Systematic Reviews
Copyright ©The Author(s) 2023.
World J Dermatol. Feb 6, 2023; 11(2): 7-29
Published online Feb 6, 2023. doi: 10.5314/wjd.v11.i2.7
Table 1 Medical subject heading terms used in each database
Database
Medical subject heading
Number of studies found
PubMed“hematidrosis”[All Fields] OR (“haematohidrosis”[All Fields] OR “hematohidrosis”[All Fields]) OR (“bloody”[All Fields] AND (“tearings”[All Fields] OR “tears”[MeSH Terms] OR “tears”[All Fields] OR “tearing”[All Fields] OR “lacrimal apparatus diseases”[MeSH Terms] OR (“lacrimal”[All Fields] AND “apparatus”[All Fields] AND “diseases”[All Fields]) OR “lacrimal apparatus diseases”[All Fields] OR “lacerations”[MeSH Terms] OR “lacerations”[All Fields])) OR (“bloody”[All Fields] AND (“sweat”[MeSH Terms] OR “sweat”[All Fields] OR “sweating”[MeSH Terms] OR “sweating”[All Fields] OR “sweats”[All Fields] OR “sweatings”[All Fields])) OR “hemolacria”[All Fields]203
Medline((hemathidrosis) OR haematohidrosis) OR bloody sweat1057
Google Scholarallintitle: hematidrosis OR haematohidrosis OR bloody sweat55
Science Direct(Hematidrosis) OR (haematohidrosis) OR (bloody sweat)200
MedRxiv(Hematidrosis) OR (haematohidrosis) OR (bloody sweat)367
BioRxiv(Hematidrosis) OR (haematohidrosis) OR (bloody sweat)839
SSRN(Hematidrosis) OR (haematohidrosis) OR (bloody sweat)0
Research Square(Hematidrosis) OR (haematohidrosis) OR (bloody sweat)134
Table 2 Characteristics of hematidrosis patients
Ref.
Patient No.
Time from onset to consultation
Triggers of bleeding
Other kinds of bleeding
Comorbidities
Bleeding frequency
Bleeding duration
Other Bleeding characteristics
Octavius et al[70], 2021 #1One monthNot mentionedNot mentionedNot mentionedThree times per dayStopped immediatelyNot mentioned
#2One monthNot mentionedNot mentionedNot mentionedNot mentionedNot mentioned Not mentioned
Bhattacharya et al[42], 2013 #1One monthNot mentionedNot mentionedNot mentioned> 10 times per dayNot mentionedNot mentioned
Manonukul et al[43], 2008 #11.5 yr agoNot mentionedNot mentionedNot mentionedRecurrent episodesNot mentionedNot mentioned
Murota et al[45], 2020#1A few monthsExcess pressure exerted while playing on the metal barNot mentionedNot mentionedNot mentionedNot mentionedIt did not occur during sleeping
Das et al[89], 2020 #1Two weeksHead traumaNot mentionedNot mentionedNot mentionedNot mentionedNot mentioned
#2One hourAnxiety symptomsAnxiety
Matsuoka et al[36], 2020#1Two yearsNot mentionedNot mentionedDissociative disorder and self-harmingFew times per weekNot mentionedBloody sweat-like fluid secretion
Chabchoub et al[49], 2013 #16 mauNot mentionedNot mentionedNot mentionedNot mentionedNot mentionedNot mentioned
Shafique et al[38], 2021#13 moNot mentionedRectal bleedSignificant psychosocial stress due to parental separation3-35 times per dayNot mentionedOccurred during sleep and wakefulness
Carvalho et al[35], 2008#1November 2004Physical exertion, increase in room temperatureNot mentionedNot mentionedNot mentionedNot mentioned Ceased spontaneously
Rossio et al[32], 2014#1Not mentionedHis family was experiencing an extremely stressful time, and his mother had just passed awayNot mentionedNot mentionedNot mentioned1-2 minNo predetermined time for recurrence
Gayal et al[90], 2020#1Six monthsHearing voices and anxietyNot mentionedSchizophrenia1-2 or 10-15 times per dayNot mentionedDepending on the intensity of psychotic symptoms
Dragan et al[91], 2017 #1Not mentionedNot mentionedEpistaxis, hemoptysisTonsillectomy, benign hypertensionNot mentionedNot mentionedNot mentioned
Alsermani et al[23], 2018#1Five monthsBullying in schoolsNot mentionedCeliac diseaseTwice weekly1-2 minSelf-limited, usually in the evening
Jayaraman et al[92], 2017#1Seven daysScolding from parents due to academic performanceNot mentionedMixed anxiety & depressive disorders, nocturnal enuresis1-2 a day3-5 minAppears when punished or stressed
Jibbawi et al[33], 2021#1Not mentionedEmotional stressNot mentionedNot mentionedNot mentionedNot mentionedAppears during stress and self-limited
Arakkal et al[24], 2016#15 mauExtreme physical exertionNot mentionedNot mentionedNot mentioned2-3 minBleeding when waking up, extreme physical activities, self-limited
Corrà et al[93], 2020#1Five yearsStress, anxious situation, exerciseNot mentionedThe peripheral visual deficit, non-hemorrhagic chronic gastritis, severe intestinal subocclusionFive times dailyFew minutesSelf-limited
Mishra[72], 2009#1Not mentionedMinor traumaHematuria, gastrointestinal bleed, epistaxisPlatelet function (PF3) dysfunctionNot mentionedNot mentionedNot mentioned
Mutanabbi et al[29], 2021 #14 mauStressHematemesis, hematuria, epistaxisNot mentioned1-2 a week3-5 minCeased spontaneously, precipitated by upcoming stress & family issues
Soliman et al[61], 2019#1Six monthsNot mentionedNot mentionedHypotension, tachycardia, syncopeNot mentioned30 secondsNever at sleep and self-remitting
Tshifularo[94], 2013 #1Not mentionedSevere mental stresses at work, study, and homeNot mentionedNot mentioned2-3 a week every 3-4 mo15-20 min2-5 ml blood-stained non-clotting discharge
#2Two yearsSevere stressFour times in 2 years (2009-2010)Not mentionedBlood-stained sweaty discharge, not clotting and watery
#3Not mentionedStressTwo times in 2 years (2010-2011)Not mentionedPainless blood-stained sweat-like discharge
#4Not mentionedSevere stress at school and home1st episode at July 2013Not mentionedNot mentioned
Meyer et al[95], 2019#1Two daysStumbled and fell on an iron toy train (trauma)Not mentionedNot mentionedAlmost dailyN/ASelf-limiting, episodic. Bleeding occurred from apparently healthy and undamaged skin; no superficial injuries
Techasatian et al[81], 2016#1April 2014Not mentionedNot mentionedNot mentioned> 10 times10-30 secondsThe time of the bleeding episode was unpredictable, not related to exertion or triggers
Yeşilova et al[96], 2016#12 dMove from cold to a warm environment, fallen out of bed (trauma) one day beforeNot mentionedNot mentioned20 times in 2 dNot mentionedNot mentioned
Quentric et al[97], 2019#1Not mentionedGeneral anxiety related to schoolEpistaxis, nipple bleedNot mentioned3-4 times a dayNot mentionedSpontaneously, mostly at night
Mora et al[98], 2013#16 moStressNot mentionedNot mentionedSeveral times a dayNot mentionedSpontaneous, self-limited, >30 episodes during hospitalization
Praveen et al[99], 2012#13 moStress (She had witnessed her elder sister being kidnapped nine months ago)EpistaxisNot mentioned2-3 times every week and had two episodes on the same day on four occasionsNot mentionedNot mentioned
Hansson et al[7], 2019#1Four weeksNot mentionedNot mentionedNot mentioned20 episodes in 4 weeksNot mentionedNot mentioned
Wang et al[44], 2014#1One monthNot mentionedNot mentionedNot mentioned2-3 times a dayNot mentionedThe bleed is described as a fresh, small amount of blood
Jerajani et al[41], 2009#1Two monthsContinuous mental stressNot mentionedDepressive disorderNot mentionedNot mentionedNot mentioned
Lipsitt et al[100], 2018#19 moExtreme emotional stress, excitement, or physical exertionNot mentionedDepression, anxiety, PTSD, non-epileptiform seizureSeven times a dayNot mentionedProgressive, episodic
Bhagwat et al[101], 2009#1Two yearsTrauma (witnessing a woman beheaded), Feeling anxiousNoneSevere depression1-2 a day15-20 minEpisodic
Sue Tin et al[40], 2015 #1Three weeksNo obvious triggerNot mentionedNot mentioned20 times a dayNot mentionedSpontaneous, worse during warmer months
Pari[102], 2018#1Five daysWithout any provoking factorNot mentionedNot mentioned2-3 a day30 minNot mentioned
Patel et al[79], 2010#1Sincmaury 2007Not mentionedNot mentionedNot mentionedOnce-daily1 minuteSpontaenous, during school hours 9-11 am
Biswas et al[76], 2013 #1Two yearsNot mentionedNot mentionedAn intelligent quotient (IQ) of 60-70 with a loss of insight1-2x daily10-15 minEpisodic, more frequent when waking up in the morning
Jafar et al[69], 2016 #1Two weeksNot mentionedEpistaxisRepeated faint attacks two years agoNot mentioned10-20 minIt was spontaneous, unpredictable, and intermittent, with no specific patterns, and it stopped spontaneously.
Wang et al[103], 2010 #13 yrEmotional excitementNot mentionedNot mentioned> 20 times during hospitalizationNot mentionedIt always occurred during the middle of the day, never when sleeping at night
Thigarajan[83], 2019#1Three daysStress fr’m father's demand on academicsNot mentionedPrimary enuresis, head tonsure, and trivial head trauma in a different location to the bleedNot mentionedFew seconds to a few minutesSelf-limited
Khalid et al[82], 2013 #1Five yearsEmotional stressNoneNoneNot mentionedNot mentionedNot mentioned
Maglie et al[104], 2017 #1Three yearsNot mentionedNot mentionedMajor depressive disorder and panic disorderNot mentioned1-5 minCould occur in sleep or during physical activity & more intense bleeding occurs during emotional stress
Uber et al[71], 2015#16 yrNot mentionedNot mentionedConversion, dissociative, and generalized anxiety disorder, high peak blood pressureNot mentionedFew minutesSpontaneous, sometimes occur during sleep, coincidental with high blood pressure peak
Salas-Alanis et al[37], 2021#1Not mentionedAcute emotional distressNot mentionedNot mentionedNumerous3-25 minFrom 6 years old, all following emotional distress
Talwar et al[80], 2021#1One yearSeparation from his auntEpistaxis, hematuria, hematocheziaAdjustment disorderOnce per weekA few minutesIt can occur during sleep
Chowdhury et al[105], 2019#1Two yearsMigraineEpistaxis, previous cesarean scar, menorrhagiaMigraineNot mentionedNot mentionedAssociated with headache, her son also had hematidrosis
Das et al[89], 2016#15 moNot mentionedNoneOtitis externaNot mentionedNot mentionedMore frequent on 06:00 A.M.
Hossain et al[77], 2018 #1One and a half yearsNot mentionedNot mentionedNot mentionedNot mentionedNot mentionedNot mentioned
#2Last two monthsNot mentionedNot mentionedRepeated episodes of stiffening of limbs with associated self-mutteringNot mentionedNot mentionedNot mentioned
#3Six monthsNot mentionedNot mentionedAcute anxiety state, repeated episodes of unresponsiveness and conduct disorderNot mentionedNot mentionedNot mentioned
Thao et al[39], 2019 #1One monthMarriageNot mentionedContinuous mental stressNot mentionedNot mentionedNot mentioned
Récher[31], 2021#1Not mentionedCOVID-19 containment induced stressNot mentionedNot mentionedNot mentionedNot mentionedA vascular pathway remains after 24 hours of bleeding
Omar[30], 2018 #1Not mentionedStressNot mentionedNot mentionedOccured in boutsNot mentionedNot mentioned
Tirthani et al[86], 2021 #1Three monthsLoss of parentsNot mentionedRecurrent panic attacks with tachycardiaNot mentioned2-3 minNot mentioned
Ricci et al[106], 2018 #1Three monthsMajor stressful event from an earthquakeNot mentionedNot mentionedNot mentionedNot mentionedNot mentioned
Bezner et al[25], 2013#1Two yearsNot mentionedHematuriaHeavy and irregular menstrual bleedUp to 20 dailyNot mentionedThe bleeding episodesreportedly decreased during her menstrual period, which typically lasted for approximately ten days
Mahamat Abderraman et al[50], 2019 #1Five years agoNot mentionedHistory of hematuria, epistaxis,Moderate anxiety-depressive disorder, acute urine retention, seizure, genu flexumNot mentioned10 minNot mentioned
Rharrabti et al[51], 2016#1Not mentionedFamily abuse, parental conflictsVulvar bleedingConversion disorder. primary enuresisNot mentionedNot mentionedMostly at day, worsening during parental conflicts
Morillo et al[52], 2019 #1Seven monthsNot mentionedNot mentionedBronchial asthma, previous history of abortionNot mentionedNot mentionedNot mentioned
Kumar et al[78], 2021#1Not mentionedAfraid, stress (during exams), anxietyNot mentionedHigh-grade anxietyNot mentionedNot mentionedSpontaneous and recurrent (4-5 times in three days)
Camargo et al[53], 2020 #1Four yearsStress and anxietyHematuriaNot mentioned2-3 times a weekNot mentionedSporadic occurrence
Shen et al[107], 2015 #1Six monthsNot mentionedNot mentionedTonic seizures1x every few days1-2 minSpontaneous, transient, self-limited
Shahriari et al[108], 2020 #1Six monthsGoing to the toilet or feeling lonelyNot mentionedNot mentionedEvery two weeksNot mentionedNot mentioned
#2Not mentionedNot mentioned EpistaxisNot mentionedEvery three weeksNot mentionedNot mentioned
#3Not mentionedFighting with her younger sister without any skin damageNot mentionedA history of aggressionNot mentionedNot mentionedNot mentioned
Badry et al[27], 2020 #1One monthStressHematemesisDepression3-5 per weekA few minutesNot mentioned
Hoover et al[54], 2019#1Almost more than seven years Illness (malaria) or distressHematemesis, hematochezia, hematuriaPrevious history of treated malaria, human immunodeficiency virus(HIV) and hepatitis B due to blood transfusionsNot mentionedNot mentionedNot mentioned
#2Not mentionedIrritability and cryingNot mentionedNot mentionedNot mentionedNot mentionedNot mentioned
Latorre Martinez et al[109], 2012#1One yearStressNot mentionedNot mentionedNot mentionedA few minutesNot mentioned
Kleymenova et al[47], 20201#1Not mentionedWeather, stressEpistaxisMenstruation problemsNot mentionedNot mentionedNot mentioned
Rani et al[84], 2018 #1Not mentionedAutonomic dysfunctionHematuriaAutonomic dysfunctionNot mentionedNot mentionedNot mentioned
#2Autonomic dysfunctionNot mentionedSpontaneous skin breaches resembling incision
#3Emotional & Psychological stressorsNot mentionedNot mentioned
#4Emotional & Psychological stressorsNot mentionedNot mentioned
#5Emotional & Psychological stressorsNot mentionedNot mentioned
#6Emotional & Psychological stressorsNot mentionedNot mentioned
Wang et al[110], 2021#1One yearNot mentionedNot mentionedNot mentionedNot mentionedNot mentionedNot mentioned
#2Four monthsNot mentionedHematemesisNot mentionedOnce every few days to dozens of times a daySeveral minutesNot mentioned
#3Five monthsCarsicknessHematuriaCanker soresUp to 60 times a daySeveral minutesNot mentioned
#4Nine monthsAnxiety or nervousness, hemolacria induced by intense lightHematemesis, hematuriaTonic-clonic seizuresOnce every few days to dozens of times a daySeveral minutesNot mentioned
#5Two monthsNot mentionedNot mentionedNot mentionedOnce every few days to dozens of times a daySeveral minutesNot mentioned
#6Four yearsMilitary training, minor emotional stress, or even during studyingNot mentionedNot mentionedOnce every few days to dozens of times a daySeveral minutesNot mentioned
#7Six monthsNot mentionedNot mentionedNot mentionedOnce every few days to dozens of times a daySeveral minutesNot mentioned
Malik et al[34], 2019 #16-8 yrEmotional stressHistory of hemolacriaGestational hypertension (after lost to follow up)Not mentionedNot mentionedBleeding started 6-8 years ago
#2Not mentionedPeriods of rage and stressEpistaxisNot mentionedNot mentionedNot mentionedUnprovoked, self-limited, and not triggered
Shahgholi[75], 2018 #1Several monthsExtreme physical or emotional stressHematemesisNot mentionedOnce or twice a day10-15 minSometimes occurred spontaneously during sleep, subsided as soon as it is wiped
#2Not mentionedUpcoming exam, playing a computer game, watching fiction TV movies or when parents not satisfying his demandsNot mentionedNot mentionedNot mentionedNot mentionedNot mentioned
#3Not mentionedNot mentionedNot mentionedNot mentionedNot mentioned5 minSpontaneous, unpredictable, and intermittent
Jagannathan et al[111], 2016 #1Not mentionedMental stressEpistaxisNot mentionedNot mentionedNot mentionedNot mentioned
#2Not mentioned
#3Not mentioned
#4Not mentioned
#5Not mentioned
#6Not mentioned
#7Not mentioned
#8Hematemesis
Agarwal et al[28], 2017 #1One monthNot mentionedNot mentionedNot mentionedNot mentioned10 secondsSelf-limited, during the day
Deshpande et al[112], 2014#1Since February 2013 (around more than one year ago)Upcoming exams, fight with parents, and parents not satisfying his demandsHemoptysis, hematuria, epistaxis, hematochezia, hematemesisOppositional defiant disorderNot mentionedNot mentionedNot mentioned
Zhang et al[16], 2004#1Four monthsNot mentionedHematemesisA previous diagnosis of Henoch-schonlein purpuraSeveral times a day or once every 3-5 dLasts secondsThe bleeding can occur during sleep
Kumar et al[78], 2019#1One year agoSuspected fear and anxiety of his lecturer Not mentionedNot mentionedThree episodes per day to 20 episodes per monthNot mentionedNot mentioned
#2Three months agoFearful dreamsNot mentionedNot mentioned> 5 episodes per dayNot mentionedEpisodes are not coinciding with dreams but occur after the event at night without any triggering factor in the day times as observed by parents at home and the hospital
Karpukhina et al[48], 2020#1One month agoNot mentionedEpistaxisHigh anxiety, neurotic behaviour, and depressionNot mentionedA few minutesNot mentioned
1There are two cases presented in this literature. However, the second case is purely a hemolacria, so we excluded it.
Table 3 Demographics of patients with hematidrosis
Variables
N (%)
Sex (n = 102)
Male 24 (23.5)
Female78 (76.5)
Age in years (n = 94) (Median - range)13 (0.17-72)
Prodromes1
Headache18 (26.9)
Dizziness3 (4.5)
Abdominal pain11 (16.4)
Nausea4 (6.0)
Vomiting4 (6.0)
Tingling5 (7.5)
Photophobia2 (3.0)
Phonophobia2 (3.0)
Pain3 (4.5)
Soreness4 (6.0)
Asthenia3 (4.5)
Easily irritable2 (3.0)
Others6 (8.7)
Presence of hemolacria34 (32.1)
Types of journals
Case report4 (5.4)
Dermatology19 (25.7)
Otorhinolaryngology4 (5.4)
General or internal medicine17 (23.0)
Hematology and oncology9 (12.2)
Pediatric (including pediatric hematology and oncology)15 (20.3)
Pharmacy1 (1.3)
Preprint (Research Square)1 (1.3)
Psychiatry3 (4.1)
Reproductive health1 (1.3)
Healthcare professionals witness the bleeding episodes
Yes 69 (65.1)
No25 (23.6)
Not explicitly mentioned12 (11.3)
Family members witness the bleeding episodes
Yes 50 (47.2)
No6 (5.6)
Not explicitly mentioned50 (47.2)
Provided bleeding pictures85 (80.2)
Laboratory tests done100 (94.3)
Radiologic and/or other tests done41 (38.7)
Normal results from all the diagnostic tests done92 (86.8)
Skin biopsies done30 (28.3)
Normal or non-specific19 (63.3)
Analyzed the blood from the bleeding site71 (67.0)
Doctors involved in the case1
Otolaryngologist25 (16.6)
Ophthalmologist6 (4)
Obstetrics and gynaecologist3 (2)
Psychiatrist29 (19.3)
Primary care4 (2.7)
Pediatrics31 (20.5)
Internal medicine19 (12.8)
Dermatologist25 (16.6)
Vascular surgeon1 (0.7)
Neurologist7 (4.8)
Tried to exclude other diagnoses90 (84.9)
Pharmacotherapy2
Beta-blockers65 (43.0)
Anxiolytic35 (23.2)
Antidepressants10 (6.6)
Atropine patches2 (1.3)
Drops of adrenaline with gauze1 (0.7)
Ascorbic acids6 (4.0)
Antifibrinolytic agents2 (1.3)
Antihistamine receptor blockers6 (4.0)
Hemostatics2 (1.3)
Others22 (14.6)
Psychosocial therapy2
Psychotherapy15 (37.5)
Behavioural therapy6 (15)
Relaxation technique6 (15)
Counseling13 (32.5)
Outcome (n = 90)
Complete resolution37 (41.1)
Time taken for complete resolution in days (median) (n = 26)60 (2-730)
Improvement of symptoms37 (41.1)
Time taken for improvement of symptoms in days (median) (n = 19)28 (10-720)
Relapse8 (8.9)
Time taken for relapse in days (mean) (n = 6)60 (53.4)
Spontaneous resolution4 (4.4)
The patient is still under follow up1 (1.1)
Loss to follow up3 (3.3)
Possibilities of hematidrosis
Highly likely21 (19.8)
Likely58 (54.7)
Unlikely27 (25.5)
1One patient can be referred to multiple specialities or have multiple prodromes.
2One patient can receive more than one modality of treatment and more than one drug.
Table 4 Differential diagnosis of hematidrosis
Hematologic disorders (Bezner et al[25], 2013; Peretz et al[68], 2016)
Acquired disorder of hemostatic mechanism
A hereditary disorder of hemostatic mechanism
Infection (Jirka, 1968; Peretz et al[68], 2016)
Chromobacterium prodigiosum
Micrococcus castellani
Disorders of vascular integrity (Bezner et al[25], 2013; Peretz et al[68], 2016)
Vasculitis
Amyloidosis
Connective tissue disorders
Gynecologic disorders (Bezner et al[25], 2013)
Ectopic endometriosis
Pathologic processes causing an acute increase in intracranial pressure
Cerebral venous thrombosis
Subarachnoid hemorrhage
Valsalva maneuver
Drugs (Girolami et al[74], 2014; Peretz et al[68], 2016)
Antiplatelets such as non-steroidal anti-inflammatory drugs
Anticoagulants
Glucocorticoids
Antidepressants (e.g. selective serotonin reuptake inhibitors)
Antiepiletics (e.g. topiramate)
Antibiotics
Chemotherapeutic agents
Intoxication with lysergic acid diethylamide (LSD)
Herbal supplement (e.g. Gingko Biloba)
Trauma (Meyer et al[95], 2019; Peretz et al[68], 2016; Rani et al[84], 2018; Yeşilova et al[96], 2017; Zhang et al[16], 2004)
Sleep disorders (parasomnia) (Peretz et al[68], 2016)
Psychiatric disorders (Bezner et al[25], 2013; Karpukhina et al[48], 2020; Peretz et al[68], 2016)
Factitious disorder/factitious disorder by proxy
Psychogenic purpura
"Artistic bleeding"
Religious stigmata or culture-bound phenomenon (Bezner et al[25], 2013; Girolami et al[74], 2014)
Sweat gland disorders (e.g. chromhidrosis) (Jerajani et al[41], 2009)