Input: Could not generate response.

Response:
In the treatment literature for children with GD, three broad approaches appear: (1) psychosocial treatments designed to reduce the GD in order to increase the likelihood that a long-term gender identity will be more congruent with the natal sex; (2) a neutral or "watchful waiting" approach in which the clinician suggests that the child be given time to sort out his or her gender identity, in one way or another, but without active interventions; and (3) psychosocial support for an "early" gender social transition, in which the child is given the opportunity to live in the social role of the "desired" gender (e.g., via a name change, presenting socially via hairstyle and clothing style in the desired 226

gender, etc.).

These three treatment approaches need to be understood in relation to underlying theoretical, philosophical, and social-value perspectives (for a broad overview of these approaches, see the guest-edited volume by Drescher & Byne, 2012, in the Journal of Homosexuality).

The first approach, which Dreger (2009) has characterized as the "therapeutic model," has the longest history. It has also taken many forms: behavior therapy, psychodynamic therapy, peer group therapy, family therapy, parent counseling, and interventions in the naturalistic environment carried out by parents with input from the clinician. From a theoretical perspective, psychosocial treatments have likely assumed that the childs GD is not yet fixed and thus amenable to change (Zucker, Wood, Singh,

& Bradley, 2012). From philosophical and social-values perspectives, clinicians probably assume that a childs life would be easier and less complicated if a gender identity that matches his or her birth sex could be achieved. For example, it would reduce the social ostracism that many of these children experience because of their cross-gender or gender-variant behaviors (Meyer-Bahlburg, 2002), and it would avoid the complex medical treatments required to achieve a gender transition. As noted by Cohen-Kettenis and Pfäfflin (2003), "Relatively little dispute exists regarding the prevention of transsexualism" (p. 120). Although much less apparent in the contemporary treatment literature, some clinicians have also advocated treatment with the expressed goal of preventing homosexuality (see Zucker, 1990). Although it has been noted for years that there is little evidence that treatment of children with GD alters their eventual sexual orientation (see Green, 1987), the contemporary clinician must be mindful that parents vary tremendously in the degree to which they would be able to accept and support their child if he or she later developed and expressed a gay or lesbian sexual identity: for some parents, it is not an issue; for other parents, it is an outcome that they eventually are able to embrace; but for other parents, it is an outcome that causes intense anxiety (for a whole host of reasons—personal, cultural, religious, etc.). Thus, there is considerable room for psychoeducational discussion with parents about what one might expect from a psychosocial treatment designed to reduce GD.

Although there is a reasonably large literature using psychosocial treatments, a perusal of it yields the sobering fact that there is not even one randomized controlled treatment trial for children with GD (Byne et al., 2012). Although there have been some treatment effectiveness studies, which might qualify as Level II standards (e.g., "evidence obtained from well-designed cohort or case-control analytic studies"), much is lacking in these investigations (Zucker, 2008). To put it plainly: There is a large empirical black hole in the treatment literature for children with GD. As a result, the therapist must rely largely on the "clinical wisdom" that has accumulated in the case report literature and the conceptual underpinnings that inform the various approaches to intervention.

In contrast to the psychosocial treatment approaches designed to reduce GD in children, the recent emergence of a therapeutic approach that supports an early gender social transition, which Dreger (2009) has termed the "accommodation model,"

provides the contemporary clinician (and parents) with a very different conceptual perspective (e.g., Vanderburgh, 2009; see also the essays in the edited volume by 227

Drescher & Byne, 2012). In this approach, there appears to be an underlying theoretical assumption that the childs gender identity is fixed and unalterable, likely rooted in some type of biological cause. Thus, any attempt to try to change the childs gender identity is viewed with great skepticism. Interestingly, this therapeutic approach nowadays is the one that receives the most media attention (Padawer, 2012; Rosin, 2008), and it certainly dominates Internet discourse.

An interesting and important empirical question is whether or not these three approaches will result in different long-term psychosexual outcomes for children with GD. Steensma, McGuire, Kreukels, Beekman, and Cohen-Kettenis (2013) have now shown that children with GD who had socially transitioned prior to puberty were more likely to persist in their GD by midadolescence than children who had not and that early social transition as a predictor variable accounted for unique variance in predicting outcome. In itself, this is an important finding. What is less clear at present is whether or not these different treatment approaches are predictive of variation in more general psychosocial and psychiatric adjustment.

**Adolescents**

Since the mid-1990s, one model of therapeutic care, developed by Dutch clinicians and researchers, has been to initiate the biomedical aspects of sex reassignment in early to mid-adolescence, rather than waiting for the legal age of adulthood (18 years in many countries) or even later. After careful psychological evaluation, adolescents deemed appropriate for such treatment are prescribed hormonal medication to delay or suppress somatic puberty (prior to the age of 16 years). If the GD persists, then cross-sex hormonal therapy is offered at the age of 16 years, and, if the adolescent so desires, surgical sex change procedures are then offered at a lower-bound age of 18 years (Cohen-Kettenis, Steensma, & de Vries, 2011; Zucker et al., 2011).

The rationale for this treatment protocol includes the following assumptions: (1) for some adolescents with GD (and perhaps even the majority), there is little systematic empirical evidence that psychological interventions can resolve it, even if the adolescent desires this; (2) the use of hormonal blockers can be helpful to the adolescent because it reduces the incongruence between the development of natal-sex secondary physical characteristics (e.g., in males, facial hair growth, hair growth on other parts of the body, deepening of the voice; in females, breast development, menstruation) and the felt psychological gender, thereby reducing stress; (3) reduction of the incongruence makes it easier for adolescents to present socially in the cross-gender identity/role (when they so desire), which is also helpful in reducing stress during the gender transition process.

Because the suspension of the patients biological puberty reduces the preoccupation with it, it has also been argued that this affords the adolescent greater opportunity to explore his or her longer term gender identity options in psychosocial counseling or psychotherapy in a more reflective and less pressured manner.

The sequence of this biomedical treatment is progressively irreversible. On the one hand, the use of hormonal medication to suppress or delay puberty is a reversible 228

procedure; on the other hand, surgical interventions (e.g., in males, vaginoplasty; in females, chest surgery) are irreversible. Accordingly, if clinicians are going to support adolescents with GD in moving down a pathway that, in the end, results in a completely irreversible intervention, it is important to have a relatively high degree of confidence that the likelihood of regret will be low.

In the Dutch model, several factors have been identified as important in deeming an adolescent eligible for early biomedical treatment. According to Cohen-Kettenis, Delemarre-van de Waal, and Gooren (2008), these include the following: (1) the presence of GD from early childhood on; (2) an increase in the GD after the first signs of puberty; (3) the absence of psychiatric comorbidity that would interfere with a diagnostic evaluation or treatment; (4) adequate psychological and social support during treatment; and (5) a demonstration of knowledge of the sex-reassignment process.

The use of hormonal blockers to treat adolescents with GD has been well received in the professional literature (Hembree et al., 2009). There are, however, a number of uncertainties that require further explication. Perhaps the most acute issue is how to best identify adolescents deemed eligible for early biomedical treatment from those who are not. As noted earlier, one criterion used by the Dutch group is a history of GD from early childhood on. Yet, in clinics such as our own, we see some adolescents with GD

who showed very little or absolutely no evidence of GD in early childhood (Zucker, Bradley, et al., 2012). In many respects, the presentations of these adolescents resemble the "late-onset" form of GD that has been described in the literature on adults (Lawrence, 2010). The GD appears to emerge, at least in the eyes of significant others (e.g., parents, therapists who have known the patient since childhood), only after the onset of puberty. It is not clear whether this late-onset group should be deemed ineligible for early hormonal therapy. Other adolescents have a history of pervasive cross-gender behavior during childhood but without apparent GD until adolescence. It is unclear whether a childhood history of pervasive cross-gender behavior without the explicit wish to be of the other gender would count as an example of "early onset" in the Dutch model.

Another issue that deserves consideration concerns the Dutch groups view on the role of psychiatric comorbidity in making treatment decisions about early biomedical interventions. It is, for example, unclear what is meant when it is stated that the presence of such comorbidity interferes with a diagnostic evaluation or in what ways the presence of such comorbidity interferes with treatment.

There are several ways to conceptualize such comorbidity. In some instances, it may be that the GD has emerged as secondary to another, more "primary" psychiatric disorder, such as autism spectrum disorder or borderline personality disorder, or as a result of a severe trauma (e.g., sexual abuse). In such situations, it could be argued that the GD would dissipate if the more primary condition was treated. In other instances, it could be that the presence of other psychopathology (e.g., substance abuse) would interfere with the adolescents ability to adhere to a biomedical treatment and that there would be risks in trying to institute a regimen of hormonal therapy until stabilization was achieved. Last, there is the thorny issue regarding the extent to which the presence of other psychopathology (e.g., depression, suicidality) is due to the stress of having GD

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or is secondary to the social ostracism and rejection that results from it. On this point, one could argue that institution of treatment of the GD may reduce the secondary psychopathology.

Given that that there are likely multiple reasons as to why adolescents with GD also present with other kinds of psychiatric issues, the clinician needs to formulate the extent to which these other difficulties are merely secondary to the GD, may actually be fueling it, or are related to other factors (e.g., a biological predisposition, family psychopathology). This kind of case formulation can help the clinician decide whether biomedical treatments should be instituted after the diagnostic assessment or delayed until the other issues can be worked through.

**Case 1**

Katie was a 15-year-old natal female with a childhood history of cross-gender behavior but without an expressed desire to be a boy. Her childhood was chaotic: Katies biological mother left the family without notice when she was 4. She was raised primarily by her biological father, who was preoccupied with his own physical health and mental health (substance abuse, depression, etc.). At the age of 12, Katie was sexually abused by a male neighbor. Three years later, Katie could still not talk about it.

During the assessment, her father fell asleep while the issue was being explored. Shortly after the abuse, Katie began to masculinize her phenotypic appearance and informed her father that she was thinking about a sex-change operation. In addition to her emerging GD, Katie began to manifest other behavioral and emotional issues: She would run away from home after arguing with her father, was often truant from school, and engaged in a lot of self-harming behavior (e.g., cutting). At the time of the assessment, Katie indicated that she was attracted sexually to girls and was trying to sort out if she was

"just a lesbian" or was "really" transsexual. Katie commented: "I know why I want to be a guy. Its to protect myself. . . . I know its related to the abuse." In Katies case, we did not recommend hormonal treatment but recommended more psychosocial treatment to stabilize her from a psychiatric and psychological point of view and to provide her with a therapeutic space to work through the trauma that resulted from the sexual abuse and to explore further her gender identity and sexual orientation.

**Case 2**

Mark was a 16-year-old natal male with no history of cross-gender behavior in childhood. He began to express a desire to be female at the age of 15. His sexual orientation was, if anything, for females, but Mark described himself as "asexual." He did not report any behaviors associated with fetishistic cross-dressing, and there was no history of taking his mothers undergarments or the clothing of other females. His parents noted that in childhood he had had a lot of social difficulties. Although a gifted youth, with a superior-range IQ, Mark had a lot of trouble with social cues and was very 230

rule bound. He preferred to stay at home with his mother (who was a homemaker) and play with his older sister. He never had a best friend in childhood. During childhood, he was seen by several psychiatrists, who questioned whether he might have Aspergers disorder. At the time of assessment, Mark had developed sufficient social skills so that he could interact with other gifted, "nerdy" youth. Much of their interactions revolved around online computer games. Mark had never actively cross-dressed in womens clothing or attempted to pass in the female gender role. At assessment, he wore his hair long, which often concealed his eyes, but this was as much to "hide" himself from others when he was anxious as it was to have a hairstyle similar to some teenage girls. He was unable to verbalize why he wanted to become a woman other than "I just dont fit in as a male." His father commented that he was puzzled as to how Mark could say that he

"felt" like a woman because he did not seem to understand the feelings of other people.

The clinical impression was that Mark met criteria for an autism spectrum disorder.

Hormonal treatment was not recommended. Mark agreed to a trial of psychosocial therapy in which his gender-dysphoric feelings could be explored further.

**Adults**

Therapeutic guidelines for adults have long adhered to the WPATH SOC (Coleman et al., 2011). Here we summarize some of the significant changes in Version 7. The guidelines no longer require a diagnosis for medical interventions, only that the GD be

"persistent and well documented." This reflects a significant shift from seeing somatic treatments as only for those with severe GD and opens the way for individuals who may identify as more "genderqueer" (i.e., as not strongly either gender or as psychologically between genders) to receive interventions. Practically speaking, this may mean more expression of gender variation through the use of hormone therapy; however, many public health care models and insurers require a diagnosis for surgical interventions.

The previous SOC had the real-life experience (RLE) eligibility criterion, which necessitated individuals to be involved in full-time work, school, or volunteer activities in their chosen gender. The idea was driven by the importance of people having a "lived informed consent," that they adjusted socially and understood the consequences of transition before beginning a medical intervention with irreversible aspects. The wording grated on many community members ("This is my real life!"), and some felt the attitude behind it paternalistic and representing an undue hardship on trans women, who had a more difficult time integrating socially without physical intervention. The criterion also de facto required people to be functional. This, too, was criticized as holding trans people to an unfairly high standard (i.e., "If cisgender people [i.e., those whose gender identities align with their natal sex] can stay at home on social assistance, why cant trans people?"). The spirit of the RLE is carried forward into the new language of a continuous gender-role experience for genital surgery only, opening up alternatives for people to document their lives and understanding of the social consequences of transition, offering time to adjust and adapt to the challenges inherent within. Readiness for hormones and surgery is captured in the stipulation that mental health issues be 231

"well controlled."

The SOC represent minimum standards and leave significant room for providers or sites as to how they will be interpreted. Because this will unavoidably lead to some amount of discrepancy in philosophy and practice, it has led to discussions in communities with multiple providers. Some providers are working on an informed consent model for hormones in the most liberal sense (i.e., "Is the person mentally capable of consenting to treatment?"), with less attention to readiness per se, which is what others consider holistically (e.g., Has a person reasonably anticipated, and is he or she mentally prepared for, the potential consequences of this decision? Is he or she sufficiently psychiatrically stable, and is social support adequate to help him or her with the ways in which his or her life might become unraveled?). These discrepancies have resulted in some psychiatrically vulnerable patients pursuing a surgery track who do not meet the higher standard for surgery and are left in physical limbo. Another tension in this work is differences in what might be considered to be medically necessary and what government health policies consider to be medically necessary and, therefore, insured services. One example of this seen in Canada is considering the mastectomy portion of a female-to-male chest surgery medically necessary, while the chest contouring is seen as cosmetic in nature. Surgeons make no distinction; they are considered part of the same surgery, and they will not provide one without the other because it would lead to poor results, such as the possibility for concave depressions in the chest. Our gender identity clinic team thinks a certain amount of cosmesis is medically necessary to relieve the dysphoria, but these are larger policy issues.

**Subtypes and Gender Differences**

There has been a significant amount of controversy in the literature and at the community level regarding the existence of "subtypes" of those with significant GD and their meaning. In brief, researchers have identified a taxonomy largely organized by natal sex (male, female) and sexual orientation (most often seen as "homosexual" vs.

"nonhomosexual" in the literature). These have also gone by other names in the literature: primary versus secondary and early versus late onset.

MALE-TO-FEMALE TRANSSEXUALS

Trans women (MTF) are considered more variable in presentation than trans men (female-to-male [FTM] transsexuals). Among these, the most controversial concept may be that of autogynephilia, based largely on the work of Blanchard (1989) and subsequently Lawrence (2009, 2013). Autogynephilia is specific to male-to-females and is characterized by sexual arousal to the idea of being a woman—an "erotic target location error" in which a male himself becomes the object of his (heterosexual) desire (Lawrence, 2009). It casts the motivation for transition in these cases as largely an erotic, and indeed paraphilic, one. Further to this, autogynephilia is proposed as a sexual 232

orientation. Features considered typical of autogynephilia include more male gender-typed childhoods, a primary attraction to women, and a history of erotic cross-dressing that evolves into a sustained cross-gender identity.

This concept has been incredibly politically laden and has also emerged within trans community circles as a divisive issue. Some high-profile women of trans experience have supported the concept as an accurate representation of their experience, whereas others have felt that the concept was "insulting to their senses of self" (see Dreger, 2008) and highly pathologizing, delegitimizing their identities. There is less debate that, for many who transition later in life, sexuality has played a developmental or mediating role in coming into their gender identities. But what this means, or what can reasonably be thought of as causal, continues to be strongly contested. Some women of trans experience describe this sexual element as a kind of phase that passes or as one that plays an important role in the exploration or clarification of their identity. Some have likened the initial practice of embodiment as having a transcendental or charged quality without being reducible to sexual desire per se (Dreger, 2008).

There has been significant narrative and clinical evidence for varied presentations, with significantly more attention paid to variations among MTFs. In clinical practice, although we may describe cases as early or late onset for the purpose of communicating someones gender identity trajectory, it serves little functional value in terms of the evaluation itself from an eligibility perspective regarding somatic treatments, such as hormone therapy and surgery. It is useful insofar as anticipating the differing life circumstances.

Early- and late-onset trans women tend to have different vulnerabilities and strengths. Early-onset trans women often recount significant histories of social exclusion and harassment over long periods of time. They tend to have high degrees of social anxiety and may be less socially skilled on account of having lesser practice within peer networks. Some of this may be resolved through transition, but there is typically quite a bit of residual work regarding grieving the experiences they have missed out on, low self-esteem, and the anxiety of being discovered as trans, as early transitioners often choose to live "stealth" (i.e., not disclose their transition history to most others). If their families are not supportive, this group is much more vulnerable to homelessness, to using substances to cope, and/or to survival sex work. Late-onset trans women have mostly grown up with fairly traditional masculine childhoods and the psychological steadiness and resilience that acceptance and "fitting in" (at least from the outside) can bring. Contemplating transition is often frightening, as the stripping of privilege and potential losses in relationship and employment can be sudden and staggering. If they lose core parts of their lives, which many do, there may also not be an easy transition into new communities or employment opportunities. Many could be helped with grief work and/or by finding new supports or activities.

FEMALE-TO-MALE TRANSSEXUALS

Most trans men are heterosexual in their sexual orientation, that is, desiring of women.

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Although some of them partner with heterosexual women, others partner with queer-identified women. The reason may be that queer-identified women are less invested in gender and bodies per se in considerations of partnering. It may also be that a significant percentage of trans men have histories in the lesbian community. Their initial identity trajectories are in many ways similar to those who later identify as lesbians (Rowniak & Chesla, 2012). Because there is a fair amount of acceptable gender variance in queer womens communities (and, indeed, sometimes an overvaluing of masculine-type gender expression and presentation), some FTMs initially believe that they belong in the lesbian community. Some will figure out, based on the degree of their body dysphoria and/or discomfort with how they are responded to culturally or sexually, that they are not similar to lesbians. The exploration of this conflict may be difficult to untangle from internalized homophobia in some cases by client and clinician alike. Some FTMs already know they are not lesbian but may take on "lesbian" as a transitory identity to "test the waters" in terms of social acceptance and/or bide some time for themselves as they strengthen their internal and external resources necessary for the often overwhelming task of coming out to others as transgender. Still, precisely because many have been involved in communities of difference and marginality, there is typically more open-mindedness in their social circles, and many can retain core aspects of their existing communities if they wish.

A significant minority of trans men identify as gay, that is, desiring of men (and an even lesser percentage as bisexual or pansexual). In our experience, this group has had less success integrating socially within gay mens communities and finding sexual partners among cisgender men whose cultures tend to be more body focused (and phallocentric, in particular). We more typically see gay trans men partnering with each other.

Trans men as a group tend to fare better psychiatrically than trans women. This may be because they have been, relatively speaking, subject to less relational trauma than trans women. Tomboyish girls enjoy a certain normativity that effeminate boys do not.

When FTMs decide to transition, they can sometimes pass for young men with changes to their appearance alone, and testosterone often creates significant masculinization over a relatively short period of time. Although this minimizes the time that they are visibly transitioning, gay trans men and trans men of color will be adapting to a new social identity role in which they may be subject to more overt discrimination than before. Because of this, they may need help navigating social spaces or responding to harassment, but their histories may have both instilled a decent sense of worth from which to work and already taught them about how discrimination operates (through previous experiences of sexism, homophobia, or racism).

**Specific Therapeutic Issues**

Clarifying sexuality from gender identity may be one of the important therapeutic tasks in working with GD, particularly in cases with young adults. Despite the fact that they are separate constructs, in practical terms the edges of their boundaries are sometimes 234

not. For example, some younger trans women interested in men have talked about a period of community and sexual exploration to better understand whether they might be a very feminine gay man or a woman. Similarly, some younger trans men interested in women have done similar exploration to better understand the shadings between being a very masculine lesbian woman and a man, especially when some amount of body dysphoria may be common among some sexual-minority subgroupings. Desire is often relational, and issues of admiration and wanting to "be like" can sometimes get confused with sexual desire. Some of our FTM clients talk about being curious and interested in men pretransition and that only in retrospect did they see their sexual involvement with them as being more an opportunity to explore mens bodies and to learn, more in a kind of role modeling or mirroring process. In getting up close to see what they wanted for themselves, it allowed them to more easily imagine how they might proceed, appear, and behave. This is not to say this is always or even mostly the case, just that it is another developmental phase for some clients in which distinctions between sexuality and gender identity may feel less clear.

**Case 3**

Edward was a 22-year-old natal female who presented for approval for hormone therapy and chest surgery. He had already undergone a legal name change and was presenting male insofar as possible. He remembered early fantasies of being a boy and being closer to his male cousins and enjoying sports that he could not play with his sisters. He was said to be quite "beautiful," received a lot of praise, and knew his parents were happy to see him presenting as more feminine. He joined the Reserves when he was old enough, resenting being in the female divisions and taking secret pleasure when people took him for a boy. Puberty was uncomfortable, but he began to realize that his discomfort was not the same as that of his peers—many of them seemed to want the changes and faster, but he did not want them at all. He felt alone in realizing that, despite the complaints they had, girls did not "hate" being girls as he did. He did not like the uneven look of his growing chest and was revolted by the idea that this was understood to be sexually desirable. He wanted, even expected, a deeper voice and was disappointed that this did not come to be and jealous of his male peers. He did not feel likeable and went through a phase of presenting as more feminine to try to attract male attention. In fiction and fantasy, male couplings were the most exciting, and he would place himself in one of the characters. He dreamed about his body being different and would be angry to wake up and find it was not so. It struck him as important that he felt normal and happy in these dreams. He did some reading on the Internet and found information about FTMs/trans men, which was not as easy to find as it was on MTFs/trans women, but for some time he did not want to think he was "like this." He understood that the surgeries would not be perfect, and this reality was also difficult for him to accept. His first true introduction was a drag performance, which included someone who identified as transgender, and he spoke about his feelings openly for the first time. He bought his first binder and packer, things that brought him much more comfort, confidence, and joy. After some failed 235

attempts at dating mostly men but some women, he began a relationship with a male who was aware of how he felt, was reportedly supportive, and addressed him using male pronouns. He wished to pursue chest surgery before hormones.

Trans people with sexual-minority identities have more identity work to do and are sometimes less understood by others (hearing iterations of "If you want to attract men, why go through the trouble of transition?"). Indeed, in this vignette, there was a small period of time that this client feminized his gender presentation to be able to enact aspects of his sexuality (albeit likely unknown to the former partners, who may have themselves experienced it as a heterosexual encounter). This tends not to be satisfying in the long term, for a holistic understanding of attractions and sexual activity involves the interaction of both gender and sexuality. The kind of sexual activity, the way it is enacted, and how one relates and is related to in the interaction, the sexual language used, and the degree to which sex can be embodied are all factors that contribute to our experience of the quality and enjoyment of sex and also can powerfully affirm or undercut our sense of gender.

Shame and loss often feature prominently in early stages of help seeking, sometimes especially so for trans women who present or transition later in life. This difficult affective combination increases the sense of internal conflict regarding a decision to live in a manner congruent with ones identity. It can also increase the risk for suicide, because the patient may feel trapped between two seemingly felt impossibilities: living what feels like a lie and keeping ones life intact, albeit fairly miserably, or living congruently and likely facing rejection by family, as well as significant losses in social status and networks. When womens gender identities have evolved from a history of what they previously felt was cross-dressing, many come to us with a sense of failure of control and/or questions about how to stop their behaviors or desires. A number of them have gone through cycles of acquiring and purging clothes and makeup and report feeling quite guilt-ridden.

**Case 4**

Fatima, a natal male in her 30s, presented as female at the assessment. It was the first time she had presented to others as female, despite many years of practice in secret, and she had told no one other than her doctor, who referred her. Growing up, she was more effeminate (and has worked to keep a petite frame), and she wished to be doing what girls of her age were doing. Her family caught her once as a teen in makeup and womens clothing and threatened to have her institutionalized. There was taunting about the incident, but the family also considered it a phase. Having immigrated from a South Asian country years ago, Fatima was now married to a woman and had two young children. She felt more comfortable being feminine and wanted to be female, but she felt transitioning would go against her Muslim faith, and she also worried about losing custody of her children. As time went on, she had felt increasingly desperate and depressed, particularly because her marriage had meant that she had had less 236

opportunity (she kept female clothes in her car), and she found thoughts of transitioning fairly preoccupying in her day-to-day life. She and her wife do not have an emotionally intimate life. Fatima tried to bring up her dysphoria indirectly: She shaved her legs and once tried on her wifes undergarments in her presence to see if she would ask her about it (she didnt). She and her wife had a sexual life, despite Fatimas discomfort having sex that involved her penis. As sexual attractions often remain stable through transition, she worried about another religious conflict if she transitioned: reconciling her attraction to women, which as a woman would be same sex, also inconsistent with her current beliefs.


Input: Could not generate response.

Response:
We felt clear in a diagnosis of GD—her feelings had been consistent and persistent, despite her concerns over them. She had significant dysphoria both for social role and her body, her genitals in particular, which she used somewhat reluctantly and with great discomfort. Living incongruently was negatively affecting her happiness and productivity at work. In initial therapy sessions, she alternated between wishing "to be cured" and hoping that we could help her be happy as a man and wanting to start hormones before she told her wife. Part of our initial work is psychoeducational, in sharing more about what the diagnosis means and that it is not something to be cured; that our role is not to rid her of these feelings but to help her come to terms with them.

This is different from having any investment in what clients then do with this diagnosis.

Some of our clients decide living congruently is the most important thing; to many, it no longer feels like a decision at some point. Some of our clients continue to live with these feelings without acting on them publicly because they decide that their marriages or relationships with their children are too important to risk (the latter of which we mean only in emotional terms—there is legal precedence whereby gender identity is not a determinant of child custody). As with working with any issue involving ambivalence, we do clients a disservice in becoming "cheerleaders"; taking up one side often only results in their taking up the other side. It is important that they come to their own resolution.

Patients may present in treatment at any one of a number of stages, but as with therapy in general, many individuals or couples are typically motivated by an urgent matter or crisis. Some may present when there has been a disclosure or discovery (e.g., if the dressing has been secret, a wife finds the clothes and believes her spouse is having an affair). Sometimes, gender variance has been an open feature of a relationship, but one or both parties have assumed it to be "just that"; having a partner consider transition puts a very different frame on the future of their lives together. In some of these cases, one or both parties knew of transgender feelings but believed they could live without transition and/or that marriage would rid them of the feelings. There may also have been a genuine evolution, from being satisfied with less gender expression to feeling that it is not enough. Levs (2004) volume reviews a number of helpful case illustrations of working with couples in which one partner has gender questions or concerns. Brown (2010) has reviewed the literature on partners experiences, both in cases of MTFs and FTMs, which may also be helpful to providers.

Many relationships break up under these circumstances, but a good minority do not.

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It is more common for FTMs in relationships with cisgender women to stay together.

We have also seen a number of MTFs in relationships with cisgender women stay together through transition, albeit infrequently. Of these couples weve seen many have had ongoing quality communication and have taken transition slowly. Some people continue on as life partners without a sexual component to the relationship. Some maintain sexual intimacy and adapt to a new life being seen as a lesbian couple. Our use of "being seen as" is not meant to demean the legitimacy of this, only to draw attention to the fact that the cisgender spouses may not identify this way and may continue to think of themselves as being part of a "mixed orientation" dyad. In other cases, they do truly renegotiate their sexual orientations.

There may also be negotiations regarding the gender expression of the trans-identified spouse and mutual compromises to come to regarding when or how much the relationship context can manage. We hear about peoples decisions to not transition or to delay transition until their children reach a certain age. An interesting case study is presented in Levine and Davis (2002), in which a trans woman made a series of returns to the male role for her wifes career events as what she could conceptualize as a "gift of love." Similarly, one of our clients dresses more plainly at home as part of honoring her partner, motivated by her interpretation of fairness in which she feels her wife deserves the right to see and connect with more of the person she agreed to marry.

If a relationship breaks up, one may also see people struggling both with their grief and with the worry of there being a smaller dating pool in the future. Our experience is that this is based in some reality; some people have partnered successfully (more so with women), whereas others have been single for years and describe significant loneliness.

A number of trans women partner with men. A number of these men appear to have a heterosexual orientation, particularly those partnered with early transitioners, and are able to incorporate a trans body into their sexuality. Men partnered with trans women who transition later in life may have paired with them because of their preoperative status. This may be an explicit aspect of their connection, having met, for example, on a trans-specific dating website. This may also be a covert aspect of a relationship, which emerges we suspect when women approaching surgery reveal that their partners are concerned and may be couching their resistance in loving terms—"I love you just the way you are. The surgery is so risky. Are you sure you want to go through with this?"

We try to prepare people for the possibility that their relationship may end as part of undergoing sex reassignment surgery. Certainly, women who date preoperatively have mixed experiences, the majority sharing relatively disappointing stories of men who appear to be interested primarily for sexual encounters rather than relationships; they may say that there is a strong fantasy or fetishistic aspect to the interaction in which the men are quite interested in the anatomy that the woman wishes to be rid of. Some see it as a concession of types—a price to pay in more of an exchange framework of intimacy.

As one of our clients said, "Its bittersweet."

**Regretters**

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In carefully evaluated patients, narrative literature reviews have estimated the regret rate for sex reassignment to be quite low—about 2%—and "dissatisfaction" with surgical treatment to be also quite low—about 4% (Gijs & Brewaeys, 2007). Gijs and Brewaeys noted, however, that these are likely lower-bound estimates of dissatisfaction and regrets, because a substantial percentage of postsurgical patients are lost to follow-up.

Dhejne et al. (2011) used a population-based registry in Sweden and found that the overall mortality for sex-reassigned patients was higher during follow-up than for controls of the same sex, with an adjusted hazard ratio of 2.8, and with death from suicide particularly high, with an adjusted hazard ratio of 19.1.

Lawrence (2003) reviewed the follow-up literature and some of the methodological problems associated with these studies. Research has identified several factors relating to an increased likelihood of postsurgical dissatisfaction or regret. Of preoperative factors relating to developmental trajectory, these included older age, late-onset desire for SRS, and a history of fetishistic cross-dressing. Of preoperative factors relating to compliance with established treatment, the evidence is more limited, but it includes a lack of social experience in the desired gender role and noncontinuous hormone use. Preoperative factors relating to the psychosocial environment included coexisting psychopathology and poor social support. Of postoperative factors, the most significant factor associated with dissatisfaction or regret relates to poor surgical outcome.

In our clinic, we try to elicit and assess the expectations of all candidates as part of our work. Where necessary, our approach is to be clear and factual. For the FTM who believes he can secure a heterosexual woman who would not know of his transsexual history, he must come to terms with the fact that he will have a scarred body that will invite questions and require explanation and that phalloplasty does not offer a phallus that looks or operates like a biological one. People who regret their transition or surgery are typically distressed and may seek counseling as part of coming to terms with their regret and deciding how to proceed with their lives. Others do not regret transitioning but have had difficult surgeries with significant complications and must come to terms with continuing dysphoria related to functional or cosmetic limitations.

As the SOC have become less stringent for approval of hormonal treatment and SRS, it will be important to continue to monitor rates of regret and satisfaction with these biomedical therapies.

**CONCLUSIONS**

In the past decade, there has been a genuine increase in the number of patients (and their families) seeking out mental health care for GD and, in the majority of adolescents and adults, biomedical treatment. Thus, for the practicing clinician who wishes to work with this population, it is critical to gain some familiarity with the extant treatment approaches, including SOC guidelines (see Byne et al., 2012; Coleman et al., 2011).

In this chapter, we have summarized the literature on therapeutics, taking into account what is known about developmental trajectories. Whereas biomedical treatment 239

for adolescents and adults is probably the most common method to ameliorate GD, it should be recognized that not all clients wish to pursue SRS and, for them, a supportive psychosocial therapeutic approach can be provided as a first-step alternative. For children, the field is currently in a great state of flux regarding best-practice principles, with a tremendous range of clinical opinion. It is important, therefore, for the practicing clinician to gain some familiarity with the different therapeutic approaches in designing a plan of care for individual children and their families.

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**CHAPTER 12**

Persistent Genital Arousal Disorder

David Goldmeier, Hossein Sadeghi-Nejad, and Thomas M. Facelle

**P** ersistent genital arousal disorder (PGAD) is a relatively "new" disorder that was originally termed persistent sexual arousal syndrome. The change in name is important as it reflects the fact that the genital arousal is not necessarily indicative of sexual arousal or pleasure. According to Goldmeier, Sadeghi-Nejad, and Facelle, women who suffer from PGAD experience relatively long periods of heightened genital sensation that are unbidden, unwanted, and highly distressing. The genital symptoms do not subside easily even after prolonged masturbation resulting in multiple orgasms. Partnered sexual activity may even worsen the symptoms. Although not a sexual disorder per se, PGAD is included in this volume because it has significantly deleterious effects on sexuality. In addition, PGAD and hypersexuality sufferers may appear similar because repeated masturbation is a symptom of both problems. This superficial resemblance, however, ignores the underlying motivations, which are very different. Goldmeier and his colleagues describe the different potential physiological (e.g., neurological) and psychological (e.g., anxiety-related) pathways that may be responsible for PGAD. The authors propose a treatment approach that involves a combination of physical and psychological remedies, including medication, pelvic floor physical therapy, and mindfulness-based CBT.

**David Goldmeier, MD,** is clinical lead for the multidisciplinary Jane Wadsworth Sexual Function Clinic at Imperial College and Imperial College NHS Healthcare in London, England. He has written 115 peer-reviewed academic papers and is chair of the sexual dysfunction special interest group of the British Association of Sexual Health and HIV. In his practice, he combines organic with psychological treatments (particularly mindfulness).

**Hossein Sadeghi-Nejad, MD,** graduated Magna Cum Laude from Bowdoin College in Brunswick, Maine, where he was the recipient of the Goodwin Commencement Prize. He received his MD from the McGill University School of Medicine in Montreal, Quebec, Canada, and served as an Intern and Resident in General Surgery at the University of California in San Francisco. Dr. Sadeghi-Nejad completed his urological residency and fellowship training in Male Reproductive Medicine (infertility and erectile dysfunction) and microsurgery at the Boston University Medical Center under the auspices of Drs. Robert Krane, Irwin Goldstein, and Robert Oates.

**Thomas M. Facelle, MD,** is a resident in Urology at Rutgers University Medical School, University Hospital, in Newark, New Jersey. He received his undergraduate degree from Wesleyan University in Middletown, Connecticut, and his medical degree from New York University School of Medicine. Additionally, he is a candidate member of the American Urologic Association.

Persistent genital arousal disorder (PGAD) is a newly recognized condition, in which the woman (or rarely man) complains of long periods of subjective genital arousal that come on unbidden (i.e., are not associated with sexual desire) and are highly distressing.

PGAD may be associated with a feeling described by some patients as "restlessness," and many women with PGAD masturbate and orgasm repeatedly in order to rid themselves of these symptoms—usually unsuccessfully. It is possible that Soranus of Ephesus (trans.

1991) alluded to the condition in his text "Midwifery and Diseases of Women" in the second century A.D., but it was first accurately described and defined by Sandra 244

Leiblum and Sharon Nathan in 2001. They presented five cases of women suffering from what they called persistent sexual arousal syndrome (PSAS; Leiblum & Nathan, 2001).

This was more accurately renamed persistent genital arousal disorder (PGAD) in light of the fact that most women did not see it as a sexually related condition (Goldmeier & Leiblum, 2006).

Fewer than 500 cases of PGAD have been described in the literature. However, some evidence suggests that it may not be such a rare condition (Garvey, West, Latch, Leiblum, & Goldmeier, 2009), and many sex therapists and psychologists anecdotally report to us at conferences that they are now seeing and managing such cases.

**WHAT PRECISELY IS PGAD AND WHAT IS IT NOT?**

Following are six features of PGAD that we believe are relevant for its diagnosis (Leiblum & Nathan, 2001; Goldmeier & Leiblum, 2006; Waldinger & Schweitzer, 2009): 1. Genital symptoms characteristic of sexual arousal (genital fullness/swelling and sensitivity with or without nipple fullness/swelling) or unpleasant genital sensations (dysesthesia), that persist for an extended period of time (hours or days) and do not subside completely on their own.

2\. Genital symptoms characteristic of sexual arousal or genital dysesthesia that do not resolve with ordinary orgasmic experience and may require multiple orgasms over hours or days to remit.

3\. Genital symptoms characteristic of sexual arousal or genital dysesthesia that are usually experienced as unrelated to any subjective sense of sexual excitement or desire.

4\. Genital symptoms that may be triggered not only by a sexual activity but also by nonsexual stimuli or by no apparent stimulus at all.

5\. Genital symptoms that are experienced as unbidden, intrusive, and unwanted.

6\. Genital symptoms that cause the woman a significant degree of distress.

PGAD needs to be differentiated from hypersexuality (Kafka, 2010), which can be defined as frequently repetitive sexual urges, fantasies or behaviors. Urges and fantasies are subjective mental events that have a desire quality—something patients with PGAD

do not report in relation to their symptoms. Repetitive sexual behavior such as masturbation may be a clinical feature of both PGAD and hypersexuality. However, only in PGAD is this undertaken because of unbidden genital arousal or dysesthesia.

Other women report spontaneous and persistent genital arousal but are not distressed by these sensations. They may feel neutral about them (Leiblum & Chivers, 2007) or may even welcome and enjoy these genital feelings, which they can either act on by engaging in sexual activity or dismiss (Leiblum, Seehuus, & Brown, 2007). Such women should not be diagnosed with PGAD. To complicate matters, some women with frequent genital arousal can be distressed about their condition some of the time but 245

welcome it at other times.

**SPECTRUM OF SYMPTOMATOLOGY**

The most common locations of symptoms of PGAD are the clitoris, vagina, labia, or a combination of all three. Less commonly it is felt in the pubic bone or around the anal area. Symptoms may be continuous or intermittent. Common symptoms include feelings of congestion, tingling, and wetness. Genital contractions and/or a feeling of imminent orgasm are commonly reported. Spontaneous vaginal and clitoral pain (28.6%) and pain with vaginal penetration (45.7%) have also been reported (Leiblum, Brown, Wan, & Rawlinson, 2005). The pain is of a throbbing quality. Original and current triggers for the onset of PGAD include physical stimulation, such as intercourse or masturbation, and psychological stress or anxiety. Seemingly innocuous stimuli such as sitting, wearing tight clothes, genital pressure, or vibrations from a motor vehicle are likely to exacerbate symptoms in most women. Erotic visual stimulation exacerbated symptoms in about 60% of women. The most frequently reported relieving factors include masturbation, orgasm, distraction, intercourse, exercise, and cold compresses.

Many of the women reported fatigue or a severe loss of energy (Leiblum et al., 2005; Waldinger, Venema, van Gils, & Schweitzer, 2009).

**CONCEPTUALIZING PGAD**

**Atypical Pudendal Nerve Neuropathy**

It has been suggested that PGAD is a neuropathy involving the pudendal and the dorsal clitoral nerves. The pudendal nerve derives from the second to fourth sacral levels of the spinal cord and sends sensory information from the perineum, anal area, and labia. A terminal branch of this nerve, the dorsal clitoral nerve, innervates the clitoris. These nerves under normal circumstances (e.g. digital stimulation) send sensory information to more central sites. It is postulated that in the pathological state they may spontaneously (i.e., without stimulation) produce pain (pudendal neuralgia) or, in the case of PGAD, a sensation of local sexual arousal and sometimes pain.

In the case of classical pudendal neuralgia, the symptoms may alter depending on posture. Similarly, many women with PGAD demonstrate exacerbation of genital symptoms with tight underwear and/or prolonged sitting (Waldinger, Venema, van Gils, & Schweitzer, 2009). Investigators have mapped mechanical "trigger points" based on hypersensitivity to static pressure in 23 volunteers with symptoms of PGAD. All women demonstrated preorgasmic or orgasmic sensations upon cotton swab examination along various points within the pudendal and dorsal clitoral nerve sensory areas. Eight women received local anesthetic blocks at one or more trigger points, and 5

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reported resolution of all symptoms of PGAD for the duration of the block. It is suggested that C (small and unmyelinated) and A delta (myelinated) pudendal and/or dorsal clitoral nerve dysfunction, possibly caused by compression or irritation, underlies PGAD (Waldinger, Venema, et al., 2009). Further evidence of a pudendal neuropathic etiology comes from a recent paper by Rosenbaum (2010), who reported the case of a 27-year-old pregnant women with PGAD and hypertonic obturator internus muscles (part of the pelvic floor group) on physical exam. Two men with PGAD have recently been described who likely had a small fiber sensory neuropathy (Waldinger, Venema, van Gils, deLint, & Schweitzer, 2011).

**Cognitive Disorder**

Leiblum and Chivers (2007) have proposed a psychological model for PGAD. This model suggests that PGAD develops when anxious individuals become overly focused on their spontaneous genital arousal. This hypervigilance results in a vicious cycle of increased anxiety, elevated sympathetic tone, and more genital arousal. In addition, anxiety itself may decrease sensory thresholds, thus increasing the perception of genital arousal. Leiblum and Chivers (2007) have speculated that a history of abuse could further contribute to anxiety and negative association with genital arousal. This theory was motivated, in part, by an online survey of PGAD sufferers (Leiblum et al., 2007) which showed a high rate of psychological symptomatology, including panic attacks (31.6%), obsessive–compulsive illnesses (24%), and major depression (57.9%). Nearly two-thirds of these women reported that their anxiety and/or depression predated their PGAD symptoms by at least 1 year. Fifty-three percent of women with PGAD in this survey reported suffering sexual abuse as children; however, only 16.7% thought that their current symptoms could be attributed to their prior abuse.

In clinical practice women with PGAD may have features suggestive of both of the preceding conceptualizations, that is, genital symptoms that are altered by posture as well as a background history of anxiety and unwanted sex. They may also complain of pain as well as unbidden arousal.

**OTHER MEDICAL CONDITIONS ASSOCIATED WITH PGAD**

**Pharmacologically Induced**

Several authors have noted a potential association between the initiation and withdrawal of selective serotonin and serotonin–norepinephrine reuptake inhibitors (SSRIs or SNRIs) and the onset of PGAD (Leiblum et al., 2007). Proposed mechanisms include rebound anxiety (on withdrawal of medication), C-nerve fiber pathology at the spinal level (Waldinger, de Lint, Venema, van Gils, & Schweitzer, 2009), genital vasodilatation following antidepressant withdrawal (Goldmeier & Leiblum, 2006), as well as a return to 247

baseline libido and newfound awareness of genital sensations previously suppressed by the medication (Goldmeier & Leiblum, 2008).

**Restless Legs and Bladder Symptoms**

Waldinger and Schweitzer (2009) investigated 18 women with PGAD and found that two-thirds of these women also complained of overactive bladder symptoms (OAB; i.e., increased urgency for micturition) and restless leg syndrome (RLS). The authors suggest that PGAD may share an underlying neuropathic pathological state with OAB and RLS.

As such, they propose that the new term "restless genital syndrome" (RGS) be adopted when PGAD is coincident with RLS.

**Miscellaneous**

There are a variety of other medical conditions that have been described in association with PGAD including epileptic foci, brain arteriovenous fistulas, and poststroke states (Anzellotti et al. 2010; Goldstein, De Elise, & Johnson, 2006); enlarged labial or pelvic veins (varices; Rosenbaum, 2010; Waldinger, van Gils, et al., 2009; Thorne & Stuckey, 2008); high soy intake (Amsterdam, Abu-Rustum, Carter, & Krychman, 2005); and sleep onset (Wylie, Levin, Hallam-Jones, & Goddard, 2005). These potentially important associations are not as yet well established, as they are based on case reports or small uncontrolled studies.

**SEXUAL DYFUNCTIONS IN PGAD**

Our clinical experience is that some women with PGAD have normal spontaneous or responsive sexual desire and enjoy genital stimulation and arousal and intercourse with their partners. They report, however, that the context of the genital stimulation is of major importance—that is, genital arousal with a partner is a positive and pleasant event, whereas unbidden genital arousal is distressing and unpleasant. Others, particularly those whose symptoms are controlled on pharmacotherapy, may have low desire and be subjectively nonresponsive but may agree to engage in sex to please their partners or because they derive emotional pleasure from such activity. Many such patients with PGAD would prefer not to have genital stimulation or vaginal intercourse, as it exacerbates or induces unpleasant genital arousal. A recent study of sexual functioning in women with a PGAD diagnosis used the Female Sexual Function Index (FSFI) questionnaire and compared their scores with those of women with female sexual arousal disorder (FSAD) and with controls (Leiblum & Seehuus, 2008). Women with PGAD showed a mean level of desire that was only slightly higher than control women, supporting the idea that PGAD is not a hypersexual state. Sexual satisfaction and pain 248

were similar in the FSAD and PGAD groups. In all other respects the scores of women with PGAD fell between those of controls and the women with FSAD. The authors conclude that frequent unprovoked sexual arousal "is not necessarily indicative of either especially good or especially poor sexual function, at least as assessed by the FSFI."

**DIAGNOSIS**

**Making a Clinical Assessment of PGAD**

Full assessment of a woman with potential PGAD may require input from more than one health care professional. The initial clinical interview should be undertaken by a health care professional with sufficient time and expertise to get a full psychosocial,

psychosexual, and medical history while making the woman feel comfortable. Figure

12.1 illustrates the crucial assessment areas that need to be covered. A psychologist or

sex therapist may often be an ideal health professional to undertake this initial assessment, as he or she will be familiar with the relevant psychosocial and psychosexual issues and methods of assessment. A psychologist or sex therapist, however, would have to familiarize him- or herself with the relevant medical domains and, if appropriate, get help from a suitable professional. The important medical areas to be assessed are also listed in Figure 12.1.  An appropriately trained physician or nurse can also carry out such an assessment. Time and comfort are essential, because many women suffering from PGAD have previously felt rejected or not listened to by clinicians or, worse still, may have been diagnosed with a primary delusional illness. The assessor needs to carefully yet compassionately obtain a full description of the PGAD symptom profile, including site, quality, duration, exacerbating and relieving factors, whether the genital arousal is preceded by desire, whether there is concomitant pain, and how long the women spends masturbating in an attempt to relieve the symptoms. Exacerbating factors include pressure on the genitals, vibrations (e.g., car driving), visual sexual stimuli, genital stimulation or intercourse, and anxiety. Relieving factors include medications, masturbation, orgasm, distraction, and physical exercise. For a clinician to make a working diagnosis of PGAD, patients should complain of unbidden sexual arousal/dysesthesia that (1) persists for an extended period of time (i.e., at least several hours); (2) does not subside spontaneously or with ordinary orgasmic experience (i.e., may require multiple orgasms over hours or days in order to gain some relief); (3) causes a clinically significant degree of distress.

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**FIGURE 12.1.** Treatment algorithm for PGAD.

**Working with the Physician/Gynecologist**

Once PGAD is suspected, the nonmedical clinician should work with a physician or gynecologist to carry out the necessary medical assessment. At a minimum, this should involve a lower genital examination (to look for sensory changes and genital varices) and a pelvic examination. Where pelvic varices or pudendal nerve entrapment is suspected, working with an experienced radiologist using MRI scanning will yield the most meaningful data. Current and past medications should be reviewed, as well as possible comorbid conditions, including voiding symptoms, urinary tract infections, restless leg symptoms, and a history suggestive of seizures, epilepsy, or temporal lobe events. Pelvic floor dysfunction is best assessed by a pelvic floor physical therapist.

**THE TREATMENT PROCESS**