1
|
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.
Collapse
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
| |
Collapse
|
2
|
Cherukuru S, Uppara SK, Raval R, Chappidi M, K DS. Crimson Tears: A Rare Case of Hematohidrosis. Cureus 2024; 16:e68233. [PMID: 39347273 PMCID: PMC11439452 DOI: 10.7759/cureus.68233] [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] [Accepted: 08/30/2024] [Indexed: 10/01/2024] Open
Abstract
Hematohidrosis is a rare clinical disorder characterized by oozing blood from intact skin and mucous membranes in the absence of a bleeding disorder. Most of the cases reported are from Asia. Although etiopathogenesis is unclear, it has been strongly linked to psychological stress. A nine-year-old girl was brought to the hospital with multiple episodes of painless bleeding from her nose and mouth for four days and eyes for three days, lasting four to five minutes each. Her symptoms and a thorough but unrevealing workup, including brain imaging, led to a clinical diagnosis of hematohidrosis, with parental disharmony as the underlying stress factor. Family therapy was recommended, and parent management training regarding the positive and negative reinforcement techniques was given. A significant improvement was observed at her one-month follow-up. This case adds to the current limited literature on hematohidrosis, highlighting its association with psychological stress and the importance of a multidisciplinary approach to management. Future research is warranted to elucidate molecular pathways involved in stress-induced vascular dysfunction and explore targeted therapeutic interventions.
Collapse
Affiliation(s)
| | | | - Rutvik Raval
- Internal Medicine, B. J. Medical College, Ahmedabad, IND
- Internal Medicine, Sri Venkateswara Medical College, Tirupati, IND
| | | | - Dileep Satya K
- Pediatrics, Sri Venkateswara Medical College, Tirupati, IND
| |
Collapse
|
3
|
Mitra D, Kishore K, Senapathi SC. Painless bleeding in a 4-year-old boy. Pediatr Dermatol 2024; 41:747-749. [PMID: 38529765 DOI: 10.1111/pde.15608] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/23/2024] [Accepted: 03/13/2024] [Indexed: 03/27/2024]
Affiliation(s)
- Debdeep Mitra
- Department of Dermatology, Command Hospital Air Force, Bengaluru, India
| | - Karthi Kishore
- Department of Dermatology, Military Hospital, Shillong, India
| | | |
Collapse
|
4
|
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.
Collapse
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
| |
Collapse
|
5
|
Gandarillas ME, Guiot-Isaac N, Larralde M. Pediatric hematohidrosis: case report of a rare clinical entity. Int J Dermatol 2024; 63:242-243. [PMID: 37994051 DOI: 10.1111/ijd.16939] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/29/2023] [Revised: 10/16/2023] [Accepted: 11/14/2023] [Indexed: 11/24/2023]
Affiliation(s)
- María E Gandarillas
- Pediatric Dermatology Department, Hospital Ramos Mejıa, Buenos Aires, Argentina
| | - Nicolle Guiot-Isaac
- Dermatology Department, Fundación Universitaria de Ciencias de la Salud, Bogotá, Colombia
| | - Margarita Larralde
- Pediatric Dermatology Department, Hospital Ramos Mejıa, Buenos Aires, Argentina
- Dermatology Department, Hospital Aleman, Buenos Aires, Argentina
| |
Collapse
|
6
|
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
| |
Collapse
|
7
|
Chen Y, Wang Y, Dong X. Genetic analysis and treatment strategy for hematohidrosis in a child. Int J Dermatol 2024; 63:e10-e12. [PMID: 37779344 DOI: 10.1111/ijd.16862] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/22/2023] [Revised: 07/21/2023] [Accepted: 09/16/2023] [Indexed: 10/03/2023]
Affiliation(s)
- Yeli Chen
- Dermatology Department, Hainan Women and Children's Medical Center, Haikou, China
| | - Yan Wang
- Dermatology Department, Hainan Women and Children's Medical Center, Haikou, China
| | - Xiujuan Dong
- Pediatric Hematology-Oncology Department, Hainan Women and Children's Medical Center, Haikou, China
| |
Collapse
|
8
|
Ogbuneke JU, Allen JC. Case Report and Review of the Pathophysiology and Therapeutics of Adult Hematohidrosis. Cureus 2023; 15:e36187. [PMID: 36937119 PMCID: PMC10016733 DOI: 10.7759/cureus.36187] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 03/15/2023] [Indexed: 03/17/2023] Open
Abstract
Hematohidrosis is an extremely rare condition characterized by the oozing or secretion of blood through intact skin and mucosa, particularly through eccrine glands. Although there is not much literature available on the condition, examples of Hematohidrosis include the crying and sweating of blood. The fluid may have a bloody tinge or may be frank blood. The anomaly has no identifiable etiology, and patients generally present in a good state of health. In this report, we present a 19-year-old female who had weekly occurrences of bloody diaphoresis that had been present consistently for one year. During her presentation at the hematology clinic, she was investigated thoroughly for alternative causes, but none were found. The patient was diagnosed with hematohidrosis and was offered treatment with propranolol, which she declined. She continues to follow up routinely in the hematology clinic with persistent symptoms.
Collapse
Affiliation(s)
| | - John C Allen
- Hematology and Oncology, Baptist Memorial Hospital, Oxford, USA
| |
Collapse
|
9
|
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.
Collapse
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
| |
Collapse
|
10
|
Takeyama M, Furukawa S, Ogiwara K, Nogami K. First case report of hematidrosis complicated with type 1 von Willebrand disease. Pediatr Int 2023; 65:e15615. [PMID: 37589374 DOI: 10.1111/ped.15615] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/15/2023] [Revised: 06/26/2023] [Accepted: 06/30/2023] [Indexed: 08/18/2023]
Affiliation(s)
| | - Shoko Furukawa
- Department of Pediatrics, Nara Medical University, Kashihara, Japan
| | - Kenichi Ogiwara
- Department of Pediatrics, Nara Medical University, Kashihara, Japan
| | - Keiji Nogami
- Department of Pediatrics, Nara Medical University, Kashihara, Japan
| |
Collapse
|
11
|
Nguyen HT, Vo PTD, Nguyen TTD, Nguyen QT, Truong DL. Hematohidrosis induced by separation anxiety disorder during COVID-19 quarantine: A case report and brief literature review. Dermatol Reports 2022. [DOI: 10.4081/dr.2023.9615] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/24/2022] Open
Abstract
Here, we report a case involving a 10-year-old Vietnamese girl who developed hematohidrosis during the coronavirus disease quarantine. She was hospitalized with a 3-week recurrent bleeding on the abdominal skin. Physical examination revealed no signs of injuries on the skin. Hematological and biochemical test results and coagulation profiles were all within normal ranges. No abnormal findings were observed on abdominal ultrasonography and computed tomography. Numerous erythrocytes were observed during microscopic examination of fluid samples from the abdominal skin. It was speculated that hematohidrosis was precipitated by separation anxiety disorder, because the onset and remission of symptoms correlated with the beginning and end of the local quarantine, respectively. Our case report and brief literature review highlight the transient and benign nature of hematohidrosis. Although specific guidelines are not well established, hematohidrosis is a transient phenomenon that is treatable with pharmaceutical and non-pharmaceutical interventions, and its overall prognosis is favorable.
Collapse
|
12
|
Biswas D, Mondal DK, Biswas R, Halder A. Hematidrosis: A rare entity - case series and review of literature. Indian J Psychiatry 2022; 64:423-425. [PMID: 36060725 PMCID: PMC9435613 DOI: 10.4103/indianjpsychiatry.indianjpsychiatry_142_22] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/16/2022] [Revised: 03/24/2022] [Accepted: 05/03/2022] [Indexed: 11/05/2022] Open
Affiliation(s)
- Dipanwita Biswas
- Department of Psychiatry, R G Kar Medical College, Kolkata, West Bengal, India
| | - Dilip K Mondal
- Department of Psychiatry, R G Kar Medical College, Kolkata, West Bengal, India
| | - Rituparna Biswas
- Department of Radiation Oncology, Chittaranjan National Cancer Institute, Kolkata, West Bengal, India
| | - Anirban Halder
- Department of Radiation Oncology, Chittaranjan National Cancer Institute, Kolkata, West Bengal, India
| |
Collapse
|
13
|
Alasfoor S, Albashari M, Alsermani A, Bakir M, Alsermani M, Almustanyir S. A Strange Occurrence of Hematohidrosis: A Case Report From Saudi Arabia. Cureus 2022; 14:e21682. [PMID: 35237478 PMCID: PMC8882245 DOI: 10.7759/cureus.21682] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 01/28/2022] [Indexed: 11/05/2022] Open
Abstract
Hematohidrosis is a rare disorder of blood excretion from the eccrine sweat gland not associated with an injury or trauma. Although several hypotheses exist to explain such a condition, the etiology of hematohidrosis remains unknown. Psychological stress is strongly linked to the condition, yet patients may present completely healthy with no identifiable etiology. The diagnosis of hematohidrosis can be difficult and requires the exclusion of bleeding disorders, vasculitis, and other disorders. Here, we present a case of hematohidrosis in a 20-year-old female who had almost weekly bloody tears, as well as gum bleeding, ear bleeding, and epistaxis for the past four months. During hospitalization, the patient was thoroughly investigated for an etiology, but no identifiable cause was found. The patient was diagnosed with hematohidrosis and treated with propranolol. Her condition had improved significantly on follow-up.
Collapse
|