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Shyness, Social Anxiety, and Impaired Self-esteem in Turner Syndrome and Premature Ovarian Failure
To the Editor: Shyness and social anxiety are reported in women with Turner syndrome (TS).1 Possible contributors include physical stigmata, such as short stature and neck-webbing, chromosomally-based deficits in social cognition, and premature ovarian failure with infertility. To investigate the potential role of premature ovarian failure and infertility, we compared measures of psychosocial distress in women with TS, women with spontaneous karyotypically normal premature ovarian failure (POF), and healthy controls.
Methods
Participants in this institutional review board–approved study were recruited through National Institutes of Health (NIH) Web sites and newspapers and provided written informed consent. Inclusion criteria for patients with TS and POF are described elsewhere.2 Daily hormone therapy was taken by 99% of the women with TS and 90% with POF but was discontinued 2 weeks before evaluation.
Control participants were recruited from the local community and were paid a small stipend. They were required to have regular menstrual cycles, take no medications, and have no current or past medical or psychiatric conditions. Control participants were interviewed during the follicular phase of their menstrual cycle.
Testing was performed in the NIH Clinical Research Center between January 2001 and January 2004. Participants completed 4 rating scales previously validated in community populations: the shyness scale,3-4 social anxiety scale,5 Rosenberg's self-esteem scale,6 and the Center for Epidemiologic Studies–Depression Scale.7 From an initial sample of 103 women with TS, 100 fully completed all the surveys; and from an initial sample of 128 women with POF, 100 completed all surveys. Age and marital status did not differ significantly between participants and nonparticipants.
Group means were compared using analysis of variance with age and body mass index (calculated as weight in kilograms divided by the square of height in meters) as covariates. Multiple linear regression was used to examine the effects of age, years of education, years of hormone therapy (as a measure of duration of ovarian failure), and marital status on scores. For comparisons of rating scale scores between women with TS and those with POF, sample sizes of 96 to 104 women per group provided 90% power to detect a difference of 10% or greater on any of the rating scales with  = .05. Analyses excluding women with TS younger than 18 years showed similar results and are not reported. All analyses were performed using Stat View 5.0.1 (SAS Institute Inc, Cary, NC).
Results
There were group differences in the percentage of participants taking thyroid or antidepressant medication (Table 1). However, all women were euthyroid, and these medications were continued during the study. More women with POF were married, likely reflecting their initial presentation of infertility.
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Table 1. Characteristics of Women With Turner Syndrome, Karyotypically Normal Premature Ovarian Failure, and Healthy Controls
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Turner syndrome and POF groups scored significantly higher on the shyness scale, social anxiety scale, and the Center for Epidemiologic Studies–Depression Scale, and lower on the self-esteem scale compared with controls (Table 2). However, there were no significant differences between the TS and the POF groups for the scores on any of these scales. Age, years of education, years of hormone therapy, and marital status did not contribute to score variations in TS or POF.
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Table 2. Results of Psychological Tests
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Comment
In this study population, 2 dissimilar groups of women who had experienced premature ovarian failure had similar psychosocial profiles, with increased shyness, social anxiety, and depression, and decreased self-esteem compared with women with healthy ovarian function. These results are not likely to be due to ovarian hormone deficiency because the majority of women in both groups were taking hormone therapy. Short-term cessation of hormone therapy should not result in changes in these scales that measure chronic social distress. Although our results do not provide an explanation for this symptom profile, uncontrolled studies8 suggest infertility as a factor.
Study limitations include the use of self-selected groups that were primarily white, relatively well-educated, and took the initiative to participate in NIH protocols; as such, they may not be representative of other women with premature ovarian failure. Control participants were screened for the absence of psychiatric illness so their rating scale scores could have been biased toward less distress than those from a general population. As a cross-sectional study, conclusions about causality cannot be made and, as an observational study, associations could be due to unmeasured confounding. To confirm these findings, it is important to study other groups, such as young women with ovarian failure secondary to cancer or cancer therapy. Nevertheless, these results suggest that clinicians should consider these psychosocial issues in addition to the medical consequences for patients with premature ovarian failure.
Author Contributions: Dr Schmidt had full access to all the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.
Study concept and design: Schmidt, Ross, Rubinow, Bondy.
Acquisition of data: Schmidt, Cardoso, Ross, Haq, Bondy.
Analysis and interpretation of data: Schmidt, Cardoso, Ross, Rubinow, Bondy.
Drafting of the manuscript: Schmidt, Cardoso, Bondy.
Critical revision of the manuscript for important intellectual content: Schmidt, Haq, Rubinow.
Statistical analysis: Schmidt, Cardoso, Ross, Haq, Rubinow, Bondy.
Obtained funding: Schmidt.
Administrative, technical, or material support: Schmidt.
Study supervision: Schmidt.
Financial Disclosures: None reported.
Funding/Support: This work was supported by the Intramural Research Programs of the National Institute of Mental Health and National Institute of Child Health and Human Development and supported in part by grant NS42777 from the National Institutes of Health (Dr Ross).
Role of the Sponsors: The sponsors had no role in the design and conduct of the study; collection, management, analysis, and interpretation of the data; and the preparation, review, or approval of the manuscript.
Acknowledgment: We thank the study participants and are grateful to Eileen Lange, RN, CCRP, and Vien Vanderhoof, LNP, for recruiting and caring for our patients. We also thank Lawrence Nelson, MD, for pioneering research on premature ovarian failure and Vladimir K. Bakalov, MD, for informal uncompensated statistical consultation.
Peter J. Schmidt, MD
peterschmidt{at}mail.nih.gov
Graca M. P. Cardoso, MD
Behavioral Endocrinology Branch
National Institute of Mental Health
National Institutes of Health
Bethesda, Md
Judith L. Ross, MD
Department of Pediatrics
Thomas Jefferson University
Philadelphia, Pa
Nazli Haq, MA;
David R. Rubinow, MD
Behavioral Endocrinology Branch
National Institute of Mental Health
Carolyn A. Bondy, MD
Developmental Endocrinology Branch
National Institute of Child Health and Human Development
National Institutes of Health
Bethesda, Md
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Letters Section Editor: Robert M. Golub, MD, Senior Editor.
JAMA. 2006;295:1374-1376.
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