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| CASE REPORT |
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| Year : 2023 | Volume
: 3
| Issue : 2 | Page : 90-92 |
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School Examination Stress Precipitating Hematohidrosis in a Girl
Anupama Vikram1, J Christy Maria Viagulum1, Poovazhagi Varadarajan2
1 Department of Paediatrics, Government Medical College and Hospital, Thiruvallur, Tamil Nadu, India
2 Department of Pediatric Intensive Care, Institute of Child Health and Hospital for Children, Madras Medical College, Chennai, Tamil Nadu, India
| Date of Submission | 14-Dec-2022 |
| Date of Decision | 09-Apr-2023 |
| Date of Acceptance | 11-Apr-2023 |
| Date of Web Publication | 24-May-2023 |
Correspondence Address:
Dr. Anupama Vikram
Flat No. 5, Kanakadhara, 20, Temple Avenue, Srinagar Colony, Saidapet, Chennai - 600 015, Tamil Nadu
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/ipcares.ipcares_287_22
Background: Hematohidrosis is a rare clinical disorder characterized by transient, self-limited bleeding from the eccrine sweat glands through intact skin in the absence of an underlying bleeding disorder. Clinical Description: We report a 10-year-old girl who presented with a history of bleeding from the dorsum of her right hand. The child has had recurrent episodes in the past. Many of the episodes were temporally related to her school examination. She did not have any other bleeding manifestations or susceptibility to bruising. Her general condition and systemic examination were unremarkable, and the skin underlying the bleeding site was normal and healthy. All investigations related to bleeding and coagulation parameters were within normal limits. Skin biopsy showed normal histology. Having ruled out all organic causes of bleeding, the typical history of transient episodes of bloody secretions from the skin, triggered by examination stress and normal skin histology, was consistent with a diagnosis of hematohidrosis. Management: The child responded dramatically to oral propranolol with a subsequent reduction in the frequency of the bloody sweating episodes, even during stressful times. Psychological counseling was also provided. Conclusion: In a child presenting with oozing of blood from the skin, a possibility of hematohidrosis may be considered, when all causes of bleeding diathesis have been ruled out by a thorough workup. Pediatricians need to counsel both parents and the child regarding the benign nature of the condition. Keywords: Bloody sweat, hematohidrosis, propranolol, stress
How to cite this article:
Vikram A, Maria Viagulum J C, Varadarajan P. School Examination Stress Precipitating Hematohidrosis in a Girl. Indian Pediatr Case Rep 2023;3:90-2 |
How to cite this URL:
Vikram A, Maria Viagulum J C, Varadarajan P. School Examination Stress Precipitating Hematohidrosis in a Girl. Indian Pediatr Case Rep [serial online] 2023 [cited 2023 Jun 3];3:90-2. Available from: http://www.ipcares.org/text.asp?2023/3/2/90/377517 |
Hematohidrosis, an intriguing and extremely rare clinical condition, is characterized by bleeding from the eccrine sweat glands of the nontraumatized skin, in the absence of any known underlying cause of bleeding.[1] Although the phenomenon has been reported over many years, the precise etiology, pathogenesis, and treatment are still unclear. We report a child who had transient episodes of bloody sweating, precipitated by stress.
| Clinical Description | |  |
A 10-year-old girl, firstborn, out of nonconsanguineous marriage, presented with a history of bleeding from the dorsum of her right hand. The child had mild epistaxis 1 month back from the right nostril and was not associated with cough, runny nose, headache, nose-picking, foreign body insertion, or allergic rhinitis. The child underwent blood sampling for the same from the dorsum of the right hand, following which parents noticed blood-tinged oozing from the dorsum of her right hand, which they perceived to be from the venipuncture site.
In addition, the girl complained of intermittent bleeding from her right hand while writing as well as difficulty in moving her right hand. There was no bleeding from any other sites in the form of petechiae, purpura, gum bleeds, hematuria, hematemesis, or melena. There was neither any history of easy bruisability or bleeding tendencies in the past, nor any history of jaundice, bone and joint pain, rashes, and prolonged fever, suggestive of hepatitis/connective tissue disorders/malignancies/Vitamin C deficiency. The family history was also not significant. The child was hospitalized for the bleeding episode due to parental anxiety and for further evaluation.
On examination, the general condition of the child was stable, with a pulse rate of 82/min, respiratory rate of 24/min, and blood pressure of 110/70 mmHg. Her built and nutrition was normal (weight - 32 kg and height - 132 cm). There was no pallor, icterus, clubbing, or lymphadenopathy. Local examination of the right hand showed bloody secretions over the dorsal surface, which on wiping off, showed an unremarkable underlying skin. The skin was intact with no evidence of trauma, purpura, scratch marks, induration, or tenderness [Figure 1]a and [Figure 1]b. Systemic examination did not reveal any organomegaly, joint swelling, bony tenderness, or oral bleeding/ulceration. The child was hospitalized for observation. | Figure 1: (a and b) shows bleeding from dorsum of right hand with intact underlying skin
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| Management and Outcome | | |
During hospitalization, the child developed another transient episode (lasting around 5 min) of bleeding from the dorsum of the right hand. The blood-tinged secretions examined under a microscope revealed plenty of red blood corpuscles (RBC) and few leukocytes (RBC - 20 hpf, white blood cell - 10 hpf). The benzidine test was positive as shown in [Figure 2]. Hemochromogen could not be done due to nonavailability. Biochemical examination of the fluid showed components of blood and sweat (pH - 5.5, Na - 28 mmol/L, K - 6.2 mmol/L [increased due to lysed RBC], Cl - 30 mmol/L). The bleeding stopped spontaneously. | Figure 2: (a) MRI of the right hand - skin, soft tissues, and muscles - normal, (b) no abnormal soft tissue STIR hyperintensities/flow voids noted. MRI: Magnetic resonance imaging, STIR-Short Tau Inversion Recovery
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Complete blood count, plasma coagulation tests, bleeding time, clotting time, factor assay, thromboelastography, platelet count with morphology, and platelet aggregation studies were all within normal limits [Table 1]. Her blood urea and creatinine levels were 31 and 0.6, respectively; serum bilirubin - 0.7 mg/dl, aspartate transaminase - 32 U/L, alanine transaminase - 25 U/L, and antinuclear antibodies - negative (1:20 dilution). A punch biopsy from the skin revealed normal histology. Computerized tomography of the paranasal sinus (for epistaxis) showed deviated nasal septum; magnetic resonance imaging of the right hand [Figure 2]a and [Figure 2]b, Doppler of the right hand [Figure 3], and ultrasound of the abdomen, were also normal, ruling out vasculitis. The child was discharged. However, she had similar bleeding episodes once in 2–3 weeks. Parents were recommended to get a psychiatric evaluation for the same. As per the psychiatrist, the girl was largely reticent throughout the consultation, communicating only when asked. Her scholastic performance was good; however, examination and writing assessment revealed a sense of fear in the child. Stress response was analyzed by the child/adolescent stress questionnaire, which revealed that the child was definitely stressed. A score of 0–13 is considered as low stress. The questionnaire revealed that the child was stressed with a score of 22. After ruling out all organic causes of bleeding, a diagnosis of hematohidrosis was made, and the child was started on propranolol at 1 mg/kg/day in two divided doses. The girl continued the treatment for 6 months. The girl responded dramatically to beta-blockers with a significant reduction in the frequency of spontaneous episodes of bloody sweating (to two episodes in 6 months). Propranolol was tapered and stopped. However, 4 months later, the child experienced a similar episode, just a week before her school examinations. She was again started on oral propranolol and antianxiety medication, and the bleeding episode stopped. The child was followed up for 2 years.
| Discussion | | |
The presentation of the child with recurrent episodes of bloody secretions, exacerbated by stress, with completely unremarkable underlying skin and normal bleeding and clotting parameters, typically fits into the diagnosis of hematohidrosis.
Hematohidrosis, variously named hematidrosis, hemidrosis, and hematidrosis, is an extremely rare condition, in which there are self-limiting episodes of spontaneous discharge of bloody secretion through intact skin or sweat gland orifices.[1] The earliest description of this condition is in the Bible, where the sweat of Jesus Christ turned into drops of blood, while he was earnestly praying the day before crucification. The exact prevalence and incidence of this condition are still unknown.
The latest systematic review by Carion-Alvarez et al. retrieved 20 articles on hematohidrosis and nine articles reporting both hematohidrosis and hemolacria (bleeding through tears).[2] The age of presentation ranged from 6 years to 72 years, with the median age being 24 years with a majority (65%) of the cases being females. Most of the cases were from India and USA. The most common sites of bleeding were the eyes (hemolacria), followed by the face and ears. Other rare sites included feet, eyelids, and axillae. In our case, bloody sweat appeared over the dorsum of the right hand, intermittently associated with writing.
The pathology in the condition is the rupture of the capillary blood vessels supplying the sweat glands.[3] The exact cause being unknown, some theories have been proposed, including: (a) increased vascular pressure resulting in the passage of blood cells into the ducts of the sweat glands; (b) activation of the sympathetic nervous system due to extreme stress, fear, or anxiety,[3],[4] leading to periglandular vessel constriction followed by dilatation, and passage of blood content into the ducts; (c) vasculitis of dermal vessels;[5] and (d) stromal weakness due to some defects in the dermis.[6]
Similar to our case, some authors have done skin biopsies immediately after bleeding. The histological findings have been largely inconsistent and nonspecific, like periglandular congested vessels, red blood cells in the follicular lumen and in between the collagen fibers, leakage of blood around the dermal capillaries, or even normal skin, just like in our case.
Diagnosis of hematohidrosis can be made only if the following criteria are met: (i) recurrent, spontaneous, painless, and self-limited oozing of bloody discharge, witnessed and confirmed by health professionals, (ii) the usual blood components are found on biochemistry studies of the discharge, and (iii) the site of bleeding is intact with no abrasion, telangiectasia, or purpura, and after wiping the area, there is no evidence of oozing. All of these criteria must be met to rule out organic bleeding disorders, self-inflicted bleeding, factitious disorder by proxy, and chromhidrosis (colored sweat).[7]
The pathogenesis of this condition being unclear, the specific treatment is also largely unknown. Authors have tried Vitamin C to restore collagen integrity, lorazepam for abating anxiety, or propranolol.[8] In our case, the girl's bleeding episodes subsided whenever she was started on beta-blockers (propranolol), highlighting the fact that beta-blockers reduce stress and anxiety, which in turn lowers the frequency of spontaneous bleeding. It also emphasizes the need for proper stress reduction through psychiatric counseling.
To conclude, hematohidrosis is a benign, self-limited condition, but psychologically alarming for parents and patients. Physicians rarely encounter this condition, and thus, this report may reiterate awareness of the existence of such a medical condition.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that names and initials will not be published, and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Bhattacharya S, Das MK, Sarkar S, et al. Hematidrosis. Indian Pediatr 2013;50:703-4.  |
| 2. | Carrion-Alvarez D, Trejo-Castro AI, Salas-Garza M, et al. Hematohidrosis, hemolacria, and “Trichorrhage”: A systematic review. Skin Appendage Disord 2022;8:179-85. |
| 3. | Biswas S, Surana T, De A, et al. A curious case of sweating blood. Indian J Dermatol 2013;58:478-80. [ PUBMED] [Full text] |
| 4. | Pari T. Hematohidrosis – A rare case. Indian Dermatol Online J 2019;10:334-5. [ PUBMED] [Full text] |
| 5. | Uber M, Robl R, Abagge KT, et al. Hematohidrosis: Insights in the pathophysiology. Int J Dermatol 2015;54:e542-3. |
| 6. | Manonukul J, Wisuthsarewong W, Chantorn R, et al. Hematidrosis: A pathologic process or stigmata. A case report with comprehensive histopathologic and immunoperoxidase studies. Am J Dermatopathol 2008;30:135-9. |
| 7. | Shahgholi E. A case series of hematohidrosis: A puzzling medical phenomenon. Turk J Pediatr 2018;60:757-61. |
| 8. | Wang Z, Yu Z, Su J, et al. A case of hematidrosis successfully treated with propranolol. Am J Clin Dermatol 2010;11:440-3. |
[Figure 1], [Figure 2], [Figure 3]
[Table 1]
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