Journal of Adolescence 43 (2015) 206e212
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Journal of Adolescence journal homepage: www.elsevier.com/locate/jado
A systematic review of literature on psychosocial aspects of gynecomastia in adolescents and young men Lynn Rew a, *, Cara Young a, Tracie Harrison a, Robert Caridi b, 1 a b
The University of Texas at Austin School of Nursing, Austin, TX 78701, USA Westlake Plastic Surgery & Austin Gynecomastia Center, Austin, TX 78746, USA
a r t i c l e i n f o
a b s t r a c t
Article history: Available online 5 July 2015
Gynecomastia represents a serious psychosocial challenge for many adolescent and young adult males, but short of surgery, little attention has been given to this concern. The purpose of this paper is to report findings from a systematic review of the literature about psychosocial correlates, psychosocial interventions, and quality of research evidence about young males with gynecomastia. From an initial 233 published papers, 10 studies were identified and reviewed. Five were descriptive case studies and included no statistical analyses. The other studies were all conducted with small samples. Despite the limited evidence, findings suggest that many young men suffer emotional distress concerning gynecomastia, but this distress has received few interventions beyond surgical removal of the breast tissue. Future studies are needed to address this problem more fully so that more aggressive measures such as frequent assessments of the physical and psychosocial aspects of the condition can be done by healthcare professionals. © 2015 The Foundation for Professionals in Services for Adolescents. Published by Elsevier Ltd. All rights reserved.
Keywords: Adolescent male Gynecomastia Emotional distress Psychosocial
Gynecomastia is a condition of increased tissue found in the breasts of males. Some estimates are that glandular proliferation occurs in roughly 65% of adolescent males, but most of these cases resolve without any medical or surgical intervention (Greydanus, Stockburger, Omar, & Dodich, 2012). Most gynecomastia (GM) in adolescents and young adults is related to pubertal development, but other causes include being overweight, use or abuse of several classes of drugs including anabolic steroids used in body-building, some antipsychotic and antidepressant drugs, cardiovascular drugs such as reserpine, as well as selected antibiotics and antiviral medications. In addition, gynecomastia may develop in response to use of recreational and illicit drugs such as marijuana, amphetamines, heroin, and alcohol (Joffe, 2008). In a study of 954 healthy males between the ages of 18e26 years randomly selected from military training centers in Greece, Georgiadis et al. (1994) found that those with GM were significantly more likely to be overweight than those without the condition. A retrospective study of 69 adolescents treated surgically for GM in the U.S. indicated that 51% were obese and 16% were overweight (Rosen et al., 2010). Although more of the overweight and obese patients had experienced psychological stress prior to surgery, there were no significant differences between the obese and overweight group and the normal weight group in surgical complications or satisfaction following the surgery. Other researchers have also shown that adolescents who
* Corresponding author. E-mail addresses: [email protected] (L. Rew), [email protected] (C. Young), [email protected] (T. Harrison), rcaridi@ westlakeplasticsurgery.com (R. Caridi). 1 Tel.: þ1 512 732 0732; fax: þ1 512 732 0735. http://dx.doi.org/10.1016/j.adolescence.2015.06.007 0140-1971/© 2015 The Foundation for Professionals in Services for Adolescents. Published by Elsevier Ltd. All rights reserved.
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are overweight are at risk for depression and may develop unhealthy behaviors to control their weight (Armstrong, Westen, & Janicke, 2014). Evans (1997) surveyed a community sample of male recreational athletes and bodybuilders (ages 16e40) and found that 64% had been using anabolic steroids for one to five years and 34% of the users experienced gynecomastia. A recent literature review showed that, overall, approximately 5% of male adolescents taking antipsychotics such as resperidone suffered from side-effects including GM (Roke, van Harten, Boot, & Buitelaar, 2009). Gynecomastia that is drug-induced by adolescents who abuse amphetamines, anabolic steroids, heroin, and marijuana generally resolves without surgical intervention when such drugs are discontinued (Goldman, 2010). Anecdotal reports have suggested that males who experience GM seriously curtail their social activities at a time when social relationships and interactions are of particular salience (Laituri et al., 2010). Given that physical maturation and selfidentity are major developmental tasks of adolescence (Rew, 2005), the purpose of this paper is to report findings from a systematic review of the literature pertaining to the psychosocial correlates, psychosocial interventions, and research evidence about adolescents (ages 10e21) and young men (ages 22e25) with GM. Method We conducted a systematic review of the literature following the general outline provided by Cooper (2010): formulation of the problem, systematic search of literature, data extraction and evaluation, data analysis and integration of findings, interpretation, and dissemination. We further followed the PRISMA Checklist for reporting systematic reviews (Liberati et al., 2009). Thus we reviewed title, abstract, introduction, methods, results, discussion, including limitations, and funding, if applicable, of those articles included in this review. For the purposes of this review, the problem of GM is situated in the context of adolescent and emerging adult development with a particular focus on males. In early and late adolescence, body image and fitting in with peers are critical to development of self-identity (Schwartz, 2008). Moreover, satisfaction with one's body has been shown to be associated with competence in using one's body, as in engaging in strength and conditioning activities, among adolescent males (Leone, Mullin, Maurer-Starks, & Rovito, 2014). Thus we sought to answer the following research questions: 1. 2. 3. 4.
What What What What
psychosocial correlates have been identified in adolescent and young adult males with GM? psychosocial interventions have been tested in adolescent males with GM? are the psychosocial clinical recommendations for practice with adolescent males with GM? is the quality of the evidence presented in studies of adolescent males with GM?
Electronic searches Four electronic databases were selected for this study and searched from August 1e24, 2014: CINAHL, Medline, PsychInfo, and PubMed. These databases were selected because they included reviews and studies about medical conditions and psychosocial correlates and interventions of GM. Terms used in the searches were “adolescent,” AND “gynecomastia,” AND “psychology;” AND “psychological;” AND “body image;” AND “self-esteem.” The subsequent reviews were conducted from August through November of 2014. Data extraction The searches returned a total of 233 papers (CINAHL ¼ 4; Medline ¼ 134; PsychInfo ¼ 34; PubMed ¼ 61). After we removed 48 duplicated articles across these databases, the remaining 185 were screened for inclusion/exclusion criteria. We included: publications written in English, those that addressed adolescents and emerging adults aged 10e25 years, and those that addressed idiopathic or medication-induced gynecomastia in males. We included studies from other countries when the full research report was available in English. We also included pertinent articles found through ancestry hand searches of references within articles included in the review. We excluded books, dissertations, letters to the editor, and articles that focused on research instrument development or were devoted solely to surgical intervention for GM. Fig. 1 shows the number of records searched, excluded, and those included in the final sample (N ¼ 10). Data were extracted from each article using the headings in Table 1: author (date and data base), methods, psychosocial correlates, psychosocial interventions, and limitations/quality and strength of evidence. Quality and strength of evidence were determined by examining the study design, sample size and representativeness, and data analysis strategies. Analysis and integration Study design Five studies were case studies of 2e4 adolescents each (Fisher & Fornari, 1990; Joseph, 2011; Money & Lewis, 1982; Schonfeld, 1962; Storch et al., 2004). One study was a retrospective chart review (Rosen et al., 2010), one was a prospective caseecontrol (Nuzzi et al., 2013) and the remaining three were non-experimental descriptive studies (Davanço et al., 2009;
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Total Citations n = 233 Duplicates Removed -n = 48
Abstracts Reviewed n = 185
Articles Reviewed n=8
Excluded due to 1) not GM focused; 2) not adolescent focused; 3) psychological correlates or treatments not addressed -n = 177
Additional Articles from Hand Searches +n = 2 Final Articles Reviewed n = 10 Fig. 1. Flow chart of literature searches on idiopathic gynecomastia in adolescents and young men.
Kasielska & Antoszewski, 2011; Kinsella et al., 2012). Four of the studies were conducted with males who were undergoing surgical correction of GM (Davanço et al., 2009; Kasielska & Antoszewski, 2011; Kinsella et al., 2012; Rosen et al., 2010) and one was conducted with males diagnosed with GM in an adolescent breast clinic that was part of a plastic surgery unit (Nuzzi et al., 2013); however, the rate and type of surgical intervention done with this sample was not discussed. Psychosocial correlates As noted in Table 1, a variety of psychosocial correlates were identified. These included several anecdotal “opinions” such as affecting personality, disturbed body image, feeling unacceptable, dissatisfaction with being male, avoiding participation in sports, excessive self-consciousness, disordered eating, social withdrawal, school disruptions, psychological stress, anxiety, fear, germ phobia, teasing, depressive symptoms, internalizing and externalizing behaviors, intense shame, emotional discomfort, interference with social relationships, feeling embarrassed, loneliness, and suicidal despair. Psychosocial correlates that were measured using psychometrically sound instruments included anxiety, depressive symptoms, and healthrelated quality of life. Psychosocial interventions Two of the studies suggested no psychosocial interventions for adolescents and young adults with gynecomastia. Psychosocial interventions that were suggested included vague terms such as psychotherapy, mental health treatment, and early intervention to improve both physical and emotional symptoms. We found no studies that reported the development or testing of psychosocial interventions specifically designed for young males with GM. The caseecontrol study by Davanço et al. (2009) found that quality of life was enhanced six months following surgical intervention for breast reduction in adolescent and adult males. In the study by Kinsella et al. (2012), all participants were offered surgical reduction, but only 50% could afford the surgery; all of the participants who did not receive surgical intervention were diagnosed with adjustment disorder, using criteria from the Diagnostic and Statistical Manual of Mental Disorders-IV (American Psychiatric Association, 1994). All of the young men surveyed by Kasielska and Antoszewski (2011) were treated surgically and significant improvements in emotional comfort and social relationships were found. Evaluation of evidence found In evaluating the evidence, the Johns Hopkins Nursing Evidence Rating Scale created by Newhouse and colleagues was used to rate both the strength (1e5), where 1 is highest (e.g., randomized controlled trial or meta analysis) and 5 is lowest
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Table 1 Data extracted for systematic review on gynecomastia in adolescence and young males arranged chronologically. Authors (date) Database 1
2
3
Methods
Descriptive case report N ¼ 284 “boys and men,” with GM, 88% first noted GM during adolescence. Illustrative case reports (n ¼ 4). Stated purpose was to “determine the effects of GM on personality adaptations”
Psychosocial correlates
Personality effects dependent upon: 1) age of onset, 2) size of breasts, 3) personality prior to onset. Described anxiety/fear; stress; ridicule; fantasies that girls were envious; feeling unacceptable; disturbed body image; self-consciousness; social withdrawal. Most affected if onset in adolescence Money and Case study type comparison of 3/10 of GM participants reported dissatisfaction with being a male, Lewis (1982) two clinical groups (i.e., idiopathic adolescent GM & identified as homosexual with Source: PsychInfo congenital virilizing cross-dressing, no history of adrenocorticism) on conformity male stereotypic sports to male stereotypes & sexual participation; orientation 4/10 with behavioral pathology N ¼ 18 (n ¼ 10 with GM, ages 14 including suicidal despair, germ e18 yrs, mean ¼ 15 yrs) phobia, and school disruptiveness. Social isolation (7/10) Teasing (8/10; 6/10 “viciously”) Case studies, N ¼ 2, ages 16.5 & Anorexia & bulimia nervosa Fisher and 1. “inadequate body” 17.5 Fornari 2. Breasts “were not going away” Weekly psychotherapy (1990) Source: PsychInfo Schonfeld (1962) Source: PsychInfo
4
Storch et al. (2004) Source: Medline
Letter to the editor. Case studies (N ¼ 2) Ages: 15 years and 17 years
5
Davanço et al. (2009) Source: From Ref. List
Non-experimental pre- postoperative measure of quality of life (SF-36) in males who had surgical intervention. Descriptive analysis. N ¼ 33, ages 18e50 (M ¼ 25.1). 91% had GM since puberty
6
Rosen et al. (2010) Source: Medline
Retrospective chart review of males who had surgical intervention. N ¼ 69 ages 12e21, patients from Adolescent Breast Clinic at Harvard Med School.
7
Joseph (2011) Source: PubMed
Case study, N ¼ 2; pair of siblings with partial androgen insensitivity syndrome (PAIS; Ambiguous genitalia). One raised as boy, other as girl
Psychosocial interventions
Limitations/quality and strength of evidence
“Psychotherapy played a significant role in the management of many of these cases.” Therapeutic goals: 1) determine significance to adolescents, 2) recognition of feelings with appropriate channeling, 3) alleviate guilt & inadequacy, 4) improve self-esteem
Not a systematic study No statistical analyses. Quality: C Strength: 5
None suggested
Small sample size Limited in focus (i.e., conformity to male stereotypes and sexual orientation) No statistical analyses. Quality: C Strength: 5
Small sample Onset of eating disorders directly related to desire to decrease breast size. No statistical analyses. Quality: C Strength: 5 Small sample size. Depressive symptoms: elevated Suggests improved routine Case study participant scores screening for psychological for both boys Anxiety symptoms: elevated in distress as a component of clinic compared to standardized visit and more focused clinical norms. 15 y/o No statistical analyses. intervention Loneliness: elevated in 17 y/o Quality: C Behavior (parent rating scale): Strength: 5 significant internalizing and externalizing symptoms reported by mom of 17 y/o Small sample Significant pre-post differences Authors suggest surgical on several domains of quality of treatment improves quality of Wide age range Wiloxon's nonparametric test of life in both physical and life: general health, p ¼ .001; emotional (i.e., mental health) pre- and post-op scores on functional capacity, p ¼ .002; domains in the SF-36 measure social aspects, p ¼ .002; mental domains of quality of life. health, p ¼ .004; vitality, p ¼ .007 Quality: B Strength: 3 None suggested Unclear how psychological 59.4% Reported psychological stress and other factors were stress; 27.9% reported reduced measured. physical activity; 78.3% of obese Chi squares, Fisher exact tests, patients reported psychological or t-tests done. stress (significantly more than Quality: C normal weight peers Strength: 4 preoperatively [p ¼ .02]) Gender identity concordant with “Boy” expressed hope related to Small sample size, N ¼ 1 of adolescent GM potential surgical correction how each reared. “Boy” developed GM at puberty; stating, “After the surgery I will No statistical analyses. reported intense shame, anxiety, be able to do anything.” (i.e., be Quality: C Strength: 5 being ridiculed/teased, avoided in public without feeling ashamed, return to school, train public/socially withdrew, quit for a better job) school grade 8. Elevated depressive symptoms, decreased quality of life per standardized tools “Girl”: no features of depression or any other psychiatric illness, “well-adjusted … with good 16.5 y/o: weekly psychotherapy; “superficially involved” terminated at 5 mos. 17.5 y/o: refused f/u appointments, further treatment or evaluation
(continued on next page)
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Table 1 (continued ) Authors (date) Database
Methods
8
Kasielska and Antoszewski (2011) Source: Medline
Descriptive, pre- and post-op. survey evaluation of males who had surgical intervention for GM. N ¼ 47 (Mean age 25.6 ± 3.5 yrs) Investigator developed survey with individual questions related to age of onset, coexisting disorders, and aspects of psychosocial effects & functioning (e.g., level of emotional discomfort, social distance, ridicule, & embarrassment)
9
Kinsella et al. (2012) Source: From Ref List
Descriptive N ¼ 24 ages 10e18 All participants had clinical interviews for psychiatric evaluation and 50% had surgery for GM. Half of sample underwent surgical correction Descriptive, comparison with healthy control group N ¼ 139 12e21 year old males (n ¼ 47 with GM and 92 healthy controls). Males with GM were recruited from a plastic surgery clinic.
10 Nuzzi et al. (2013) PubMed
Psychosocial correlates
functioning in social, personal, and academic realms” Pre-surgery: almost all reported emotional discomfort & limitations of daily activities. Majority reported interference with entry into social relationships & relationships with women, being teased, & feeling embarrassed. Statistically sig. improvements in psychosocial effects seen across all questions post-surgery (e.g., less emotional discomfort, more physical activity, improved social and romantic relationships) 100% of Participants diagnosed with DSM IV diagnoses: adjustment disorder (72.9%), anxiety d/o (16.7%), dysthymia (16.7%) “Most” self-reported decreased self-confidence & negative selfimage Health-related quality of life: GM adolescents scored lower than controls in: self-esteem, general health, vitality, social functioning, role-emotional, & mental health scored lower than controls Disordered eating behaviors: higher in GM adolescents than controls
Psychosocial interventions
Limitations/quality and strength of evidence
6 Participants had consulted psychologist prior to surgery (41 had not) After surgical treatment: 100% of participants reported improvement in emotional discomfort level & endorsed surgery as right decision.
Small sample from one surgery center. No psychometric properties established for survey. Chi-square tests. Quality: B Strength: 3
Mental health treatment recommended for all participants
Individual participants' scores on standardized measures compared to standardized norms, no group analyses Pearson chi squares and linear regressions done. Quality: B Strength: 3 Statistical testing controlled for BMI: differences in general health, social functioning, mental health, and self-esteem remained statistically significant. Quality: Level A Strength: 2
Early intervention and treatment recommended to improve negative physical and emotional symptoms
(e.g., opinion, personal experience, case study) and the quality (AeC) of the evidence, where A is high, and C is low (Newhouse, Dearholt, Poe, Pugh, & White, 2007). This allowed the team to review both the types of research being done in the area and the value of the research in understanding how GM influences the psychological development of adolescent males. It also provided insight into ways in which this problem may be best addressed in future studies. The strength of the 10 studies ranged from 2 to 5, with the majority (n ¼ 5) rated as a level 5 due to the emphasis on case studies, which used clinical expertise/experience as the foundation for the interpretation of findings. There were 3 studies that were rated level 3 due to their use of descriptive data, which were used to delineate the psychological correlates found in this group of adolescent males. Only one study met criteria for receiving an A level of quality and strength of 2; it was a quasi-experimental study that controlled for BMI and included adolescent males without GM (n ¼ 92) as a comparison group to study mental and physical health outcomes for those with GM (n ¼ 47) (Nuzzi et al., 2013). In this study, self-esteem and several domains of health (i.e., general health, vitality, mental health, emotional role, and social functioning) were significantly lower for adolescents (age 12e21) with GM, but physical functioning, physical roles, and pain, as assessed by physicians, did not significantly vary. Moreover, while no significant difference in prevalence of disordered eating behaviors was found, the adolescents with GM reported significantly worse attitudes towards eating than those without the condition. These authors used standardized, valid measures of psychological outcomes. The remaining studies were given an Evidence Rating Scale quality score of B or C. As shown in Table 1, these studies lacked methodological description, emphasis on expert opinion without evidential backing, and/or small sample sizes without randomization or independent sampling. Discussion The studies included in our review reflect a beginning level of knowledge development on the topic of adolescent GM. Overall the 10 articles reviewed included small samples ranging from the single-participant case study to a sample of 139. All studies were limited to single sites, such as surgery centers where young men went to have surgical breast reductions. Half of the studies (n ¼ 5) included no statistical analyses of the data. Most statistical analyses were limited to Chi-squares and linear
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regressions. The study by Kinsella et al. (2012) compared participants' scores on standardized measures to population norms. The population estimates for GM and its correlates varied widely within the reports. There was strong evidence to suggest that young adolescent males with GM are reporting emotional problems to their clinical providers. These providers have worked to gain the attention of health researchers and public health officials through their case reports. Regardless, over the last 50 years, little attention has been shown to GM. Schonfeld's seminal study (1962) introduced the topic over 5 decades ago, but it has received minimal response in the way of knowledge development that could guide professional medical care beyond surgical repair. Indeed, although date parameters were not placed on this review to enable the reader to see the shift in studies over time, the studies conducted from 1962 to 2004 demonstrated only the continued efforts by clinicians to publish case reportsdbringing repeated focus on the lack of evidence available. From 2009 until 2013, sample sizes increased, highlighting the possibly growing number of youth affected, but still, no large scale studies were conducted. In the process of including and excluding papers for this review, no studies were found that established a population prevalence for GM in adolescents. For this reason, there remains a lack of epidemiologic evidence of the incidence, prevalence and reoccurrence of gynecomastia and its associated psychosocial factors among adolescents and young adults. Consistently, over the past 50 years, young adolescent males have presented to their providers with GM, which does not resolve over time in as many as 10% of the population. The result has been that young men experience problems with selfesteem, eating disorders (Fisher & Fornari, 1990; Nuzzi et al., 2013), social withdrawal, body image disturbances (Money & Lewis, 1982), depression, anxiety (Storch et al., 2004), and lessened quality of life (Davanço et al., 2009). The only known intervention routinely offered has been surgical (Kasielska & Antoszewski, 2011); however, few data document its effectiveness in addressing the mental health problems that accompany this condition. Further, owing to the impact of psychosocial outcomes on long-term developmental trajectories, surgical interventions have been suggested to begin at early ages before body image disturbances, eating disorders, and low socialization overwhelm the adolescent's ability to cope (Davanço et al., 2009; Schonfeld, 1962). Yet, alternative treatments have not been studied systematically. Further studies are warranted to determine the population prevalence of idiopathic gynecomastia in adolescent males. Given the dearth of evidence found in the literature, evaluations of psychosocial symptoms and problems in adolescents should be investigated at intervals in those who also display symptoms of GM so that healthcare providers could determine for whom symptoms resolve and for whom they do not. Such knowledge is important so that healthcare providers can intervene in a timely manner. Moreover, interventions that specifically evaluate the pre- and post-operative symptoms of emotional and psychosocial distress may be warranted. Implications for practitioners Healthcare providers in primary care have been consistently educated that GM in adolescents is developmentally normal and in a majority of cases will resolve on its own within three years. Indeed, a leading pediatric primary care textbook addressed GM only once with the recommendation that providers question an adolescent regarding alcohol, marijuana, and anabolic steroid use if the condition persists (Burns, Dunn, Brady, Starr, & Blosser, 2013). The findings from this review, however, suggest this limited approach to management is missing the multiple psychological factors potentially associated with this condition. This review indicates that there are young men whose GM does not resolve and requires breast reduction due to emotional distress at varying points in their lives as well as those who develop severe and lasting mental health conditions. Healthcare providers working with adolescents need also consider the implications for the socio-emotional development that characterizes adolescence as well as the impact on establishment of health behaviors that may be carried into adulthood. Normal adolescent transitions involve developmental tasks associated with identity vs. role confusion and sexual maturation where issues related to gender-role socialization, body image, social relationships, attitudes, values, and self-esteem are addressed (Erikson, 1968). A condition such as GM that involves shame, embarrassment, ridicule, and negative affect towards self may have a devastating impact on many of these crucial developmental tasks. Even with spontaneous resolution of this idiopathic condition by the end of adolescence, the developmental consequences may affect that individual for the rest of their lives. The general lack of evidence regarding how often the condition occurs, how often it resolves, probability of recurrence, and associations with long term health outcomes necessitate, we believe, a more aggressive approach to ongoing assessment by healthcare providers than has traditionally been recommended. The findings of this review call for primary care providers to consider maintaining a closer evaluation of their patients' psychological and developmental status within the context of GM in order to prevent or curtail secondary developmental and mental health consequences. Conclusions The findings from this systematic review of literature about the psychosocial correlates of and interventions for adolescents and young adult males with idiopathic gynecomastia are meager. Spanning 50 years of literature, aside from descriptions of surgical interventions, very little is known about how this condition, if unresolved during adolescence, affects the psychosocial development and mental health of young males. More rigorous research with large and diverse samples is sorely needed to address this serious concern.
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Contents lists available at ScienceDirect
Journal of Adolescence journal homepage: www.elsevier.com/locate/jado
A systematic review of literature on psychosocial aspects of gynecomastia in adolescents and young men Lynn Rew a, *, Cara Young a, Tracie Harrison a, Robert Caridi b, 1 a b
The University of Texas at Austin School of Nursing, Austin, TX 78701, USA Westlake Plastic Surgery & Austin Gynecomastia Center, Austin, TX 78746, USA
a r t i c l e i n f o
a b s t r a c t
Article history: Available online 5 July 2015
Gynecomastia represents a serious psychosocial challenge for many adolescent and young adult males, but short of surgery, little attention has been given to this concern. The purpose of this paper is to report findings from a systematic review of the literature about psychosocial correlates, psychosocial interventions, and quality of research evidence about young males with gynecomastia. From an initial 233 published papers, 10 studies were identified and reviewed. Five were descriptive case studies and included no statistical analyses. The other studies were all conducted with small samples. Despite the limited evidence, findings suggest that many young men suffer emotional distress concerning gynecomastia, but this distress has received few interventions beyond surgical removal of the breast tissue. Future studies are needed to address this problem more fully so that more aggressive measures such as frequent assessments of the physical and psychosocial aspects of the condition can be done by healthcare professionals. © 2015 The Foundation for Professionals in Services for Adolescents. Published by Elsevier Ltd. All rights reserved.
Keywords: Adolescent male Gynecomastia Emotional distress Psychosocial
Gynecomastia is a condition of increased tissue found in the breasts of males. Some estimates are that glandular proliferation occurs in roughly 65% of adolescent males, but most of these cases resolve without any medical or surgical intervention (Greydanus, Stockburger, Omar, & Dodich, 2012). Most gynecomastia (GM) in adolescents and young adults is related to pubertal development, but other causes include being overweight, use or abuse of several classes of drugs including anabolic steroids used in body-building, some antipsychotic and antidepressant drugs, cardiovascular drugs such as reserpine, as well as selected antibiotics and antiviral medications. In addition, gynecomastia may develop in response to use of recreational and illicit drugs such as marijuana, amphetamines, heroin, and alcohol (Joffe, 2008). In a study of 954 healthy males between the ages of 18e26 years randomly selected from military training centers in Greece, Georgiadis et al. (1994) found that those with GM were significantly more likely to be overweight than those without the condition. A retrospective study of 69 adolescents treated surgically for GM in the U.S. indicated that 51% were obese and 16% were overweight (Rosen et al., 2010). Although more of the overweight and obese patients had experienced psychological stress prior to surgery, there were no significant differences between the obese and overweight group and the normal weight group in surgical complications or satisfaction following the surgery. Other researchers have also shown that adolescents who
* Corresponding author. E-mail addresses: [email protected] (L. Rew), [email protected] (C. Young), [email protected] (T. Harrison), rcaridi@ westlakeplasticsurgery.com (R. Caridi). 1 Tel.: þ1 512 732 0732; fax: þ1 512 732 0735. http://dx.doi.org/10.1016/j.adolescence.2015.06.007 0140-1971/© 2015 The Foundation for Professionals in Services for Adolescents. Published by Elsevier Ltd. All rights reserved.
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are overweight are at risk for depression and may develop unhealthy behaviors to control their weight (Armstrong, Westen, & Janicke, 2014). Evans (1997) surveyed a community sample of male recreational athletes and bodybuilders (ages 16e40) and found that 64% had been using anabolic steroids for one to five years and 34% of the users experienced gynecomastia. A recent literature review showed that, overall, approximately 5% of male adolescents taking antipsychotics such as resperidone suffered from side-effects including GM (Roke, van Harten, Boot, & Buitelaar, 2009). Gynecomastia that is drug-induced by adolescents who abuse amphetamines, anabolic steroids, heroin, and marijuana generally resolves without surgical intervention when such drugs are discontinued (Goldman, 2010). Anecdotal reports have suggested that males who experience GM seriously curtail their social activities at a time when social relationships and interactions are of particular salience (Laituri et al., 2010). Given that physical maturation and selfidentity are major developmental tasks of adolescence (Rew, 2005), the purpose of this paper is to report findings from a systematic review of the literature pertaining to the psychosocial correlates, psychosocial interventions, and research evidence about adolescents (ages 10e21) and young men (ages 22e25) with GM. Method We conducted a systematic review of the literature following the general outline provided by Cooper (2010): formulation of the problem, systematic search of literature, data extraction and evaluation, data analysis and integration of findings, interpretation, and dissemination. We further followed the PRISMA Checklist for reporting systematic reviews (Liberati et al., 2009). Thus we reviewed title, abstract, introduction, methods, results, discussion, including limitations, and funding, if applicable, of those articles included in this review. For the purposes of this review, the problem of GM is situated in the context of adolescent and emerging adult development with a particular focus on males. In early and late adolescence, body image and fitting in with peers are critical to development of self-identity (Schwartz, 2008). Moreover, satisfaction with one's body has been shown to be associated with competence in using one's body, as in engaging in strength and conditioning activities, among adolescent males (Leone, Mullin, Maurer-Starks, & Rovito, 2014). Thus we sought to answer the following research questions: 1. 2. 3. 4.
What What What What
psychosocial correlates have been identified in adolescent and young adult males with GM? psychosocial interventions have been tested in adolescent males with GM? are the psychosocial clinical recommendations for practice with adolescent males with GM? is the quality of the evidence presented in studies of adolescent males with GM?
Electronic searches Four electronic databases were selected for this study and searched from August 1e24, 2014: CINAHL, Medline, PsychInfo, and PubMed. These databases were selected because they included reviews and studies about medical conditions and psychosocial correlates and interventions of GM. Terms used in the searches were “adolescent,” AND “gynecomastia,” AND “psychology;” AND “psychological;” AND “body image;” AND “self-esteem.” The subsequent reviews were conducted from August through November of 2014. Data extraction The searches returned a total of 233 papers (CINAHL ¼ 4; Medline ¼ 134; PsychInfo ¼ 34; PubMed ¼ 61). After we removed 48 duplicated articles across these databases, the remaining 185 were screened for inclusion/exclusion criteria. We included: publications written in English, those that addressed adolescents and emerging adults aged 10e25 years, and those that addressed idiopathic or medication-induced gynecomastia in males. We included studies from other countries when the full research report was available in English. We also included pertinent articles found through ancestry hand searches of references within articles included in the review. We excluded books, dissertations, letters to the editor, and articles that focused on research instrument development or were devoted solely to surgical intervention for GM. Fig. 1 shows the number of records searched, excluded, and those included in the final sample (N ¼ 10). Data were extracted from each article using the headings in Table 1: author (date and data base), methods, psychosocial correlates, psychosocial interventions, and limitations/quality and strength of evidence. Quality and strength of evidence were determined by examining the study design, sample size and representativeness, and data analysis strategies. Analysis and integration Study design Five studies were case studies of 2e4 adolescents each (Fisher & Fornari, 1990; Joseph, 2011; Money & Lewis, 1982; Schonfeld, 1962; Storch et al., 2004). One study was a retrospective chart review (Rosen et al., 2010), one was a prospective caseecontrol (Nuzzi et al., 2013) and the remaining three were non-experimental descriptive studies (Davanço et al., 2009;
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Total Citations n = 233 Duplicates Removed -n = 48
Abstracts Reviewed n = 185
Articles Reviewed n=8
Excluded due to 1) not GM focused; 2) not adolescent focused; 3) psychological correlates or treatments not addressed -n = 177
Additional Articles from Hand Searches +n = 2 Final Articles Reviewed n = 10 Fig. 1. Flow chart of literature searches on idiopathic gynecomastia in adolescents and young men.
Kasielska & Antoszewski, 2011; Kinsella et al., 2012). Four of the studies were conducted with males who were undergoing surgical correction of GM (Davanço et al., 2009; Kasielska & Antoszewski, 2011; Kinsella et al., 2012; Rosen et al., 2010) and one was conducted with males diagnosed with GM in an adolescent breast clinic that was part of a plastic surgery unit (Nuzzi et al., 2013); however, the rate and type of surgical intervention done with this sample was not discussed. Psychosocial correlates As noted in Table 1, a variety of psychosocial correlates were identified. These included several anecdotal “opinions” such as affecting personality, disturbed body image, feeling unacceptable, dissatisfaction with being male, avoiding participation in sports, excessive self-consciousness, disordered eating, social withdrawal, school disruptions, psychological stress, anxiety, fear, germ phobia, teasing, depressive symptoms, internalizing and externalizing behaviors, intense shame, emotional discomfort, interference with social relationships, feeling embarrassed, loneliness, and suicidal despair. Psychosocial correlates that were measured using psychometrically sound instruments included anxiety, depressive symptoms, and healthrelated quality of life. Psychosocial interventions Two of the studies suggested no psychosocial interventions for adolescents and young adults with gynecomastia. Psychosocial interventions that were suggested included vague terms such as psychotherapy, mental health treatment, and early intervention to improve both physical and emotional symptoms. We found no studies that reported the development or testing of psychosocial interventions specifically designed for young males with GM. The caseecontrol study by Davanço et al. (2009) found that quality of life was enhanced six months following surgical intervention for breast reduction in adolescent and adult males. In the study by Kinsella et al. (2012), all participants were offered surgical reduction, but only 50% could afford the surgery; all of the participants who did not receive surgical intervention were diagnosed with adjustment disorder, using criteria from the Diagnostic and Statistical Manual of Mental Disorders-IV (American Psychiatric Association, 1994). All of the young men surveyed by Kasielska and Antoszewski (2011) were treated surgically and significant improvements in emotional comfort and social relationships were found. Evaluation of evidence found In evaluating the evidence, the Johns Hopkins Nursing Evidence Rating Scale created by Newhouse and colleagues was used to rate both the strength (1e5), where 1 is highest (e.g., randomized controlled trial or meta analysis) and 5 is lowest
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Table 1 Data extracted for systematic review on gynecomastia in adolescence and young males arranged chronologically. Authors (date) Database 1
2
3
Methods
Descriptive case report N ¼ 284 “boys and men,” with GM, 88% first noted GM during adolescence. Illustrative case reports (n ¼ 4). Stated purpose was to “determine the effects of GM on personality adaptations”
Psychosocial correlates
Personality effects dependent upon: 1) age of onset, 2) size of breasts, 3) personality prior to onset. Described anxiety/fear; stress; ridicule; fantasies that girls were envious; feeling unacceptable; disturbed body image; self-consciousness; social withdrawal. Most affected if onset in adolescence Money and Case study type comparison of 3/10 of GM participants reported dissatisfaction with being a male, Lewis (1982) two clinical groups (i.e., idiopathic adolescent GM & identified as homosexual with Source: PsychInfo congenital virilizing cross-dressing, no history of adrenocorticism) on conformity male stereotypic sports to male stereotypes & sexual participation; orientation 4/10 with behavioral pathology N ¼ 18 (n ¼ 10 with GM, ages 14 including suicidal despair, germ e18 yrs, mean ¼ 15 yrs) phobia, and school disruptiveness. Social isolation (7/10) Teasing (8/10; 6/10 “viciously”) Case studies, N ¼ 2, ages 16.5 & Anorexia & bulimia nervosa Fisher and 1. “inadequate body” 17.5 Fornari 2. Breasts “were not going away” Weekly psychotherapy (1990) Source: PsychInfo Schonfeld (1962) Source: PsychInfo
4
Storch et al. (2004) Source: Medline
Letter to the editor. Case studies (N ¼ 2) Ages: 15 years and 17 years
5
Davanço et al. (2009) Source: From Ref. List
Non-experimental pre- postoperative measure of quality of life (SF-36) in males who had surgical intervention. Descriptive analysis. N ¼ 33, ages 18e50 (M ¼ 25.1). 91% had GM since puberty
6
Rosen et al. (2010) Source: Medline
Retrospective chart review of males who had surgical intervention. N ¼ 69 ages 12e21, patients from Adolescent Breast Clinic at Harvard Med School.
7
Joseph (2011) Source: PubMed
Case study, N ¼ 2; pair of siblings with partial androgen insensitivity syndrome (PAIS; Ambiguous genitalia). One raised as boy, other as girl
Psychosocial interventions
Limitations/quality and strength of evidence
“Psychotherapy played a significant role in the management of many of these cases.” Therapeutic goals: 1) determine significance to adolescents, 2) recognition of feelings with appropriate channeling, 3) alleviate guilt & inadequacy, 4) improve self-esteem
Not a systematic study No statistical analyses. Quality: C Strength: 5
None suggested
Small sample size Limited in focus (i.e., conformity to male stereotypes and sexual orientation) No statistical analyses. Quality: C Strength: 5
Small sample Onset of eating disorders directly related to desire to decrease breast size. No statistical analyses. Quality: C Strength: 5 Small sample size. Depressive symptoms: elevated Suggests improved routine Case study participant scores screening for psychological for both boys Anxiety symptoms: elevated in distress as a component of clinic compared to standardized visit and more focused clinical norms. 15 y/o No statistical analyses. intervention Loneliness: elevated in 17 y/o Quality: C Behavior (parent rating scale): Strength: 5 significant internalizing and externalizing symptoms reported by mom of 17 y/o Small sample Significant pre-post differences Authors suggest surgical on several domains of quality of treatment improves quality of Wide age range Wiloxon's nonparametric test of life in both physical and life: general health, p ¼ .001; emotional (i.e., mental health) pre- and post-op scores on functional capacity, p ¼ .002; domains in the SF-36 measure social aspects, p ¼ .002; mental domains of quality of life. health, p ¼ .004; vitality, p ¼ .007 Quality: B Strength: 3 None suggested Unclear how psychological 59.4% Reported psychological stress and other factors were stress; 27.9% reported reduced measured. physical activity; 78.3% of obese Chi squares, Fisher exact tests, patients reported psychological or t-tests done. stress (significantly more than Quality: C normal weight peers Strength: 4 preoperatively [p ¼ .02]) Gender identity concordant with “Boy” expressed hope related to Small sample size, N ¼ 1 of adolescent GM potential surgical correction how each reared. “Boy” developed GM at puberty; stating, “After the surgery I will No statistical analyses. reported intense shame, anxiety, be able to do anything.” (i.e., be Quality: C Strength: 5 being ridiculed/teased, avoided in public without feeling ashamed, return to school, train public/socially withdrew, quit for a better job) school grade 8. Elevated depressive symptoms, decreased quality of life per standardized tools “Girl”: no features of depression or any other psychiatric illness, “well-adjusted … with good 16.5 y/o: weekly psychotherapy; “superficially involved” terminated at 5 mos. 17.5 y/o: refused f/u appointments, further treatment or evaluation
(continued on next page)
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Table 1 (continued ) Authors (date) Database
Methods
8
Kasielska and Antoszewski (2011) Source: Medline
Descriptive, pre- and post-op. survey evaluation of males who had surgical intervention for GM. N ¼ 47 (Mean age 25.6 ± 3.5 yrs) Investigator developed survey with individual questions related to age of onset, coexisting disorders, and aspects of psychosocial effects & functioning (e.g., level of emotional discomfort, social distance, ridicule, & embarrassment)
9
Kinsella et al. (2012) Source: From Ref List
Descriptive N ¼ 24 ages 10e18 All participants had clinical interviews for psychiatric evaluation and 50% had surgery for GM. Half of sample underwent surgical correction Descriptive, comparison with healthy control group N ¼ 139 12e21 year old males (n ¼ 47 with GM and 92 healthy controls). Males with GM were recruited from a plastic surgery clinic.
10 Nuzzi et al. (2013) PubMed
Psychosocial correlates
functioning in social, personal, and academic realms” Pre-surgery: almost all reported emotional discomfort & limitations of daily activities. Majority reported interference with entry into social relationships & relationships with women, being teased, & feeling embarrassed. Statistically sig. improvements in psychosocial effects seen across all questions post-surgery (e.g., less emotional discomfort, more physical activity, improved social and romantic relationships) 100% of Participants diagnosed with DSM IV diagnoses: adjustment disorder (72.9%), anxiety d/o (16.7%), dysthymia (16.7%) “Most” self-reported decreased self-confidence & negative selfimage Health-related quality of life: GM adolescents scored lower than controls in: self-esteem, general health, vitality, social functioning, role-emotional, & mental health scored lower than controls Disordered eating behaviors: higher in GM adolescents than controls
Psychosocial interventions
Limitations/quality and strength of evidence
6 Participants had consulted psychologist prior to surgery (41 had not) After surgical treatment: 100% of participants reported improvement in emotional discomfort level & endorsed surgery as right decision.
Small sample from one surgery center. No psychometric properties established for survey. Chi-square tests. Quality: B Strength: 3
Mental health treatment recommended for all participants
Individual participants' scores on standardized measures compared to standardized norms, no group analyses Pearson chi squares and linear regressions done. Quality: B Strength: 3 Statistical testing controlled for BMI: differences in general health, social functioning, mental health, and self-esteem remained statistically significant. Quality: Level A Strength: 2
Early intervention and treatment recommended to improve negative physical and emotional symptoms
(e.g., opinion, personal experience, case study) and the quality (AeC) of the evidence, where A is high, and C is low (Newhouse, Dearholt, Poe, Pugh, & White, 2007). This allowed the team to review both the types of research being done in the area and the value of the research in understanding how GM influences the psychological development of adolescent males. It also provided insight into ways in which this problem may be best addressed in future studies. The strength of the 10 studies ranged from 2 to 5, with the majority (n ¼ 5) rated as a level 5 due to the emphasis on case studies, which used clinical expertise/experience as the foundation for the interpretation of findings. There were 3 studies that were rated level 3 due to their use of descriptive data, which were used to delineate the psychological correlates found in this group of adolescent males. Only one study met criteria for receiving an A level of quality and strength of 2; it was a quasi-experimental study that controlled for BMI and included adolescent males without GM (n ¼ 92) as a comparison group to study mental and physical health outcomes for those with GM (n ¼ 47) (Nuzzi et al., 2013). In this study, self-esteem and several domains of health (i.e., general health, vitality, mental health, emotional role, and social functioning) were significantly lower for adolescents (age 12e21) with GM, but physical functioning, physical roles, and pain, as assessed by physicians, did not significantly vary. Moreover, while no significant difference in prevalence of disordered eating behaviors was found, the adolescents with GM reported significantly worse attitudes towards eating than those without the condition. These authors used standardized, valid measures of psychological outcomes. The remaining studies were given an Evidence Rating Scale quality score of B or C. As shown in Table 1, these studies lacked methodological description, emphasis on expert opinion without evidential backing, and/or small sample sizes without randomization or independent sampling. Discussion The studies included in our review reflect a beginning level of knowledge development on the topic of adolescent GM. Overall the 10 articles reviewed included small samples ranging from the single-participant case study to a sample of 139. All studies were limited to single sites, such as surgery centers where young men went to have surgical breast reductions. Half of the studies (n ¼ 5) included no statistical analyses of the data. Most statistical analyses were limited to Chi-squares and linear
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regressions. The study by Kinsella et al. (2012) compared participants' scores on standardized measures to population norms. The population estimates for GM and its correlates varied widely within the reports. There was strong evidence to suggest that young adolescent males with GM are reporting emotional problems to their clinical providers. These providers have worked to gain the attention of health researchers and public health officials through their case reports. Regardless, over the last 50 years, little attention has been shown to GM. Schonfeld's seminal study (1962) introduced the topic over 5 decades ago, but it has received minimal response in the way of knowledge development that could guide professional medical care beyond surgical repair. Indeed, although date parameters were not placed on this review to enable the reader to see the shift in studies over time, the studies conducted from 1962 to 2004 demonstrated only the continued efforts by clinicians to publish case reportsdbringing repeated focus on the lack of evidence available. From 2009 until 2013, sample sizes increased, highlighting the possibly growing number of youth affected, but still, no large scale studies were conducted. In the process of including and excluding papers for this review, no studies were found that established a population prevalence for GM in adolescents. For this reason, there remains a lack of epidemiologic evidence of the incidence, prevalence and reoccurrence of gynecomastia and its associated psychosocial factors among adolescents and young adults. Consistently, over the past 50 years, young adolescent males have presented to their providers with GM, which does not resolve over time in as many as 10% of the population. The result has been that young men experience problems with selfesteem, eating disorders (Fisher & Fornari, 1990; Nuzzi et al., 2013), social withdrawal, body image disturbances (Money & Lewis, 1982), depression, anxiety (Storch et al., 2004), and lessened quality of life (Davanço et al., 2009). The only known intervention routinely offered has been surgical (Kasielska & Antoszewski, 2011); however, few data document its effectiveness in addressing the mental health problems that accompany this condition. Further, owing to the impact of psychosocial outcomes on long-term developmental trajectories, surgical interventions have been suggested to begin at early ages before body image disturbances, eating disorders, and low socialization overwhelm the adolescent's ability to cope (Davanço et al., 2009; Schonfeld, 1962). Yet, alternative treatments have not been studied systematically. Further studies are warranted to determine the population prevalence of idiopathic gynecomastia in adolescent males. Given the dearth of evidence found in the literature, evaluations of psychosocial symptoms and problems in adolescents should be investigated at intervals in those who also display symptoms of GM so that healthcare providers could determine for whom symptoms resolve and for whom they do not. Such knowledge is important so that healthcare providers can intervene in a timely manner. Moreover, interventions that specifically evaluate the pre- and post-operative symptoms of emotional and psychosocial distress may be warranted. Implications for practitioners Healthcare providers in primary care have been consistently educated that GM in adolescents is developmentally normal and in a majority of cases will resolve on its own within three years. Indeed, a leading pediatric primary care textbook addressed GM only once with the recommendation that providers question an adolescent regarding alcohol, marijuana, and anabolic steroid use if the condition persists (Burns, Dunn, Brady, Starr, & Blosser, 2013). The findings from this review, however, suggest this limited approach to management is missing the multiple psychological factors potentially associated with this condition. This review indicates that there are young men whose GM does not resolve and requires breast reduction due to emotional distress at varying points in their lives as well as those who develop severe and lasting mental health conditions. Healthcare providers working with adolescents need also consider the implications for the socio-emotional development that characterizes adolescence as well as the impact on establishment of health behaviors that may be carried into adulthood. Normal adolescent transitions involve developmental tasks associated with identity vs. role confusion and sexual maturation where issues related to gender-role socialization, body image, social relationships, attitudes, values, and self-esteem are addressed (Erikson, 1968). A condition such as GM that involves shame, embarrassment, ridicule, and negative affect towards self may have a devastating impact on many of these crucial developmental tasks. Even with spontaneous resolution of this idiopathic condition by the end of adolescence, the developmental consequences may affect that individual for the rest of their lives. The general lack of evidence regarding how often the condition occurs, how often it resolves, probability of recurrence, and associations with long term health outcomes necessitate, we believe, a more aggressive approach to ongoing assessment by healthcare providers than has traditionally been recommended. The findings of this review call for primary care providers to consider maintaining a closer evaluation of their patients' psychological and developmental status within the context of GM in order to prevent or curtail secondary developmental and mental health consequences. Conclusions The findings from this systematic review of literature about the psychosocial correlates of and interventions for adolescents and young adult males with idiopathic gynecomastia are meager. Spanning 50 years of literature, aside from descriptions of surgical interventions, very little is known about how this condition, if unresolved during adolescence, affects the psychosocial development and mental health of young males. More rigorous research with large and diverse samples is sorely needed to address this serious concern.
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