Ambiguity in a masculine world: Being a BRCA1/2 mutation carrier and a man with prostate cancer

Abstract Objective Increased risk of prostate cancer (PCa) is observed in men with BRCA1/BRCA2 mutations. Sex and gender are key determinants of health and disease although unequal care exists between the sexes. Stereotypical male attitudes are shown to lead to poor health outcomes. Methods Men with BRCA1/2 mutations and diagnosed with PCa were identified and invited to participate in a qualitative interview study. Data were analysed using a framework approach. “Masculinity theory” was used to report the impact of having both a BRCA1/2 mutation and PCa. Results Eleven of 15 eligible men were interviewed. The umbrella concept of “Ambiguity in a Masculine World” was evident. Men's responses often matched those of women in a genetic context. Men's BRCA experience was described, as “on the back burner” but “a bonus” enabling familial detection and early diagnosis of PCa. Embodiment of PCa took precedence as men revealed stereotypical “ideal” masculine responses such as stoicism and control while creating new “masculinities” when faced with the vicissitudes of having 2 gendered conditions. Conclusion Health workers are urged to take a reflexive approach, void of masculine ideals, a belief in which obfuscates men's experience. Research is required regarding men's support needs in the name of equality of care.

Men with breast cancer experience stigmatisation, embarrassment, and altered body image, exacerbating their shock at diagnosis. 20,21 Formal support programmes are lacking, information is sparse, and deliverance gendered. 20,21 Both sexes at high-risk of BRCA1/2 are similar in their responses regarding genetic risk. 17 Both use family histories to evaluate risk and decision-making 22,23 although women are likely to influence male decisions. 22 A lower uptake of testing and higher drop out in men 12,17 is counterbalanced by a greater interest in testing. 17 Men are thought to be more vulnerable to psychological stress than women when undergoing testing 17 although low levels of psychological symptoms are reported in both sexes undergoing testing for BRCA1/2 mutations. 17,24 A gender approach is invisible notwithstanding a few exceptions where it is used as a backdrop to men's responses. 12,18,19,25 No genetic study has used "masculinity theory" as an integral aspect of research, yet sex and gender are key determinants of health. 11

| Aims
The experiences of male BRCA1/2 carriers who have prostate cancer are not reported. The aim of this study was to highlight men's social characteristics to explain behaviour and attitudes where both conditions have gendered connotations by using masculinity theory 8,9,[26][27][28][29] and empirical work. 10 Eligibility relied on a diagnosis of PCa and a pathogenic germline mutation in either BRCA1 or BRCA2. All patients were "counselled" regarding their disease status. Eligible patients were invited to undergo a semistructured interview, given an information sheet and reply slip to express interest. Interested men were contacted, interviews arranged, consent obtained. One-to two-hour interviews were conducted in a venue of the patients' choice. Interviews were audio recorded, transcribed, and analysed. Interviewees were at liberty to stall or curtail interviews.
A female sociologist outside the clinical team, with relevant experience, asked, "Has BRCA mutation status and having PCa impacted on your life?" Topics informed by interviewees, clinical practice, and literature were covered flexibly (Table S1). 20,21

| Method of analyses
A "Framework Analysis" 33 (allowing for the use of relevant topics of interest) was used to code items at face value, followed by conceptual coding identifying overarching themes and "deviant" accounts. 33,34 Interviewing and analysis was an iterative process. Constant comparison was used; interpretations made by checking patients' perceptions within and across verbatim transcripts. Interpretation relied on context, literature, field notes, and expert opinion. 33,34 3 | RESULTS

| Sample
Twenty-nine participants (11 men, 9 partners, and 9 children) were interviewed. This analysis draws on the interview data of 11 men.
Two partners were present but remained silent.
Fifteen eligible men were identified from the database; 13 men accepted; one later declined; one was ineligible. Most men had children (Table S2). There were no discernible demographic or social differences in responses (Table S2).

| Themes
The umbrella theme "Ambiguity in a Masculine World" wove its way through men's responses. Four subthemes were evident. Shock at carrier status or having PCa (Storm Clouds) was juxtaposed by the Vulnerability and a so-called feminine need for "attachment" were juxtaposed by a stereotypical "masculine" response of self-determination, stoicism, control, and "normality."

| Storm Clouds: the BRCA mutation
Participants were initially reluctant to test, claiming "low risk" (or no risk), a "hazy" perception of hereditary transmission and the gendering of breast cancer. 21 No reference was made regarding the risk of ovarian cancer associated with the BRCA1/2 genes. Far from being coerced to test, 17 men felt a need to pay back "work" of family members:

| Storm Clouds: prostate cancer
Men with PCa focus on its physical aspects. 10 Diagnosis, treatment, and side effects loomed large in our participant's accounts. Constant self-referral wrapped itself around stoicism, optimism, and self-determination. 10,[13][14][15][16] Distress was most profound amongst younger participants who referred to longevity; older men described the ramifications of treatment, any "crisis" normalised, 10  Martin: "My wife helped me through and through along with the hospital staff… Incontinence was very difficult but it is all back to normal" The "double whammy" was seldom addressed, and when it was, prostate cancer took precedence.
Edward:"I was probably more upset about prostate cancer although I didn't show it…I've thrown the dice, got the double whammy… get shot of it, get on with life." Nick: "I take the view that I would prefer to deal with prostate cancer than any others because I've heard that it's more likely treated successfully if caught early...." Treatment effects and fear of relapse, responses that did not apparently warrant formal support, were stoically presented. 9,10 "Counselling" might provide knowledge and advice, never to express emotions. When emotion was articulated, it was defended: George: "After prostate cancer (PCa) surgery, I threatened to throw myself from the window...a counsellor came…it was nice having a chat…breaking up days…he left realising I wasn't mad…just reacting to the situation."

| Silver Linings: the BRCA mutation
Shock and repercussions of the BRCA mutation were accompanied by a sense of "bonus." Like women, men appreciated early recognition of BRCA as a preventative measure, not for themselves but for gaining knowledge and scientific progress benefitting mankind, mainly daughters. 17,19,25,[30][31][32] Roger: "We are terribly lucky to have found out (about the gene) and it offered solutions…knowledge is power…if I'd got it…my daughter could find out too and do something" Reproductive options were seldom mentioned, although Edward, who distanced himself from family problems, broached the subject.
His concerns counter the suggestion that emotional distancing necessarily leads to downplaying risk. 17 however. This is evident amongst affected women and men in other studies. 12,17,19,22,[30][31][32] Martin: "to think that our daughter might have to make a decision! It's harder worrying that (daughter) had it rather than myself" Men's sadness at passing on the mutation was sometimes visceral but suppressed.
Rupert: "we love our children... in that respect one feels responsibility...I said to (daughter)..."You've got the wrong Daddy"....sorry I'll stop." (crying) Despite occasional lapses into pathos and ongoing distress, men remained optimistic "putting this BRCA stuff on the back burner."

| Silver Linings: prostate cancer
The BRCA mutation led to concern for others. 19,22,25,31,32 In contrast, PCa narratives manifested highly subjective accounts. 10 If PCa was intrinsically worrying, there was a strong sense of "bonus"-care through monitoring and early diagnosis. Clinical psychological symptoms were absent, 10,12 and formal support seldom offered or accessed. 10,13 Men showed no hesitation in rallying health providers, seeking information, and gaining knowledge. 13,35 Appreciation was continuously shown for personal aspects of supportive care received from medical personnel, the rudiments of "attachment" emphasised. 36 Nick: "the doctors are brilliant…wonderful… (Nurse) was magnificent…you can ring her up, she helps you… knows you…just what I needed…I think (nurse) likes me…they really looked after you."

| Brushing under Carpets: the BRCA mutation
Men put BRCA on the "back burner" despite the importance of alerting family members. Few men recounted BRCA1/2 status, its virulence never addressed, and its low risk status in men reiterated and/or gendered. Addressing the consequences of having the mutation was overshadowed by a PCa discourse-the embodied disease.
Jeff: "BRCA2?… it doesn't have much impact (on me)… being male it wasn't significant… it is the prostate that worries me."

| Brushing under Carpets: prostate cancer
While PCa narratives held sway, 10 disclosure rarely went beyond close family and friends. 10,13,30,37 Men chose their confidants with care, withholding details, maintaining normality, strength, and activity in the face of shame. 10,37 A reticence to "speak" out is found amongst women in the genetic context 24  There was evidence, however, that men did not have the words to say or when to say them mirroring problems in the male BRCA arena. 19,30 Jeff: "how do I tell (children) and when? That's not today's problem...( )...I haven't thought how to tell my son..."

| Facing the Music: the BRCA mutation
Studies have shown that in contrast to men's apparent reluctance to exchange information, and the ambiguity men display in terms of transmitting genetic information to family members, 17 women become the "gatekeepers of health," 17,24,30 resulting in the "gendering of responsibility." 19,30,31 In contrast, our male participants transmitted BRCA1/2 information to family including female children, 17,19,24,25, Fatalism sometimes overshadowed responsibility and guilt. 31 Multiple genes and or a simple mutation status was argued for, as disclosure was left to partners. In a few cases, family disharmony precluded disclosure, and this is found amongst both sexes. 24,30 Stephen: "I am not going to tell my 30 year old daughter about the breast...we don't speak to each other"

| Facing the Music: prostate cancer
Mutation status led to "facing the music" as men disclosed information to family members while remaining silent in respect of the ways it impinged on themselves. In contrast, subjective elements of PCa were conveyed, syphoned through constant descriptions of activity, good health, perspective, enabling self-preservation. 9,10,[13][14][15][16]29 Jeff: "(then) prostate cancer!…I thought of myself as Restorative physical training found in other populations 10,38,39 was not preferred amongst our participants. Finding new hobbies and being proactive were indicative of the ways that men restored "face" with optimism and fortitude. 10

| DISCUSSION
A qualitative methodology has enabled us to "dig deep" into how men respond when facing two gendered conditions. By using masculinity theory, we have revealed dynamic and ambiguous responses that are sometimes similar to women in this context, challenging "innate" stereotypical ways of viewing men. Men present themselves as not only ideally masculine, but vulnerable too, sometimes simultaneously but always in ways that reinstate masculinity.
Our participants formed a homogenous group. Caution is advocated when working with diverse populations within and outside developed countries where "hegemonic masculinity" may be perceived in differing ways. 40 Globalisation is changing the existence of gender orders in less developed countries, often mimicking Western versions of hegemonic masculinities. 40 Where ambiguity exists, gender as a "relational" activity, occurring in diverse locations and differing contexts requires investigation. 28,40 Men with breast cancer respond in visceral ways. 20,21 Men in our sample experienced mutation status, possibly accounting for "silence" in the wake of an invisible disease. The gendering of breast cancer 20,21 and the lack of targeted information for men 20,21 may also have contributed to self-confessed and relative "ignorance" concerning that condition. Like women, however, 17 and men in other genetic studies, 17,25 we found men were mindful of families' wellbeing, especially that of daughters. Difficulties were articulated in relation to when and how to inform children in terms of both BRCA status and PCa. 17 While men require help in that respect, reticence was balanced by an overwhelming need to inform genetic transmission to wider family members-countering the belief that men necessarily exclude themselves from health matters. 5,9 A "head of family" role is, however, indicative of the ways that men reinvent themselves as "masculine" in a context where stereotypical identities may be undermined. 10 While women with BRCA1/2 mutations 20,21 and men with PCa 10,17 do not generally exhibit clinical psychological symptoms, we were surprised that men were void of severe distress in the wake of a "double whammy," seldom mentioned as a single entity-one that may have been too difficult to contemplate for all its gendered connotations, or because men were eager to put BRCA on a "back burner." Men in our sample appreciated the bonus of early PCa diagnosis and the possibility of passing on knowledge regarding BRCA1/2. This may have allayed significant emotional fallout 17 although serious psychological symptoms are not generally an issue for men with cancer and in larger samples than our own. 10,13,17 By focusing on the treatment and aftermath of PCa, and by placing importance on an embodied disease, we suggest that a conduit was "produced" for normalising damaged male identities especially in the light of carrying a BRCA1/2 mutation. As Wenger suggests, men with PCa are challenged "to manage a fully blown cancer that disrupts an embodied masculinity, the latter associated with action and strength." 10 Interestingly, sexual dysfunction was rarely evoked, unlike men in other studies, [14][15][16] but nor was it a principle focus of our enquiry. Reticence may have relied on early stage PCa where nerve sparing prostatectomies preserve potency. Men did however exhibit distress in relation to both conditions. 10,13,17,18,20,21 Lives are changed, identity issues abound, responsibility and guilt towards families is stark, wrapped in stoicism, activity, positive self-referral, knowledge building, and a need and appreciation of an "attachment" to health clinicians with whom they seemed to form a measure of equality and trust. In contrast to this, men shunned formal support. 10 Men may wish to guard vulnerability while re-establishing some "normality" and a sense of unscathed masculinity. 10 This has to be respected but as Wenger says men may use help seeking such as knowledge gathering and eliciting the help of professionals 10,35 "to demonstrate power, control and even self-reliance" as well as being "a possible indirect request for an intervention where men's help seeking is 'socially problematic.'" 10 There is an urgent need to research ways of providing support for men where physical 38,39 and/or informational resources and "good relationships" with health personnel 10,35 are addressed and where there is a recognition of men's gendered responses such as a need to maintain strength and identity 10,38,39 as well as acknowledging so-called feminine responses such as expression of emotion. 5,8 Men in our sample did not display overt emotion but underneath expressed bravado there lurked a palpable sense of sadness and fear. 17 We do not believe that men (or women) necessarily experience specific and exclusive feelings when facing difficulty. The ways in which a man expresses distress may differ, usually to suit expectations for all the connotations of "masculinity" that we have alluded to. These caveats need to be cast in recognition of the pitfalls of essentialism as they lead to unequal care.

| Clinical implications
Men appear to need help regarding disclosure to children especially boys; to understand BRCA1/2 cancer risks and inheritance patterns; to access information and reassurance from clinicians, while being mindful of men's need to reinstate a sense of their masculine selves.
Research is required regarding support whilst recognising the possibility of differing needs in various populations of men. "Masculine behaviour may mask vulnerability to reinstate male identities and especially in the wake of bearing the "stigma" of having a so-called "female" mutation as well as a full blown "masculine" cancer. Health professionals are asked to question their own gendered expectations, the latter possibly obfuscating men's experience, rendering invisible the "masculinities" that may be operating in this context and a need to recognise equal care.

| Limitations
This study took place in one centre with a small homogeneous sample.
More research using gender analyses is required that includes age, ethnicity, cultural diversity, sexuality, and socio-economic status.

SUPPORTING INFORMATION
Additional Supporting Information may be found online in the supporting information tab for this article.