Discussion Questions

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  What were the study objectives?

  
  
  
  

  
  

  See related article, page 484

  
  
  
  

  
  

  For a summary and analysis of this discussion, see page 504

  
  
  
  
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  What was the study design?

  
  
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  What were the key findings of the study?

  
  
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  What were the study’s strengths and weaknesses?

  
  
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  Will this study change your practice?

  
  
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  What are some next steps for research in this area?

Introduction

Patients with Turner’s syndrome (TS), who number 1 in 2500 females, are missing all or part of 1 X chromosome. The disorder is marked by short stature, gonadal dysfunction, and dysmorphic traits. While intelligence and verbal abilities are usually normal, some patients have nonverbal learning disabilities. Other congenital hypogonadisms (OCH) in patients with a 46,XX karyotype may not be attended by physical or neurocognitive abnormalities. Comparable psychosocial difficulties have been reported for women in both groups. Ros and colleagues further explored these links and described quality of life (QoL) and sexual function in women with these diagnoses.

Camaryn Chrisman Robbins, MD, MPH and George A. Macones, MD, MSCE, Associate Editor

Introduction

Patients with Turner’s syndrome (TS), who number 1 in 2500 females, are missing all or part of 1 X chromosome. The disorder is marked by short stature, gonadal dysfunction, and dysmorphic traits. While intelligence and verbal abilities are usually normal, some patients have nonverbal learning disabilities. Other congenital hypogonadisms (OCH) in patients with a 46,XX karyotype may not be attended by physical or neurocognitive abnormalities. Comparable psychosocial difficulties have been reported for women in both groups. Ros and colleagues further explored these links and described quality of life (QoL) and sexual function in women with these diagnoses.

Camaryn Chrisman Robbins, MD, MPH and George A. Macones, MD, MSCE, Associate Editor

Study Design

Chrisman Robbins: What were the primary and secondary objectives of this study?

Wolfe: The primary objective of the study was to describe quality of life (QoL) and sexual function among women with TS and OCH. These were measured using the Medical Outcome Study Short Form-36 (SF-36) questionnaire in all patients and the Female Sexual Function Index (FSFI) questionnaire in sexually active patients. Scores were compared to questionnaire responses in controls. Secondarily, the authors examined whether the presence of dysmorphic features or other comorbidities affected sexual activity or sexual function in TS patients.

Chrisman Robbins: Please describe the SF-36 and the FSFI.

Squires: The SF-36 is a questionnaire containing 36 self-administered questions developed to assess QoL, including 4 physical health domains (physical functioning, role physical functioning, bodily pain, and general health) and 4 mental health domains (vitality, role emotional functioning, social functioning, and mental health). Domain scores range from 0–100 and higher scores correlate with better QoL. The questionnaires were scored using both standard and country-specific algorithms, and the results for each domain were calculated to provide an average for each domain; averages were then normalized for the Spanish population.

The FSFI contains 19 self-administered questions used to assess 6 sexual domains (desire, lubrication, arousal, orgasm, global satisfaction, and pain). Each question is scored from 0-5 with higher scores being associated with better sexual status. The final score is calculated by adding the scores of each domain. Both questionnaires are standardized and previously validated.

Chrisman Robbins: Do you believe the FSFI is a useful tool in assessing female sexual function ?

Weiner: The FSFI is generally acknowledged to be an extremely well-worded and easily understood screening measure for female sexual dysfunction with good psychometric characteristics. Its use provided a quick and easy measure of the 6 sexual domains. While it is a generally useful tool, there have been some concerns about scoring procedures creating conceptual and statistical problems. For example, subjects’ responses to question 4 of the FSFI—“within the last 4 weeks, how would you rate your level of sexual arousal during sexual activity or intercourse”—can be misleading. There are many reasons besides sexual dysfunction that could account for a 4-week lapse in sexual activity. The Derogatis Sexual Functioning Inventory contains 254 items and is somewhat longer to complete, but it is also self-administered and available in Spanish. It would be interesting to compare results using this instrument.

Chrisman Robbins: Who were the study participants? What was the study design?

Wolfe: Forty seven women, between 20 and 50 years of age, were recruited from an endocrinological gynecology outpatient clinic in Barcelona and identified based on primary amenorrhea. The first cohort was made up of 26 women with TS. A second cohort of 21 women with OCH consisted of 7 with pure gonadal dysgenesis and 14 with idiopathic hypogonadotropic hypogonadism. Diagnoses in these 2 cohorts were based on American Society for Reproductive Medicine criteria. All were receiving hormone replacement therapy for development of secondary sexual characteristics. In addition, 41 age-matched controls who were being treated with hormonal contraception or replacement therapy for more than 1 year were enrolled in the study. Further details on identification and recruitment of subjects were not provided.

According to the researchers, this was a “prospective cohort study” in which they identified patients with certain exposures (in this case, TS or OCH) and those without (controls) and evaluated the effect of the disorders on QoL and sexual functioning. However, the data were questionnaire responses from a single point in time as opposed to data accumulated by following patients over an extended period and seeing how the outcome was affected by the exposure. Were we to classify this as a cohort study, it would be a retrospective cohort, as the questionnaires specifically asked about health and function in the weeks and years prior to filling out the form. Data were not collected on a prospective basis.