s
Home About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Subscribe Contacts Login 
Home Print this page Email this page Small font size Default font size Increase font size Users Online: 340

 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 18  |  Issue : 4  |  Page : 327-329

Hematohidrosis: A rare case of a female child who sweat blood


Department of DVL, Gandhi Medical College, Hyderabad, Telangana, India

Date of Web Publication29-Sep-2017

Correspondence Address:
Geeta Kiran Arakkal
H-No: 2, Vayupuri, Sainikpuri, Secunderabad, Telangana - 500 094
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2319-7250.193031

Rights and Permissions
  Abstract 


Hematohidrosis is a rare clinical entity characterized by excretion of blood in the sweat. Hematohidrosis presents as unpredictable, recurrent episodes of spontaneous and self-limited bloody discharge from the intact skin. Diagnosis of hematohidrosis is confirmed by testing the secretions for the presence of blood components. It is an extremely rare condition, with very few case reports in literature. Herein, we describe a case of hematohidrosis in an 11-year-old female child with pain and tenderness who responded to propranolol.

Keywords: Hematohidrosis, propranolol, tenderness


How to cite this article:
Arakkal GK, Poojari S, Netha G N, Kumar B U. Hematohidrosis: A rare case of a female child who sweat blood. Indian J Paediatr Dermatol 2017;18:327-9

How to cite this URL:
Arakkal GK, Poojari S, Netha G N, Kumar B U. Hematohidrosis: A rare case of a female child who sweat blood. Indian J Paediatr Dermatol [serial online] 2017 [cited 2018 Jun 23];18:327-9. Available from: http://www.ijpd.in/text.asp?2017/18/4/327/193031




  Introduction Top


Hematohidrosis is the name given to the clinical phenomenon in which an individual sweats blood.[1] It is a rare condition of excreting blood in the sweat and is attributed to varied etiological factors such as component of systemic disorders, vicarious menstruation, excessive exertion, and psychogenic and idiopathic causes.[2] The term “hematofolliculohidrosis” was proposed because blood appeared along with sweat-like fluid and exuded through the follicular canals.[3]


  Case Report Top


An 11-year-old female child presented to the Department of DVL, Gandhi Hospital, with a history of episodic bleeding from the intact skin over her left side of the face, especially left cheek and temple area for 5 months. The bleeding occurred in episodes, most frequently in the morning after waking up and after extreme physical exertion such as playing. Each episode is preceded with an aura of pain and tingling sensation over the left side of the face, followed by bleeding that lasts for 2–3 min and ceases spontaneously, but the pricking pain over the left side of face persists for few hours. The patient does not give a history of ingestion of anticoagulants and other drugs, topical application of medication, or exposure to dyes. There is no history of any chronic medical condition or similar history in the family. The patient has not attained menarche.

The child was active and no abnormality was detected on general examination. On palpation, tenderness was present on the left side of the face and blood could not be extruded on pressure. Systemic examination was normal. We witnessed one episode of bleeding [Figure 1] and [Figure 2] which occurred early in the morning. On examination, the secretion was bright red, less viscous than blood. There was no sign of injury and the skin over the face appeared normal. The secretion was immediately collected for examination and later mopped after which bleeding did not occur.
Figure 1: Clinical photograph showing hematohidrosis from the left cheek

Click here to view
Figure 2: Clinical photograph showing hematohidrosis from the left temple and cheek

Click here to view


Microscopic examination of the secretions revealed plenty of red blood cells [Figure 3] and [Figure 4]. Bleeding stopped within few minutes, and thereafter, examination revealed no breach in the continuity of the skin and no other abnormalities, except tenderness over left side of the face. Hemogram, bleeding and clotting time, prothrombin time, active partial thrombin time, liver and renal function tests, and computed tomography scan of the head were normal. The child was managed with 0.5 mg/kg body weight of propranolol (10 mg) for the past 2 months, with no further episodes of hematohidrosis.
Figure 3: Photomicrograph of smear of the secretions showing numerous red blood cells (×10, Leishman)

Click here to view
Figure 4: Photomicrograph of smear of the secretions showing numerous red blood cells (×40, Leishman)

Click here to view



  Discussion Top


Hematohidrosis is an enigmatic disorder characterized by recurrent episodes of self-limited bleeding from intact skin.[4] It is also known as hematidrosis and hemidrosis. It is a well-recognized diagnosis according to the International Classification of Diseases (ICD9-CM 705-89). It is an extremely rare condition, in which capillary blood vessels that feed the sweat glands rupture causing them to exude blood. It can occur at any part of the body and at several points simultaneously and the amount of bleeding is usually small. Most common site is face with some reports of bleeding from eyes and ears also leading to bloody tears and blood otorrhea. Other sites include trunk, limbs, and rarely palms, soles, and mucosa. Exact etiology is not known though various factors are attributed from bleeding disorders to psychogenic and unknown causes.[2] Hysterical mechanisms and psychosomatic disorders are also believed to induce bleeding.[3] Acute physical or emotional stress is the most common cause. In our case, extreme physical exertion was a trigger for bleeding.

Etiopathogenesis of hematohidrosis has been explored in some of the previous studies with no clear conclusive evidence. It has been proposed by Dr. Frederick Zugibe that multiple bloods vessels present in a net-like form around the sweat glands constrict under the pressure of stress and as the anxiety passes out vessels dilate to point of rupture and blood goes into glands and pushed to the surface.[5] Severe anxiety activates sympathetic system to invoke the stress-fight or flight reaction to such a degree as to cause hemorrhage of the vessels supplying sweat glands into their ducts.[6] In our case, the persistent pain and tenderness for few hours after each episode of bleeding may have been due to the underlying vasoconstriction.

Manonukul et al. have proposed that there may be some dermal defects that communicate with dermal vascular spaces and eventually dilate as blood comes in and later exude this blood through follicular canals to collapse without leaving any scar. This phenomenon acts like a balloon, waxing, and waning, thus causing intermittent and self-limited episodes of bleeding. Immediate biopsy soon after an episode of bleeding may reveal the dermal defects as blood filled spaces.[3] A study by Zhang et al.[6] revealed some intradermal bleeding and obstructed capillaries with no abnormality in sweat glands, hair follicles, and sebaceous glands. They concluded that a distinctive vasculitis might be the pathological basis for hematohidrosis.[7]

Diagnosis of hematohidrosis is made on the presence of bloody discharge without any obvious cause, through intact skin, witnessed and confirmed by health professional and presence of blood components on testing the bloody discharge.[8] This condition is usually self-limiting in nature with a good prognosis.[9] Currently, there is no convincing specific therapy available for this rare condition though there are reports of good response to various drugs such as anxiolytics, especially in cases triggered by extreme stress.[3] Various authors have reported good response to propranolol given in a dose of 1 mg/kg/day in two divided doses.[4],[7],[9] Successful use of beta-blockers supports the role of sympathetic nerve activity in the pathogenesis of this disease. Recently, Biswas et al. have reported a case successfully treated with atropine transdermal patch.[10]

We report this case for its rarity and the presence of localized pain and tenderness after each episode of bleeding which has not been documented so far in literature and also for its clinical response to propranolol.

Declaration of Patient Consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Freddrick Z. Hematidrosis. Available from: http://en.wikipedia.org/wiki/hematidrosis. [Last accessed on 2016 Aug 16].  Back to cited text no. 1
    
2.
Holoubek JE, Holoubek AB. Blood, sweat and fear. “A classification of hematidrosis”. J Med 1996;27:115-33.  Back to cited text no. 2
    
3.
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.  Back to cited text no. 3
    
4.
Bhattacharya S, Das MK, Sarkar S, De A. Hematidrosis. Indian Pediatr 2013;50:703-4.  Back to cited text no. 4
    
5.
Jerajani HR, Jaju B, Phiske MM, Lade N. Hematohidrosis – A rare clinical phenomenon. Indian J Dermatol 2009;54:290-2.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Zhang FK, Zheng YL, Liu JH, Chen HS, Liu SH, Xu MQ, et al. Clinical and laboratory study of a case of hematidrosis. Zhonghua Xue Ye Xue Za Zhi 2004;25:147-50.  Back to cited text no. 6
    
7.
Wang Z, Yu Z, Su J, Cao L, Zhao X, Bai X, et al. Acase of hematidrosis successfully treated with propranolol. Am J Clin Dermatol 2010;11:440-3.  Back to cited text no. 7
    
8.
Tshifularo M. Blood otorrhea: Blood stained sweaty ear discharges: Hematohidrosis; four case series (2001-2013). Am J Otolaryngol 2014;35:271-3.  Back to cited text no. 8
    
9.
Patel RM, Mahajan S. Hematohidrosis: A rare clinical entity. Indian Dermatol Online J 2010;1:30-2.  Back to cited text no. 9
[PUBMED]  [Full text]  
10.
Biswas S, Surana T, De A, Nag F. A curious case of sweating blood. Indian J Dermatol 2013;58:478-80.  Back to cited text no. 10
[PUBMED]  [Full text]  


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
Abstract
Introduction
Case Report
Discussion
References
Article Figures

 Article Access Statistics
    Viewed994    
    Printed28    
    Emailed0    
    PDF Downloaded65    
    Comments [Add]    

Recommend this journal