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Anyfantakis D, Tsoucalas G, Karelis A, Papazoglou N, Spandidos DA, Krasagakis K, Symvoulakis E. Diapedesis leading to hematidrosis due to abrupt emotional suffering (Review). Exp Ther Med 2024; 28:453. [PMID: 39478740 PMCID: PMC11523038 DOI: 10.3892/etm.2024.12743] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/21/2023] [Accepted: 09/13/2024] [Indexed: 11/02/2024] Open
Abstract
Hematidrosis constitutes a rare imposing phenomenon encountered millennia ago. It involves the plexus of small, mostly superficial arteries, veins and sweat glands of various parts of the human body. Bloody sweat implies skin pathology. It has been connected to psychogenic impairment and lacks a clear etiology. The present study conducted a historical review using the Thesaurus Linguae Graecae®, Gallica-BNF and Google Books databases. The year 1900 was the mark for full-text works to be included. A second review of the modern literature, over the last 20 years, was carried out using the PubMed/MedLine database. In both eras, the terms h[a]emat[h]idrosis, diapedesis, sudor sanguineus, plus various others vocables of Hellenic and Latin origin, were used as key words. Cases of persons suffering hematidrosis during stigmata as partly connected to this study were surveyed and discussed separately. References were obtained from the classical Greece, late Hellenic antiquity and Roman era. A number of 70 cases were registered from the Renaissance to 1900, and 44 cases between 2007 and 2022. The later time interval is considered as the selection of references of all time, while the 1870 to 1884 interval followed. Differences in sex were observed (males, 60.6%; and females, 39.4%; vs. males, 25.6%; and females, 74.4%). The scalp, forehead, face, trunk, arms and legs were the most common areas of appearance in both time intervals. The eyes and ears were the most common areas of appearance in the modern era. As regards the limitations of the present review, it should be emphasized that not all historical cases were available for survey. Stigmata were studied independently. Records in etiology hypothesis, pathological description and the manifestation of a cluster of post-triggering effects described were similar over time. The modern way of life, major stressful events, a fear of death, hematological disorders, vessel derangements and deep faith may trigger hematidrosis. This spectacular acute event stimulates alertness in patients, family, caregivers and physicians, and may lead to social isolation and unnecessary diagnostic and/or therapeutic interventions. The present narrative review aimed to connect science with history and religion, promote contemplation among health professionals and highlight a medical peculiarity. An open mind is warranted in order to comprehend phenomena in religion. Education for the family and a knowledge update for health professionals is also necessary.
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Affiliation(s)
| | - Gregory Tsoucalas
- Department of History of Medicine and Deontology of Medicine, School of Medicine, University of Crete, 71003 Heraklion, Greece
| | - Andreas Karelis
- Department of Social Medicine, School of Medicine, University of Crete, 71003 Heraklion, Greece
| | - Nikolaos Papazoglou
- Department of Nursing, School of Health Sciences, Hellenic Mediterranean University, 71004 Heraklion, Greece
| | - Demetrios A. Spandidos
- Laboratory of Clinical Virology, School of Medicine, University of Crete, 71003 Heraklion, Greece
| | | | - Emmanouil Symvoulakis
- Clinic of Social and Family Medicine, School of Medicine, University of Crete, 71003 Heraklion, Greece
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Matsyura O, Besh L, Jefimova S, Khrystyna SK, Gerasymov S. Hematohidrosis in Pediatric Practice - a Case Report and Review of the Literature. Acta Med Litu 2024; 31:12-21. [PMID: 38978871 PMCID: PMC11227682 DOI: 10.15388/amed.2024.31.1.2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/03/2022] [Revised: 06/26/2023] [Accepted: 11/16/2023] [Indexed: 07/10/2024] Open
Abstract
Hematohidrosis (bloody sweat) is a symptom of trophic damage to the vascular wall, in which sweat mixes with blood and seeps onto undamaged areas of the skin in the form of red or pink liquid (depending on the ratio of sweat to blood). In our study we have analyzed 25 case reports of hematohidrosis in children, reported throughout the world using PubMed, ResearchGate with detailed description and opened access. We took into consideration: age of the patient, sex, location of bloody excretion, cause or trigger, treatment and its effectiveness. Our clinical case present a 9-year-old girl complained of a periodic bleeding from the intact skin of the face, neck, thighs (without visible damage to the skin) manifested by red or pink liquid, nosebleeds, and bloody discharge from the mucous membrane of the eyes. The secretions were of varying intensity and lasted up to several hours. Most of all episodes are associated with a strong emotional exertion. One of the theories of hematohidrosis pathogenesis is evident vasoconstriction of the blood vessels surrounding the sweat glands, provoked by hyperactivation of the sympathetic nervous system, which is followed by their excessive vasodilation up to rupture and blood entering the sweat gland ducts. Capillary endothelial cells are known to contain β2-adrenoceptors, which, through the modulation of nitric oxide release, cause endothelium-dependent vasodilation. Blocking β-adrenoceptors (for example, propranolol) prevents excessive vasodilation of blood vessels and, accordingly, their rupture and blood flow to the sweat gland. We managed to find out that the patient's bloody sweat was a manifestation of a separate pathological phenomenon, and not one of the symptoms of another disease. A properly formed treatment complex and the great trust of the parents enabled to stabilize the child's condition, and later to cure her. Currently, hematohidrosis is recognized as an independent disease that requires in-depth study of the triggering mechanisms of development, pathogenetic and clinical features.
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Affiliation(s)
- Oksana Matsyura
- Danylo Halytsky Lviv National Medical University, Lviv, Ukraine Communal Nonprofit Enterprise “City Children’s Clinical Hospital of Lviv”, Lviv, Ukraine
| | - Lesya Besh
- Danylo Halytsky Lviv National Medical University, Lviv, Ukraine Communal Nonprofit Enterprise “City Children’s Clinical Hospital of Lviv”, Lviv, Ukraine
| | - Svitlana Jefimova
- Danylo Halytsky Lviv National Medical University, Lviv, Ukraine Communal Nonprofit Enterprise “City Children’s Clinical Hospital of Lviv”, Lviv, Ukraine
| | - Slivinska-Kurchak Khrystyna
- Danylo Halytsky Lviv National Medical University, Lviv, Ukraine Communal Nonprofit Enterprise “City Children’s Clinical Hospital of Lviv”, Lviv, Ukraine
| | - Sergiy Gerasymov
- Communal Nonprofit Enterprise “City Children’s Clinical Hospital of Lviv”, Lviv, Ukraine
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Goh SW, Wee WHD, Chua WXR, Fan BE. Hematohidrosis in 15th Century Renaissance Art and a Review of Modern Literature. Semin Thromb Hemost 2024; 50:119-129. [PMID: 37192650 DOI: 10.1055/s-0043-1768937] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 05/18/2023]
Affiliation(s)
- Shyer Wern Goh
- Yong Loo Lin School of Medicine, National University of Singapore, Singapore, Singapore
| | - Wen Hon Darren Wee
- Yong Loo Lin School of Medicine, National University of Singapore, Singapore, Singapore
| | - Wen Xin Rachel Chua
- Yong Loo Lin School of Medicine, National University of Singapore, Singapore, Singapore
| | - Bingwen Eugene Fan
- Yong Loo Lin School of Medicine, National University of Singapore, Singapore, Singapore
- Department of Haematology, Tan Tock Seng Hospital, Singapore, Singapore
- Department of Laboratory Medicine, Khoo Teck Puat Hospital, Singapore, Singapore
- Lee Kong Chian School of Medicine, Nanyang Technological University, Singapore, Singapore
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Octavius GS, Meliani F, Heriyanto RS, Yanto TA. Systematic review of hematidrosis: Time for clinicians to recognize this entity. World J Dermatol 2023; 11:7-29. [DOI: 10.5314/wjd.v11.i2.7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/22/2022] [Revised: 10/12/2022] [Accepted: 11/29/2022] [Indexed: 02/03/2023] Open
Abstract
BACKGROUND Hematidrosis is a sporadic disease, to a point where its existence is still denied up to date. It is also linked to stigmata, psychological roots, and religious beliefs, whih has strengthened clinicians' disbelief in hematidrosis.
AIM To conduct a thorough review to classify the likelihood of hematidrosis cases.
METHODS We searched PubMed, Science Direct, Medline, and Google Scholar, as well as four different preprint databases, including Medrxiv, Research Square, SSRN, and Biorxiv. We included studies from 1996 onwards, with no limitation on language. Hematidrosis was classified as "unlikely", "likely", and "highly likely".
RESULTS There are 74 articles with 106 hematidrosis cases. India (n = 40) and China (n = 11) report the most cases. Patients are mostly female (76.5%) with a median age of 13 years. The head region is the most common bleeding site (n = 168/254). Headaches (26.9%) and abdominal pain (16.4%) are the most common prodromes. Beta-blockers (43%) and anxiolytic (23.2%) are the most commonly prescribed pharmacotherapy. Psychotherapy (37.5%) and counseling (32.5%) are the most utilized non-pharmacotherapy measures. Only 41.1% and 19.8% of all cases reach complete resolution and are highly likely to be hematidrosis, respectively.
CONCLUSION Although hematidrosis is rare and the pathophysiology is still largely unknown, that does not mean hematidrosis does not exist. It is important to note that the most frequent trigger factors are either anxiety, fear, or excessive stress. Clinicians need to exclude other diagnoses and search for stressors to alleviate the bleeding.
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Affiliation(s)
| | - Fellisa Meliani
- Department of Medicine, Universitas Pelita Harapan, Tangerang 15811, Indonesia
| | | | - Theo Audi Yanto
- Department of Medicine, Universitas Pelita Harapan, Tangerang 15811, Indonesia
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Carrion-Alvarez D, Trejo-Castro AI, Salas-Garza M, Fajardo-Ramirez OR, Salas-Alanis JC. Hematohidrosis, Hemolacria, and “Trichorrhage”: A Systematic Review. Skin Appendage Disord 2022; 8:179-185. [PMID: 35707284 PMCID: PMC9149430 DOI: 10.1159/000520648] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/26/2021] [Accepted: 10/16/2021] [Indexed: 11/19/2022] Open
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.
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Affiliation(s)
- Diego Carrion-Alvarez
- Dystrophic Epidermolysis Bullosa Research Association, Guadalupe, Mexico
- Basic Science Department, School of Health Sciences, Universidad de Monterrey, San Pedro Garza García, Mexico
| | - Alejandro I. Trejo-Castro
- School of Engineering and Sciences, Tecnológico de Monterrey, Monterrey, Mexico
- *Alejandro I. Trejo-Castro,
| | - Mauricio Salas-Garza
- School of Medicine and Health Sciences, Tecnológico de Monterrey, Monterrey, Mexico
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Shafique DA, Hickman AW, Thorne A, Elwood HR, Zlotoff BJ. Pediatric hematidrosis - A case report and review of the literature and pathogenesis. Pediatr Dermatol 2021; 38:994-1003. [PMID: 34515356 DOI: 10.1111/pde.14792] [Citation(s) in RCA: 13] [Impact Index Per Article: 3.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/03/2023]
Abstract
Hematidrosis is a disorder in which blood-tinged fluid exudes from uninjured skin or mucosa. It is often classified as an eccrine sweat disorder, though the precise mechanism-including involvement of sweat glands-has yet to be proven. In contemporary case reports, hematidrosis appears most frequently in the pediatric population, with 83% of cases in the literature since 2008 occurring in individuals 18 years old or younger. We present here a case of a 10-year-old girl with hematidrosis followed by a review of the literature, with an emphasis on the features of this condition in the pediatric population.
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Affiliation(s)
| | | | - Amy Thorne
- University of Incarnate Word School of Medicine, San Antonio, Texas, USA
| | - Hillary R Elwood
- Department of Pathology, University of New Mexico, Albuquerque, New Mexico, USA
| | - Barrett J Zlotoff
- Department of Dermatology, University of Virginia, Charlottesville, Virginia, USA
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