Now that we have captured your attention with our “snappy” title, we should apologize
if the title suggests we are trivializing a potentially serious, and certainly emotive
and controversial topic. Hematidrosis (also called blood sweat), is reported to be
a very rare condition in which a human “sweats blood.” Hematidrosis is purportedly
caused by the rupture of capillaries that feed the sweat glands, causing them to include
blood elements in the secreted sweat. The condition most frequently seems to occur
at times of substantial physical or emotional stress and fear. In the Christian faith,
Jesus is said to have undergone hematidrosis before the crucifixion, during the Agony
in the Garden (New Testament; Luke 22:44). Hematidrosis has also been proposed as
a possible explanation for “stigmata.”
Hematidrosis is a condition that only a portion of medical professionals believe in.
Certainly, as a condition, there is both plausibility and plausible deniability, with
many alternate explanations, potentially including self-inflicted or other injuries
(e.g., Munchausen's syndrome or Munchausen's by proxy). Neither of the authors to
the current commentary, also guest editors to the current issue of the journal, have
ever been personally involved in such a case, although perhaps pertinent, in terms
of “similar” physical etiology, we have seen cases of “skin hemorrhages,” albeit always
associated with hemorrhagic conditions, either congenital or acquired.
Nevertheless, a proposed case of hematidrosis is described in this issue of the journal,[1] and the authors of that case report certainly believe they are describing a case
of hematidrosis. This case report was sent to several hematologists for review ahead
of publication, and skepticism was expressed, having like us, never seen such a case
during their careers. Some of the potential differential diagnoses and concerns expressed
in these reviews are identified in [Table 1]. Despite this healthy skepticism, we decided to permit the publication of this case
in this issue of the journal. The intention here is to permit potential future dialogue
not only on hematidrosis but also on other potential controversies in thrombosis and/or
hemostasis, as the situation may present in the future. Our take on the topic of hematidrosis
follows.
Table 1
Plausible deniability for hematidrosis[a]
|
• Rare; most of us have never seen such a case
|
|
• Unconscious “avoidance” of pathologies for which there is no clear cause or treatment
|
|
• Most patients are under stress (therefore high probability of self-inflicted wounds)
|
|
• Munchausen's syndrome or Munchausen's by proxy
|
|
• Most reports are from older literature, in foreign language, in nonhematology journals,
and there is a lack of publications in high-quality journals (limited “trust” factor
for modern day hematology workers)
|
|
• Self-inflicted injuries/ecchymoses never occur at sites not reachable by the hands,
such as in between the scapula
|
|
• Most reported cases seem to be from tropical areas–significance?
|
|
• Insufficient scientific proof that hematidrosis is an entity
|
a Including alternate explanations offered by reviewers of the case study.
A search of the literature uncovers only sporadic publications on the topic, with
the search term “hematidrosis” or “haematidrosis” in PubMed uncovering a total of
only 22 reports, with an additional report on “hematidrosis” in Russian. Of the total
23 reports, 10 were in a non-English language source, and another 3 appeared in English
within Indian journals (i.e., tallying more than half of the total). The majority
of the publications appear in dermatological or pediatric journals, with one article
published in a psychological journal, and only a single report published in a hematological
journal. Among some recent articles were a case published in 2017 involving a 10-year-old
girl with mixed anxiety and depressive disorder,[2] another case of a young girl with epilepsy who was successfully treated using an
antiepileptic drug (i.e., oxcarbazepine) published in 2015,[3] and a case published in 2013 in an American Society of Hematology (ASH) Image Bank
article.[4] No other publication in the hematology literature. Not many publications in English
geography journals. Apart from two short (single page) publications in Blood[4] and the British Medical Journal (BMJ),[5] no publication in a high-impact journal is available. Of further interest, the BMJ
report (in a newborn) was published exactly 100 years ago![5] No surprise, then, that hematologists, particularly in developed English geography
locations, may not believe hematidrosis to be real.
Why is hematidrosis plausible? As mentioned, hematidrosis is reportedly caused by
the rupture of capillaries that feed the sweat glands, particularly under periods
of stress, causing them to include blood elements in the secreted sweat. This is perhaps
why most hematidrosis cases are published in dermatology journals. We are not dermatologists,
nor experts in sweat gland physiology. However, that capillaries feed the sweat glands,
and may rupture under periods of stress, certainly sounds plausible to us. For the
case published in Blood: “Physical examination showed no signs of self-inflicted lesions.
All laboratory, image, and coagulation tests, including platelet aggregation, were
normal. More than 30 bleeding episodes were witnessed during hospitalization and samples
of the fluid contained all normal blood cells.”[4] That case was successfully managed using propranolol. For another reported case
of a 12-year-old girl: “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.”[6] Again, laboratory testing failed to identify any hemostatic or other defect, and
the case was again successfully managed using propranolol.
Importantly, hematidrosis is not the only kind of “blood leakage” phenomenon. For
example, hemolacria is a condition in which tears are partially composed of blood,
and otorrhagia represents bleeding from the ear. A PubMed search of “hemolacria” and
“haemolacria” identified 17 articles, unsurprisingly with most in ophthalmology journals.
More interestingly, a PubMed search of “otorrhagia” uncovered 69 publications, and
we suspect that otorrhagia is more common than either “hematidrosis” or “hemolacria,”
and thus more plausibly acceptable to workers in hematology.
Why is hematidrosis plausibly deniable? Any rare condition, should it truly exist,
will rarely be seen by workers in the field. Neither of the authors of this commentary
nor the reviewers to the case study have ever personally seen a typical case of hematidrosis.
Should we then send the case for review by someone who has seen such a case? Naturally,
given the proposed rarity, that would be difficult; however, we would suspect in this
situation that the reviews would be more favorable at least. The literature on hematidrosis
is scarce, not generally published in the English literature and in high impact journals.
Working in developed countries, both authors have personally not seen many of the
more established rare bleeding disorders, and yet, we believe they exist. Nevertheless,
with hematidrosis, there are also plausible alternative diagnoses or explanations
([Table 1]). So, we can personally never be sure of the truth or otherwise of such a diagnosis
in any given individual.
We hope that this commentary, and the accompanying case report, encourage further
dialogue, not only in regards to hematidrosis but also as related to other potential
controversies in thrombosis and/or hemostasis, as the situation may present in the
future.
