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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 26  |  Issue : 2  |  Page : 99-102

Hematohidrosis: Reports and update of clinically mysterious phenomenon


1 Department of Otolaryngology-Head and Neck Surgery, Faculty of Medicine, Sohag University, Sohag, Egypt
2 Department of Internal Medicine, Division of Hematology, Faculty of Medicine, Sohag University, Sohag, Egypt
3 Department of Radiology, Faculty of Medicine, Sohag University, Sohag, Egypt

Date of Submission08-Dec-2019
Date of Decision17-Feb-2020
Date of Acceptance26-Feb-2020
Date of Web Publication17-Jul-2020

Correspondence Address:
Dr. Mahmoud I Elbadry
Department of Internal Medicine, Division of Hematology, Faculty of Medicine, Sohag University, Nasr City, Eastern Avenue, University Street, Sohag
Egypt
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/indianjotol.INDIANJOTOL_135_19

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  Abstract 


Hematohidrosis is a mysterious and rare disorder characterized by one or more attacks of spontaneous, bloody sweating from intact surfaces of skin and/or mucous membranes. In the current literature, we faced a case of a 16-year old female who presented with recurrent attacks of right-sided bloody otorrhea upon exposure to extreme stress and anxiety. The patient had no history of bleeding disorders, trauma, or drug intake. Ear examination was completely normal with intact skin and tympanic membrane. All bloody investigations of the patient were within the normal range. The outcome was favorable in this case with medical treatment (beta-blocker and anxiolytics) and psychological support. In this article, we highlight with a detailed description of clinical characteristics and treatment options of this disease, followed by a review of the possible pathophysiological mechanisms. Our case together with previous reports should raise awareness of hematohidrosis occurrence, especially among ENT patients.

Keywords: Bloody otorrhea, hematohidrosis, pathogenesis, stress


How to cite this article:
Badry MS, Elbadry MI, Ragab ARA, Ahmed ME. Hematohidrosis: Reports and update of clinically mysterious phenomenon. Indian J Otol 2020;26:99-102

How to cite this URL:
Badry MS, Elbadry MI, Ragab ARA, Ahmed ME. Hematohidrosis: Reports and update of clinically mysterious phenomenon. Indian J Otol [serial online] 2020 [cited 2020 Aug 12];26:99-102. Available from: http://www.indianjotol.org/text.asp?2020/26/2/99/289945




  Introduction Top


Hematidrosis or hematohidrosis is a rare phenomenon in which blood is excreted with sweat upon exposure to extreme physical or emotional stress. The exact explanation of this condition is not so clear, but activation of the sympathetic nervous system has been suggested.[1] Upon exposure to extreme anxiety, the nets like form of multiple blood vessels that feed the sweat glands undergo constriction and then dilatation to the point of rupture. Then, the blood goes into the sweat glands, which push it along with sweat to the surface, presenting as droplets of blood mixed with sweat.[1] Hematohidrosis is diagnosed when there is bloody discharge without any obvious cause through intact skin, witnessed and confirmed by a doctor and the presence of blood components on biochemical and microscopic studies of the discharge. The updated registry of this disease is important for data collection to achieve a good sample size for prospective epidemiological and clinical studies for understanding the pathophysiology of this rare disease.


  Case Report Compared With Published Cases Top


Here, we report a case of a 16-year-old female patient who presented to our department complaining of recurrent attacks of right-sided bleeding per ear of 1-month duration. She suffered from 3-5 self-limited attacks per week which occur spontaneously and take few minutes. The onset of bleeding in each time was preceded with a history of severe occipital headache which was relieved with initiation of bleeding. She claimed that nearly every attack was precipitated by exposure to surrounding stressful situation. One week before admission, the patient mentioned the occurrence of attack of right-sided epistaxis which has the same criteria mentioned above. The patient also has a history of recurrent attacks of hematemesis for which the patient underwent upper gastrointestinal tract (GIT) endoscopy and revealed the presence of active erosive gastritis and duodenitis. She denied any other orificial bleeding or bleeding from any other skin surface or mucous membrane. There was no history of trauma, drug intake, or any other medical problems. There was no history of other ENT symptoms or family history of similar conditions.

Physical examination revealed normal vital signs and average body built. Ear examination was completely normal with intact skin surface of external auditory canal without evidence of prior trauma or even abrasion and intact tympanic membrane, only a few blood clots. The patient witnessed during the attack three times during her 7-day hospital stay which revealed excretion of bloody sweat [Figure 1]a. Written informed consent by the patient was obtained. The color of other body fluids such as saliva and urine was normal. The results of laboratory tests were normal, regarding platelets count (220 × 1000 ML), activated partial thromboplastin time (32.9 s), prothrombin time (11.7 s), and normal bleeding and coagulation time. Microscopic examination of the excreted fluid revealed red blood cells and other components of peripheral blood. Computed tomography angiogram and audiogram were completely free [Figure 1]b, [Figure 1]c, [Figure 1]d. After psychiatric consultation the patient was diagnosed to have depression for which received medical treatment in the form of Tegretol tablet once daily and propranolol 10 mg twice daily. After 2 weeks of treatment the episodes started to decrease.
Figure 1: Our case report. (a) Bloody secretion of the right ear. Computed tomography angiography showing carotid and vertebrobasilar system showing branches to external ear through external carotid artery (gray solid arrow) and branches to middle/inner ear through internal carotid artery/vertebrobasilar artery (solid black arrows) with no aneurysmal dilatation or arteriovenous fistula. (b) Axial image. (c) Reconstruction image. (d) Three-dimensional reconstruction image

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A comprehensive analysis was conducted on reports documenting hematohidrosis patients. 37 patients met the hematidrosis diagnostic criteria [Supplementary Table 1], the majority were female (31 of 37) [Figure 2]a. Ten (27%) patients were aged above 18 years, and most of the adult patients were aged between 18 and 35 years [Figure 2]b, with only one case over the age of 70 years suffering from mental stress.[2] More than 75% of the patients had bleeding from multiple locations; the most common locations were forehead and ear [Figure 2]c. Bleeding from limbs and forehead were the most common location among males in contrast the most bleeding location among females occurred from eyes and ears [Figure 2]d.{Table 1}
Figure 2: Clinical characteristics of reviewed cases. (a) The bar graph showed the percentage of sex differences among reported cases. (b) The mean age comparison between males and females. (c) Percentage of site of bleeding in all patients. (d) Comparison percentage of site of bleeding between males and females

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Our case together with other report findings indicates that treatment with beta-blockers and antidepressants may be effective for the treatment of hematohidrosis.


  Discussion Top


Hematohidrosis or hematidrosis is a very rare and strange condition, with only a few cases have been reported [Supplementary [Table 1]. Reported patients are usually young girls[3] and have the clinical presentation of oozing bloody fluid from intact skin or mucous upon exposure to stressful and anxious situations.[2] The attacks last for few minutes and resolve almost spontaneously.[4] Investigations required for the diagnosis of bleeding disorders are usually normal, and the examination of the bloody fluid demonstrates the presence of blood elements. It is crucial to examine the skin carefully to exclude self-inflicted or any other accidental injuries and to also differentiate this entity from other conditions which may have similar presentation such as chromhidrosis (color pigment in sweat), vasculitis, or other connective tissue disorders where vascular wall weakness can lead to easy bleeding. Although hematidrosis usually has a great and distressful effect on the patient and his surroundings, its diagnosis is usually delayed due to the rarity of the condition and the presence of multiple differential diagnoses. Frequently, patients will undergo extensive evaluations including hematology consultation before reaching a definitive diagnosis. The exact etiology remains mostly unknown but it has been proposed that stress-induced vasoconstriction (through sympathetic stimulation) that affects blood vessels around the sweat glands leads to vascular rupture with blood extravasation with the sweat.[2] It has been found that when patients treated with anxiolytics and beta-blockers such as propranolol 10 mg twice daily, most of them experienced great improvement, and this is a great proof in the favor of the suggested theory.[5] As in all reported cases, the attack of bleeding is usually preceded with exposure to stressful condition. The stimulus in our case was exposure to family social problems. The association of hematidrosis with recurrent upper GIT bleeding due to erosive gastritis in our case might be related to the current condition of the patient in that both of them may be stress induced, especially that some attacks of bleeding per ear occurred in concurrent with those of hematemesis. The previous finding could support by the study reported that depression may be an independent risk factor for erosive gastritis.[6]

The etiopathogenesis for bleeding in hematohidrosis is still unknown, and a single explanation could not cover the source of bleeding in hematohidrosis [Figure 3]. The first hypothesis could be blood extravasation into the sweat glands which push (through adrenergic stimulation) to the surface and presenting as droplets of bloody fluid.[5] The fact that beta-blockers controlled the bleeding in our patient, and several others reviewed that cases could make presence this hypothesis plausible. Manonukul et al. suggested that defects in the dermis that communicate with the vascular spaces can cause areas filled with blood. These blood-filled spaces opened in follicular canals or directly on the skin surface.[7]
Figure 3: Possible theories of hematohidrosis pathophysiology. Although there is no single explanation of the cause of bleeding in hematohidrosis, this figure stresses the possible abnormal constrictions and expansions roles of periglandular vessels together with the other causes in comparison to normal skin. (1) Blood could pass through eccrine ducts to the surface of the skin after expansions of periglandular vessels or through apocrine ducts to canals of hair follicles or direct in the canals. (2) The areas without sweat glands could be exuding bloody fluid through the presence blood-filled spaces of dermal defects openings into the surface of the skin (3) or directly into the skin surface (4)

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Zhang et al. reported obstructed capillaries and some intradermal bleeding with nonspecific changes in sweat and sebaceous glands or hair follicles. They suggested that vasculitis could be the pathological basis for hematohidrosis.[8] Many studies with detailed clinical characteristics, series of laboratory examinations, and immediate skin biopsy are important more than a late biopsy during remission to determine the skin pathohistologic features and possible changes in the ultramicroscopic structures.


  Conclusion Top


The simultaneous occurrence of hematohidrosis and hematemesis in our case together with the clinicopathological finding of the reviewed cases treated with beta-blockers and psychiatric counseling provide the new insights to the relationship between psychogenic causes and pathological basis of hematohidrosis.

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.

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  References Top

1.
Hansson K, Johansson EK, Albåge M, Ballardini N. Paediatric haematohidrosis: An overview of a rare but clinically distinct condition. Acta Paediatr 2019;108:1023-7.  Back to cited text no. 1
    
2.
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. 2
[PUBMED]  [Full text]  
3.
Shahgholi E. A case series of hematohidrosis: A puzzling medical phenomenon. Turk J Pediatr 2018;60:757-61.  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.
Wang Z, Yu Z, Su J, Cao L, Zhao X, Bai X, et al. A case of hematidrosis successfully treated with propranolol. Am J Clin Dermatol 2010;11:440-3.  Back to cited text no. 5
    
6.
Cho YJ, Jung TH, Jin HM, Kim JH, Kim DY, Kim SJ. Relationship between depression and endoscopic erosive gastritis in men and women aged over 40 years. Korean J Fam Pract 2017;7:681-7.  Back to cited text no. 6
    
7.
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. 7
    
8.
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. 8
    


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