Summary
There are few topics in pediatric urology that have attracted the attention of the general public, as well as the scrutiny of human rights organizations — genital surgery in infants and children with a disorder/difference of sex development (DSD) is such a topic. This article may easily be overlooked by surgeons and other clinical specialists caring for patients with DSD and their families, yet a close reading will help clarify why the controversies regarding surgical decision-making appear intractable.

Liao and colleagues, focusing on clitoral surgery, note that the young “patient” cannot consent to procedures; instead, providers rely on proxy permission of parents or legal guardians. These procedures, while considered therapeutic are, by and large, elective and could be conducted later in life should the person make that choice. Yet the recommended standard since the surgical procedures were available has been to perform these in early life predicated on the assumption that elimination (or reduction) of the atypicality of the visible sex organs will be an important factor in stabilizing gender identity, supporting positive psychosocial adaptation and promoting full sexual function, largely conceived as penetrative intercourse {1}. It is also believed (without systematic evidence) that performing these procedures earlier rather than later in life results in superior surgical outcomes {2-3}.

The reader may be surprised by the authors’ claim that studies of long-term psychosexual outcomes following feminizing genitoplasty have yet to be performed when they go on to summarize a number of reports of poor sexual outcomes in adult women (i.e., anorgasmia, loss of clitoral sensitivity) who had received feminizing procedures early in life.

Highlighted among the studies showing negative long-term consequences of early feminizing genitoplasty is a report from the German multi-site DSD Network Working Group, restricted to individuals with 46, XY karyotype reared as girls. Missing from this limited review are other studies reporting more positive outcomes or, at least, lack of regret that procedures had been performed early {4-6}.

In order to examine the considerations “surrounding clitoral surgery on minors,” the authors conducted a qualitative methods study involving interviews with a range of specialists involved in the clinical management of DSD, all working in either England or Sweden. Study participants were either members of, or affiliated with, multidisciplinary teams for pediatric or adult patients with DSD. Study investigators coded the interview transcripts using a “theoretical thematic analysis.” Key findings included the observation that some participants viewed themselves as “conservative,” i.e. rejecting early surgery, but others less so. The qualitative analysis generated four overlapping strategies the authors perceived as maintaining the status quo of early surgery.

The authors view delay of genital surgery until the patient is old enough to make their own decision as being aligned with the pillars of bioethics. They also note “(t)he consensus statement (referring to {7}) acknowledged the absence of evidence that clitoral surgery succeeded in normalizing anatomy, identity and relationships.” This is unquestionably true, but represents a shifting of the burden of proof; in other words, the absence of evidence is not evidence of absence. In this context, Parkhurst {8} wrote an informative analysis of what is referred to as “wicked policy problems,” i.e. problems characterized by their “intractability,” whereby appeals to evidence are unable to provide policy resolution. To make the current impasse regarding the timing of surgery and related issues in the clinical management of DSD more understandable (even if not soluble), the reader is encouraged to examine Parkhurst’s analysis and detailing of both “technical” and “issue” biases.

7. Consensus statement on management of intersex disorders. International Consensus Conference on Intersex. Lee PA, Houk CP, Ahmed SF, Hughes IA, International Consensus Conference on Intersex organized by the Lawson Wilkins Pediatric Endocrine Society and the European Society for Paediatric Endocrinology. Pediatrics. 2006 Aug; 118(2):e488-500
https://doi.org/10.1542/peds.2006-0738 
PMID: 16882788 (/prime/pubmed/ref/16882788)

8. Appeals to evidence for the resolution of wicked problems: the origins and mechanisms of evidentiary bias. Parkhurst JO. Policy Sci. 2016 Dec; 49(4):373-393 https://doi.org/10.1007/s11077-016-9263-z