A curious case of sweating blood.

Hematohidrosis is a very rare condition in which an individual sweats blood. It may occur in an individual who is suffering from extreme levels of stress. Various causative factors have been suggested like component of systemic disease, vicarious menstruation, excessive exertion, psychogenic, and unknown causes. Fear and intense mental contemplation are the most frequent causes. It may also occur in bleeding disorders. We here report a case where bloody sweat was discharged from the forehead, face, and body episodically in a 12-year-old healthy girl with no bleeding disorder or any other underlying cause. All investigations done were within normal limits, except low intelligent quotient and loss of insight. The patient was given atropine sulphate transdermal patch with marked improvement in severity.

What was known?

Hematohidrosis is known to be precipitated by stress, strain, or any sort of exertion, and it may occur in individuals with underlying bleeding disorders.

Introduction

Bloody sweating is called hematohidrosis; true hematohidrosis occur in bleeding disorders.[1] It may occur in individuals suffering from extreme levels of stress. Around the sweat glands, there are multiple blood vessels in a net-like form, which constrict under the pressure of great stress. Then, as the anxiety passes, the blood vessels dilate to the point of rupture and goes into the sweat glands. As the sweat glands produce a lot of sweat, they push the blood to the surface, which comes out as droplets of blood mixed with sweat.[23] Duan et al., reported hematohidrosis associated with primary thrombocytopenic purpura.[4] Migliorini described a case of hematidrosis, which is otorrhea with otoerythrosis.[5] We hereby report a case where bloody sweat discharged from the forehead, face, and body episodically in a healthy young girl who did not have any underlying disorders.

Case Report

A 12-year-old girl visited us with a history of bleeding from the intact skin over the forehead, scalp, cheek, nose, and trunk [Figures 1 and 2] for the last 2 years. The bleeding occurred in episodes, once or twice a day, sometimes more frequently, especially on waking up in the morning. No preceding history of stress or anxiety and no preceding episode of tingling sensation were found. Each episode started with mild watery secretion over the forehead or other body parts, followed immediately with bright-red colored secretion. Each episode lasted for about 10-15 min, and the patient remained perfectly alright during the post-episode period until the next episode. There was no history of bleeding from any other site. No history of ingestion of any anticoagulants, dyes, or other drugs was obtained from her. She did not have any history of major medical or surgical illness in the past. No family member had similar complaints. Menstrual cycles were regular and normal.

Figure 1

Sweat admixed with blood from the right side of the face

Figure 2

Sweat admixed with blood from nose

Her general physical examination and systemic examinations did not reveal any abnormality. The skin over the forehead was normal. There was no local tenderness. Blood or red colored secretion could not be extruded on manipulation. On gross examination, the secretion was bright-red in color, less viscous than blood, and it was not frank blood. On collection of the secretion and examination of its smeared preparation under a microscope, plenty of erythrocytes were observed; the sample was benzidine test positive [Figure 3] and alkaptonuria negative. Her routine hemogram, blood counts, platelet count, bleeding time (2 min), clotting time (3 min 30 sec), active partial thrombin time (25 sec; normal range 24-32 sec), prothrombin time, liver function tests, and renal function tests were within normal limits. Peripheral smear of the secretion showed multiple red blood cells (RBCs) along with numerous gram-positive cocci and bacilli [Figure 4]. Urine and stool examination did not reveal erythrocytes or any abnormalities.

Figure 3

Benzidine test of the secretion, confirming presence of blood

Figure 4

Peripheral smear of the secretion showing RBCs and numerous cocci and bacilli

Psychiatric analysis revealed Intelligent Quotient (I.Q.) between 60 and 70 and a loss of insight. Histopathological examination revealed no abnormalities in blood vessels and intact walls. Hair follicles, sweat glands, and sebaceous glands are normal. Direct immunofluorescence study ruled out vasculitis.

The patient was given atropine transdermal patch over the involved sites and she noticed gradual improvement in both the severity and frequency of the episodes. Gradually, after 1 month, the patient stopped getting similar episodes. We followed the patient for 2 months after stopping the atropine treatment. During this period, she did not have any more episode.

Discussion

Hematohidrosis also known as hematidrosis, hemidrosis, and hematidrosis is a condition in which capillary blood vessels that feed the sweat glands rupture, causing them to exude blood; it occurs under conditions of extreme physical or emotional stress.[1]

Various causative factors have been suggested by Holoubek, like component of systemic disease, vicarious menstruation, excessive exertion, psychogenic, psychogenic purpura, and unknown causes.[2] Acute fear and intense mental contemplation are the most frequent causes, as reported in six cases in a study.[6] In our case, no causative factor was identified.

One hypothesis proposed for etiopathogenesis of hematohidrosis, as suggested by some authors,[7] is that multiple blood vessels present in a net-like form around the sweat gland constrict under pressure of stress. As the anxiety passes out, the blood vessels dilate to the point of rupture. The blood goes into the sweat glands, which push it along with sweat to the surface, presenting as droplets of blood mixed with sweat. Severe mental anxiety activates the sympathetic nervous system to invoke stress-fight reaction to such a degree as to cause hemorrhage of the vessels supplying the sweat glands into the ducts of the sweat glands.[8] Skin histopathological study by Zhang et al., revealed some intradermal bleeding and obstructed capillaries. No abnormality was found in sweat glands, hair follicles, and sebaceous glands. They concluded that pathological basis for hematohidrosis might be distinctive vasculitis,[8] but direct immunofluorescence study did not reveal any abnormality in our case.

Biopsy during symptom free period did not reveal any blood-filled vascular spaces, intradermal bleeding or abnormality in hair follicle, sebaceous, or sweat glands. Benzidine test is an important tool in diagnosis, where hemoglobin in blood reacts with hydrogen peroxide-liberating oxygen, which then reacts with organic reagent producing a green to blue colored compound.[7] Peripheral blood smear confirms that the blood is of human origin in our case.

Treatment of this condition is not so convincing. There is one report of successful use of propranolol.[9] In our case, the patient was treated with atropine transdermal patch, followed by complete remission. We believe that a further insight in the etiopathogenesis may help develop more comprehensive management protocol of this rare and unusual condition.

What is new?

1. In our case, no underlying precipitating factors such as stress, strain, or any bleeding disorder was identified.

2. No evidence of vasculitis.

3. Dramatic response with atropine sulphate transdermal patch.