The
exact cause of hematohidrosis is unknown, but it has been associated with
anxiety disorders. Laboratory investigations, including blood workup and
systemic examination, are usually unremarkable. Hematohidrosis can also be
associated with systemic diseases and extreme exertion [7,8]. It
may sometimes be triggered by specific events, such as school examinations or
quarantine periods [9]. Treatment with propranolol has shown
successful outcomes in some cases. Beta-blockers, such as propranolol, are
useful in managing hematohidrosis [10]. However, no definitive
treatment is available for this condition [11]. Psychological
anxiety has been identified as a predisposing factor for hematohidrosis [12].
The condition can be effectively managed if the underlying cause is correctly
identified and addressed. Healthcare professionals need to provide
psychological counseling and reassurance to both the patient and their parents,
as hematohidrosis is a benign condition [13]. Although specific
guidelines for treatment are not well established, hematohidrosis can be
successfully treated with a combination of pharmacotherapy and
non-pharmaceutical interventions. We now present a case of a 12-year-old Indian
girl who presented with recurrent spontaneous bleeding while walking mixed with
sweat from intact skin, diagnosed as hematohidrosis, to understand the
condition better.
CASE
PRESENTATION-
A 12 years old female child resident of Raipur, Chhattisgarh, India, was
presented to the Dept of General Medicine, Shri Balaji Institute of Medical
Science, Raipur, on 25 July 2023 with complaints of spontaneous bleeding from
intact skin of the tendoachilles region right ankle on walking. The bleeding
was noted to be spontaneous and recurring. The patient had a history of trauma
15 days back, but there is no abrasion or discoloration associated with bleeding
or any other bleeding sites such as gums/ oral mucosa/ sub conjunctiva/
epistaxis/ hematoma; scurvy and other bleeding dyscrasias were ruled out.
Bleeding was found to be not associated with menstruation. Also, no family
history of any bleeding tendency was noted. The patient had complaints for 15
days that occurred while walking (Fig. 1). She gives a history of recurrent
bleeding on walking multiple times a day. She advised me to follow in OPD.
.jpg)
.jpg)
A
B
Fig.
1:
(A) Bleeding starts on walking (B) Normal skin when not walking
The patient was alert and no abnormality
was noted on general examination. She was oriented, comprehended, and
communicated well with people nearby. No psychotic symptoms were elicited and
her intelligence was observed to be within normal limits. On detailed
examination, no physical or gynaecological abnormality was noted. The secretion
from the area was collected and processed for benzidine test and microscopic
examination (Fig. 2). The bleeding was cleaned and the area was examined for
trauma or self-inflicted wounds. Absence of injury was noted and the skin over
the tendoachilles region right ankle appeared normal. The absence of systemic
disease and allergy towards drugs or food was noted. Later, in the OPD another
episode of bleeding was observed and was witnessed by three physicians. The
blood secretion was found to be spontaneous for one to two minutes. On
biochemical examinations, the secretion from the tendoachilles region right
ankle was positive for blood components with no other abnormalities. Complete
blood count, bleeding and clotting time, prothrombin time, active partial
thrombin time, liver function tests, and renal function tests are within normal
limits. Ultrasonography of the abdomen and pelvis was found to be normal. The
secretion was confirmed to be blood by benzidine test and microscopic
examination also revealed blood elements in the sample.
As informed by the patient and her family,
symptoms usually appear when she walks, and she is afraid or stressed,
particularly in the morning. Also, in the hospital, while she walks and
expressed fear and stress regarding her academic performance. The patient was
prescribed vitamin C daily and propranolol 10 mg twice daily for ten days and
tapered for 1 month. No recurrence of the bleeding episode was noted until two
months during her academic exams. The possibility of exacerbation associated
with anxiety and stress was noted. Propranolol repeat dose was advised and the
patient did not report any such bleeding complaints for six months.
.jpg)
Fig. 2:
Smear prepared from the oozing blood
DISCUSSION- Hematidrosis
is an extremely rare and enigmatic disorder characterised by recurrent episodes
of spontaneous bleeding from intact skin, which is usually self-limiting. The
etiopathogenesis for the condition, as proposed by a few authors, includes post-systemic
disease, excessive exertion or stress, vicarious menstruation psychogenic and
unknown [2,14-16]. In most of the cases, acute stress and anxiety
were the precipitating factors. In our case, the patient was advised vitamin C
owing to its probable role in sympathetic activity and anxiety. The phenomenon
was found to be isolated and was not found to be associated with general health
conditions.
Duan et
al. [17] reported hematohidrosis associated with primary
thrombocytopenic purpura. Diagnosis was made by exclusion and clinical findings
of peripheral blood exudates of intact skin. In our study, no such abnormality
was found. There is a lack of knowledge about the pathology of the condition.
However, some studies have suggested increased vascular pressure leading to the
passage of blood cells through the duct of sweat glands, vasculitis of dermal
vessels and exacerbated sympathetic activation leading to peri glandular vessel
constriction and subsequent expansion, allowing the passage of blood content
into ducts [15].
Hematohidrosis
should be differentiated from chromatidrosis, meaning coloured sweat due to
coloured apocrine secretion and also from other haemorrhagic disorders,
including platelet abnormality, coagulation or vasculature abnormality, which
usually is found to be associated with petechae, purpura, mucosal bleeding or
hematoma. Differential diagnosis for the disorder must include chromhidrosis,
factitious dermatitis, vicarious menstruation, vasculitis, and platelet and
coagulation disorders. Lab investigations are of utmost importance.
To
date there, no specific treatment for the disease has been identified. However,
Manolukul et al. [18] used
lorazepam as anxiolytic and significant results were obtained. Badry et al. [19] treated their
case of a 16-year-old female patient complaining of recurrent attacks of right-sided
bleeding with propranolol 10 mg twice daily. After 2 weeks of the treatment,
the episodes were found to have decreased. This case and treatment methodology
used to decrease the episode is similar to our case and results [20].
The sympathetic system has a certain role in the recurrent episodes of
hematohidrosis, which is usually common during emotional or physical stress, as
in our case. The patients were found to be relieved, and recurrence of
hematohidrosis decreased on administration of beta blockers, similar to our
study. Bhattacharya et al. [21]
noted diazepam's ineffectiveness and reported effective results with
propranolol, thus recommending propranolol and supporting the treatment used in
our study.
CONTRIBUTION OF AUTHORS
Research
concept- Naresh Verma
Research design- Naresh
Verma
Supervision- Aum Bapat
Materials- Naresh Verma
Data collection- Naresh
Verma
Data analysis and Interpretation- Deepak
Jaiswal
Literature search- Priyanka
Khatri
Writing article- Priyanka
Khatri
Critical review- Dr
Kamala Kant Bhoi
Article editing- Dr
Kamala Kant Bhoi
Final approval- Dr
Kamala Kant Bhoi
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