Male genital self-mutilation: a systematic review of psychiatric disorders and psychosocial factors

    Male Genital Self-Mutilation: A Systematic Review of Psychiatric Disorders and Psychosocial Factors Thomas A. Veeder M.D., Raphael J. Leo M.A., M.D. PII: DOI: Reference:

S0163-8343(16)30145-1 doi: 10.1016/j.genhosppsych.2016.09.003 GHP 7139

To appear in:

General Hospital Psychiatry

Received date: Revised date: Accepted date:

18 June 2016 8 September 2016 9 September 2016

Please cite this article as: Veeder Thomas A., Leo Raphael J., Male Genital SelfMutilation: A Systematic Review of Psychiatric Disorders and Psychosocial Factors, General Hospital Psychiatry (2016), doi: 10.1016/j.genhosppsych.2016.09.003

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ACCEPTED MANUSCRIPT Male Genital Self-Mutilation: A Systematic Review of Psychiatric Disorders and Psychosocial Factors

Thomas A. Veeder, M.D.

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Assistant Professor Department of Psychiatry

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Oregon Health and Science University, Portland, Oregon

Raphael J. Leo, M.A., M.D.

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Associate Professor

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Department of Psychiatry Jacobs School of Medicine and Biomedical Sciences

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State University of New York at Buffalo, Buffalo, New York

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Running head: Genital Self-Mutilation among Males

Corresponding Author Information: Thomas A. Veeder, MD, Department of Psychiatry, Oregon Health & Science University, 3181 SW Sam Jackson Park Road, UHN 80, Portland, OR 97239, Phone: 503-494-8144, Email: [email protected] Article Data: Number of Figures: 1; Number of Tables: 3; Number of Supplementary Tables: 3; Word Count: 4572; Abstract Word Count: 200

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ACCEPTED MANUSCRIPT Abstract Objective: To identify psychiatric diagnoses and psychosocial factors associated with intentional male genital self-mutilation (GSM) of specific injury subtypes.

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Methods: A search of MEDLINE, EMBASE, PsycINFO, PubMed, Web of Science, and CINAHL for cases of GSM was conducted until December 2015, based on genital selfmutilation and related terms. Cases were examined for injury sub-type, psychiatric diagnosis, and psychosocial factors. Chi-square analyses were employed to determine differences in frequency of such factors across injury subtypes.

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Results: Data were obtained from 173 cases, genital mutilation (n=21); penile amputation (n=62); castration (n=56); and combined amputation/castration (n=34). Common psychiatric disorders included schizophrenia spectrum (49%); substance use (18.5%); personality (15.9%); and gender dysphoric disorders (15.3%). Chi-square analyses revealed that schizophrenia spectrum disorders occurred significantly more often among auto-amputates as compared with self-castrators or mutilators. Gender dysphoria occurred significantly more often among selfcastrators than auto-amputates.

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No significant differences emerged regarding psychosocial factors across GSM subtypes. However, associations were observed between psychosocial factors and psychiatric diagnoses. Although, altogether not commonly reported, experiential factors were reported in 82% of psychotic individuals. Treatment inaccessibility was noted among 71% of gender dysphorics engaging in auto-castration.

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Conclusion: Clinicians must consider the diverse range of psychiatric disorders and psychosocial factors underlying GSM.

Key Words: Genital mutilation; self-amputation, self-castration, Klingsor syndrome

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ACCEPTED MANUSCRIPT Male Genital Self-Mutilation: A Comprehensive Review of Psychiatric Disorders and Psychosocial Factors

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1. Introduction

Intentional self-mutilation, i.e., deliberate and direct physical self-injury, has long been

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observed to occur in a variety of cultural and psychopathologic states [1]. Often classified by the

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degree of injury, the most common forms include those which involve superficial to moderate tissue injury of low lethality such as cutting and stereotypic actions, e.g., head banging. Major

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self-mutilation, i.e., involving major trauma and tissue injury, is less common; genital selfmutilation (GSM) has been among the most dramatic examples of the latter.

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Intentional GSM constitutes catastrophic events that are often, but not solely,

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encountered within the context of severe mental illness. Although described in both men and women, GSM is thought to occur predominantly among men [2]. Cases of genital self-injury

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have been described in a number of ethnic and racial groups, religious contexts and have been

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culturally or even legally sanctioned [3-5]. The first case of male GSM [6] attributable to psychopathology was published in 1901, describing an auto-castrate who believed that his lack of success in life was due to the presence of his sexual organ. Several cases have since been reported in the psychiatric and urologic literature. Although considered to occur infrequently, the prevalence rates of GSM are difficult to estimate as many cases go unreported [7,8]. The contemporaneous factors associated with GSM have remained elusive [5]. GSM has been described through mostly a thin narrative, gleaned largely from case reports and small case series. Such reports provide incomplete descriptions of the diagnostic conditions and 3

ACCEPTED MANUSCRIPT psychosocial factors underlying male GSM. In a seminal publication [9], based on four cases, several factors were postulated to account for this behavior, e.g., absence of a competent male

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figure for identification, over-controlling mothers who encouraged their sons’ masochistic

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behaviors, and unresolved sexual conflicts, among others. These conclusions reflected the prevailing views of sexuality, sexual pathology and personality development rooted in

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psychoanalytic perspectives of the time, and influenced subsequent conceptualizations

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surrounding the reporting of male GSM for several years [10-13]. Because of concerns that such perspectives have become outmoded, others have suggested the need to explore the evolving

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factors and other influences associated with GSM in light of more contemporary views of psychopathology and psychosocial mediators [14]. For example, since homosexuality has long

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been removed from the Diagnostic and Statistical Manual of Mental Disorders (DSM) as

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reflective of a form of psychopathology and societal attitudes regarding homosexual expression have changed, it is anticipated that homosexual guilt would become less of a factor influencing

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one to engage in GSM [14].

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Controversy attends the factors reportedly underlying GSM. Some have contended that GSM is an expression of suicidal intent [8,15], or that the self-injury represented a form of focal suicide, i.e., presumably eliminating part of one’s self as a substitute for the whole [16], whereas others suggested that it was a non-suicidal self-injury [17,18]. Experiential factors underlying the self-injury have reportedly varied as well and have included religious delusions [13,19-21] (previously referred to as Klingsor syndrome [19-21]); guilt related to sexual impulses or activity [22,23]; shame [10]; and somatic preoccupations [15]. Two subsequent reviews [14,24] identified that perpetrators of genital self-injury were primarily suffering from psychosis or transsexualism, however, the methodology undertaken to identify cases from which data were 4

ACCEPTED MANUSCRIPT gathered had not been clearly defined. In both reviews, an attempt was made to correlate severity of self-inflicted injury to underlying psychopathology, but no association between injury

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severity and diagnosis emerged. The aforementioned reviews have treated all forms of GSM equally. As with the broader

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rubric of self-mutilation, GSM can range extensively from superficial cuts to partial/total removal of the penis and/or testicles [25]. It may be presumptuous to treat such a diverse array

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of self-injurious actions as unitary; doing so may fail to identify the diversity of

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psychopathological, experiential and precipitating factors that may underlie these acts. Herein, a comprehensive review of cases of GSM in the literature was undertaken; the intent was to

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synthesize the existing data to identify psychiatric diagnoses and describe the major psychosocial factors associated with various forms of GSM.

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2. Methods

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The goal of the study was to systematically review the available literature related to male

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GSM. Case reports and case series were identified from the psychiatric and urological literature. Instead of treating all forms of male GSM homogenously, identified cases were separated according to the type of self-injury. Unlike previous reviews, it was hypothesized that psychiatric diagnoses associated with different categories of genital injury vary. In addition, attempts were made to characterize the common experiential and precipitating factors associated with the different classes of GSM. A comprehensive search of MEDLINE (1946-current), EMBASE (1946-current), PsycINFO (1967-current), PubMed (1977-current), Web of Science (1900-current) and CINAHL (1946-current), was conducted until December 2015. The search strategy was based on title, 5

ACCEPTED MANUSCRIPT abstract, and MeSH terms, for genital self-mutilation OR genital self-amputation OR penile selfmutilation OR penile self-amputation OR autocastration OR auto-castration OR self-castration

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OR Klingsor syndrome OR emasculation OR self- emasculation OR major self-mutilation.

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Limits were placed on those articles focusing on humans and published in the English language. Articles generated by the searches were screened by title and abstract review. The search

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strategy employed can be found in Figure 1. Subsequently, full text review of publications

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---------------------------------

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Insert Figure 1 about here

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--------------------------------was conducted; a manual review of relevant article reference lists was also conducted for eligible

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cases.

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Individual cases were selected for the review if they included: (1) case descriptions of men who engaged in some form of non-accidental GSM, (2) some description of a discernable

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psychiatric diagnosis, and (3) some combination of experiential and/or precipitating factors temporally linked to the self-injury. It was recognized, however, that there would be variability in the degree to which the latter were described in the available case reports, and as such, the presence of all of these was not essential for inclusion. Case histories were subcategorized as reflecting self-amputation (defined as removal of the glans penis), castration (defined as self-removal of one or both testes) and genital mutilation (defined as any other type of genital self-injury that did not result in amputation or castration). Cases in which both amputation and castration co-occurred were classified as a fourth category.

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ACCEPTED MANUSCRIPT 2.1 Data Extraction The cases were reviewed for psychiatric history and concurrent psychiatric illness at the

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time of self-injury. The original diagnosis was converted to an equivalent DSM-5 diagnosis whenever applicable, e.g., transsexualism and gender identity disorders were both categorized as

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gender dysphoria. In such cases when the diagnosis was not explicitly stated, a diagnosis of

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unknown was recorded and no other diagnosis was inferred from the available data. Whenever possible, other facets of the clinical history were extracted from the case

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histories based on psychosocial factors reported in previous literature [9-13]. Case histories were examined for psychosocial factors deemed to be linked with the GSM, including experiential

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factors (e.g., delusional content, hallucinations/perceptual disturbances, religious preoccupation, guilt, perceived need for atonement, and suicidality); and precipitating factors (e.g., perceived

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rejection, changes in one’s social support, access to health care, acute substance intoxication

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and/or withdrawal).

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The authors independently examined the studies to determine eligibility, and conflicts were resolved by discussion. Because this study consisted of a review of published, publicly available research data, institutional review board approval was not required. 3. Results As illustrated in Figure 1, the initial database search yielded 398 articles after duplicates were removed (MEDLINE = 73, EMBASE = 50, PsycINFO = 125, PubMed = 0, Web of Science = 138, CINAHL = 0, Other = 12). Of these, 162 had titles and/or abstracts suggesting they might be eligible for inclusion (MEDLINE = 48, PsycINFO = 66, EMBASE = 24, Web of Science = 12, Manual = 12); the remaining articles were deemed to be unrelated, e.g., addressing 7

ACCEPTED MANUSCRIPT non-genital self-mutilation, or focused on urologic management of genital injury. From this pool, 148 articles were examined by the two authors as they specifically addressed one of the

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three forms of GSM. Articles were excluded for several reasons, e.g., failing to contain case

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descriptions [1,5,17,20,26-45], focusing on a complication of mutilation [46], or the case involved accidental injury [47]. Articles presenting the same case information from previously

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published work were included only once in this review [48,49]. Most of the retained articles

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(74/121; 61.2%) were derived from psychiatric journals; the remaining (47/121; 38.8%) were

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obtained from journals pertaining to urology, medicine, or other sub-specialties.

3.1 Sample Characteristics

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One hundred seventy-three cases of genital self-injury were assessed [2,9-14,18,21,23-

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25,49-157]; 12.1% (n = 21) were classified as having focused on genital mutilation [49,51,52,5860,75,79,93,97,98,100,127,133,137,138,142,151,152,155]; 35.8% (n = 62), penile amputation

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[2,9,14,23-25,50,52,56,58,59,63,67,70-72,76,77,80,84,86,87,89,95,96,102,104,106,107,117,118,

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121,124,128,129,131,134,141,142,144-146,148,150,151,154-156]; 32.4 % (n = 56), castration [9-11,13,23,49,53-56,59,62,64,66,69,73,74,77,78,82,85,88,91,92,99,101,103,106,109-112,115, 116,119,120,122,123,125,126,130,136,137,139,143,147,148,157]; and 19.7 % (n = 34), combined amputation/castration [2,10,12,18,21,58,61,64,65,68,74,77,81,83,88,90,93,94,105, 106,108,109,113,114,118,135,140,149]. The mean age (+ standard deviation) of the entire sample was 36.3 years + 12.9; and for the mutilation, amputation, castration and combined amputation/castration groups, these were 38.2 + 15.1, 33.9 + 12.1, 35.8 + 12.1, and 39.2 + 14.0, respectively. There were no significant differences in the ages between the four groups, F(3,167) = 1.23, p = 0.30. Other demographic data, e.g., ethnicity, religious affiliation, marital and

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ACCEPTED MANUSCRIPT socioeconomic status, as well as developmental factors, were inconsistently reported in published reports and, therefore, were not amenable to meaningful summary here. Other features

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of the cases across the four categories are summarized in detail below. 3.2 Psychiatric Diagnostic Characteristics

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Psychiatric diagnoses identified among the subcategories of GSM are summarized in

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Table 1. Psychiatric disorders were either not reported/unknown in 9.2% (16/173) of the

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------------------------------Insert Table 1 about here

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cases [49,57,60,74,81,85,86,91,92,106,114,133,134,144]. Among the 157 cases in which psychiatric disorders were reported, the most commonly identified included 49% (77/157) with

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schizophrenia spectrum disorders [2,9-14,18,21,23-25,49,52,58,59,61,63,68,70,71,77,78,80,

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83,84,86,87,89,94,96,99,105-109,113,115-118,128,131,136,140,142,143,148-151,155,156]; 18.5% (29/157) with substance use disorders [2,53,55,58,62,65,68,69,74,77,79,82,93-95,106, 109,115,116,124,131,149,155,156]; 15.9% (25/157) with personality disorders [2,50,55,67,73, 76,77,82,84,88,97,100,104,112,118,128,137,138,146,152]; and 15.3% (24/157) with gender dysphoria [56,64,67,75,77,79,88,101,102,103,105,111,123,125,130,137,139,147]. Psychosis was identified among 54.8% (86/157) of cases; a majority 89.5% (77/86) were diagnosed with a schizophrenia spectrum disorder, relatively few were diagnosed with depression [72,119,120] (3.5%; 3/86), substance-induced psychosis [2,62,65,124] (4.7%; 4/86), and bipolar disorder and neurocognitive disorder (1.2%; 1/86, respectively). Individuals within 9

ACCEPTED MANUSCRIPT the schizophrenia spectrum disorder encompassed unspecified psychosis [14,84,116,131] (n = 4); brief reactive psychosis [11,49] (n = 2); schizoaffective disorder [80,107,108,117] (n = 4);

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schizophrenia [2,9-13,18,21,23-25,52,58,59,61,63,68,70,71,77,78,83,86,87,89,94,96,99,105,

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106,109,113,115,118,128,131,136,140,142,143,148-151,155,156] (n = 65); and schizophreniform disorder [155,156] (n = 2). Among the four categories of GSM employed

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here, psychotic disorders were reported among mutilation [52,58,59,142,156] (5/21; 23.8%);

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amputation [2,9,14,23-25,52,58,59,63,70-72,80,84,86,87,89,96,107,117,124,128,131,142,148, 150,151,155, 156] (39/62; 62.9%); castration [9-11,13,23,49,59,62,77,78,99,109,115,116,119,

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120,136,143,148] (21/56; 37.5%); and combined amputation/castration [2,10,12,18,21,61,65,

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68,83,94,105,106,108,109,113,118,140,149] (19/34; 55.9%) groups. Regarding the schizophrenia spectrum disorders specifically, chi-square analysis revealed

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significant differences among the four injury sub-types, χ2 = 12.67 (df = 3; p = 0.005). These

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disorders were most common among cases in which amputation (either alone or in combination with castration) was self-inflicted; a significantly greater proportion of individuals were

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diagnosed with schizophrenia spectrum disorders within the amputation group versus the mutilation (χ2 = 6.91; df = 1; p = 0.009) or castration groups (χ2 = 8.36; df = 1; p = 0.004) and among those within the combined amputation/castration group versus the mutilation (χ2 = 4.19; df = 1; p = 0.04) or the castration groups (χ2 = 4.06; df = 1; p = 0.043). Other sub-group comparisons were not significantly different. Substance use disorders were the second most frequently encountered psychiatric condition. Chi-square analysis failed to reveal significant differences among the mutilation [58,79,93,156], amputation [77,95,124,131,155], castration [53,55,62,69,77,82,106,109,

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ACCEPTED MANUSCRIPT 115,116], and combined amputation/castration [2,65,68,74,93,94,106,149] groups with regard to the proportions of individuals carrying these diagnoses, (χ2 = 6.35; df = 3; p = 0.09).

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The proportions of cases for whom personality disorders were diagnosed within the mutilation [97,100,138,152], amputation [50,67,76,77,84,104,118,128,146], castration

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[55,73,77,82,88,112,137], and combined amputation/castration [2,77] groups are summarized in the Table. Chi-square analysis failed to reveal significant differences across the four injury

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groups, (χ2 = 3.13; df = 3; p = 0.37).

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Gender dysphoria [56,64,64,67,75,77,79,88,101,102,103,105,111,123,125,130,137, 139,147] was present in 15.3% of cases; it was diagnosed slightly less frequently than personality

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disorders. Chi-square analysis revealed significant differences across the four groups, χ2 = 14.22 (df = 3; p = 0.003). Gender dysphoria appeared to be most common among cases in which

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castration was self-inflicted; sub-analyses revealed that a significantly greater proportion were

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diagnosed with gender dysphoria within the castration [56,64,77,88,101,103,111,123,125,130,

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137,139,147] versus the amputation group [67,102] (χ2 = 13.62; df = 1; p = 0.0002) as well as the combined amputation/castration group [64,77,88,105] as compared with the amputation group, (χ2 = 4.37; df = 1; p = 0.037). Other sub-group comparisons were not significantly different. Mood disorders were relatively uncommonly reported. Depression [54,66,72,90,106,109, 119,120,126,127,129,130,135,153] comprised 8.9% (14/157) and bipolar disorder [23,58] 1.3% (2/157) of the cases reviewed. 3.3 Psychiatric Diagnostic Comorbidities More than one primary psychiatric disorder was noted in 17.8% (28/157) of the cases in which a diagnosis was identified. Substance use and personality disorders were conditions often 11

ACCEPTED MANUSCRIPT recognized as secondary conditions complicating the presentation of men engaging in GSM. For 48.3% (14/29) of individuals, a substance use disorder was the sole diagnosis [53,58,62,65,68,74,

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93-95,106,109,124,131,155]. The remaining 51.7% (15/29) of cases had comorbid psychiatric

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conditions including 42.9% (6/14) with schizophrenia spectrum disorders [2,106,115,116,149, 155]; 35.7% (5/14), personality disorders [2,55,77,82]; 14.3% (2/14), gender dysphoria [77,79];

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and 21.4% (3/14) with miscellaneous conditions, e.g., anxiety or depression [93,106,109]. Sole

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diagnoses of substance use disorders included alcohol [58,62,68,69,74,106,109,131] (53.3%; 8/15); cannabis [53,65,93] (20%; 3/15); stimulant [93,124,155] (20%; 3/15); and hallucinogen

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[62] (6.7%; 1/15) use disorders.

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A personality disorder was the sole diagnosis for 48% (12/25) of cases [50,53,76,77,97, 100,112,118,128,138,146,152], i.e., 41.7% (5/12) with Cluster A and 58.3% (7/12) with Cluster

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B personality subtypes. The remaining 52% (13/25) had identifiable comorbid psychiatric

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conditions including 53.8% (7/13) with gender dysphoria [67,77,88,137]; 38.5% (5/13), substance use disorders [2,55,77,82]; 15.4% (2/13), schizophrenia spectrum disorders [2,84]; and

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7.7% (1/13) with mild mental retardation [104]. 3.4 Psychosocial Factors

Experiential and precipitating factors associated with GSM were inconsistently reported. Generally, these factors were reported in cases published within psychiatric journals more frequently (79.7% and 44.6%, respectively) than for those published within non-psychiatric journals (44.7% and 27.7%, respectively). 3.4.1 Experiential Factors

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ACCEPTED MANUSCRIPT Experiential factors were identified in the histories of 62.4% (108/173) of cases, see

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Table 2; 53% (57/108) had more than one identifiable experiential factor at the time of the self-

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injury. Chi-square analysis failed to reveal significant differences among the four groups with

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regard to the proportion with which any one of the aforementioned experiential factors were identified.

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Psychiatric diagnoses associated with each of the aforementioned experiential factors are available in a supplemental table on-line. Few notable associations between experiential factors

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and psychiatric diagnosis appeared to emerge. Most of these were reported in association with

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psychosis, i.e., 80.2% (69/86) of individuals with some form of psychosis were determined to have displayed one of the aforementioned experiential factors, and 55.8% (48/86) had more than

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one of the experiential factors. Additionally, although suicidal ideation/intent was encountered in 15% (26/173) of men engaging in GSM, 31% (8/26) had these in association with psychotic symptoms, i.e., delusions and/or hallucinations [2,14,84,108,136]. Thirty-five percent (9/26) had ended their lives, four of whom were diagnosed with schizophrenia spectrum disorders [2,62, 126,136]; three, with major depression [126,129,135]; and two with unspecified diagnoses [81,106]. It was noteworthy that none of the suicidal individuals had gender dysphoria. 3.4.2 Precipitating Factors Precipitating factors were identified in the histories of 42.2% (73/173) of the cases, see Table 3. Chi-square analyses failed to reveal significant differences among the four groups with 13

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regard to the proportion with which any one of the aforementioned precipitating factors were

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identified.

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Psychiatric diagnoses associated with the aforementioned precipitating factors are summarized in a supplementary table online. A diagnosable substance use disorder was present

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among 78.6% (22/28) of those reporting an acute substance-related precipitating factor [2,53,55,58,62,65,68,69,74,79,82,93-95,106,109,115,116,149,156]. Additionally, 71% (10/14)

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[64,77,88,111,125,130,137,139].

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of those identified with lack of access to treatment carried a diagnosis of gender dysphoria

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4. Discussion

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Although an extensive number of case reports of male GSM have been published since the initial case [6] documented in 1901, there have been few advances in identifying the characteristics of men attempting to divest themselves of part or all of their external genitalia. Previous attempts at delineating characteristics of such individuals engaging in GSM were limited by small samples upon which the reviews were based [9,55,104,127]. With an overreliance on sexuality, gender development and sexual dysfunction that prevailed at the time of publication, the theoretical conceptualizations which formed the basis for characterizing individuals performing GSM have since been deemed to be outmoded [9,84]. We systematically compiled the most comprehensive review to date of published reports focused on men engaging in such behaviors. 14

ACCEPTED MANUSCRIPT Overall, the presence of some form of psychopathology elevates the probability of GSM; rarely were cases reported in which the individual did not have a psychiatric illness [57,60,85,91,

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92,114]. In the present review, nearly half of cases suffered from psychosis. Among these, a

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majority carried a schizophrenic spectrum disorder diagnosis. Although generally in line with estimates reported in previous research, the rate of psychosis among GSM cases herein is

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somewhat more conservative. Psychiatric disorders accompanying GSM in previous reviews

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may have been unreliably delineated, e.g., gender disordered individuals were assumed to be delusional and may have been preferentially diagnosed as psychotic [24,67,105], inflating rates

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of psychosis while underestimating rates of gender dysphoria [24].

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In contrast to previous reviews indicating that transsexualism, i.e., gender dysphoria, was the next common condition associated with GSM among non-psychotic individuals; we found

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that substance use disorders, personality disorders, and then gender dysphoria, were the

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conditions next most commonly encountered. Substance use and personality disorders may have been underestimated or overlooked in prior reviews as those reports were based on the patient’s

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primary disorder [14,24].

The present investigation demonstrates that GSM, like the broader rubric of selfmutilation, is a heterogeneous form of self-injury. Notably, statistically significant differences in the psychiatric diagnoses associated with self-inflicted injuries of different types were identified. Individuals who are unduly influenced by a schizophrenic spectrum disorder tended to perform self-amputation whereas those experiencing gender dysphoria tended to engage in self-castration. Personality and substance use disorders were often comorbid with other psychiatric conditions, and no particular association with GSM subtype was found.

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ACCEPTED MANUSCRIPT The psychosocial factors underlying GSM have been less well understood. Although distinctions could not be made for psychosocial factors associated with type of injury incurred,

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some trends became apparent for those with diverse psychiatric disorders. Experiential factors

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were predominantly reported for those with psychotic disorders. Such factors were less often reported for individuals self-inflicting other types of genital injury, and seldom for persons with

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non-psychotic disorders. These data suggest that severe forms of GSM, such as amputation,

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appear to occur among individuals who manifest more symptoms and greater severity of

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psychopathology.

In addition, the present data highlight the significant role of substance use, both as a

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psychiatric disorder and as a precipitating factor, increasing the vulnerability toward GSM, a characteristic which had been largely ignored in previous reviews [9,14,24]. These data are

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consistent with trends observed among individuals engaging in a variety of self-injurious

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behaviors generally [29,158], and may have contributed to disinhibition or a dissociative state

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thereby enabling individuals to engage in self-injury [158]. The relationship between self-injurious behaviors and suicide is complex; empirical research has suggested that a substantial proportion of those engaging in some form of selfinjurious behavior entertain thoughts of suicide at the time of injury and more than one-half have made at least one attempt at suicide [159,160]. In the present review, suicidal ideation/intent was encountered more often than had been reported in an early review of GSM cases [24], and was notably encountered among individuals with schizophrenia spectrum disorders and psychotic mood disorders. For these individuals, the suicidal ideation co-occurred in the context of delusions, hallucinations and pathological guilt preoccupations. The fact that individuals would engage in significant self-injury can be a harbinger of levels of distress that, if sub-optimally 16

ACCEPTED MANUSCRIPT treated and addressed, can ultimately lead to significant morbidity and even death. In contrast to individuals with psychosis, none of the individuals with suicidal ideas/intent had gender

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dysphoria. It is of note that many persons with gender dysphoria tended to perform self-

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castration in a premeditated manner with low suicidal intent, often related to a lack of, or refusal

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for, gender confirmation surgery [64,91,101,111,123,125,130,137,139,147]. In the course of conducting this review, we discovered reports suggesting that there is a

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sub-population of non-psychotic and non-gender dysphoric men who desire castration. Although

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not encountered often in the present review, these individuals, referred to as “eunuchs” [91,92,161,162], seek castration for cosmetic concerns, to reduce sexual desire, or because the

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prospect of castration was sexually arousing [162]. Such persons have reportedly turned to internet sites dedicated to the subject [163,164] providing a forum for the exchange of ideas

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regarding how one can pursue self‐castration, castration by nonmedical professionals, or

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self‐inflict testicular damage so that urological castration becomes inevitable [92]. It is uncertain how many of these persons may, on further evaluation, have another as yet unrecognized

5. Limitations

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disorder, e.g., a personality disorder or paraphilic disorder.

In previous work, attempts were made to correlate psychopathologic conditions with GSM based on injury severity [14,24] but no association emerged. The parameters of injury severity had not been explicated and may have been subjectively determined. In order to avoid subjectivity, classification of injury subtype was instead employed here. It is conceivable that classification of self-injury subtype may have been arbitrary and limited our ability to adequately distinguish persons engaging in GSM with regard to psychiatric diagnosis and/or psychosocial

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ACCEPTED MANUSCRIPT factors. For example, although persons within the mutilation category might have intended a more extensive injury, they may have been impeded from executing the plan for a variety of

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reasons such as pain, misjudgment of the “effectiveness” of the measures used, or interruption by

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others. Yet, determining intended versus actual injury outcome was not readily decipherable in the cases reviewed here, and would, like the problems encountered in rating injury severity, have

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been subjectively based.

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Second, publication bias may have limited the scope of data extraction to cases involving

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extreme types of injury. Cases warranting publication may have been of a more extreme nature and may only represent a subset of individuals engaging in GSM, whereas less dramatic injuries

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would not receive the same level of attention. Hence, it is unsurprising that cases involving mutilation may have been underrepresented here, as compared with those involving amputation

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and/or castration.

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Third, this review was limited by missing data, e.g., experiential and precipitating factors,

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particularly for cases derived from non-psychiatric journals. The latter cases emphasized urologic and surgical procedures to remediate sustained injuries; it is unsurprising that many of the psychosocial variables that we intended to quantify went unreported as such points would have been superfluous to the intended focus of the journals within which those cases were published. Lastly, the diagnostic nosology employed to identify psychiatric conditions within the cases reviewed may have varied depending on the version of the DSM, or the International Classification of Diseases, employed at the time of publication. The differences in evolving diagnostic classifications over time may have influenced conceptualizations of pathology and the

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ACCEPTED MANUSCRIPT rates at which psychiatric diagnoses may have been reported. For many of the cases, it was impossible to determine the accuracy of psychiatric diagnoses, i.e., the longitudinal and historical

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clinical information that guide diagnosis, were often not provided. 6. Conclusions

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In sum, these data suggest that clinicians encountering men engaging in GSM need to

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entertain the diversity of psychiatric disorders and psychosocial mediators underlying such selfinflicted injuries and contour psychiatric interventional approaches accordingly. Clinicians must

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resist assuming that such behaviors invariably reflect psychosis as had been suggested by previous publications [9,14,24,147,165].

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The macabre nature of GSM can elicit strong reactions in clinicians, evoking intense fear,

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perplexity, helplessness, frustration among other strong counter-transference responses [166168]. The reactions engendered in the responses of providers to the behaviors of individuals

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inflicting GSM can impede therapeutic endeavors and the formation of therapeutic alliances.

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Individuals engaging in GSM require immediate crisis intervention necessitating collaboration between psychiatrists, surgeons, and emergency medical professionals. It is hoped these data assist clinicians encountering persons who engage in GSM to moderate potential negative emotional responses and to develop an effective treatment plan. Despite the dearth of literature available on the long-term course of men engaging in GSM, significant advancements have been made in surgical interventions [124]. However, a more informative biopsychosocial approach to psychiatric and psychotherapeutic interventions is necessary in order to assist such individuals after initial injury and subsequent surgical management [75,84,97,139,156,169,170]. Pharmacologic interventions advocated for assisting 19

ACCEPTED MANUSCRIPT patients with recurrent histories of self-injurious behaviors have included naltrexone, topiramate, and clozapine [171-176], although the utility of these agents in the management of GSM has not

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been specifically explored. Working collaboratively, psychiatrists and urologists can enhance

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the care provided to persons engaging in GSM through exploring the exploration of the meanings and motivations underlying the behavior, enhancing social support, and providing long-term risk

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monitoring [177]. Future publications will need to detail the various treatment approaches and

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the impact these have on the longitudinal outcomes of persons engaging in GSM.

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ACCEPTED MANUSCRIPT Funding: This research did not receive any specific grant from funding agencies in the public,

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commercial, or not-for-profit sectors.

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[141]. Sudarshan CY, Rao KN, Santosh SV. Genital self-mutilation in erectile disorder. Indian J Psychiatry. 2006; 48(1): 64-5.

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[142]. Suraya Y, Saw KC. Psychiatric and surgical management of male genital self-mutilation.

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[143]. Tang WN. The first case of autocastration from east Asia. Can J Psychiatry. 1996;

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[144]. Tharoor H. A case of genital self-mutilation in an elderly man. Prim Care Companion J

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[146]. Tsao CI, Negrette G, Riley A. Self-amputation of the nipples and penis in a nonpsychotic, non-gender-dysphoric man. Psychosomatics. 2009; 50(2):178-80. [147]. Van Kammen DP, Money J. Erotic imagery in self-castration in transvestism/transsexualism: A case report. J Homosex. 1977; 2(4):359-66. [148]. Varman A, Moirangthem S, Nambi S, Gopala Krishnan SB, Surianarayanan U. Genital self-mutilation: Two case reports. Indian J Psychiatry. 2012; 54(suppl 1):114. [149]. Vender S, Bianchi L, Callegari C, Poloni N, Diurni M. Cannabis use and genital selfmutilation: An update of case reports. Riv Psichiatr. 2015; 50:3:148-50.

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[151]. Walter PJ, Krauss DJ, Nsouli IS. Repeat male genital self-mutilation precipitated by urinary complications of prior repair. J Urol. 1993; 149(6):1551-2.

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[153]. Wan SP, Soderdahl DW, Blight EM Jr. Nonpsychotic genital self-mutilation. Urology.

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12 Additional records identified through other sources (review articles and manual search)

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Identification

Figure 1: Flow Diagram for Case Selection

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Screening

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398 Records identified after duplicates were removed

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148 Full-text articles assessed for eligibility

Included

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Eligibility

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162 Records screened

27 Full-text articles excluded 24 Were not Case Reports 1 Accidental Injuries 1 Article focusing on Post-Injury Infestation 1 Duplicate Data

121 Studies included in review

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Mutilation (N = 21)

Amputation (N = 62)

Unknown/Not Reporteda

3 (14.3%)

5 (8.1%)

5 (8.9%)

Castration + Amputation (N= 34) 3 (8.8%)

Anxiety & OCD-Related Disordersb Obsessive Compulsive Disorder Post-Traumatic Stress Disorder

1 (4.8%) 0

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0 1 (1.8%)

0 0

Gender Dysphoriac

2 (9.5%)

2 (3.2%)

15 (26.8%)

5 (14.7%)

Mood Disordersd Bipolar Disorder Depression

0 2 (9.5%)

1 (1.6%) 3 (4.8%)

0 6 (10.7%)

1 (2.9%) 3 (8.8%)

0 2 (9.5%)

0 1 (1.6%)

0 1 (1.8%)

1 (2.9%) 0

Neurodevelopmental Disorderf

1 (4.8%)

1 (1.6%)

2 (3.6%)

0

Personality Disordersg

4 (19%)

9 (14.5%)

10 (17.9%)

2 (5.9%)

Schizophrenia Spectrum Disordersh Psychosis (Unspecified) Brief Reactive Psychosis Schizoaffective Disorder Schizophrenia Schizophreniform Disorder

0 0 0 5 (23.8%) 0

3 (4.8%) 0 3 (4.8%) 28 (45.2%) 2 (3.2%)

1 (1.8%) 2 (3.6%) 0 15 (26.8%) 0

0 0 1 (2.9%) 17 (50%) 0

Sexual/Paraphilic Disordersi Erectile Disorder Excess Sexual Drive Transvestism

0 0 0

1 (1.6%) 1 (1.6%) 0

0 0 1 (1.8%)

0 0 0

Substance Use Disordersj

4 (19%)

5 (8.1%)

11 (19.6%)

9 (26.5%)

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2 (3.2%) 0

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Neurocognitive Disorderse Delirium Dementia

Castration (N = 56)

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Diagnosis

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Table 1: Psychiatric Diagnoses Associated with GSM Reported in the Literature

More than 1 Diagnosis 3 (14.3%) 5 (8.1%) 13 (23.2%) 7 (20.6%) Notes: References (available online in an E-Supplement Appendix) a = [35,38,53,60,64,65,71, 72,89,97,121,122,133,134]; b = [73,105,128,145]; c = [34,43,46,54,56,58,68,84-86,88,94,108, 111,117,126,128,137]; d = [32,36,45,51,70,89,92,100,103,104,112,113,116,117,124,143]; e = 42

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[29,36,47,107,126]; f = [33,80,87,93]; g = [28,33,46,52,55,56,61,63,68,78,82,87,95,102,114, 120,126,127,136,141]; h = [30,36,37,39-41,47,49,50,56,57,59,62,63,65-67,69,74,75,77,79,81, 83,88-92,96,98,99-102,106,109,114,115,118-120,123,125,129,131,132,134,138-140,142,144, 146,147]; i = [130,135,137]; j = [31,33,36,40,44,47,48,53,56,58,61,73,75,76,89,92,98,99,110, 118,120,139,146,147]

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Mutilation (N = 21)

Amputation Castration (N = 62) (N = 56)

Unknown/Not Reported

13 (61.9%)

14 (22.6%)

26 (46.4%)

Castration + Amputation (N= 34) 12 (35.3%)

Delusionsa Erotomanic Grandiose Paranoid Religious Other

4 (19%) 0 1 (4.8%) 3 (14.3%) 0 0

23 (37.1%) 1 (1.6%) 6 (9.7%) 9 (14.5%) 8 (12.9%) 5 (8.1%)

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16 (28.6%) 0 4 (7.1%) 4 (7.1%) 7 (12.5%) 3 (5.3%)

12 (35.3%) 1 (2.9%) 1 (2.9%) 5 (14.7%) 5 (14.7%) 2 (5.9%)

Guiltb

3 (14.3%)

23 (37.1%)

14 (25%)

9 (26.5%)

Hallucinationsc

2 (9.5%)

18 (29%)

4 (7.1%)

9 (26.5%)

Need for Atonementd

0

8 (12.9%)

12 (21.4%)

5 (14.7%)

1 (4.8%)

3 (4.8%)

2 (3.6%)

0

0

14 (22.6%)

6 (10.7%)

6 (17.6%)

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Experiential Factors

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Table 2: Experiential Factors Associated with GSM Reported in the Literature

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Religious Preoccupationse Suicidal ideas/plansf

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More than 1 Experiential Factorg 2 (9.5%) 29 (46.8%) 15 (26.8%) 11 (32.4%) Notes: References (available online in an E-Supplement Appendix) a = [29,30,40-42,44,47, 49,50,56,59,62,63,65,66,69,74,76,77,79,83,88,89,91,92,96,98-100,102-104,106,109,114,115, 120,123,125,131,134,138,144,147,148]; b = [30,35,36,37,39,41,45-47,50,52-56,59,63,65,66,69, 71,78,79,87,91,96-100,102-104,106,108,115,116,119,120,131,135,143,144,146]; c = [30,41,42, 44,47,48,50,51,59,62,65,66,69,76,77,100,102,109,110,120,123,129,131,138,146]; d = [36,37, 69,71,79,83,87,98,99,100,103,104,106,109,114,120,123,144,148]; e = [35,38,55,83,95,131]; f = [28,32,37,41,55-57,60,63,70,75,89-92,105,112,115,116,120,124,125,133,146]; g = [30,35,37,41, 42,44,47,50,55,59,62,63,65,66,69,71,76,77,79,83,87, 91,96,98-100,102-104,106,109,114-116, 120,123,125,131,138,144,146-148]

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Precipitating Factors

Mutilation (N = 21)

Amputation Castration (N = 62) (N = 56)

Unknown/Not Reported

13 (61.9%)

47 (75.8%)

Change/Loss of Social Supporta

3 (14.3%)

4 (6.5%)

8 (14.3%)

6 (17.6%)

Perceived Rejectionb

1 (4.8%)

7 (11.3%)

4 (7.1%)

2 (5.9%)

Substance Intoxication/Withdrawalc

4 (19%)

3 (4.8%)

14 (25%)

7 (20.6%)

Treatment Issuesd Discontinuation Access Prevented by Others

0 0 0

2 (3.2%) 1 (1.6%) 1 (1.6%)

6 (10.7%) 0 7 (12.5%)

5 (14.7%) 1 (2.9%) 4 (11.8%)

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26 (46.4%)

Castration + Amputation (N= 34) 14 (41.2%)

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Table 3: Precipitating Factors Associated with GSM Reported in the Literature

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More than 1 Precipitating Factor 0 (0%) 1 (1.6%) 3 (5.4%) 0 (0%) Notes: References (available online in an E-Supplement Appendix); a = [30,32,39,41,48,68,79, 82,89,96,97,109,112,114,116,125,132,134,147]; b = [34,36,56,63,75,100,122,126,127,133,136, 146]; c = [31,33,36,40,44,45,47,48,52,53,58,61,68,71,73,75,76,89,92,98,99,120,139,147]; d = [43,47,55,56,68,72,94,111,117,126,128,139]

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