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Can we assess risk of psychiatric sequelae from perinatal injuries?
International Journal of Law and Psychiatry 49 (2016) 233–237
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International Journal of Law and Psychiatry
Can we assess risk of psychiatric sequelae from perinatal injuries? Seth A. Eappen, M.D. a,⁎, Elissa P. Benedek, M.D. b, Kerrie Murphy, Ph.D. c a b c
Chief Fellow, Division of Child & Adolescent Psychiatry, Institute of Psychiatry, Medical University of South Carolina, Charleston, SC, United States Adjunct Professor of Psychiatry at the University of Michigan, Ann Arbor, MI, United States. She is also in private practice Clinical Assistant Professor, Division of Child & Adolescent Psychiatry, Institute of Psychiatry, Medical University of South Carolina, Charleston, SC, United States
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Available online 8 November 2016 Keywords: Forensic child psychiatry Circumcision injury Urological injury Medical malpractice Personal injury
a b s t r a c t Medical malpractice litigation involving seven to nine figure settlements has been on the rise over the past decade. Given the life-altering scope of these incidents, forensic child psychiatrists may be asked to opine on the effects of a permanent perinatal injury on normal human development. Whether forensic child psychiatrists can assess risk of future psychiatric effects from such incidents is an important question. This paper is spurred by two separate cases, each involving a male infant having a portion of their genitals accidentally amputated by an obstetrician–gynecologist (OB/GYN) during a routine circumcision. In each case, the genitals were repaired, but permanent defects remained. The question arises how such injuries would affect these children as they matured. An analysis of the literature supported that there are risks of psychiatric effects of such birth-related injuries. Such predictions are made with reference to what is already known about human development, and by an exploration of the literature on the psychiatric effects of comparable injuries to children. © 2016 Elsevier Ltd. All rights reserved.
1. Introduction Medical malpractice litigation involving seven to nine figure settlements has been on the rise over the past decade; many of these cases have involved obstetrical accidents (American Medical Association, 2012). This paper was influenced by two separate cases, each involving children who had a part of the glans of their penis inadvertently amputated during a routine circumcision. For each child, the amputated glans was reattached, but the reattachment resulted in permanent deficits, including deviated urinary stream and scarring at the site of injury. Each of the parents filed lawsuits against the physicians who caused the injuries. Each of the plaintiff's lawyers contacted a forensic child psychiatrist to evaluate each case as an impartial expert, to assess future risk of psychiatric sequelae as a result of these injuries. The authors of this article reviewed these two cases for research purposes, which led us to ask: should we predict future psychiatric sequelae from repair of early penile injury, and if so, how do we do it? 2. How do we assess future risk? Based on the work of Lidz, Mulvey, and Gardner (1993), a threepronged approach is required in ascertaining predictive outcome, and we applied this template to our work on the type of perinatal injury under investigation. It permitted us to: 1) pursue literature examining long term psychiatric effects of similar injuries occurring at a similar ⁎ Corresponding author. E-mail addresses: [email protected] (S.A. Eappen), [email protected] (E.P. Benedek), [email protected] (K. Murphy).
http://dx.doi.org/10.1016/j.ijlp.2016.10.011 0160-2527/© 2016 Elsevier Ltd. All rights reserved.
age, 2) interview the family and/or child and to 3) utilize previously written documents in the case to form a final opinion. We expanded the procedure to include the five following classes of information to help gathering information on the long term effects of penile injury in cases going to court: Steps 1 and 2: Corroborating information and record review; Step 3: Literature review; Step 4: Interviewing the family and/or child; and Step 5: Utilizing past patient experience to form opinion. 3. Applying our model to cases 3.1. Steps 1 and 2: corroborating information and record review For the two cases in our practice that spurred the present review of the literature, depositions were reviewed from the physician who caused the injury, outpatient pediatrician, pediatric urologist who reattached the penis and maintained follow-up care, mother and any significant other, such as babysitter. No mental health professionals, aside from the forensic child psychiatrist who was consulted, were involved. Medical records were reviewed. 3.2. Step 3: literature review 3.2.1. Introduction The literature review focused on the long-term, post-operative, psychological and psychiatric course of children born with hypospadias. Hypospadias is a common congenital abnormality of the penis, where the opening of the urethra appears at various levels on the undersurface of the penis; this is seen in 1 out of 250 of newborn boys (Subramaniam, Spinoit, & Hoebeke, 2011). With the exception of less injurious cases,
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most individuals with this condition receive surgical reconstruction, the aim of which is to void in a forward stream, obtain normal penile appearance, and have a straight erection (Sharma, 2005). This congenital condition was chosen as a template in the cases at hand due to its similar anatomic location, the fact that surgical intervention is often necessary, for its similar post-operative complaints (functional and cosmetic issues of the penis), and its extensive applicable literature base, that specifically commented on the psychological and psychiatric course of these injured individuals during the decades after their operation. The search was limited to all articles published before June 31, 2014: on hypospadias, and various combinations of depression, psychiatric, psychological, mental, anxiety, plus various combinations related to urological/genital surgery pediatric and controls. All 40 articles that were found by the search were reviewed, but only nine met the ultimate needs of the search, having positive results. Two more had negative results and they are reported first. The remaining 31 articles were unrelated to our question or were not written in English. In order to orient the reader, before beginning the literature review, we provide the direction of general findings in the review. The results of the literature review are summarized below. Overall, after surgical repair of this condition, there was a greater incidence of negative post-operative effects, including timidity/social relation problems, difficulty initiating sexual relationships, and poor self-esteem. Further, while results on negative psychiatric adjustment were mixed, some studies also noted increased risk of depression, anxiety, behavior problems, and health-related quality of life. Because the literature is sparse, we describe each study in depth, so that their quality is more evident. We do so in the chronological order of their publication date, pointing out at times their strengths and weaknesses as we proceed. 3.2.2. The research 3.2.2.1. Correlational findings. Some research on the topic had found only minimal psychological effects from the need to surgically repair hypospadias. For example, Mureau, Slijper, Slob, and Verhulst (1997) found that the more the 9- to 18-year-old participants had a negative genital body perception, the worse was their psychosocial function, e.g., as measured by a subscale for inadequacy on the DPQ-J (Dutch Personality Questionnaire, Junior version; Luteijn, Van Dijk, & Van der Ploeg, 1989) and total problem score on the YSR (Youth Self Report, Achenbach, 1991). Schonbucher, Landolt, Gobet, and Weber (2008a) found that the later the corrective surgery in boys aged 7–17 years, the more likely they were insecure in their gender role behavior, as measured by the GRQ (Gender Role Questionnaire, Cohen-Kettenis & Pfäfflin, 2003, German version, see Medical Outcomes Trust, 2005). Liu et al. (2006) found a relationship between age of corrective surgery and an unvalidated measure of sexual inhibition (in seeking girlfriends or sexual contact). These findings concern correlations for which causation cannot be determined. The studies reviewed below report statistical differences involving groups of participants, although they do have the methodological weaknesses indicated. 3.2.2.2. Group findings 3.2.2.2.1. Negative results. Two studies that found pertinent correlational results on the question did not find any group differences of note and these are reviewed. These negative findings do not alter the general thrust of the literature that early penile injury does have some long term psychological consequences, e.g., related to self-esteem. Mureau et al. (1997) examined 118 children and adolescents from 9 to 18 years of age using standardized questionnaires on the psychosocial effects of hypospadias surgery. The age-matched comparison group consisted of 88 males who were operated for inguinal hernia. The tests used included the DPQ-J, the SAS-C (Social Anxiety Scale for Children (Dekking, 1983), and the CBCL (Child Behavior Checklist) or YSR (Achenbach, 1991; Dutch versions; Ferdinand, Verhulst, &
Witznitzer, 1995), depending on the age of the participant. Research indicating acceptable reliability and validity was reported for these measures. Upon statistical analysis, no group differences in psychosocial function were found, although the authors cautioned that in individual cases, the surgery involved might have psychological effects. Schonbucher et al. (2008a) examined sixty-eight 7- to 17-year olds in comparison to age-matched controls who underwent hernia repair on measures pertaining to psychosexual function. The measures involved included the PPPS (Pediatric Penile Perception Scale, Weber, Schonbucher, Landolt, & Gobet, 2008), the GRQ, and an unvalidated questionnaire on sexual attitudes. No group differences were found. However, further psychological investigation by this research group did find positive results on the question (see below). 3.2.2.2.2. Positive results. Several studies utilizing the same sample of 34 adult men operated for hypospadias in childhood and 36 controls who underwent an operation for appendicitis examined a variety of psychological and sexual outcomes (Berg, Berg, & Svensson, 1982; G. Berg & R. Berg, 1983; R. Berg & G. Berg, 1983; Svensson, Berg, & Berg, 1981). Participants were administered a semistructured psychiatric interview conducted by an experienced psychiatrist and developed by the authors (Berg & Berg, 1983a) and several psychological tests, including an intelligence test (Swedish Psychometric Battery; Dureman & Sälde, 1959) and projective personality assessments (the Rorschach Inkblot Test Rorschach, 1921); the Franck Drawing Completion Test (FDCT; Franck & Rosen, 1952); and the Gough Femininity Scale (GFS; Gough, 1952, 1966). Overall, these studies showed that individuals with hypospadias were generally well-adjusted young adults, although several notable psychological and social difficulties emerged consistently. Specifically, during childhood, individuals with hypospadias reported increased enuresis, shyness, increased social isolation and being timid with peers at school, as well as more frequent teasing compared to controls (Berg et al., 1982; Svensson et al., 1981). Regarding psychosexual outcomes, individuals in the hypospadias group had later age of first intercourse and fewer partners relative to controls, but a majority reported a satisfactory sex life (Svensson et al., 1981). There were no significant differences in the groups related to severity of hypospadias and psychosexual outcomes (e.g., sex drive, number of sexual partners, frequency of sexual activity), and participants in the hypospadias group showed normal social adjustment (Svensson et al., 1981). However, individuals with hypospadias showed a more doubtful affected sexual identity, among other findings, combined with lesser utilization of cognitive and emotional resources than controls (G. Berg & R. Berg, 1983; R. Berg & G. Berg, 1983; Svensson et al., 1981). Regarding adult psychological outcomes, these group of three early studies suggested that individuals operated for hypospadias in their childhood reported increased psychiatric symptoms, including depression and anxiety, as well as decreased stress tolerance and more difficulties utilizing coping skills or resources (Berg et al., 1982). Further, individuals with hypospadias reported more impaired capacity for social relations and overall poorer interpersonal relationships compared to controls (G. Berg & R. Berg, 1983; Berg et al., 1982). However, the research used projective instruments for the most part, which have reliability and validity issues. In a study by Sandberg, Meyer-Bahlburg, Aranoff, Sconzo, and Hensle (1989), a sample of 69 boys (ages 6 to 10) was selected from the files of a pediatric urology department at a major medical center in New York. The boys were chosen for being born with hypospadias with or without additional genital or non-genital malformations. Parents completed several behavior rating scales [e.g., (CBCL), Achenbach, 1991; Child Behavior and Attitude Questionnaire (CBAQ), Bates, Bentler, & Thompson, 1973] to assess behavior problems, social competency, academic achievement, gender role atypicality (e.g., displaying ambiguous or atypical gender-role behaviors), and family adjustment. Parent ratings were statistically compared to both the clinical and
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non-clinical standardization samples used in the development of these measures. The latter did not include cases like the ones in the sample studied. Results indicated that boys with hypospadias were rated as having significantly more behavior problems, lower social competency, and increased gender atypical scores when compared to the non-clinical standardization samples. However, the index cases did not arrive at clinical levels in their results. There were no effects noted in the index cases on academic achievement. The pattern of these results suggests that parents of boys with hypospadias reported significantly more behavior problems and more social competency difficulties compared to a general sample of children. Mondaini et al. (2002) examined the incidence of hypospadias and psychosexual functioning of young adult men from a populationbased sample of 18-year-old Italian males examined as part of their evaluation for enlistment for compulsory National Service (N = 11,649). A group of 500 males were randomly selected from the 11,649 subjects to serve as a control group matched on age, socioeconomic status, and geographic region. The assessment included a medical evaluation, administration of the Minnesota Multiphasic Personality Inventory (MMPI; a standardized assessment of personality psychopathology and psychological functioning, Hathaway & McKinley, 1940), and an interview about psychosexual functioning. Results indicated the incidence of hypospadias was 3.6/1000 (N = 42). Individuals with hypospadias reported increased inhibition in approaching the opposite sex and increased negative genital appraisals compared to the control group. Further, significantly fewer individuals with hypospadias (7/42 vs. 210/500 controls) reported having had sexual intercourse. There were no significant differences between groups on measures of psychological functioning. Overall, this study provided some mixed results regarding the psychosexual functioning of young adults with hypospadias, in that the individuals reported increased inhibition and more limited sexual experience relative to controls, but reported no significant differences in psychological functioning. This study was weakened by an unclear description of measures used to assess sexual functioning and a limited assessment of psychological functioning. For instance, the authors did not specify whether they used an Italian version of the MMPI and provided a vague description of results from the MMPI. That is, they noted that “subjects whose answers were considered nonappropriate [on the MMPI] were further examined by psychiatric staff” (p. 82). The authors reported that 38% of hypospadics gave an overall nonappropriate response on the MMPI compared to 28.6% of controls; however, this difference was not statistically significant and the authors did not define how a “nonappropriate response” was conceptualized, making the results difficult to interpret. In a study by Schonbucher, Landolt, Gobet, and Weber (2008b), the health-related quality of life (HRQoL) and psychological adjustment of 77 boys (aged 7–17) living in Switzerland who underwent hypospadias repair were compared to a group of 77 boys who had undergone surgery for hernia repair (control group). This cross-sectional study included both child- and mother-report on several standardized questionnaires of HRQoL [(TNO-AZL Child Quality of Life Questionnaire, Child Form) (TACQOL child form, Verrips et al., 1999); (TACQOL parent form, Vogels et al., 1998)] and psychological adjustment (CBCL, Achenbach, 1991). The results found that self-reported HRQoL of child and adolescent patients with hypospadias was lower in most dimensions than the control group. There were no significant differences in self-reported psychological adjustment. Interestingly, while there were no significant differences in HRQoL between groups on mother-reported HRQoL or psychological adjustment, mothers of boys with hypospadias rated their sons as having significantly higher HRQoL compared to the child's self-report. The authors hypothesized that this surprising effect could be the result of mothers psychologically suppressing the impact of hypospadias and is worthy of further inquiry (p. 871). Further, results indicated that younger age may be a risk factor for self-reported impairments in HRQoL and maladjustment.
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In a study by Wang et al. (2010), the psychosexual functioning of adults who had hypospadias surgery at different ages was evaluated. A detailed questionnaire was completed by 130 patients who underwent hypospadias surgery between January 1988 and December 2007 in a Chinese hospital, and 50 healthy males who served as the control group. The men in both groups were between 24 and 35 years old. Symptoms of depression and anxiety [as measured by the Zung SelfRating Anxiety Scale (Zung, 1971) and the Zung Self-Rating Depression Scale (Zung, 1965)], satisfaction with sexual functioning [as measured by the International Index of Erectile Function-5 (IIEF) (Rosen et al., 1997)], and measurements of penile length and circumference were used to assess psychosexual functioning and adjustment. Results indicated that individuals in the hypospadias group reported significantly higher levels of depression and anxiety compared to controls. Further, hypospadias patients reported increased social difficulties related to hypospadias. There were no significant differences between patients and controls regarding libido strength, overall sexual satisfaction, and erectile function. In a study by Kiss et al. (2011), the consequences of hypospadias repair on psychosexual functioning in Hungarian adult males were investigated. Mailed surveys were completed by a homogenous group of middle-aged adult men (N = 104; Mage = 31.6, SD = 6.0) who all underwent the same type of operation (Denis–Browne repair, which utilized a tubularized ventral penis skin procedure) to correct penile hypospadias of the same severity (proximal hypospadias) in early childhood. Age-matched, healthy controls (N = 63; Mage = 32.7, SD = 4.7) with no history of genital malformations were recruited. A 15-item questionnaire developed by the authors was given, which included questions regarding satisfaction with sex life, penile appearance, and body image. Results indicated that hypospadias patients reported significantly lower satisfaction with genital appearance, although significantly higher sexual satisfaction compared to controls. There were no significant differences in satisfaction with body image, general or sexual disadvantages due to the operation, regular sexual contact, masturbation, age at first kiss, age of first sexual intercourse, or having a child. Further, analyses suggested that hypospadias patients who were unsatisfied with their surgery reported lower scores on all questions relative to hypospadias patients who were satisfied with surgery. While this study was limited by the use of an unvalidated questionnaire and did not directly examine any psychological functioning, results give some preliminary evidence that an individual's satisfaction with surgical outcomes may play an important role in the individual's later adjustment and sexual functioning. At the same time, the risk is that the results are taken as definitive in court when this is far from the case. That is, more research is needed on the matter before arriving at any definitive conclusions in these regards. The paucity of the research, in general, in the area, suggests that this is the case for any conclusions reached. 3.2.2.3. Summary of the research findings. Taken together, results of this literature review consistently found that individuals operated for hypospadias in childhood reported impaired psychosexual and social functioning, particularly related to difficulties developing and initiating sexual relationships, increased timidity and shyness, and poor selfesteem related to body image and genital appearances (R. Berg & G. Berg, 1983; Berg et al., 1982; Kiss et al., 2011; Mondaini et al., 2002; Sandberg et al., 1989; Svensson et al., 1981; Wang et al., 2010). Interestingly, most studies noted no significant deficits related to actual sexual functioning (e.g., Svensson et al., 1981; Wang et al., 2010). Results related to psychopathology among individuals with hypospadias were mixed, however, as some studies noted increased rates of depressive and anxiety symptoms among individuals with hypospadias (Berg et al., 1982; Wang et al., 2010), others noted increased behavior problems (Sandberg et al., 1989), and still others noted decreased health-related quality of life based on self-report (Schonbucher et al.,
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2008b). These mixed results may be due in part to inconsistency in the literature regarding psychiatric outcomes, in that the studies reviewed often used different informants (e.g., adult retrospective report on childhood symptoms, adult report on current symptoms, parentreport on childhood symptoms), and varied measures of psychiatric functioning (e.g., standardized measures, interviews, or unvalidated measures), making it difficult to draw broad conclusions about the long-term psychiatric effects of hypospadias. While additional research is warranted, these results suggest a consistent pattern of impaired social functioning, particularly related to developing sexual relationships, as well as deficits in self-esteem and health-related quality of life. 3.3. Step 4: interviewing the family and/or child The decision of whether to interview a child is a controversial topic, and must be made on a case-by-case basis. The age of the child (the decision not to interview a 2-year-old is different from the decision not to interview a 13-year-old) and the parents' wishes both play a role. Parents are legal guardians of children unless a court says otherwise. Therefore, the parents make the final decision about any treatment or interview their child receives. For both case studies, the families were interviewed, but the children—who were between ages 3 and 5—were not. The child psychiatrist who testified believed that if a child was not yet developmentally capable of understanding the significance of their injury, then a focused interview could place the child at risk of excessively worrying about the injury, as supported by Friedberg (2015). 3.4. Step 5: utilizing past patient experience to form opinion In certain malpractice cases, there may not be any formal research studies done examining the psychiatric effects of a rare injury on an individual. Therefore, aside from extrapolating from closely-related literature, the next best source of information is the forensic psychiatrist's past experience with other patients who have gone through similar injuries. Limitations to this include sample bias and small sample size, and to a degree even moreso than found in the sparse literature. 4. Discussion The literature review undertaken on perinatal penile injury and its psychological effects has shown that the research is sparse and the conclusions that could derive from them needs further investigation to confirm them. Nevertheless, a body of literature is emerging that suggests some long term consequences in such cases. It is always difficult to generalize from the literature to individual cases in court, but the supplementary clinical information that is gathered along with available documentation, for example, indicates the plausibility if the outcomes of the literature review that long term outcomes might be detrimental to those undergoing early penile corrective surgery for obstetrical errors in routine circumcision. In this regard, these injuries are actionable in court on the basis of varied negative outcomes, for example, in selfesteem and sexuality. The literature bears legal relevance in court because, the studies, even if sparse, can be connected to the cases at hand, and illustrate the negative consequences of the medical malpractice involved. The studies have “appreciable probative value” (Garner, 2009, p. 1404), and so help the trier of fact in deliberations toward proving or disproving a matter at issue, in this case, proving the long term negative psychological consequences involved. The assessment of damages in a perinatal injury case—when there is no clear outcome data for that particular injury—is an incredibly complex undertaking. This article was not written to serve as a guideline, but rather as a point of reference on how the authors and this particular forensic child psychiatrist tackled this particular
question. This could have been handled in multiple different ways by other forensic psychiatrists. Further, there are several limitations to testifying in cases like this. The expert's testimony might rely on extrapolating psychiatric sequelae from his past patients rather than focusing on the post-operative psychiatric sequelae of hypospadias found in the literature. This is a source of bias, but so is focusing on the literature—there might be sample bias and small sample size, which limits extrapolating from them. We expect that continued research and practice in using this approach to the type of assessment under discussion will allow us to refine it further, as it is challenged over time. We believe that it is reasonable to assess risk of future psychiatric symptoms if given enough data, because physicians already make assessments regarding suicide risk, violence risk and life expectancy—and often do so with a fraction of the time and data that we were given with both cases. As for the ethical question of “can we?”: given that we can assess risk of future psychiatric symptoms to a reasonable degree of medical certainty, and that persistent practice and research will improve the accuracy of such an assessment, we believe that it is our duty to continue to attempt such assessments, as long as we adhere to the basic forensic psychiatric principles discussed in this paper as we do so.
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Contents lists available at ScienceDirect
International Journal of Law and Psychiatry
Can we assess risk of psychiatric sequelae from perinatal injuries? Seth A. Eappen, M.D. a,⁎, Elissa P. Benedek, M.D. b, Kerrie Murphy, Ph.D. c a b c
Chief Fellow, Division of Child & Adolescent Psychiatry, Institute of Psychiatry, Medical University of South Carolina, Charleston, SC, United States Adjunct Professor of Psychiatry at the University of Michigan, Ann Arbor, MI, United States. She is also in private practice Clinical Assistant Professor, Division of Child & Adolescent Psychiatry, Institute of Psychiatry, Medical University of South Carolina, Charleston, SC, United States
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Available online 8 November 2016 Keywords: Forensic child psychiatry Circumcision injury Urological injury Medical malpractice Personal injury
a b s t r a c t Medical malpractice litigation involving seven to nine figure settlements has been on the rise over the past decade. Given the life-altering scope of these incidents, forensic child psychiatrists may be asked to opine on the effects of a permanent perinatal injury on normal human development. Whether forensic child psychiatrists can assess risk of future psychiatric effects from such incidents is an important question. This paper is spurred by two separate cases, each involving a male infant having a portion of their genitals accidentally amputated by an obstetrician–gynecologist (OB/GYN) during a routine circumcision. In each case, the genitals were repaired, but permanent defects remained. The question arises how such injuries would affect these children as they matured. An analysis of the literature supported that there are risks of psychiatric effects of such birth-related injuries. Such predictions are made with reference to what is already known about human development, and by an exploration of the literature on the psychiatric effects of comparable injuries to children. © 2016 Elsevier Ltd. All rights reserved.
1. Introduction Medical malpractice litigation involving seven to nine figure settlements has been on the rise over the past decade; many of these cases have involved obstetrical accidents (American Medical Association, 2012). This paper was influenced by two separate cases, each involving children who had a part of the glans of their penis inadvertently amputated during a routine circumcision. For each child, the amputated glans was reattached, but the reattachment resulted in permanent deficits, including deviated urinary stream and scarring at the site of injury. Each of the parents filed lawsuits against the physicians who caused the injuries. Each of the plaintiff's lawyers contacted a forensic child psychiatrist to evaluate each case as an impartial expert, to assess future risk of psychiatric sequelae as a result of these injuries. The authors of this article reviewed these two cases for research purposes, which led us to ask: should we predict future psychiatric sequelae from repair of early penile injury, and if so, how do we do it? 2. How do we assess future risk? Based on the work of Lidz, Mulvey, and Gardner (1993), a threepronged approach is required in ascertaining predictive outcome, and we applied this template to our work on the type of perinatal injury under investigation. It permitted us to: 1) pursue literature examining long term psychiatric effects of similar injuries occurring at a similar ⁎ Corresponding author. E-mail addresses: [email protected] (S.A. Eappen), [email protected] (E.P. Benedek), [email protected] (K. Murphy).
http://dx.doi.org/10.1016/j.ijlp.2016.10.011 0160-2527/© 2016 Elsevier Ltd. All rights reserved.
age, 2) interview the family and/or child and to 3) utilize previously written documents in the case to form a final opinion. We expanded the procedure to include the five following classes of information to help gathering information on the long term effects of penile injury in cases going to court: Steps 1 and 2: Corroborating information and record review; Step 3: Literature review; Step 4: Interviewing the family and/or child; and Step 5: Utilizing past patient experience to form opinion. 3. Applying our model to cases 3.1. Steps 1 and 2: corroborating information and record review For the two cases in our practice that spurred the present review of the literature, depositions were reviewed from the physician who caused the injury, outpatient pediatrician, pediatric urologist who reattached the penis and maintained follow-up care, mother and any significant other, such as babysitter. No mental health professionals, aside from the forensic child psychiatrist who was consulted, were involved. Medical records were reviewed. 3.2. Step 3: literature review 3.2.1. Introduction The literature review focused on the long-term, post-operative, psychological and psychiatric course of children born with hypospadias. Hypospadias is a common congenital abnormality of the penis, where the opening of the urethra appears at various levels on the undersurface of the penis; this is seen in 1 out of 250 of newborn boys (Subramaniam, Spinoit, & Hoebeke, 2011). With the exception of less injurious cases,
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most individuals with this condition receive surgical reconstruction, the aim of which is to void in a forward stream, obtain normal penile appearance, and have a straight erection (Sharma, 2005). This congenital condition was chosen as a template in the cases at hand due to its similar anatomic location, the fact that surgical intervention is often necessary, for its similar post-operative complaints (functional and cosmetic issues of the penis), and its extensive applicable literature base, that specifically commented on the psychological and psychiatric course of these injured individuals during the decades after their operation. The search was limited to all articles published before June 31, 2014: on hypospadias, and various combinations of depression, psychiatric, psychological, mental, anxiety, plus various combinations related to urological/genital surgery pediatric and controls. All 40 articles that were found by the search were reviewed, but only nine met the ultimate needs of the search, having positive results. Two more had negative results and they are reported first. The remaining 31 articles were unrelated to our question or were not written in English. In order to orient the reader, before beginning the literature review, we provide the direction of general findings in the review. The results of the literature review are summarized below. Overall, after surgical repair of this condition, there was a greater incidence of negative post-operative effects, including timidity/social relation problems, difficulty initiating sexual relationships, and poor self-esteem. Further, while results on negative psychiatric adjustment were mixed, some studies also noted increased risk of depression, anxiety, behavior problems, and health-related quality of life. Because the literature is sparse, we describe each study in depth, so that their quality is more evident. We do so in the chronological order of their publication date, pointing out at times their strengths and weaknesses as we proceed. 3.2.2. The research 3.2.2.1. Correlational findings. Some research on the topic had found only minimal psychological effects from the need to surgically repair hypospadias. For example, Mureau, Slijper, Slob, and Verhulst (1997) found that the more the 9- to 18-year-old participants had a negative genital body perception, the worse was their psychosocial function, e.g., as measured by a subscale for inadequacy on the DPQ-J (Dutch Personality Questionnaire, Junior version; Luteijn, Van Dijk, & Van der Ploeg, 1989) and total problem score on the YSR (Youth Self Report, Achenbach, 1991). Schonbucher, Landolt, Gobet, and Weber (2008a) found that the later the corrective surgery in boys aged 7–17 years, the more likely they were insecure in their gender role behavior, as measured by the GRQ (Gender Role Questionnaire, Cohen-Kettenis & Pfäfflin, 2003, German version, see Medical Outcomes Trust, 2005). Liu et al. (2006) found a relationship between age of corrective surgery and an unvalidated measure of sexual inhibition (in seeking girlfriends or sexual contact). These findings concern correlations for which causation cannot be determined. The studies reviewed below report statistical differences involving groups of participants, although they do have the methodological weaknesses indicated. 3.2.2.2. Group findings 3.2.2.2.1. Negative results. Two studies that found pertinent correlational results on the question did not find any group differences of note and these are reviewed. These negative findings do not alter the general thrust of the literature that early penile injury does have some long term psychological consequences, e.g., related to self-esteem. Mureau et al. (1997) examined 118 children and adolescents from 9 to 18 years of age using standardized questionnaires on the psychosocial effects of hypospadias surgery. The age-matched comparison group consisted of 88 males who were operated for inguinal hernia. The tests used included the DPQ-J, the SAS-C (Social Anxiety Scale for Children (Dekking, 1983), and the CBCL (Child Behavior Checklist) or YSR (Achenbach, 1991; Dutch versions; Ferdinand, Verhulst, &
Witznitzer, 1995), depending on the age of the participant. Research indicating acceptable reliability and validity was reported for these measures. Upon statistical analysis, no group differences in psychosocial function were found, although the authors cautioned that in individual cases, the surgery involved might have psychological effects. Schonbucher et al. (2008a) examined sixty-eight 7- to 17-year olds in comparison to age-matched controls who underwent hernia repair on measures pertaining to psychosexual function. The measures involved included the PPPS (Pediatric Penile Perception Scale, Weber, Schonbucher, Landolt, & Gobet, 2008), the GRQ, and an unvalidated questionnaire on sexual attitudes. No group differences were found. However, further psychological investigation by this research group did find positive results on the question (see below). 3.2.2.2.2. Positive results. Several studies utilizing the same sample of 34 adult men operated for hypospadias in childhood and 36 controls who underwent an operation for appendicitis examined a variety of psychological and sexual outcomes (Berg, Berg, & Svensson, 1982; G. Berg & R. Berg, 1983; R. Berg & G. Berg, 1983; Svensson, Berg, & Berg, 1981). Participants were administered a semistructured psychiatric interview conducted by an experienced psychiatrist and developed by the authors (Berg & Berg, 1983a) and several psychological tests, including an intelligence test (Swedish Psychometric Battery; Dureman & Sälde, 1959) and projective personality assessments (the Rorschach Inkblot Test Rorschach, 1921); the Franck Drawing Completion Test (FDCT; Franck & Rosen, 1952); and the Gough Femininity Scale (GFS; Gough, 1952, 1966). Overall, these studies showed that individuals with hypospadias were generally well-adjusted young adults, although several notable psychological and social difficulties emerged consistently. Specifically, during childhood, individuals with hypospadias reported increased enuresis, shyness, increased social isolation and being timid with peers at school, as well as more frequent teasing compared to controls (Berg et al., 1982; Svensson et al., 1981). Regarding psychosexual outcomes, individuals in the hypospadias group had later age of first intercourse and fewer partners relative to controls, but a majority reported a satisfactory sex life (Svensson et al., 1981). There were no significant differences in the groups related to severity of hypospadias and psychosexual outcomes (e.g., sex drive, number of sexual partners, frequency of sexual activity), and participants in the hypospadias group showed normal social adjustment (Svensson et al., 1981). However, individuals with hypospadias showed a more doubtful affected sexual identity, among other findings, combined with lesser utilization of cognitive and emotional resources than controls (G. Berg & R. Berg, 1983; R. Berg & G. Berg, 1983; Svensson et al., 1981). Regarding adult psychological outcomes, these group of three early studies suggested that individuals operated for hypospadias in their childhood reported increased psychiatric symptoms, including depression and anxiety, as well as decreased stress tolerance and more difficulties utilizing coping skills or resources (Berg et al., 1982). Further, individuals with hypospadias reported more impaired capacity for social relations and overall poorer interpersonal relationships compared to controls (G. Berg & R. Berg, 1983; Berg et al., 1982). However, the research used projective instruments for the most part, which have reliability and validity issues. In a study by Sandberg, Meyer-Bahlburg, Aranoff, Sconzo, and Hensle (1989), a sample of 69 boys (ages 6 to 10) was selected from the files of a pediatric urology department at a major medical center in New York. The boys were chosen for being born with hypospadias with or without additional genital or non-genital malformations. Parents completed several behavior rating scales [e.g., (CBCL), Achenbach, 1991; Child Behavior and Attitude Questionnaire (CBAQ), Bates, Bentler, & Thompson, 1973] to assess behavior problems, social competency, academic achievement, gender role atypicality (e.g., displaying ambiguous or atypical gender-role behaviors), and family adjustment. Parent ratings were statistically compared to both the clinical and
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non-clinical standardization samples used in the development of these measures. The latter did not include cases like the ones in the sample studied. Results indicated that boys with hypospadias were rated as having significantly more behavior problems, lower social competency, and increased gender atypical scores when compared to the non-clinical standardization samples. However, the index cases did not arrive at clinical levels in their results. There were no effects noted in the index cases on academic achievement. The pattern of these results suggests that parents of boys with hypospadias reported significantly more behavior problems and more social competency difficulties compared to a general sample of children. Mondaini et al. (2002) examined the incidence of hypospadias and psychosexual functioning of young adult men from a populationbased sample of 18-year-old Italian males examined as part of their evaluation for enlistment for compulsory National Service (N = 11,649). A group of 500 males were randomly selected from the 11,649 subjects to serve as a control group matched on age, socioeconomic status, and geographic region. The assessment included a medical evaluation, administration of the Minnesota Multiphasic Personality Inventory (MMPI; a standardized assessment of personality psychopathology and psychological functioning, Hathaway & McKinley, 1940), and an interview about psychosexual functioning. Results indicated the incidence of hypospadias was 3.6/1000 (N = 42). Individuals with hypospadias reported increased inhibition in approaching the opposite sex and increased negative genital appraisals compared to the control group. Further, significantly fewer individuals with hypospadias (7/42 vs. 210/500 controls) reported having had sexual intercourse. There were no significant differences between groups on measures of psychological functioning. Overall, this study provided some mixed results regarding the psychosexual functioning of young adults with hypospadias, in that the individuals reported increased inhibition and more limited sexual experience relative to controls, but reported no significant differences in psychological functioning. This study was weakened by an unclear description of measures used to assess sexual functioning and a limited assessment of psychological functioning. For instance, the authors did not specify whether they used an Italian version of the MMPI and provided a vague description of results from the MMPI. That is, they noted that “subjects whose answers were considered nonappropriate [on the MMPI] were further examined by psychiatric staff” (p. 82). The authors reported that 38% of hypospadics gave an overall nonappropriate response on the MMPI compared to 28.6% of controls; however, this difference was not statistically significant and the authors did not define how a “nonappropriate response” was conceptualized, making the results difficult to interpret. In a study by Schonbucher, Landolt, Gobet, and Weber (2008b), the health-related quality of life (HRQoL) and psychological adjustment of 77 boys (aged 7–17) living in Switzerland who underwent hypospadias repair were compared to a group of 77 boys who had undergone surgery for hernia repair (control group). This cross-sectional study included both child- and mother-report on several standardized questionnaires of HRQoL [(TNO-AZL Child Quality of Life Questionnaire, Child Form) (TACQOL child form, Verrips et al., 1999); (TACQOL parent form, Vogels et al., 1998)] and psychological adjustment (CBCL, Achenbach, 1991). The results found that self-reported HRQoL of child and adolescent patients with hypospadias was lower in most dimensions than the control group. There were no significant differences in self-reported psychological adjustment. Interestingly, while there were no significant differences in HRQoL between groups on mother-reported HRQoL or psychological adjustment, mothers of boys with hypospadias rated their sons as having significantly higher HRQoL compared to the child's self-report. The authors hypothesized that this surprising effect could be the result of mothers psychologically suppressing the impact of hypospadias and is worthy of further inquiry (p. 871). Further, results indicated that younger age may be a risk factor for self-reported impairments in HRQoL and maladjustment.
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In a study by Wang et al. (2010), the psychosexual functioning of adults who had hypospadias surgery at different ages was evaluated. A detailed questionnaire was completed by 130 patients who underwent hypospadias surgery between January 1988 and December 2007 in a Chinese hospital, and 50 healthy males who served as the control group. The men in both groups were between 24 and 35 years old. Symptoms of depression and anxiety [as measured by the Zung SelfRating Anxiety Scale (Zung, 1971) and the Zung Self-Rating Depression Scale (Zung, 1965)], satisfaction with sexual functioning [as measured by the International Index of Erectile Function-5 (IIEF) (Rosen et al., 1997)], and measurements of penile length and circumference were used to assess psychosexual functioning and adjustment. Results indicated that individuals in the hypospadias group reported significantly higher levels of depression and anxiety compared to controls. Further, hypospadias patients reported increased social difficulties related to hypospadias. There were no significant differences between patients and controls regarding libido strength, overall sexual satisfaction, and erectile function. In a study by Kiss et al. (2011), the consequences of hypospadias repair on psychosexual functioning in Hungarian adult males were investigated. Mailed surveys were completed by a homogenous group of middle-aged adult men (N = 104; Mage = 31.6, SD = 6.0) who all underwent the same type of operation (Denis–Browne repair, which utilized a tubularized ventral penis skin procedure) to correct penile hypospadias of the same severity (proximal hypospadias) in early childhood. Age-matched, healthy controls (N = 63; Mage = 32.7, SD = 4.7) with no history of genital malformations were recruited. A 15-item questionnaire developed by the authors was given, which included questions regarding satisfaction with sex life, penile appearance, and body image. Results indicated that hypospadias patients reported significantly lower satisfaction with genital appearance, although significantly higher sexual satisfaction compared to controls. There were no significant differences in satisfaction with body image, general or sexual disadvantages due to the operation, regular sexual contact, masturbation, age at first kiss, age of first sexual intercourse, or having a child. Further, analyses suggested that hypospadias patients who were unsatisfied with their surgery reported lower scores on all questions relative to hypospadias patients who were satisfied with surgery. While this study was limited by the use of an unvalidated questionnaire and did not directly examine any psychological functioning, results give some preliminary evidence that an individual's satisfaction with surgical outcomes may play an important role in the individual's later adjustment and sexual functioning. At the same time, the risk is that the results are taken as definitive in court when this is far from the case. That is, more research is needed on the matter before arriving at any definitive conclusions in these regards. The paucity of the research, in general, in the area, suggests that this is the case for any conclusions reached. 3.2.2.3. Summary of the research findings. Taken together, results of this literature review consistently found that individuals operated for hypospadias in childhood reported impaired psychosexual and social functioning, particularly related to difficulties developing and initiating sexual relationships, increased timidity and shyness, and poor selfesteem related to body image and genital appearances (R. Berg & G. Berg, 1983; Berg et al., 1982; Kiss et al., 2011; Mondaini et al., 2002; Sandberg et al., 1989; Svensson et al., 1981; Wang et al., 2010). Interestingly, most studies noted no significant deficits related to actual sexual functioning (e.g., Svensson et al., 1981; Wang et al., 2010). Results related to psychopathology among individuals with hypospadias were mixed, however, as some studies noted increased rates of depressive and anxiety symptoms among individuals with hypospadias (Berg et al., 1982; Wang et al., 2010), others noted increased behavior problems (Sandberg et al., 1989), and still others noted decreased health-related quality of life based on self-report (Schonbucher et al.,
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2008b). These mixed results may be due in part to inconsistency in the literature regarding psychiatric outcomes, in that the studies reviewed often used different informants (e.g., adult retrospective report on childhood symptoms, adult report on current symptoms, parentreport on childhood symptoms), and varied measures of psychiatric functioning (e.g., standardized measures, interviews, or unvalidated measures), making it difficult to draw broad conclusions about the long-term psychiatric effects of hypospadias. While additional research is warranted, these results suggest a consistent pattern of impaired social functioning, particularly related to developing sexual relationships, as well as deficits in self-esteem and health-related quality of life. 3.3. Step 4: interviewing the family and/or child The decision of whether to interview a child is a controversial topic, and must be made on a case-by-case basis. The age of the child (the decision not to interview a 2-year-old is different from the decision not to interview a 13-year-old) and the parents' wishes both play a role. Parents are legal guardians of children unless a court says otherwise. Therefore, the parents make the final decision about any treatment or interview their child receives. For both case studies, the families were interviewed, but the children—who were between ages 3 and 5—were not. The child psychiatrist who testified believed that if a child was not yet developmentally capable of understanding the significance of their injury, then a focused interview could place the child at risk of excessively worrying about the injury, as supported by Friedberg (2015). 3.4. Step 5: utilizing past patient experience to form opinion In certain malpractice cases, there may not be any formal research studies done examining the psychiatric effects of a rare injury on an individual. Therefore, aside from extrapolating from closely-related literature, the next best source of information is the forensic psychiatrist's past experience with other patients who have gone through similar injuries. Limitations to this include sample bias and small sample size, and to a degree even moreso than found in the sparse literature. 4. Discussion The literature review undertaken on perinatal penile injury and its psychological effects has shown that the research is sparse and the conclusions that could derive from them needs further investigation to confirm them. Nevertheless, a body of literature is emerging that suggests some long term consequences in such cases. It is always difficult to generalize from the literature to individual cases in court, but the supplementary clinical information that is gathered along with available documentation, for example, indicates the plausibility if the outcomes of the literature review that long term outcomes might be detrimental to those undergoing early penile corrective surgery for obstetrical errors in routine circumcision. In this regard, these injuries are actionable in court on the basis of varied negative outcomes, for example, in selfesteem and sexuality. The literature bears legal relevance in court because, the studies, even if sparse, can be connected to the cases at hand, and illustrate the negative consequences of the medical malpractice involved. The studies have “appreciable probative value” (Garner, 2009, p. 1404), and so help the trier of fact in deliberations toward proving or disproving a matter at issue, in this case, proving the long term negative psychological consequences involved. The assessment of damages in a perinatal injury case—when there is no clear outcome data for that particular injury—is an incredibly complex undertaking. This article was not written to serve as a guideline, but rather as a point of reference on how the authors and this particular forensic child psychiatrist tackled this particular
question. This could have been handled in multiple different ways by other forensic psychiatrists. Further, there are several limitations to testifying in cases like this. The expert's testimony might rely on extrapolating psychiatric sequelae from his past patients rather than focusing on the post-operative psychiatric sequelae of hypospadias found in the literature. This is a source of bias, but so is focusing on the literature—there might be sample bias and small sample size, which limits extrapolating from them. We expect that continued research and practice in using this approach to the type of assessment under discussion will allow us to refine it further, as it is challenged over time. We believe that it is reasonable to assess risk of future psychiatric symptoms if given enough data, because physicians already make assessments regarding suicide risk, violence risk and life expectancy—and often do so with a fraction of the time and data that we were given with both cases. As for the ethical question of “can we?”: given that we can assess risk of future psychiatric symptoms to a reasonable degree of medical certainty, and that persistent practice and research will improve the accuracy of such an assessment, we believe that it is our duty to continue to attempt such assessments, as long as we adhere to the basic forensic psychiatric principles discussed in this paper as we do so.
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