ORIGINAL ARTICLE

Endocrine Care—Brief Report

Psychosexual and Psychosocial Functions of Anorchid Young Adults

Preamrudee Poomthavorn, Robyn Stargatt, and Margaret Zacharin

Department of Endocrinology and Diabetes (P.P., M.Z.), The Royal Children’s Hospital, Melbourne 3052, Australia; Department of Psychology (R.S.), La Trobe University, Melbourne 3086, Australia; and Department of Pediatrics (P.P.), Faculty of Medicine, Ramathibodi Hospital, Mahidol University, Bangkok 10400, Thailand Downloaded from https://academic.oup.com/jcem/article/94/7/2502/2597091 by guest on 27 September 2021

Context: Prenatal exposure of the male fetus to at wk 8–24 of gestation is essential for expression of male sex-typed behaviors in later life. Another peak of in male infants is demonstrated 1–5 months postnatally. The significance of this postnatal testosterone on male sex-typed behaviors remains to be elucidated. Bilateral congenital anorchia represents an example of an individual lacking postnatal testosterone. If postnatal testosterone surge is critical for male sex-typed behaviors, differences should be seen in this group of patients in comparison with men with functioning testes.

Objective: The objective of the study was to examine the psychosexual function of males with anorchia.

Design: This was a cross-sectional study.

Setting: The study was conducted at the Royal Children’s Hospital, Melbourne, Australia, a referral center.

Patients and Interventions: Fifteen young male adults with anorchia and 15 healthy young males were enrolled in the study. All of them completed the same set of questionnaires evaluating general health and psychological health as well as sexuality. All had androgen replacement treat- ment to induce puberty and maintain adult androgen effect.

Main Outcome Measures: General health, psychosocial, and psychosexual functionings according to the validated questionnaires were measured.

Results: There were no differences in age, education, marital status, and identified gender between subjects and controls. No significant differences of general health, psychosocial, and psychosexual functionings of the subjects with anorchia and healthy young adults were demonstrated.

Conclusion: A postnatal testosterone surge does not seem to be critical for male-typed sexual behaviors in men with anorchia when adolescent testosterone replacement has occurred. (J Clin Endocrinol Metab 94: 2502–2505, 2009)

renatal androgen has been known to influence sexual-role with complete androgen insensitivity syndrome, who do not re- P behaviors via its effect on brain organization (1–4). The spond to testosterone, have female-typed behavior (6, 7). These effects of sex hormones on human behaviors have been demon- features support the role of in the development of strated in a variety of conditions. Females with congenital ad- sex-typed behaviors. Moreover, most genetic male cloacal ex- renal hyperplasia due to 21-hydroxylase deficiency have been strophy patients with severe phallic inadequacy but normal reported to exhibit male-typed behaviors with some demonstrat- androgens have moderate to marked male-typed behaviors ing altered sexual orientation (3, 5). In contrast, genetic males despite often having been reared as and living as females (4). Almost all individuals born with hypogonadotropic hypogo- ISSN Print 0021-972X ISSN Online 1945-7197 nadism such as Kallmann syndrome are reported to have nor- Printed in U.S.A. mal sex-typed behavior according to their genetic sex (8). Copyright © 2009 by The Endocrine Society doi: 10.1210/jc.2008-1886 Received August 26, 2008. Accepted April 8, 2009. However, male-to-female transsexual individuals who are ge- First Published Online April 14, 2009 netically male have a female gender identity (9). This may be

2502 jcem.endojournals.org J Clin Endocrinol Metab, July 2009, 94(7):2502–2505 J Clin Endocrinol Metab, July 2009, 94(7):2502–2505 jcem.endojournals.org 2503 evidence against the organizational effect of pre- and postna- Subjects and Methods tal androgens, but gender identity is quite complex in the transsexual population. Fifteen young male adults with anorchia were recruited into the study. All had been treated at the Royal Children’s Hospital between birth and late Weeks 8–24 of gestation have been regarded as a sensitive teenage years and all were seen by one of the authors (M.Z.) for adult period for maximal brain organizational effects, during which follow-up care. All had previously been treated with im and/or sc tes- time there is a peak of testosterone (10). Another peak of tes- tosterone from age 13 to 14 yr to induce normal pubertal growth and tosterone in male infants is demonstrated during months 1–5 virilization. All had continued replacement with sc pellet or im testos- postnatally, which may be another sensitive period for a brain terone as adults after cessation of linear growth for maintenance of adult sexual function and secondary sexual characteristics. All had testicular organizational effect (2, 10). However, the significance of post- prostheses surgically inserted during adolescence. All potential partici- natal testosterone peak for human behaviors has not been well pants were seen for regular care and were physically normal with no studied. other general health problems. Downloaded from https://academic.oup.com/jcem/article/94/7/2502/2597091 by guest on 27 September 2021 The study of genetic males with bilateral congenital anorchia All subjects were sent a letter of invitation and consent form explain- may provide insights into the influence of the early postnatal ing the study. Follow-up phone calls were undertaken to enroll all the testosterone peak on future male sex-typed behaviors. subjects. Participation rate was 100%. Consenting participants received a set of questionnaires that they were asked to complete and return by Bilateral congenital anorchia is a rare condition. It is defined mail in the provided stamped envelopes. All questionnaires were dei- as the absence of testicular tissue in a karyotypically and other- dentified. Written consent was obtained separately at clinic visits for all wise phenotypically male individual. The etiology has not been men before the study. The questionnaires collected sociodemographic completely defined. However, normal male genitalia develop; information along with validated inventories measuring general health therefore, functional testes must have been present early in life and psychological health including interpersonal problems, personality, self-esteem, anxiety, stress, depression, and sexuality (13–23). Details of during the first half of gestation, with testicular loss after 14–16 each inventory are provided in Table 1. wk gestation (11, 12). Patients who have anorchia are raised as Fifteen healthy young male adults were recruited as controls. They males as they are phenotypically male, although some of them were recruited from patients’ healthy siblings when possible or from may have had associated . healthy friends and relatives of Royal Children’s Hospital patients. Sub- If early postnatal testosterone exposure has an influence on jects and controls were matched for age and educational level. The con- trol males were administered the same set of questionnaires and re- male sexual behavior, an alteration in male psychosexual de- quested to complete and return in the same manner. velopment should be seen in this group of patients. There have Completion of the questionnaires was estimated to take 1–2 h. The been no previous reports of psychosexual function in males study was approved by the Royal Children’s Hospital Ethics in Human with anorchia. Research Committee (EHRC 25127).

TABLE 1. Study measures, description, and reliability statistics

Variables Measures Descriptions Reliability General health Rand-36 Health Status Inventory (13) Assesses physical, mental and global health. Total score. 0.95 Relationships Inventory of Interpersonal Problems (14) Assesses interpersonal characteristics such as 0.85 compassion, sociability, assertiveness. Total score. Personality Eysenck Personality Questionnaire Revised Three scales assessing psychoticism, neuroticism, and 0.61–0.88 Psychoticism (15) extroversion. Three subscale scores. Neuroticism Extroversion Self-esteem Coopersmith Self-Esteem Inventory (16) Assesses self esteem. Total score. 0.86 Anxiety state trait State-Trait Anxiety Inventory, form Y (17) Assesses state anxiety, a level of emotional intensity at a 0.93 and 0.94 given moment in time, and trait anxiety, a stable personality characteristic of anxiety-proneness. Two scores. Stress Impact of Events Scale-Revised (18) Rates impact of stressful life events. Total score. 0.94 Depression Beck Depression Inventory, Short Form (19) Assesses severity of symptoms depression. Total raw 0.89 score. Body image Body Parts Satisfaction Scale (20) The scale measures satisfaction with 24 body parts. A 0.91 total raw score provides a measure of overall body satisfaction. Sexual awareness The Sexual Awareness Questionnaire (21) The questionnaire assesses aspects of sexual awareness, 0.89 sexual consciousness, monitoring, assertiveness, and appeal and provides a total summary score. Gender identity Personal Attributes Questionnaire (22) The questionnaire consists of assessment of masculine 0.57 and 0.72 Masculinity Bem Sex Role Inventory- Short Form (23) and feminine personality attributes. 0.86 Femininity The inventory measures gender identity on two subscales: masculinity and femininity.

All scores are raw scores. 2504 Poomthavorn et al. Psychosexual Function in Anorchia J Clin Endocrinol Metab, July 2009, 94(7):2502–2505

TABLE 2. Demographic characteristics of the subject and tionships, extroversion, and body image did not reach criteria for control groups homogeneity of variance. This was accounted for in the ANOVA. Table 3 illustrates general health, psychosocial, and psycho- Patients Controls sexual functionings of the subject and the control groups. Gen- ؍ ؍ Characteristics (n 15) (n 15) eral health and psychosocial well-being, including interpersonal Age, mean (SD) 23.8 (5.2) 23.0 (5.5) problems, personality, self-esteem, anxiety, depression, trauma Education symptoms, and stress were not different between the subjects Secondary school, n (%) 8 (53.3) 7 (46.7) Postsecondary school, n (%) 7 (46.7) 7 (46.7) with anorchia and the control sample. In addition, body image, N/A 0 (0) 1 (6.6) including size and appearance of the sex organs, sexual satisfac- Marital status tion, sexuality, masculinity, and femininity were comparable be- Married/de facto, n (%) 3 (20.0) 4 (26.7) tween the subjects and control group. There was no significant Single/other, n (%) 12 (80.0) 11 (73.3) difference between the two groups identified on any of the vari- Downloaded from https://academic.oup.com/jcem/article/94/7/2502/2597091 by guest on 27 September 2021 Identified gender Male, n (%) 15 (100.0) 15 (100.0) ables examined. Female, n (%) 0 (0) 0 (0) Sexual preference Heterosexual, n (%) 13 (86.6) 13 (86.7) Bisexual, n (%) 1 (6.7) 0 (0) Discussion Homosexual, n (%) 0 (0) 2 (13.3) N/A, n (%) 1 (6.7) 0 (0) Psychosocial outcomes in men with anorchia were not different N/A, Not applicable. from the controls. In our participants, and as is generally the case with other men with anorchia, functional secretory testes had Results been present during the first 14–16 wk of gestation before van- ishing (24). All participants had a male gender identity and gen- Demographic characteristics of the subjects with anorchia and der role. All had androgen replacement during adolescence and controls are summarized in Table 2. One of the controls did not adulthood to induce puberty and to maintain adult androgen indicate his level of education. There were no significant differ- levels and effects. Sexual orientation was predominantly hetero- ences in age, education, marital status, and identified gender sexual with one man reporting he was bisexual. Although the between subjects and controls. One man with anorchia described sample size was small, these features were not different from an himself as bisexual and added the comment that he was very age- and education-matched cohort of normal men. confused and possibly homosexual. Two of the control men de- Organizational effects of testosterone on the human brain scribed themselves as homosexual. take place during wk 8–24 of gestation (10). Our findings imply There were three incomplete data sets, and three separate that the brain patterning of sexual behavior of the participants in participants did not complete the relationships, self-esteem, and this study developed similar to that of normal controls. stress questionnaires. Missing data were not substituted. All vari- Genetic male cloacal exstrophy with early neonatal orchiec- ables were tested for homogeneity of variance using the Levene test. tomy is an another example of similar prenatal and postnatal sex ANOVA compared subjects with controls on outcome measures hormones milieu as those of anorchia. Reiner and Gearhart (4) that met criteria for homogeneity of variance. The variables rela- reported moderate to marked male-typed interests and attitudes

TABLE 3. Results of general health, psychosocial, and psychosexual questionnaires in the subject and control groups

Measures Patients mean (SD) Controls mean (SD) F Significance General health 99.47 (15.43) 102.80 (5.51) 0.645 0.429 Relationshipsa 30.20 (17.51) 36.79 (8.50) Ϫ1.302 0.207 Neuroticism 7.13 (3.06) 5.93 (3.56) 0.986 0.329 Psychoticism 5.07 (1.66) 5.07 (1.62) 0.000 1.000 Extroversiona 4.93 (2.65) 5.07 (1.16) Ϫ1.78 0.861 Self-esteem 13.20 (3.36) 12.00 (3.92) 0.220 0.884 State anxiety 39.00 (14.66) 39.33 (10.47) 0.005 0.943 Trait anxiety 41.20 (15.69) 40.02 (10.15) 0.043 0.837 Stress 34.13 (20.41) 28.50 (15.91) 0.680 0.417 Depression 5.27 (9.36) 5.87 (6.50) 0.042 0.840 Body imagea 69.33 (16.37) 64.40 (10.08) 0.994 0.329 Sexual awareness 64.86 (12.89) 66.20 (17.19) 0.056 0.815 Masculinity PAQ 28.47 (4.82) 27.20 (3.95) 0.619 0.438 Femininity PAQ 29.40 (4.59) 28.40 (3.96) 0.408 0.528 Masculinity BSRI 47.67 (8.33) 44.53 (7.18) 1.216 0.279 Femininity BSRI 48.07 (8.62) 52.40 (7.78) 2.088 0.160

For the scores of sexual awareness, masculinity and femininity Personal Attributes Questionnaire (PAQ) and masculinity and femininity Bem Sex Role Inventory-Short Form (BSRI), higher scores represent more of that characteristic. All scores are raw scores. a These variables did not fulfill criteria for homogeneity of variance. T scores rather than the F statistic are reported. J Clin Endocrinol Metab, July 2009, 94(7):2502–2505 jcem.endojournals.org 2505

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