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      Experiment of Nurture: Ablatio Penis at 2 Months, Sex Reassignment at 7 Months, and a Psychosexual Follow-up in Young Adulthood

      1 , 2 , 3 , 4
      Pediatrics
      American Academy of Pediatrics (AAP)

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          Abstract

          Guidelines of psychosexual management for infants born with physical intersex conditions are intended to assist physicians and parents in making decisions about sex of assignment and rearing including the following: 1) sex assignment should be to the gender that carries the best prognosis for good reproductive function, good sexual function, normal-looking external genitalia and physical appearance, and a stable gender identity; 2) the decision regarding sex assignment should be made as early as possible, preferably during the newborn period, with an upper age limit for reversal of an initial sex assignment no later than 18 to 24 months; and 3) there should be minimal uncertainty and ambiguity on the part of parents and professionals regarding the final decision about sex assignment and rearing.

          J. Money used these guidelines in a case of a biologically normal male infant (one of a pair of monozygotic twins) whose penis was accidentally ablated during a circumcision at the age of 7 months. The decision to reassign the infant boy to the female sex and to rear him as a girl was made at 17 months, with surgical castration and initial genital reconstruction occurring at 21 months.

          Money reported follow-up data on this child through the age of 9 years. Although the girl was described as having many “tomboyish” behavioral traits, a female gender identity had apparently differentiated. Thus, it was concluded that gender identity is sufficiently incompletely differentiated at birth as to permit successful assignment of a genetic male as a girl, in keeping with the experiences of rearing.

          Subsequent follow-up by other investigators reported that by early adolescence the patient had rejected the female identity and began to live as a male at the age of 14 years. In adulthood, the patient recalled that he had never felt comfortable as a girl, and his mother reported similar recollections. At age 25, the patient married a woman and adopted her children. The patient reported exclusive sexual attraction to females.

          The present case report is a long-term psychosexual follow-up on a second case of ablatio penis in a 46 XY male. During an electrocautery circumcision at the age of 2 months, the patient sustained a burn of the skin of the entire penile shaft, and the penis eventually sloughed off. At age 7 months, the remainder of the penis and the testes were removed. By age 7 months, if not earlier, the decision was made to reassign the patient as a female and to raise the infant as a girl.

          The patient was interviewed on two occasions: at 16 years and twice while in the hospital for additional surgery at 26 years of age. At ages 16 and 26, the patient was living socially as a woman and denied any uncertainty about being a female. During childhood, the patient recalled that she self-identified as a “tomboy” and enjoyed stereotypically masculine toys and games; however, the patient also recalled that her favorite playmates were usually girls and that her best friend was always a girl.

          When seen at age 16, the patient had been admitted to the hospital for vaginoplasty. At that time, she wished to proceed with the further repair of her genitalia to make them suitable for sexual intercourse with males. At age 26, the patient returned to the hospital for further vaginoplasty.

          Regarding the patient's sexual orientation, she was attracted predominantly to women in fantasy, but had had sexual experiences with both women and men. At the time of the second surgery, she was in a relationship with a man and wished to be able to have intercourse. The patient's self-described sexual identity was “bisexual.” After surgery at age 26 years, the patient developed a rectovaginal fistula. Within a few months of the surgery, the patient and her male partner separated for reasons other than the patient's physical problems. The patient subsequently began living with a new partner, a woman, in a lesbian relationship.

          The psychosexual development of our patient was both similar to and different from the patient described earlier. Our patient differentiated a female gender identity; in contrast, the other patient had adopted a male gender identity after experiencing intense discomfort living as a female, apparently around the beginning of adolescence. At the time of interview at age 26, our patient was living with a man, but they subsequently separated and she began a new relationship with a woman; the other patient was married to a woman. Our patient had a “bisexual” sexual identity; the other patient had a “heterosexual” sexual identity. The patients were similar in that they had a childhood history of “tomboyism.” Our patient was predominantly sexually attracted to women; the other patient was exclusively sexually attracted to women. Our patient had sexual experiences with both women and men; the other patient had sexual experiences only with women.

          The most plausible explanation of our patient's differentiation of a female gender identity is that sex of rearing as a female, beginning at around age 7 months, overrode any putative influences of a normal prenatal masculine sexual biology.

          Because cases of ablatio penis in infancy are so rare and long-term follow-up data are scant, it is obviously impossible to know whether our patient or the previous case would be more typical of the psychosexual outcome in a larger sample of such individuals. However, our case suggests that it is possible for a female gender identity to differentiate in a biologically “normal” genetic male. At present, however, the clinical literature is deeply divided on the best way to manage cases of traumatic loss of the penis during infancy. Further study is clearly required to decide on the optimal model of psychosocial and psychosexual management for affected individuals.

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          Most cited references27

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          Human behavioral sex differences: a role for gonadal hormones during early development?

          Evidence that gonadal hormones during prenatal and neonatal development influence behavior is reviewed. Several theoretical models of hormonal influences, derived from research in other species, are described. These models are evaluated on the basis of data from humans with either normal or abnormal hormonal exposure. It is concluded that the evidence is insufficient to determine which model best explains the data. Sexual differentiation may involve several dimensions, and different models may apply to different behaviors. Gonadal hormones appear to influence development of some human behaviors that show sex differences. The evidence is strongest for childhood play behavior and is relatively strong for sexual orientation and tendencies toward aggression. Also, high levels of hormones do not enhance intelligence, although a minimum level may be needed for optimal development of some cognitive processes. Directions for future research are proposed.
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            Psychosexual development of women with congenital adrenal hyperplasia.

            Women with congenital adrenal hyperplasia (CAH) (N = 31) and their unaffected sisters or female cousins (N = 15) participated in a study of psychosexual development. All participants were > or = 18 years of age (mean age, 25 years; range, 18-40). Comparisons were also made between the CAH women with the salt-wasting (SW) form of the disorder and those with simple virilization (SV). A psychosexual assessment protocol examined six variables: (1) sex assignment at birth (probands only); (2) recalled sex-typed behavior during childhood; (3) gender identity and gender role identification in adulthood; (4) relationship status; (5) sexual orientation in fantasy; and (6) sexual orientation in behavior. Salt-wasting status and sex assignment at birth were also ascertained for the CAH women who either refused to participate in the study (N = 10) or could not be traced (N = 13). Compared to the controls, the women with CAH recalled more cross-gender role behavior and less comfort with their sense of "femininity" during childhood. The two groups did not differ in degree of gender dysphoria in adulthood, although the probands showed more cross-gender role identification. Three of the nonparticipant probands were living, as adults, in the male social role (2 reared from birth as boys and 1 who changed from the female to the male social role during adolescence). The CAH women and the controls did not differ in relationship status (married/cohabiting vs. single). The CAH women had lower rates of exclusive heterosexual fantasy and fewer sexual experiences with men than the controls; however, the CAH women did not have more sexual experiences with women than the controls. Comparisons between the SW and SV revealed several differences: the SW were less likely to be assigned to the female sex at birth, recalled more cross-gender role behavior during childhood, were less likely to be married or cohabiting, and had lower rates of sexual experiences with men. The results were discussed in relation to the effects of prenatal androgens on psychosexual differentiation.
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              Early Androgens Are Related to Childhood Sex-Typed Toy Preferences

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                Author and article information

                Journal
                Pediatrics
                American Academy of Pediatrics (AAP)
                1098-4275
                0031-4005
                July 01 1998
                July 01 1998
                July 01 1998
                July 01 1998
                : 102
                : 1
                : e9
                Affiliations
                [1 ]From the Department of Psychiatry and
                [2 ]Division of Gynaecology, Hospital for Sick Children, Toronto, Ontario, Canada;
                [3 ]Private Practice, London, Ontario, Canada; and the
                [4 ]Child and Adolescent Gender Identity Clinic, Child and Family Studies Centre, Clarke Institute of Psychiatry, Toronto, Ontario, Canada.
                Article
                10.1542/peds.102.1.e9
                9651461
                31709911-0349-424c-8f59-5b124c150496
                © 1998

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