and hair regrowth. No recurrence was noted after 6 months of follow-up. This case is remarkable as it constitutes the first occurrence of hematidrosis, hemolacria, and subsequently, trichotillomania in an adolescent girl with depressive disorder, thus possibly representing a model of complex interactions between skin and psyche. Indeed, anxiety and stress were described as triggers of hematidrosis.1 Hematidrosis is a disorder occurring in any part of the body characterized by the excretion of blood components in sweat from intact skin, including red blood cells, white blood cells, and platelets. Clinically, the bleeding appears pink, clearer, and more fluid than blood. Females are more frequently affected than males, with an average age of onset between 10 and 15 years.2 As in our patient, hemolacria, or bloody epiphora, can be associated with hematidrosis. Differentials include self-inflicted or secondary injuries, chromhidrosis, vasculitis, scurvy, or connective tissue disorders with vascular fragility. The mechanism of hematidrosis remains poorly understood. One hypothesis is that blood “escapes” from capillaries by diapedesis to adnexal structures (sweat glands and ducts or through follicular openings).3 Conventional histology and electron microscopy showed no evidence of vasculitis or ultrastructural abnormalities of the skin appendages. Another possible explanation is related to the overactivation of the sympathetic nervous system triggered by acute stress with initial severe vasoconstriction of the vessels. After stress relief, significant vascular relaxation may cause microruptures in the vessel walls, allowing blood to leak into the dermis and sweat glands and subsequently to the skin's surface, mixing with and carried by sweat.3 Episodes of hematidrosis can be associated with high blood pressure and anxiety.3 Therefore, anxiolytics (by controlling causative stress) and beta-blockers (by inhibiting the sympathetic nervous system) are usually used to treat symptoms and prevent recurrences. Atropine sulfate transdermal patches were also used successfully.4 Tap-water ionophoresis was reported as a successful intervention for hematidrosis of the palms.5 In conclusion, this case highlights the psychosomatic nature of hematidrosis and emphasizes the importance of a multidisciplinary approach involving dermatologists, internists, and psychiatrists. Informed consent was obtained from the patient included in this article. Details of the included cases are described in the manuscript. Additional data will be made available upon reasonable request from the corresponding author.