Hematohidrosis, Hemolacria, and “Trichorrhage”: A Systematic Review

Abstract

<b><i>Introduction:</i></b> Hematohidrosis and hemolacria are 2 conditions surrounded in religiousness, mysticism, and supernatural superstitions. While the mechanism is still unclear, these cases have amazed physicians for centuries. <b><i>Methods:</i></b> We performed a systematic review in PubMed from 2000 to mid-2021 accounting for 75 studies from which we included 60 cases in 53 articles which were described. <b><i>Results:</i></b> The median age of apparition was 24 years with the youngest case being 12 and the oldest 81. Some of the diseases were secondary to other causes such as hemangiomas and other neoplasias or epistaxis episodes. Most of the cases have been reported in India and the USA; most of them correspond to hemolacria alone (51.6%). <b><i>Discussion:</i></b> We have stated the basics of the substances involved in the coagulation process that have been described as genetically altered in some patients such as mucins, metalloproteinases, and fibrinogen, as well as propose a mechanism that can explain the signs of this particular entity and approach to its treatment as well as provide the first trichoscopy image of a patient with hemolacria.

Hematohidrosis in 15th Century Renaissance Art and a Review of Modern Literature

“…Genetic mutations related to ECM function and development can affect vessel integrity and formation and can lead to a bleeding phenotype. 5 Cases 37 and 38 are siblings, suggesting potential familial association in patients who experience hematohidrosis. The mother of cases 37 and 38 is reported to have a sibling who passed away due to some unknown bleeding complication, potentially additionally suggestive of familial association in cases of hematohidrosis.…”

“…2 There seems to be increasing literature on hematohidrosis (given the tempo of case reports published) as noted by Duffin, 3 when comparing articles published between 1880 and 1935, and 1952 and 2016. As compared with 76 cases analyzed by Holoubek and Holoubek in 1996 (many of which were from before the 20th century) 4 and 25 cases by Kluger 2 in 2017 and 28 peer-reviewed cases by Duffin 3 in 2017, Carrion-Alvarez et al 5 identified 60 cases; however, this review included cases of trichorrhage (bleeding from hair follicles) and hemolacria. This could be attributed to a true increase in incidence of the condition, or an increase in the recognition or reporting of such cases.…”

A young girl crying out blood: Haemolacria

“…Only 23 paediatric cases of idiopathic haemolacria have been reported in the literature. 6 It may be associated with haematidrosis, epistaxis, haematemesis and bloody otorrhoea. 7 One case of vaginal bleeding was reported in a 9-year-old girl.…”

Pediatric hematohidrosis: case report of a rare clinical entity

“…It has been historically linked to mysticism and spirituality. 1 Its incidence and prevalence are unknown, but a recently published study by Shafique et al 2 found 36 cases reported since 1952, mainly pediatric patients (83%) with a mean age of 11 years and female predominance.…”

“…However, its clinical relevance is not clear yet. 1 This is a diagnosis of exclusion. It is essential to witness the episodes and rule out disorders associated with vascular fragility (i.e., vasculitis and connective tissue disorders), factitious disorders, and chromhidrosis.…”