It was considered a medical triumph 33 years ago when John became Joan.
John was 8 months old when his penis was accidentally destroyed during minor surgery to correct a problem with the foreskin. Doctors advised his horrified parents to raise the child as a girl, and surgery to create female anatomy followed shortly thereafter.
The 1964 operation made headlines around the world. It also became the standard of practice in cases involving freak accidents like John's or when children are born with severely deformed genitals, a rare defect occurring in about 1 out of every 100,000 births.
Now, however, two doctors involved with John's case from the start have tracked the results, and they say the decision to raise John as a girl was all wrong.
John, who reverted to being a male as a teen-ager, had always felt he was a boy, even though he was called Joan, wore dresses and had female genitals, say the authors of the report, published Friday in the Archives of Pediatric & Adolescent Medicine.
In addition to calling into question the practice of sex reassignment, as the treatment is called, the case yields valuable insight about what it means to be male or female. It also provides stark evidence that a person's brain predetermines sexual identity - not one's anatomy or social environment, experts say.
"The question this case doesn't answer but lends data to is: What is the origin of gender identity? How do we know we are a girl? How do we know we are a boy? And when do we know it?" says Dr. William Reiner, a child psychiatrist at the Johns Hopkins Hospital in Baltimore, where John was treated. "John, in spite of being raised as a girl and being treated with hormones and estrogen, said, `Forget it. I'm a boy.' "
Children who undergo sex reassignment provide a unique opportunity to explore the complexities of sexuality, says Reiner, who wrote an editorial accompanying the report. While only one case, John's story is particularly strong testimony that the roots of sexual identity lie deep inside the brain, as more recent scientific studies have attested.
Even as a child, Joan, who had not been told what had happened, said she "felt like a trapped animal."
That was not what doctors would have predicted three decades ago, when notions of sexuality were much different. In proposing the sex-change surgery, doctors told John's parents that infants were sexually neutral at birth and that children learned their gender as they grew up and as hormonal influences kicked in at puberty.
"Doctors haven't been doing this willy-nilly. They have tried to do what they think is the right thing," says Milton Diamond, co-author of the report and an expert in anatomy and psychology at the Pacific Center for Sex and Society, University of Hawaii-Manoa. Diamond served as a consultant to the British Broadcasting Co. in its coverage of the case in the 1960s and 1970s.
Indeed, the surgery on John was followed by a host of reports on the case touting it as a success. The case was quickly written into textbooks on pediatrics, psychiatry and sexuality - where it remains today - as an example that gender should be based on an infant's anatomy, not on chromosomes or the individual's feelings about his or her sexuality.
However, the case received little more attention until the retrospection published Friday, which is heartbreaking in its portrayal of John's and his family's suffering.
The article is based on interviews in 1994 and 1995 with John, his wife and his mother. According to Diamond, John wanted his story told, although he requested the use of the pseudonyms John and Joan.
The article suggests that the sex reassignment was wrong from the outset.
John's mother recalls: "As soon as he had the surgery, the doctor said I should now start treating him as a girl, doing girl things, and putting him in girl's clothes. But that was a disaster. I put this beautiful little dress on him ... and he (immediately tried) to rip it off. I think he knew it was a dress and that it was for girls and he wasn't a girl."
As a girl, Joan rejected almost everything feminine. When her twin brother refused to share his toys, Joan saved her allowance and bought herself a toy truck. She began to suspect she was a boy around the ages of 9 to 11.
"There were little things from early on," John recalled in the 1994-'95 interviews. "I began to see how different I felt and was from what I was supposed to be. But I didn't know what it meant."
Life became miserable for Joan as she neared puberty. She was given estrogen, which made her feel awful. Despite being a nice-looking girl, her voice and movements were strange. So compelling were her feelings of maleness, Joan even attempted to stand to urinate. She was teased mercilessly by other children about her odd looks and behavior.
She continued to be seen by a team of experts at Johns Hopkins and, at 14, told her endocrinologist that she suspected she was a boy. The team, alarmed by her threats of suicide, discussed conversion to being male with Joan and her family.
Joan gladly embraced the suggestion, began taking male hormone shots and completed a mastectomy and phallus construction by 16. She learned the truth shortly thereafter in a tearful discussion with her father.
"John said, `All of a sudden everything clicked. For the first time, things made sense and I understood who and what I was,' " note Diamond and co-author Dr. H. Keith Sigmundson. (Sigmundson, who now works at the Ministry of Health, Victoria, Canada, supervised John's case at Johns Hopkins and kept in contact with the family over the years.)
The family rejected doctors' advice to relocate upon Joan's switch to John. And after some initial sensational reaction, John's peers rallied around him. He developed into a muscular, good-looking young man who attracted girls. While the surgery to create male anatomy was only partially successful, John married at 25 and adopted his wife's children.
Diamond and Sigmundson describe him now as a well-adjusted person with a good sense of humor. He is bitter about what happened to him, Diamond says, but has thrived with the love and support of his family and friends.
Friday's paper should rekindle a discussion of what approach to take with infants born with genital abnormalities, as well as cases of sex chromosome abnormalities, gender identity disorders and metabolic adrenal or testicular errors - all conditions in which sexual identity and orientation can be unclear.
"We need to have a strong discussion on what are our data. Are we doing the right thing (with sex reassignment)? John gives us reason to pause and question what we're doing," Reiner says.