Hair-thread strangulation syndrome in childhood: a systematic review

DOI: https://doi.org/https://doi.org/10.57187/smw.2023.40124

Daniela Djokic a, Gregorio P. Milanibc, Sebastiano A. G. Lavade, Gianluca Gualcoaf, Teresa Coriglianoaf, Mario G. Bianchettiaf, Camilla Lavagnog

a Pediatric Institute of Southern Switzerland, Ente Ospedaliero Cantonale, Bellinzona, Switzerland

b Pediatric Unit, Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, Milan, Italy

c Department of Clinical Sciences and Community Health, Università degli Studi di Milano, Milan, Italy

d Pediatric Cardiology Unit, Department of Pediatrics, Centre Hospitalier Universitaire Vaudois and University of Lausanne, Lausanne, Switzerland

e Heart Failure and Transplantation, Department of Pediatric Cardiology, Great Ormond Street Hospital, London, United Kingdom

f Faculty of Biomedical Sciences, Università della Svizzera Italiana, Lugano, Switzerland

g Pediatric Emergency Department, University Children’s Hospital Zurich, Zurich, Switzerland

Summary

INTRODUCTION: Hair-thread strangulation syndrome describes the constriction of a body part by a tightly wound hair or thread. This research aims to review the literature about this entity.

METHODS: A systematic review was performed to characterise hair-thread strangulation syndrome in subjects aged ≤16 years. This pre-registered review (PROSPERO ID: CRD42022363996) followed the PRISMA methodology.

RESULTS: Subjects with digital strangulation were significantly younger (median = 4.0 [interquartile range: 2.0–6.1] months; n = 143) than females with genital strangulation (9.0 [6.8–11] years; n = 36), males with genital strangulation (5.1 [1.9–8.0] years; n = 36), and subjects with non-digital and non-genital strangulation (24 [13–48] months; n = 11). Digital strangulation was followed by an amputation in five (3.5%) and a reconstructive surgical intervention in seven (4.9%) cases. Sequelae occurred in four (11%) cases after female genital strangulation: clitoris autoamputation (n = 2) and surgical removal of a necrotic labium minus (n = 2). Severe complications were observed in 14 (39%) cases with male genital strangulation: urethral fistula (n = 7), urethral transection (n = 2), and partial penile autoamputation (n = 5). A partial uvular autoamputation was observed in one case (9.0%) with non-digital and non-genital strangulation.

CONCLUSIONS: Early recognition and management are crucial to avoid sequelae or long-term care in hair-thread strangulation syndrome.

Introduction

Hair-thread strangulation syndrome describes the acute constriction of a body part, such as digits or external genitalia, by a tightly wound hair or thread in a young infant, a child, or an adolescent [1]. Narrow constriction reduces lymphatic and venous drainage, resulting in pain, swelling, oedema, and, if recognised late, ischemia [1]. This condition was first observed in the 19th century in a 4-week-old English boy with a painful penis swelling induced by a circumferential hair proximal to the swelling [2].

Hair-thread strangulation syndrome is either not mentioned or only briefly discussed in currently available paediatric textbooks. Its symptoms and presentation may be misdiagnosed, leading to delayed treatment and potentially adverse outcomes. A comprehensive literature review on this condition could help raise awareness among healthcare providers, ultimately improving diagnostic accuracy and patient outcomes. Therefore, we conducted a systematic review on this paediatric syndrome.

Methods

Search strategy

This literature review did not require Institutional Review Board approval. It was pre-registered at the International Prospective Register of Systematic Reviews (PROSPERO ID: CRD42022363996) and followed the 2020 edition [3] of the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines (PRISMA) according to the Joanna-Briggs Manual. The search was undertaken in the National Library of Medicine, Excerpta Medica, and Web of Science databases without restriction. The search strategy combined the following terms: “hair strangulation syndrome” OR “hair tourniquet syndrome” OR “hair-thread strangulation syndrome” OR “hair-thread tourniquet syndrome” OR “strangulation syndrome” OR “tourniquet syndrome”.

Articles available in Google Scholar or already known to the authors were also considered.The search was undertaken in February 2022 and repeated before submission (November 2022).

Following a first round of selection based on title and abstract, the full text of the selected articles was evaluated for eligibility.

Eligibility criteria and data extraction

Eligible articles were original reports and letters published in peer-review journals describing individual subjects aged ≤16 years with hair-thread strangulation affecting the digits, the external female genitalia, the penis, or other appendages. Cases with rubber band syndrome around the wrist were excluded [4]. Cases published only as abstracts and case series not detailing information on individual cases were also excluded.

The extracted data included demographics, the anatomic location, the source of strangulation (hair, threads, or elastic bands), the possible deliberate strangulation (by a caregiver or self-inflicted), the necessity of a reconstructive surgical procedure, and the outcome (development of long-term sequelae).

Two investigators independently (and in duplicate) performed the literature search, the selection of reports retained for analysis, and the data extraction in an unblinded manner with the support of a senior investigator. Any disagreements were discussed. One investigator entered the data into a pilot-tested database, and another verified them for accuracy.

Analysis

Pairwise deletion was used for missing data. Discrete variables are presented as counts and compared using Fisher’s exact test. Numerical variables are presented as medians and interquartile ranges or as box-and-whisker plots and compared using Kruskal–Wallis and Tukey’s tests [5, 6]. Two-sided P-values of <0.05 were considered statistically significant. Data were analysed using GraphPad Prism (version 9.5.1).

Results

Search output

The literature search identified 231 potentially relevant articles (figure 1). After removing irrelevant articles, 168 full-text articles were reviewed for eligibility. One hundred fifty-two articles [2, 7–157] describing individual cases of hair-thread strangulation syndrome published between 1951 and 2022 in English (n = 147), German (n = 2), French (n = 1), Spanish (n = 1), and Dutch (n = 1) were retained for the final analysis.

Figure 1Digital, female genital, male genital, and non-digital non-genital hair-thread strangulation syndrome in childhood. PRISMA flowchart of the literature search.

Fifty-eight articles reported cases from America, 45 from Asia, 39 from Europe, six from Oceania, and four from Africa. The articles provided information on 226 cases: 143 cases of digital strangulation [2, 7–90], 36 of female genital strangulation [22, 36, 49, 86, 91–119], 36 of male genital strangulation [7, 12, 24, 120–147], and 11 cases of non-digital and non-genital strangulation [148–157]. Characteristics of the individual cases are provided in the supplementary material (supplementary tables 1–4).

Clinical data

General data

Subjects with digital hair-thread strangulation syndrome (females: 50%; males: 50%) were significantly (p < 0.01) younger (4.0 [2.0–6.1] months) than females with genital strangulation (9.0 [6.8–11] years), males with genital strangulation (5.1 [1.9–8.0] years), and subjects (females: 58%; males: 42%) with non-digital and non-genital strangulation (24 [13–48] months; figure 2). Females with genital strangulation, males with genital strangulation, and subjects with non-digital and non-genital strangulation did not differ significantly in age.

Figure 2Age at presentation in paediatric patients with the various forms of hair-thread strangulation syndrome. The results are presented as box and whisker plots. Please note that the vertical axis is on a logarithmic scale. The age at presentation was significantly lower in children with digital strangulation than in children with strangulation of genitalia (p <0.005) or other appendages (p <0.01).

Twenty-nine (13%) of the 226 cases were explicitly reported as not induced by hairs but by threads arising from footed pyjamas, mittens, socks, underwear, and a nylon or elastic band. Three (1.3%) further cases were caused by hairs and threads.

The prevalence of cases not associated only with hairs was significantly higher (p = 0.0003) for digital strangulation (n = 29; 20%) than for the other forms of strangulation syndrome (n = 3; 3.6%).

A non-accidental cause of strangulation was suspected in 21 cases [12, 17, 36, 132, 133, 143]. Voluntary penile constriction was induced to manage enuresis in three cases [132, 133, 143].

Digital strangulation syndrome

Patients with digital hair-thread strangulation syndrome presented with irritability, crying, digital swelling, discolouration, and pain. About four fifths of the 143 cases of digital strangulation syndrome were observed on the toes (table 1). The female-to-male ratio did not differ significantly (p = 0.336) between infants with toe compared to finger strangulation syndrome. Cases with toe strangulation were slightly but significantly older than those with finger strangulation by 1.5 months on average (p = 0.034).

Table 1Fingers and toes with hair-thread strangulation syndrome in 136 paediatric cases.

Total Right Left
Fingers
Female:male ratio 0.6*
Age 2.4** [2.0–3.1] months
n 21 10 11
Thumb 7 3 4
Forefinger 3 1 2
Middle finger 6 4 2
Ring finger 5 2 3
Little finger 0 0 0
One finger 21 10 11
Two fingers 0 0 0
Three fingers 0 0 0
Toes
Female:male ratio 1.1
Age 4.0 [2.6–5.0] months
n 115 67 48
Big toe 3 2 1
Long toe 28 19 9
Middle toe 82 46 36
Ring toe 45 24 21
Little toe 8 5 3
One toe 79 47 32
Two toes 21 11 10
Three toes 15 9 6
*p = 0.343 and **p = 0.034 versus toe strangulation.

A detailed description of the affected digits was available for 136 (95%) of the 143 cases. Finger strangulation always affected only one finger. In contrast, toe strangulation affected at least two toes in about a third of cases. The little finger, the big toe, and the little toe were relatively rarely affected. Digital strangulation was induced by threads in eight (38%) of 21 cases with finger strangulation and in 12 (10%) of 115 cases with toe strangulation (p = 0.0037).

An amputation was performed in five (3.5%) cases: left thumb (n = 1), left middle toe (n = 1), right big toe (n = 1), right middle toe (n = 1), and right little toe (n = 1) [12, 26, 30, 53, 88]. A reconstructive surgical intervention was necessary in seven (4.9%) cases [12, 28, 29, 41, 70, 74, 81]. Removal of hair thread either mechanically or by applying a depilatory agent was followed by a full recovery in the remaining 131 (92%) cases.

Female genital strangulation syndrome

Hair-thread strangulation syndrome of the female external genital was reported in 36 cases, who presented with genital pain (sometimes associated with wide-base gait or painful micturition), vulvar swelling, or both genital pain and vulvar swelling. The strangulation was localised as follows (two structures were concurrently affected in two cases): clitoris (n = 19), labium minus (n = 16; bilateral in one case), labium majus (n = 2), and mons pubis (n = 1).

Sequelae were observed in four (11%) cases: total clitoris autoamputation in a 14-year-old adolescent, partial clitoris autoamputation in a 5-year-old girl, and surgical removal of a necrotic labium minus in a 6-year-old and a 10-year-old girl [95, 97, 105, 119].The remaining 32 cases recovered uneventfully.

Male genital strangulation syndrome

Hair-thread strangulation syndrome of the male external genital was reported in 36 cases. Young infants presented with irritability and crying, often associated with swelling; older children presented with pain and swelling.

Severe complications were observed in 14 (39%) cases [121, 123, 127, 128, 130, 134–136, 141, 143, 145–147]: urethral fistula (n = 7), urethral transection (n = 2), and partial penile autoamputation (n = 5).

Non-digital and non-genital strangulation syndrome

A non-digital and non-genital strangulation syndrome was noted in 11 cases (female-to-male ratio = 1.3): uvula (n = 5), neck (n = 3), circumvallate tongue papilla (n = 2), and tooth (n = 1). A partial uvular autoamputation was observed in one case [157].

Discussion

This literature review addressed the entire spectrum of conditions that may be caused by hair-thread strangulation in paediatric patients. While it found that they mainly affected the digits of young infants, they could also occur in older children. It also demonstrated that severe sequelae or the need for long-term care are possible (especially in cases with digital or penile strangulation). Furthermore, strangulation is sometimes non-accidental (e.g. intentional application to manage bedwetting). Finally, some cases were not caused by hairs but by threads or an elastic band.

Infants with hair strangulation often presented with a history of acute excessive crying [158, 159]. While the cause of acute and unexplained infantile crying is often benign, a careful history and physical examination remain the cornerstone of the evaluation. However, many cases present with local symptoms and signs such as pain, swelling, or discolouration. Detecting the encircling hair (which might be more challenging with light-coloured hairs) or thread is critical for diagnosis.

Because of the risk of ischemic damage, rapid management of hair-thread strangulation syndrome is crucial [160, 161]. Our review did not specifically address this issue. After taking a history and performing an examination to make the diagnosis, the first management step is currently appropriate analgesia [162]. Chemical depilatory agents, which break hairs (or threads) and subsequently lead to a weakening of the fibres and eventual dissolution, are currently the first-line treatment choice because they are generally painless and noninvasive [160, 161]. Mechanical release should be performed if strangulation release is not achieved after two attempts with a depilatory agent. Cases with strangulation near mucous membranes or with signs of ischemia should be treated with primary mechanical release [160, 161].

In infants, strangulation was caused mainly by maternal hair related to increased hair loss after birth [163]. It is assumed that hair may fall unnoticed into the bath water during routine care or diaper changing.

Our review had both weaknesses and strengths. Its main weakness was the low number of identified cases. The second weakness was its inability to address potential risk factors, such as socioeconomic status or ethnicity. Its most relevant strength was that it covered the entire spectrum of conditions that may be caused by hair-thread strangulation in paediatric patients. For example, two recent reviews focused explicitly on the hair-thread strangulation syndrome of the female genital tract or both the female and the male genital tracts [118, 164].

Conclusions

Early recognition and management are crucial to avoid sequelae or long-term care in hair-thread strangulation syndrome. Children and infants with unexpected acute onset of crying or local symptoms and signs such as pain, swelling, or discolouration require rapid medical evaluation. Healthcare professionals should be aware of this condition, with a particular focus on digital strangulation in infants and genital strangulation in children.

Data sharing statement

The data underlying this article can be shared upon reasonable request to the corresponding author.

Acknowledgments

Dr. Lava is the current recipient of research grants from Fonds de perfectionnement, Centre Hospitalier Universitaire Vaudois, Lausanne, Switzerland; Fondation SICPA, Prilly, Switzerland; Fondazione Dr. Ettore Balli, Bellinzona, Switzerland, Fondazione per il bambino malato della Svizzera italiana, Bellinzona, Switzerland and Frieda Locher-Hofmann Stiftung, Zürich, Switzerland.

Author contributions: Drs. Milani, Bianchetti and Lavagno conceptualized and designed the review. Drs. Djokic, Milani, Lava and Lavagno acquired the data and performed the analysis. Drs. Djokic, Bianchetti and Lavagno drafted the initial manuscript. All authors reviewed and revised the manuscript and approved the final manuscript as submitted.

Notes

Financial disclosure

This research received no external funding.

Potential competing interests

All authors have completed and submitted the International Committee of Medical Journal Editors form for disclosure of potential conflicts of interest. No potential conflict of interest related to the content of this manuscript was disclosed.

Dr. Gregorio P Milani

Fondazione IRCCS Ca’ Granda

Ospedale Maggiore Policlinico

IT-20122 Milan

milani.gregoriop[at]gmail.com

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64. Alruwaili N, Alshehri HA, Halimeh B. Hair tourniquet syndrome: successful management with a painless technique. Int J Pediatr Adolesc Med. 2015 Mar;2(1):34–7. 10.1016/j.ijpam.2015.02.003 

65. Baloch N, Atif M, Rashid RH, Hashmi PM. Toe-tourniquet syndrome: A rare potentially devastating entity. Malays Orthop J. 2015 Nov;9(3):55–7. 10.5704/MOJ.1511.008 

66. Baştuğ O, Korkmaz L, Korkut S, Halis H, Kurtoğlu S. Hair-thread tourniquet syndrome in a preterm baby. Turk Pediatri Ars. 2015 Dec;50(4):245–7. 10.5152/TurkPediatriArs.2015.1301 

67. El-Kehdy J, Nasr J, Bitar C, El-Habr C, Abi-Rached Megarbane H, Sammour R, et al. Don’t miss this blue toe: hair tourniquet syndrome. J Eur Acad Dermatol Venereol. 2015 Jan;29(1):191–2. 10.1111/jdv.12393 

68. Hussin P, Mawardi M, Masran MS, Ganaisan P. Hair tourniquet syndrome: revisited. G Chir. 2015;36(5):219–21. 10.11138/gchir/2015.36.5.219 

69. Kuiper JW, de Korte N. Hair thread tourniquet syndrome in a toe of an 18 mo old girl. World J Clin Cases. 2015 Apr;3(4):368–70. 10.12998/wjcc.v3.i4.368 

70. Pinkowsky GJ, Hennrikus WL. Toe tourniquet syndrome caused by cable tie. Pediatr Emerg Care. 2015 Jun;31(6):431–2. 10.1097/PEC.0000000000000461 

71. Pye S, Banks J, Pal C. Loom bands: a case of post-tourniquet syndrome. Arch Dis Child. 2015 May;100(5):481. 10.1136/archdischild-2014-307438 

72. van Meurs A, Korpershoek M, van Herwaarden-Lindenboom M. Female infant with toe swelling. Ann Emerg Med. 2016 Jun;67(6):785–96. 10.1016/j.annemergmed.2015.12.002 

73. Dunphy L, Verma Y, Morhij R, Lamyman M. Hair thread tourniquet syndrome in a male infant: a rare surgical emergency. BMJ Case Rep. 2017 Nov;2017:bcr2017221002. 10.1136/bcr-2017-221002 

74. Ojha S, Pandey VK, Garg G, Bansal R. “Missed band under the band”: don’t miss it! Indian J Pediatr. 2017 May;84(5):410–1. 10.1007/s12098-016-2275-z 

75. Rahul SK, Behera S, Suman P, Mohapatra S. Hair tourniquet syndrome. Med J DY Patil Univ. 2017;10(1):79–81. 10.4103/0975-2870.197919 

76. Tengattini V, Magnano M, Patrizi A, Neri I. Sudden swelling and redness of the toe. Clin Case Rep. 2017 Sep;5(11):1901–2. 10.1002/ccr3.1115 

77. Uygur E, Çarkçi E, Ünkar E. Can washing socks without flipping inside out cause hair tourniquet syndrome? A claim with two case reports. J Pediatr Orthop B. 2017 Mar;26(2):193–4. 10.1097/BPB.0000000000000348 

78. Yadav R, Rahul SK, Kumar V, Zaheer H, Thakur K, Agarwal A. Toe tourniquet syndrome. J Indira Gandhi Inst Med Sci. 2017;3(1):34–5. 

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80. Kesu Belani L, Leong JF, Narin Singh PSG, Abdullah S. Hair thread tourniquet syndrome in an infant: emergency exploration saves limbs. 2018;26(2):31-35. https://dx.doi.org/10.7748/en.2018.e1756. 

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82. Singh AP, Tanger R, Mathur V, Gupta AK. Hair tourniquet syndrome: A rare case report. Menoufia Med J. 2018;30(4):1260–1. 10.4103/mmj.mmj_111_17 

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133. Pahwa HS, Kumar A, Srivastava R, Kumar S, Goel A, Ahmad A. Partial penile amputation due to penile tourniquet syndrome in a child troubled with primary nocturnal enuresis—a rare emergency. Urology. 2013 Mar;81(3):653–4. 10.1016/j.urology.2012.11.012 

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135. Dikshit VK, Gupta RK, Kothari PR, Gupta AR, Kamble RS, Kesan KV. Near total penile amputation caused by hair tourniquet, managed with the URAGPI procedure. Afr J Urol. 2015;21(4):254–7. 10.1016/j.afju.2014.11.010 

136. Gazali ZA, Singal AK. Hair Thread Tourniquet Syndrome of Penis Causing Urethral Fistula. Indian Pediatr. 2015 Jun;52(6):538. 

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139. Sheikh Andalibi MS, Azarfar A, Esmaeeli M, Sheykh Andalibi N, Alizadeh A, Ravanshad Y. Penile hair tourniquet syndrome due to a coil of hair: first report in Iran. Nephrourol Mon. 2017;9(4):e59788. 10.5812/numonthly.59788 

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Appendix: Supplementary tables

Supplementary table 1Characteristics of 143 individuals affected by digital hair-thread strangulation syndrome. The country of publication is identified using the International vehicle registration code.

Case First author Year of publication Country Sex Age (months)
1 Alpert JJ [7] 1965 USA Male 2.0
2 Male 19
3 Curran JP [8] 1966 USA Male 2.5
4 Chonwn [9] 1967 CDN 12
5 Hill RH [10] 1967 CDN 5.0
6 Hack M [11] 1972 IL Female 0.8
7 Kerry RL [12] 1973 USA Male 4.0
8 Male 1.8
9 Male 5.0
10 Female 2.3
11 Male 1.5
12 Female 1.8
13 Female 3.5
14 Mack JW [13] 1976 USA Female 11
15 Male 5.0
16 Douglas DD [14] 1977 USA 10
17 Female 11
18 Miller PR [15] 1977 USA Female 15
19 Arato A [16] 1978 H 6.0
20 Narkewicz RM [17] 1978 USA Male 4.0
21 Male 3.0
22 Male 1.0
23 Mosely LH [18] 1979 NZ Female 2.0
24 Bérard J [19] 1981 F Male 6.0
25 Male 3.0
26 Male 3.0
27 Manfemaleredi SJ [20] 1981 USA Female 7.0
28 Male 3.0
29 Barton DJ [1] 1988 USA Male 5.0
30 Female 2.0
31 Female 4.0
32 Male 1.8
33 Male 144
34 Female 0.6
35 Collins AG [21] 1990 AUS Female 14
36 Beasley SW [22] 1993 USA 2.5
37 Liow RY [23] 1996 UK Female 18
38 Male 3.0
39 Vazquez Rueda F [24] 1996 E Female 2.0
40 Male 4.0
41 Male 2.0
42 Female 5.0
43 Male 3.0
44 Male 7.0
45 Conners G [25] 1997 USA Male 5.0
46 Lefemaleerink VJ [26] 1997 NL Male 4.0
47 Male 4.0
48 Female 2.0
49 Male 0.8
50 Debo HF [27] 1999 USA Male 2.0
51 Male 4.0
52 Sudhan ST [28] 2000 UK 5.0
53 Sunil TM [29] 2001 IND Female 8.0
54 Wang M [30] 2001 USA 1.5
55 Corazza M [31] 2002 I Female 72
56 Harris EJ [32] 2002 Female 5.0
57 Male 5.0
58 Smith AM [33] 2003 UK Male 7.0
59 Strahlman RS [34] 2003 USA Male 3.0
60 Karalezli K [35] 2004 B Male 3.0
61 Klusmann A [36] 2004 D 4.0
62 4.0
63 1.5
64 2.0
65 Mackey S [37] 2005 UK Female 3.0
66 Gutiérrez-Gómez C [38] 2006 MEX Female 2.0
67 Kurup HV [39] 2006 UK Male 3.0
68 Lohana P [40] 2006 UK 1.5
69 Mat Saad AZ [41] 2006 IRL Female 3.0
70 O'Quinn JC [42] 2006 USA Female 2.3
71 von Känel OE [43] 2006 CH Male 2.0
72 Anwar MU [44] 2007 UK Female 4.0
73 Haene RA [45] 2007 UK 2.8
74 Kumaravel S [46] 2008 IND 24
75 Singh V [47] 2008 UK Female 14
76 Al-Buainain H [48] 2009 KSA Male 3.0
77 Cutrone M [49] 2009 I Female 2.0
78 Female 2.0
79 Garcia-Mata S [50] 2009 USA Female 18
80 Male 5.0
81 Ünlü C [51] 2009 NL Male 5.0
82 Cevik Y [52] 2010 TR Female 4.0
83 Heather J [53] 2011 NZ Female 36
84 O'Gorman A [54] 2011 USA Male 3.0
85 Onyeama CO [55] 2011 USA Male 3.0
86 Sivathasan N [57] 2012 UK
87
88 Shankar R [56] 2012 IND Female 1.0
89 Wan WCY [58] 2012 CHN Female 2.0
90 Alvarez-Perez A [59] 2013 E Male 8.0
91 Hickey BA [60] 2013 UK Male 48
92 Au S [61] 2014 USA Male 6.0
93 Bizzotto N [62] 2014 I Female 2.0
94 Kamal NM [63] 2014 KSA Female 6.0
95 Male 6.0
98 Baloch N [65] 2015 PK Female 4.0
99 Bastug O [66] 2015 TR Female 0.5
100 El-Kehdy J [67] 2015 RL Female 6.0
101
102 Hussin P [68] 2015 MAL Female 3.0
103 Male 5.0
104 Kuiper JW [69] 2015 NL Female 18
105 Pinkowsky GJ [70] 2015 USA Female 84
106 Pye S [71] 2015 UK Female 48
Alruwaili N [64] 2016 KSA Male 6.0
0.5
107 van Meurs A [72] 2016 USA Female 4.0
108 Dunphy L [73] 2017 UK Male 2.0
109 Ojha S [74] 2017 IND Female 12
110 Rahul SK [75] 2017 IND Male 3.0
111 Tengattini V [76] 2017 I Female 8.0
112 Uygur E [77] 2017 TR Female 4.0
113 Female 6.0
114 Yadav R [78] 2017 IND Male 2.0
115 Booth J [79] 2018 UK Male 3.0
116 14
117 Ihara T [86] 2018 J Female 1.3
118 Sebaratnam D [81] 2018 E Female 24
119 Singh AP [82] 2018 IND Male 1.5
120 Agherbi F [83] 2019 MA
121 Aslantürk O [84] 2019 TR Male 2.0
122 Male 5.6
123 Female 1.9
124 Female 3.5
125 Female 0.6
126 Male 4.3
127 Male 2.0
128 Female 4.0
129 Male 1.0
130 Male 13
131 Male 0.8
132 Male 3.9
133 Female 6.0
134 Female 2.8
135 Female 2.6
136 Female 9.1
137 Basheer SM [85] 2019 BRN Female 5.0
138 Kesu Belani L [80] 2019 MAL Female 2.0
139 Azizalrahman A [87] 2020 KSA Female 3.0
140 McGrath RB [88] 2020 IRL Male 12
141 Shafemaleiq MA [89] 2020 MAL 2.0
142 Sutcu M [90] 2020 TR Female 2.0
143 Male 0.5

Supplementary table 2Characteristics of 36 female subjects affected by genital hair-thread strangulation syndrome. The country of publication is identified using the International vehicle registration code.

Case First author Year of publication Country Age (years)
1 Chapman HL [91] 1968 CDN 9
2 Summers JL [93] 1973 USA 11
3 Webley JA [94] 1973 USA 7
4 Press S [92] 1980 USA 8
5 Beasley S.W [22] 1991 AUS 8
6 Rich MA [95] 1999 USA 5
7 Sylvestzrak MS [96] 2000 USA 5
8 Kuo JH [97] 2001 USA 14
9 Golshevsky J [100] 2004 AUS 14
10 Klusmann A [36] 2004 D 12
11 Bacon JL [99] 2005 USA 13
12 Osterhoudt KC [101] 2005 USA 8
13 Alverson B [102] 2007 USA 9
14 Serour F [98] 2007 IL 4
15 Cutrone M [49] 2009 I 0.5
16 Pomeranz M [103] 2009 IL 11
17 Stock C [104] 2012 USA 12
18 Bannier MA [105] 2013 NL 6
19 Dua A [106] 2013 USA 12
20 Lee JH [107] 2013 UK 11
22 Ozcakir E [108] 2014 TR 9
22 Panagidis A [109] 2015 GR 14
23 10
24 Parlak M [110] 2015 TR 6
25 Zimmerman LN [111] 2015 USA 5
26 Zhong N [112] 2017 USA 10
27 Ihara T [86] 2018 J 14
28 Roman E [113] 2018 AUS 12
29 Erikci VS [114] 2019 TR 8
30 7
31 9
32 Imam AS [115] 2019 USA 6
33 Diaz-Morales O [116] 2020 E 10
34 Minodier P [117] 2020 F 5
35 Chawla RK [119] 2021 USA 10
36 Adjei N [118] 2021 USA 11

Supplementary table 3Characteristics of 36 male subjects affected by genital hair-thread strangulation syndrome. The country of publication is identified using the International vehicle registration code.

Case First author Year of publication Country Age (months)
1 DeWeese JA [120] 1952 USA 22
2 Alpert JJ [7] 1965 USA 13
3 Farah R [121] 1973 USA 132
4 Kerry RL [12] 1973 USA 2
5 4
6 10
7 Haddad FS [122] 1982 USA 24
8 24
9 McClure WJ [123] 1985 USA 60
10 Sheinfeld J [124] 1985 USA 84
11 Mariani PJ [125] 1986 USA 60
12 Vazquez Rueda F [24] 1996 E 24
13 48
14 Pantuck AJ [126] 1997 USA 36
15 Bangroo AK [127] 2005 IND 120
16 Chaware SM [128] 2006 IND 84
17 Dar NR [129] 2007 PK 60
18 Hussain HM [130] 2008 AUS 72
19 Okeke LI [131] 2008 NGR 108
20 Basner A [132] 2012 D 2
21 Pahwa HS [133] 2013 IND 96
22 Jesus LE [134] 2014 BR 120
23 Dikshit VK [135] 2015 IND 120
24 Gazali ZA [136] 2015 IND 72
25 Zengin K [137] 2015 TR 96
26 Sekhon V [138] 2017 IND 84
27 Sheikh Andalibi MS [139] 2017 IR 54
28 Arshadi H [141] 2019 IR 108
29 Degheili JA [142] 2019 RL 48
30 Ozcift B [140] 2019 TR 48
31 Bouassida K [143] 2020 TN 108
32 Rawls WF [144] 2020 PK 24
33 17
34 Loloi J [147] 2021 USA 96
35 Patterson JW [145] 2022 USA 0.5
36 Rampersad B [146] 2022 TT 24

Supplementary table 4Characteristics of 11 individuals affected by non-digital and non-genital hair-thread strangulation syndrome. The country of publication is identified using the International vehicle registration code.

Case First author Year of publication Country Sex Age (months)
1 McNeal RM [148] 1987 USA Male 13
2 Krishna S [149] 2003 USA 3
3 Chegwidden HJ [150] 2005 USA Female 11
4 Schneider K [151] 2013 USA Female 84
5 Flores JR [152] 2014 MEX 24
6 48
7 Alghamdi F [153] 2018 KSA Female 48
8 Wong K [154] 2019 USA Male 13
9 Chan LYF [156] 2020 HK Male 21
10 Stansfield J [155] 2020 UK 9
11 Leighton J [157] 2021 UK Female 36