Ghost Spell or Hematohidrosis

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Hematohidrosis is one such rare condition in which blood is excreted in sweat, tears or any other part of the body with varied underlying etiologies.2 Successful.
CASE REPORT

Ghost Spell or Hematohidrosis

Shahid Raza Khalid1, Shahzad Maqbool2, Nusrat Raza2, Tahir Mukhtar1, Aamer Ikram3 and Shahbaz Qureshi1

ABSTRACT Hematohidrosis is a rare condition of excreting blood in sweat, tears or any other part of the body, with varied underlying etiologies and variable success to different available treatment modalities. We are reporting one such case of an adult female who could secrete blood from eyes and ears simultaneously either due to crying as a result of emotional outburst or even sometimes voluntarily. Considering her origin from tribal culture, she was often thought to be victim of some evil spell. The lady had taken various spiritual treatments from Pirs and various forms of medical treatments from homeopaths, quacks and medical doctors, but of no relief. She was treated for 2 months with non-selective beta blocker (propanolol 10 mg thrice daily) along with psychotherapy. She did improve symptomatically but did not get complete cure. Eventually she was lost to follow-up as is common in female patients of tribal background. To our knowledge, this is the first such reported case from Pakistan. Key words:

Bloody tears. Hematohidrosis. Hematidrosis. Sweat.

INTRODUCTION Secretion of blood through eyes, ears or any other part of the body may be taken as spell-bound especially in an uneducated and superstitious region. However, like many other interesting phenomena which were initially thought to be due to some super-natural origin, it may ultimately turn out to be of purely scientific origin.1 Hematohidrosis is one such rare condition in which blood is excreted in sweat, tears or any other part of the body with varied underlying etiologies.2 Successful management lies in prompt recognition and early diagnosis followed by convincing the patient and relatives that the symptoms have a pure scientific explanation and the condition can be managed medically.3 Once the most difficult part of successfully convincing the patient and relatives has been achieved (particularly in a tribal society), various available modalities of treatment can be tried with variable successes. This case report describes this rare phenomenon in an adult lady of tribal origin.

CASE REPORT A 35-year-old married lady presented with 5 years history of shedding blood in tears and through ears. Complaints used to occur whenever she was emotionally disturbed otherwise she might not have this problem for months. There was no history of

bleeding tendencies, hematuria, petechiae, ecchymoses, menorrhagia or trauma. There was no history of excessive consumption of coloured diets or allergy to food or medicine. On examination, blood drops were seen rolling down her cheeks and below the ear lobes (Figure 1). There was no apparent bleeding site in the external auditory meatus upto the ear drums which were intact on both sides. No conjunctival congestion, red eye or evidence of bleeding site in the eyes was noticed. Fundoscopy of both the eyes was unremarkable. Blood complete picture was within normal limits with platelet count of 270 x 109/L. Bleeding time, prothrombin time, partial thromboplastin time and biochemical investigations including LFTs were within normal limits. Platelet functional studies were normal. Urine microscopy revealed no abnormality. Benzidine test was positive for presence of blood in tears. She was treated for 2 months with non-selective beta blocker (propanolol 10 mg thrice daily) along with psychotherapy. She did get some symptomatic relief but her complaints never got fully cured. As is often the problem in our society, particularly in female patients coming from tribal background, she was eventually lost to follow-up.

Department of Medicine1 / ENT2 / Pathology3, Combined Military Hospital, Quetta. Correspondence: Dr. Shahid Raza Khalid, Department of Medicine, Combined Military Hospital, Quetta Cantt. E-mail: [email protected] Received June 15, 2011; accepted July 10, 2012.

Figure 1: Blood coming out of eyes and ears of the patient.

Journal of the College of Physicians and Surgeons Pakistan 2013, Vol. 23 (4): 293-294

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Shahid Raza Khalid, Shahzad Maqbool, Nusrat Raza, Tahir Mukhtar, Aamer Ikram and Shahbaz Qureshi

DISCUSSION Hematohidrosis is a rare condition in which human beings sweat blood. In this condition almost any part of the body can be the seat of manifestation, and it may occur at several points simultaneously.4 The skin may be perfectly normal in appearance, or the 'bleeding' may be momentarily preceded by slight elevation of the integument. Somewhat allied cases have been described in which the bleeding was preceded by vesicle or bleb formation and also by erythematous areas, sometimes becoming superficially abraded or gangrenous. In this part of the world, it can easily be taken as ghostly activity. Causes of hematidrosis can be divided into clotting factor deficiencies, platelet factor 3 (PF3) deficiency,2 vasculitis, sympathetic over activity3 or idiopathic.5 It is presumed that extravasation of blood takes place into and around the sweat-coils that mixes with sweat which is discharged through sweat-pores.2 The amount of blood discharged is usually small. Concurrent increase in the sweat secretion is also a possibility. Aggravating factors known to-date include emotional stress and excitement. A distinct predisposition is seen in young females. An attack usually appears during some intense emotional excitement and in some instances has been observed to be preceded by neuralgic pain or hyperesthesia of the part. A case report from China showed multiple blood-filled spaces that opened directly into the follicular canals or on to the skin.4 In a case series from USA,6 three out of four patients were females. No cause of blood in tears could be ascertained in this study even after exhaustive investigations. There are two case reports from India; first, a 13-year-old female shedding blood in the tears due to PF3 deficiency, and the other was a 72-year-old male.1,7 Another study from China, which focused on the pathogenesis of hematidrosis, labeled distinctive vasculitis as a cause of hematidrosis.2 Presently, there is no universally accepted treatment for this condition probably due to its varied etiologies; however, successful treatment has been reported for some of the cases. In a case report from China, a girl

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having spontaneous bleeding episodes from intact skin was successfully treated with propanolol.3 Another study focused on the pathology, diagnosis and treatment of a patient with hematidrosis concluded that sympathetic nerve activation might play some role in hematidrosis and beta-blockers might be an effective drug for treatment.8 The 72-year-old male patient from India, responded to psychotherapy alone. The 4 cases reported from USA recovered spontaneously. This case did respond to propanolol given for more than 2 months along with psychotherapy, but could not be completely cured of the symptoms. Long-term results of treatment could not be evaluated as the patient was lost to follow-up. Hematidrosis is a rare condition and treatable provided underlying pathology is identified. However, in our culture, key to successful treatment also includes convincing the patient about the possible treatment and cure of this condition, and ensuring patient's adherence to treatment.

REFERENCES 1.

Mishra KL. Bloody tears and hematohidrosis in a patient of PF3 dysfunction: a case report. Cases J 2009; 15:9029.

2.

Manonukul J, Wisuthsarewong W, Chantorn R, Vongirad A, Omeapinyan P. Hematidrosis: a pathologic process or stigmata. A case report with comprehensive histopathologic and immunoperoxidase studies. Am J Dermatopathol 2008; 30:135-9.

3.

Wang Z, Yu J, Cao L, Zhao X, Zhan S, Wu T, et al. A case report of hematidrosis successfully treated with propanolol. Am J Clin Dermatol 2010; 11:440-3.

4.

Jin LJ, Chen HF, Wu TQ, Shen HS, Li ZY, Zou P, et al. Pathology, diagnosis and treatment of a patient with hematidrosis. Zhonghua Xue Ye Xue Za Zhi 2010; 31:157-60.

5.

Freddrick Z. Sweating Blood. Annacheriyan-ga 2006; 34:14-6.

6.

Ho VH, Wilson MW, Linder JS, Fleming JC, Haik BG. Bloody tears of unknown cause: case series and review of literature. Ophthal Plast Reconstr Surg 2004; 20:442-7.

7.

Jerajani HR, Jaju B, Phiske MM, Lade N. Hematohidrosis: A rare clinical phenomenon. Indian J Dermatol 2009; 54:290-2.

8.

Zhang FK, Zheng YL, Liu JH, Chen HS, Liu HS, Xu MQ, et al. Clinical and laboratory study of a case of hematidrosis. Zhonghua Xue Ye Xue Zhi 2004; 25:147-50.

Journal of the College of Physicians and Surgeons Pakistan 2013, Vol. 23 (4): 293-294