A Case Report Of Hematohidrosis: A Rare Phenomenon Of Sweating Blood

Background

Hematohidrosis, sometimes also referred to as ‘Hematidrosis’, is an extremely rare clinical condition of sweating blood. Such phenomenon has long been associated with Christian religious belief, when Jesus Christ sweated blood before his crucifixion in the garden of Gethsemane. Instances are also found in the description of a legendary soldier by Leonardo Da Vinci who sweated blood before battle and there are also claims of such phenomena in some people awaiting execution [1]. But only a few well documented case reports are available till now in modern medical literature.

Most of the cases are reported to have blood sweat with excessive exertion or anxiety. Reports are also available with otorrhoea and otoerythrosis [2], with epilepsy [3] and with primary thrombocytopenic purpura [4].

The exact pathogenesis behind hematohidrosis is not yet established. It is proposed that under pressure from sympathetic stimulation due to excessive stress or anxiety the vessels around sweat gland contracts. When the stress passes the vessels dilate to the point of rupture to cause blood being mixed with sweat [5].

We are here reporting a case who presented with episodes of blood mixed sweat from different sites of her body on the top of severe headache with strong family history of such condition.

Case report

A 25-year-old lady presented with episodes of headache which comes with blood mixed sweat for 2 years. Her headache episodes have increased in both frequency and severity in last 2 months to about 3 to 4 episodes a day. She was having severe, dull aching headache with photophobia and phonophobia. Headache usually started in one side, mostly left side of her head and neck and gradually spreaded over whole of the cranium. At this stage, some headache episodes were associated with blood mixed sweat from different sites of her body through intact skin. This blood mixed sweat commonly came from forehead, neck or hand and less commonly from scalp, chest or arms.

She also had a few episodes of epistaxis and bleeding from a scar of previous caesarian section in her abdomen with such headache. The amount of blood was usually only a few drops. She remained drowsy after that for one or two hours. Then the headache usually subsides after the bloody sweat episode. Her menstruation was a bit irregular with occasional menorrhagia. No history of any previous bleeding manifestation was available. No drug history was to mention except occasional NSAIDs for headache. The colour of tear, saliva, stool and urine has always been normal. While exploring her family history, we astonishingly found that 1 of her 3 children, who is a boy of 4 years, was showing the same type of bloody sweat through intact skin for last 1 year. And another family member, who is son of her sister experienced the same.

Nothing significant was found from her general examination and all the systemic examinations. Thorough neurological examination including fundoscopy was also normal. She had some of such episodes of headache with bloody sweat while she was kept for observation in our hospital. The skin over the bleeding sites just after bleeding episodes was absolutely normal. There was no bleeding point or cut mark. Neither was there any tenderness or any other skin abnormality. The blood mixed sweat was bright red and appeared as ‘blood mixed with water or sweat’ in gross examination. It was unclotted when secreted but clotted on the intact skin as it was left there for few minutes.

Everything was found normal after extensive investigations. Complete Blood Count showed mild normocytic normochromic anemia with normal platelet count (Hb: 11.5 gm/dL, Platelet count: 275x109/L). Bleeding Time and Clotting Time were normal (2 minutes and 30 seconds and 5 minutes 30 seconds respectively). Prothrombin Time was 12 seconds (INR 1.01) and Activated Partial Thromboplastin Time was 30 seconds which were also in normal range. Other investigations including serum electrolytes, renal and liver function tests were also normal. Peripheral Smear was prepared from the secretion with Lishmann stain, microscopic examination of which revealed numerous RBCs along with some WBCs and platelets with a few epitheloid cells. ABO blood group was also done taking samples from both venous blood and from bloody sweat. Both were found to be B positive.

Her headache she was treated as migraine. Acute episodes were treated with Paracetamol 1gm or Naproxen 500mg. As some previous reports showed that hematohidrosis episodes partially responded to beta blocker, Propranolol was started in low dose and increased gradually both as migraine prophylaxis and to cover hematohidrosis. With dose of Propranolol 40mg 12hourly, she showed significant improvement of headache. Bloody sweat episodes also declined to 1 episodes in 3 or 4 days.

Discussion

The patient described here is an otherwise healthy female of 25 years who was experiencing severe headache along with bloody sweat for 2 years. Thorough clinical examinations and investigations were normal. There was no clue of any bleeding diathesis or any other systemic disease. All her bloody sweat episodes occurred on the top of severe headache. Some episodes occurred when she was in the hospital in front of attending doctors and nurses. Thus the chance of factitious disorder is excluded.

As an explanation for such phenomenon in previous cases, activation of sympathetic nervous system has been implicated [5]. It was proposed that multiple blood vessels are present in a net-like form around the sweat glands. The vessels initially constricts with activation of sympathetic nervous system. As the sympathetic stimulus passes away the vessels dilate to the point of rupture. The blood goes into the sweat gland which is then pushed along with sweat to the surface presenting as droplets of blood being mixed with sweat. Then the vessels are collapsed leaving no scar on the skin. Extreme physical or emotional stress are most common causes for such activation. In our case severe headache is probably the cause of sympathetic activation. Other causes are ruled out by thorough history taking and examination.

Some tests like Benzidine test and Haemochromogen test could be performed to identify haemoglobin in the sample. In our patient, these tests were not done as presence of numerous RBCs along with all other components of blood were visualized in microscopic examination. ABO and Rh typing was also done taking sample from bloody sweat which matches patient’s blood group. Thus the blood was confirmed to be originated from the patient’s own peripheral blood.

Immediate biopsy would show multiple blood filled spaces that open directly to the follicular canals or to the skin surface. After exudation, the blood vessels collapses and biopsy will not be helpful. Biopsy in our patient was not done as it was refused by the patient.

In our patient, we noticed strong family history of the condition. One of three of her children, and a nephew of her are affected. To our knowledge family history was not present in the previously reported cases.

Treatment options not yet have very convincing evidences. Remission of symptom with Propranolol are commonly reported in patients with prominent features of sympathetic activation [6]. Administration of Benzodiazepines to reduce patient’s anxiety is also found in some cases [5]. Another report showed attenuation of symptoms with transdermal atropine patch [7]. Our patient was treated for her headache as migraine. Propranolol was given both as migraine prophylaxis and as treatment for hematohidrosis. The patient showed significant improvement of both her headache and bloody sweats.

Conclusion

Bloody sweat is an extremely rare symptom which presented with headache in this patient. This poorly understood phenomenon needs more medical data to acquire further knowledge about the pathogenesis and treatment options. As it was with previous reports, our patient also showed partial relieve of symptoms with Propranolol and Paracetamol. The most unique thing we noticed in this patient was a very strong familial affiliation which we believe will assist in further research.