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Indian Pediatr 2013;50: 703-704 |
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Hematidrosis
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Subham Bhattacharya, Mrinal Kanti Das, Suman Sarkar,
and *Avishek De
From Department of Pediatric Medicine and
*Dermatology, IPGMER & SSKM Hospital, Kolkata, West Bengal, India.
Correspondence to: Dr Subham Bhattacharya, 39
Bhadreswar Station Road(S). Hooghly. 712124. P.O-Saradapally, India.
Email: [email protected]
Received: January 08, 2013;
Initial review: February 05, 2013;
Accepted: March 30, 2013.
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Hematidrosis is an extremely rare
clinical entity characterized by recurrent episodes of skin bleeding
mixed with sweat. We report a case of hematidrosis in a 10-year-old girl
where detailed laboratory and histopathological investigation revealed
no abnormality. The girl was successfully treated with propranolol with
no recurrence of bleeding over a follow-up of 3 months.
Keywords: Child, Hematidrosis , Propranolol.
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Hematidrosis is a extremely rare and
enigmatic disorder characterized by recurrent episodes of
self limited bleeding from skin. Though, classically
hematidrosis means blood in sweat, practically blood is
mixed with sweat like material rather than true sweat in
this condition [1]. Till date only nine cases are reported
in literature. We report a case of hematidrosis diagnosed
and treated successfully.
Case Report
A 12-year-old girl was referred to our
hospital with complaints of recurrent episodes of
spontaneous skin bleeding for last one month. The bleeding
was spontaneous from different sites of body including face,
limb, palm and sole but not from mucous membrane. The
consistency of bleeding was little thinner than blood and
stopped as soon as the site was wiped, leaving behind no
oozing site. It was occurring both day and night but never
after she slept.
During hospitalization she had more than
10 instances of spontaneous intermittent bleeding per day
that was evidenced by almost all of our on duty doctors and
nurses. Her medical records were non-contributory with no
known underlying disease. Her development was normal with
normal tanners staging and menarche not yet started.
Complete blood count, and blood
biochemistry was normal, with normal coagulation screening
tests. Platelet aggregation test and estimation of von-Willebrand
factor revealed no abnormality. Assay of anti-nuclear
antibody and interleukins was done with a view to exclude
underlying autoimmune conditions like vasculitis, but
revealed no abnormality. Microscopic examination of bloody
exudates from face revealed the same components as of
peripheral blood including red blood cells, leukocytes and
platelets mixed with epithelial cells. Skin biopsy performed
from the bleeding area in palm immediately after the
bleeding revealed irregular acanthosis of epidermis with
broadening of rete pigs along with hypergranulosis and
marked hyperkeratosis with edema of superficial dermis,
similar description toin a previous case
[3]. There was no abnormality in sweat
gland or sebaceous gland.
The girl was treated initially with
diazepam without any significant improvement. Finally, based
on a previous case [5], she was treated with propranolol in
a dose of 1 mg/kg/day in two divided dose with a baseline
ECG and monitoring of heart rate. The bleeding episodes
started to reduce within 2 days of therapy and stopped
completely within 6 days. There was no recurrence of
bleeding within three months of follow-up.
Discussion
Hematidrosis is a condition in which
capillary blood vessels that feed the sweat glands rupture,
causing them to exude blood [2].
The term hematofolliculohidrosis has
been proposed because it appears along with sweat-like fluid
and the blood exudes via the follicular canals [3].
Although, historically it was described to occur under
conditions of extreme physical or emotional stress, various
case report have shown that it may occur without stressful
situations, as in our case. Recently, sympathetic nerve
ending activation and vasculitis have been proposed as an
important cause. The etiopahogenesis of this condition was
described as bleeding from the blood vessels that supply the
sweat gland [4]. Common differential diagnosis is
self-injury and chromhidrosis (sweat containing color
pigment). In our case there was no evidence of self-injury
and diagnosis established with demonstration of blood
corpuscles in the secretion along with negative tests for
bleeding diathesis. Vasculitis was also excluded in this
case with normal ANA and interleukin levels and finally
normal skin biopsy.
Till date there is no specific
management. Vitamin C and hemostatic drugs are not
effective. Manolukul, et al. [2] used lorazepam as
anxiolytic in a case and got excellent result. Zhaoyue,
et al. [5]
used propranolol with the hypothesis of sympathetic
overactivity and it was found to be effective. In our case
diazepam did not work, but we got excellent result with
proapranolol. Bleeding stopped completely within one week of
starting treatment and did not recur over next three months
of follow up.
Contributors: SB, MKD, SS, AD were
involved in diagnosis and management of the case. SB drafted
the paper and searched literature. SB, MKD & AD contributed
to paper writing. All author approved the final manuscript.
Funding: None; Competing interests:
None stated.
References
1. Holoubek JE, Holoubek AB. Blood, sweat
and fear. "A classification of hematidrosis". J Med.
1996;27:115-33.
2. Manonukul J, Wisuthsarewong W,
Chantorn R, Vongirad A, Omeapinyan P. Hematidrosis: A
pathologic process or stigmata. A case report with
comprehensive histopatho-logic and immunoperoxidase studies.
Am J Dermato-pathol. 2008;30:135-9.
3. Jerajani HR, Jaju B, Phiske MM, Lade
N. Hematohidrosis - A rare clinical phenomenon. Indian J
Dermatol. 2009;54:290-2.
4. 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.
5. Zhaoyue W, Ziqiang Yu, Jian S, Lijuan C, Xiaojuan Z,
Xia B, et al. A case of hematidrosis successfully
treated with propranolol. Am J Clin Dermatol. 2010;11:440-3.
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