Systematic Review and Meta-Analysis

Skin Appendage Disord 2022;8:179–185 Received: August 26, 2021

Accepted: October 16, 2021
DOI: 10.1159/000520648 Published online: January 3, 2022

Hematohidrosis, Hemolacria, and

“Trichorrhage”: A Systematic Review
Diego Carrion-Alvarez a, b Alejandro I. Trejo-Castro c Mauricio Salas-Garza d
     

Oscar Raul Fajardo-Ramirez d Julio Cesar Salas-Alanis e

   

aDystrophic
Epidermolysis Bullosa Research Association, Guadalupe, Mexico; bBasic Science Department, School
of Health Sciences, Universidad de Monterrey, San Pedro Garza García, Mexico; cSchool of Engineering and
Sciences, Tecnológico de Monterrey, Monterrey, Mexico; dSchool of Medicine and Health Sciences, Tecnológico de
Monterrey, Monterrey, Mexico; eInstituto Dermatológico de Jalisco, Guadalajara, Mexico

Keywords this particular entity and approach to its treatment as well as

Hematohidrosis · Hemolacria · Trichorrhage provide the first trichoscopy image of a patient with hemo-
lacria. © 2022 S. Karger AG, Basel

Abstract
Introduction: Hematohidrosis and hemolacria are 2 condi-
tions surrounded in religiousness, mysticism, and supernat- Introduction
ural superstitions. While the mechanism is still unclear, these
cases have amazed physicians for centuries. Methods: We Hematohidrosis is defined as the sweeting of blood,
performed a systematic review in PubMed from 2000 to mid- while hemolacria is stated as the crying of blood with-
2021 accounting for 75 studies from which we included 60 out any clinical explanation such as a surgery. Both
cases in 53 articles which were described. Results: The me- clinical entities are strongly linked to mysticism and
dian age of apparition was 24 years with the youngest case spirituality. The first description of this dermatological
being 12 and the oldest 81. Some of the diseases were sec- manifestation comes from the Bible, in which is de-
ondary to other causes such as hemangiomas and other neo- scribed how Jesus of Nazareth appeared the day before
plasias or epistaxis episodes. Most of the cases have been the crucifixion as stated by the evangelist Luke “and be-
reported in India and the USA; most of them correspond to ing in anguish he prayed more earnestly, and his sweat
hemolacria alone (51.6%). Discussion: We have stated the was like drops of blood falling to the ground.” Since
basics of the substances involved in the coagulation process then, this phenomenon has been related to appearance
that have been described as genetically altered in some pa- of stigmas and is often looked like a supernatural phe-
tients such as mucins, metalloproteinases, and fibrinogen, as nomenon. Several reports have been published through-
well as propose a mechanism that can explain the signs of out history, especially during the 18th and 19th centu-

karger@karger.com © 2022 S. Karger AG, Basel Correspondence to:

www.karger.com/sad Alejandro I. Trejo-Castro, a00818219 @ itesm.mx
Table 1. Articles and characteristics of the patients included in the systematic review

Author Year Country Gender Age Hematohidrosis Hemolacria Place of blood sweeting

Bhagwat et al. [5] 2009 India Female 12 X Forehead, umbilical area

Jaju et al. [3] 2009 India Male 72 X Abdomen

Mishra [6] 2009 India Female 13 X X Eyes

Patel and Mahajan [7] 2010 India Male 13 X Face, arm, trunk

Jablenska et al. [8] 2010 United Kingdom Female 39 X Eyes

Praveen and Vincent [9] 2011 India Female 10 X X Face, neck, umbilical, wrists, legs

Özcan et al. [10] 2012 Turkey Female 11 X Eyes

Biswas et al. [11] 2013 India Female 12 X Face, trunk

Dillivan [12] 2013 USA Male 81 X Eyes

Tshifularo [13] 2013 South Africa Female 30 X Ear

Female 26 X Ear
Female 34 X Ear
Female 18 X Ear

Deshpande et al. [14] 2014 India Male 10 X Face

Iovieno et al. [15] 2015 Italy Male 28 X Eyes

Shen et al. [16] 2015 China Female 9 X Canthi, nails, tongue, umbilical area

Uber et al. [17] 2015 Brazil Female 18 X Palms, back of hands, face

Doshi et al. [18] 2015 India Female 10 X Eyes

Fowler et al. [19] 2015 USA Male 16 X Eyes

Female 20 X Eyes

Jafar and Ahmad [20] 2016 Kuwait Female 12 X Eyes

Pujari and Bajaj [21] 2016 India Female 15 X Eyes

Bakhurji et al. [22] 2017 Saudi Arabia Female 4 months X Eyes

Gião Antunes et al. [23] 2017 Portugal Male 66 X Eyes

Jayaraman et al. [24] 2017 India Female 10 X Scalp

Maglie and Caproni [25] 2017 Italy Female 21 X Palms, face

Sobol and Barmettler [26] 2017 USA Female 11 X Eyes

James et al. [27] 2018 India Male 22 X Eyes

Ali and Naik [28] 2018 India Female 35 X Eyes

Di Maria and Fama [29] 2018 Italy Male 52 X Eyes

Khanna et al. [30] 2018 France Female 67 X Eyes

Manzano et al. [31] 2018 USA Male 11 months X Eyes

Shahgholi [32] 2018 Iran Female 11 X Face, umbilical area, vagina, back
Male 8 X Nail, hand, and feet
Female 9 X Face

Ricci et al. [4] 2018 Italy Female 15 X X Eyelids, palms

Das et al. [33] 2019 India Female 15 X X Ears, nose, eyes

Female 13 X X Face, eyes, shoulders, forearm

180 Skin Appendage Disord 2022;8:179–185 Carrion-Alvarez et al.

DOI: 10.1159/000520648
Table 1 (continued)

Author Year Country Gender Age Hematohidrosis Hemolacria Place of blood sweeting

Hansson et al. [34] 2019 Sweden Male 9 X Forehead, neck

Murota et al. [35] 2019 Japan Female 6 X Palms

Pari [36] 2019 India Female 10 X Forehead, chest

Corrà et al. [37] 2020 Italy Female 15 X X Oral cavity

Drake and Packer [38] 2020 USA Male 81 X Eyes

Seethapathy and Jethani [39] 2020 India Male 62 X Eyes

Agrawal et al. [40] 2020 India Female 13 X Eyes

Das et al. [41] 2020 India Female 11 X Eyes

Boffa et al. [42] 2020 Malta Male 11 days X Eyes

Garcia et al. [43] 2020 USA Male 12 X Eyes

Khan et al. [44] 2020 Bangladesh Female 20 X Eyes

Li et al. [45] 2020 USA Male 22 X Eyes

Salas-Alanis et al. [46] 2020 Mexico Male 22 X X Scalp, palms, eyes

AlGoiriani et al. [47] 2021 Saudi Arabia Female 12 X Eyes

Hoover et al. [48] 2021 USA Male 9 X X Gum, eyes, umbilical area, axillae
Female 6 months X X Hairline, ears

Kosałka-Węgiel et al. [49] 2021 Poland Male 18 X Eyes

Idowu et al. [50] 2021 USA Female 21 X Eyes

Nandakumal et al. [51] 2021 Malaysia Male 12 X Eyes

Singh et al. [52] 2021 India Female 60 X Eyes

Tirthani et al. [53] 2021 India Female 13 X Face

Ghosh et al. [54] 2021 India Female 25 X Eyes

ries [1]. However, the first published medical report of Methods

hematohidrosis accessible in PubMed dates from 1918
We performed a systematic search following the PRISMA flow-
in the British Medical Journal [2]. In this disease, the chart (online suppl. material; for all online suppl. material, see
secretion of a blood-like substance is observed in situa- www.karger.com/doi/10.1159/000520648). An electronic data-
tions of extreme stress such as facing death [3]. It is a base search was conducted in PubMed from January 1, 2021, to
rare disease, and its mechanism is still unknown, while July 31, 2021. The words included were “hematohidrosis” OR “he-
the use of some medications like propranolol has re- molacria” OR “haematohidrosis” OR “haemolacria.” Only case re-
ports and case series were included. Articles on English and Span-
ported a clinical benefit in some case reports [4]. A clin- ish were included. The exclusion criteria included inability to find
ical case was previously reported by our colleague Salas- the full text even after contacting the corresponding author and
Alanis et al. in June 2020. Considering the small num- lack of information. All duplicates were removed using a citation
ber of cases reported so far, we performed a systematic manager program.
review of published cases in order to systematize the
current information of this condition.

Hematohidrosis, Hemolacria, and Skin Appendage Disord 2022;8:179–185 181

“Trichorrhage”: A Systematic Review DOI: 10.1159/000520648
 Fig. 1. Action places of the coagulation-al-
tered mechanisms. The figure was created
in the Mind the Graph platform.

a c

Fig. 2. a Trichorrhage starting from the scalp that bleeds down to the cheek. b Trichoscopy: localized bleeding
around the hair follicle forming small ponds. c Arrow shows erythrocytic clusters located in the stratum corneum
from hair skin.

Results tears was associated with a systemic disease such as un-

derlying hemangiomas or meningiomas [29, 30]. In
A total of 66 results were found, and after duplica- some of the cases, the clinical manifestations appeared
tion removal and identification, only 53 articles which after traumatizing events; however, the magnitude of
accounted for 60 cases were included. All the articles these events significantly varied between the cases,
can be found in Table 1 [3–54]. From the retrieved ar- while some patients have been exposed to life threats
ticles, 20 presented only hematohidrosis while 31 pre- [46], witnessed a violent death [5], or outlived a natural
sented hemolacria and 9 cases presented both manifes- disaster [4], other detonates included familiar conflicts
tations. In some articles, the presence of the bloody [18].

182 Skin Appendage Disord 2022;8:179–185 Carrion-Alvarez et al.

DOI: 10.1159/000520648
Table 2. Role of gene-affected factors in coagulation

Component Role Reference

Extracellular matrix The extracellular matrix contains several proteins which activate platelets once the blood vessels become Bergmeier and
damaged; some of those proteins include integrins and fibrinogen; there are also several nonintegrin Hynes et al. [56]
receptors in the matrix that contribute significantly to platelet adhesion
Collagen chains Collagen chains’ role in coagulation has been largely studied, exercising regulatory functions within the Farndale et al.
vasculature. Several types of collagens exist, some of them being responsible for platelet adhesion and [57]
promoting the interaction with coagulation factors
A-disintegrin These molecules discovered from snake venom were later also described in mammals; its role is to adhere to Gieber and
integrins and prevent the platelet inhibition, being responsible for the hemorrhage in snake bite victims Zigrino [61]
Metalloproteinase Similar to disintegrins, they play a role in inhibiting platelet aggregation, which helps to decrease coagulation
and the formation of blood clots
Metallopeptidases The metallopeptidases have also been described in both snake venom and within the human; the role they Das et al. [62]
play is similar to metallopeptidase inhibiting platelet adhesion. However, this one has also been shown to
participate in breast cancer migration or invasion
Integrins Integrins are responsible for regulation in the hemostasis process affecting the endothelial cell contractility Gregg et al.
and barrier as well as modifying the adhesion of platelets [63]
Laminins These are noncollagenous proteins present in the basement membrane of the cells; they interact with the White-Adams
platelets promoting adhesion, activation, and aggregation in the initial steps of hemostasis depending on the et al. [64]
XII factor
Mucins While their most important job is related to the control of mineralization and in the immune response to Shao et al. [65]
certain pathogens, they have also shown a role in coagulation in certain types of cancer such as carcinoma
allowing them to promote thrombosis through the manipulating signaling in neutrophils and platelets
von Willebrand factor This glycoprotein has been identified as a major player in hemostasis since the mid-1950s; its functions include Peyvandi et al.
binding to the FVIII, platelets, and constituents of connective tissue, thus promoting clot formations [60]

From the published cases, the median age was 23.51 years, genic mutations in genes related to the extracellular ma-
with the youngest case being a newborn of only 11 days [42] trix, as well as collagen chains, A-disintegrin, metallopro-
and the oldest an 81-year-old patient [38]. The cases were teinase, metallopeptidases, mucins, and von Willebrand
more predominant in females (65%) than in males (35%). factor. All these components play a role in would healing
Most of the cases were reported in India (20/60), USA (10/60), and coagulation (shown in Fig. 1). The extracellular ma-
and Italy (5/60). Only 2 cases have been reported in Latin trix provides platelets with proteins necessary for their
America [17, 46] as well as other 2 in Africa [10, 13]. The most activation [55]; collagen chains have been associated with
common place of bleeding was the eyes (36), followed by the wound healing since several years, with their role in the
face (10) and ears (8), and uncommon places included the feet formation of blood clots [56]; A-disintegrin and metal-
[32], eyelids [4], and axillae [48]. loproteinases have an active part in the activation of
platelets [57]. Mucins have been shown to facilitate the
platelet activation by circulating tumor cells in certain
Discussion types of cancer thus having an active play in coagulation
as well [58]. Finally, von Willebrand factor is a glycopro-
As we have stated before, hematohidrosis and hemo- tein whose part in hemostasia has been described since
lacria are still diseases that continue to be surrounded by the mid-1950s [59]. In-depth functions of these compo-
mystery, mystics, and a certain lack of information about nents can be found in Table 2 [55, 56, 59–64]. Alterations
the fisiopatholgy, leading to an overall lack of under- like this may explain how the disease works. These condi-
standing. tions both surrounded in mystery and syncretism have a
It is important to add up that even when information physiological explanation, characterized by an increase in
is still needed in the mechanisms behind the disease, ge- heart rate, erythrocytic extravasation in small caliber ves-
netic testing has been performed finding possible patho- sels such as capillaries, and leaking in both sweat glands

Hematohidrosis, Hemolacria, and Skin Appendage Disord 2022;8:179–185 183

“Trichorrhage”: A Systematic Review DOI: 10.1159/000520648
and hair follicles. Furthermore, here we reported the first Conflict of Interest Statement
clinical, biopsy, and trichoscopy images of trichorrhage,
The authors have no conflicts of interest to declare.
a novel medical term that describes the flow of blood from
hair follicles in patients with hematohidrosis (as shown in
Fig. 2). While more research is needed, the genetic insight
Funding Sources
allows us to draft a general picture of the possible path-
ways involved in hemolacria and hematohidrosis. No funding was needed.

Acknowledgment Author Contributions

The figure was created in the Mind the Graph platform. D.C.-A., A.I.T.-C., J.C.S.-A., M.S.-G., and O.R.F.-R.: all 5
worked in the ideation, writing, and corrections of the manuscript.

Statement of Ethics
Data Availability Statement
Ethical approval was not required for this study in accordance
with national guidelines. Written informed consent was obtained All data can be accessed in Table 2.
from the patient for publication of this case report and any accom-
panying images.

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Hematohidrosis, Hemolacria, and Skin Appendage Disord 2022;8:179–185 185

“Trichorrhage”: A Systematic Review DOI: 10.1159/000520648