Materials and Methods
Study design, outcome variables, and population
This was a prospective, longitudinal, single-center study of all women with vaginal hypoplasia and without a previous history of vaginal surgery, who were prescribed vaginal dilation treatment, between March 2010 and April 2013. Participants followed the treatment protocol outlined in Figure 2 . The outpatient program was designed based on previous experiences and careful exploration of the literature, especially motivational and behavioral psychology.
At baseline (T0), at the stop of treatment (T1), and at 1 year follow-up (T2), validated standardized questionnaires on emotional and sexual well-being were completed ( Table 1 ), and gynecological examinations were performed to measure vaginal length. Semistructured interviews included questions about the participant’s reaction toward the diagnosis and experience of dilator treatment (advantages and disadvantages), information provision, and compliance with dilation treatment. The study was approved by the local ethics committee (EC 2010/030), and a written informed consent had been obtained at T0 from all participants.
| Outcome variable | Definition | Method | Reference |
|---|---|---|---|
| Gynecological assessment | |||
| Vaginal length | Length in centimeters from the posterior fourchette to the most proximal part of the blind ending vagina | Measured by inserting a cotton bud into the vagina by an experienced gynecologist | |
| Vaginal perceptions | How do you perceive your vagina? | Participants are asked to choose (1 or several) from the following 6 statements: (1) my vagina is more or less normal; (2) I do not know or I am not sure; (3) my vagina is tiny or nonexistent; (4) my vagina is small (short or narrow); (5) a sexual partner would notice it is different from other women; (6) I would like a bigger vagina (longer or wider) | |
| Psychological assessment | |||
| Validated questionnaires | |||
| Female Sexual Function Index (FSFI) | Six domains of female sexual function: desire, arousal, orgasm, pain during intercourse, vaginal lubrication, and global sexual and relationship satisfaction | 19-item questionnaire with 0-5 rating denoting variations in frequency, intensity or degree of satisfaction with the sexual domains (each with a total score between 0 and 6). Higher scores indicate more normal sexual function during the last month preceding the survey | |
| Female Sexual Distress Scale, revised (FSDS-R) | Different aspects of sexual distress | 13-item questionnaire with 0–4 rating (0 never- 4 always). The higher the total score (0-52), the higher the level of sexual distress during the last month preceding the survey | |
| TNO-AZL Questionnaire for Adult’s Health-Related Quality of Life (TAAQoL) | Ten quality-of-life domains: cognition, sleep, pain, social contacts, daily activities, sex, vitality, happiness, depressive moods, and anger moods | Occurrence of problems on the different domains with 1-4 rating (1, never, to 4, often) and degree to which the participant was actually emotionally bothered by that problem (1, not at all to 4, very much) during the last month. Scores for each scale are transformed to scores between 0 and 100, with higher scores indicating a higher occurrence of health-related quality-of-life problems and distress | |
| Youth Self-Report (YSR; ages 11-18 years) and Adult Self-Report (ASR; ages 18-59 years) | Internalizing behavior (ie, overcontrol of emotions, such as social withdrawal or feelings of inferiority) and externalizing behavior problems (ie, undercontrol of emotions, such as rule-breaking behavior) and a combined total problem score | Participants are presented with statements related to emotional and behavioral problems using a 3-point format to establish the frequency of problem behaviors (0, not true; 1, somewhat true; 2, very true). Scores for externalizing and internalizing behavior problems and a total problem score are converted to T-scores with M = 50 and SD 10. Problems are clinically relevant when T-scores are greater than 65 | |
| Body Image Scale | Satisfaction with body image and genital image | Satisfaction regarding the genital area (mean of 4 aspects: vagina, clitoris, labiae, pubic hair) and total body image (mean of 28 aspects [eg, arms, legs, nose]) coded on 1 (very satisfied) to 5 (very dissatisfied) scale | |
| Rosenberg Self-Esteem (RSE) | Self-esteem | Participants are presented with 10 self-worth statements, coded on a 0 (strongly disagree) to 3 scale (strongly agree). Higher total scores (0-30) indicate a higher global self-esteem | |
| Semistructured interview | |||
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Statistical analysis
Statistical analyses were performed using SPSS version 20.0 (SPSS Inc, Chicago, IL). Nonparametric Wilcoxon signed-rank tests were conducted with a Bonferroni correction for comparing mean rank scores for emotional and sexual well-being at T0, T1, and T2. Nonparametric effect size (ES) estimates and 95% confidence intervals were calculated. Mann-Whitney U tests assessed differences in anatomical success when controlling for the frequency of dilation (≤4 or >4 times a week), sexual activity during dilation program (yes or no), and karyotype (46, XY or 46, XX). Associations between variables were sought using Spearman correlation coefficients. A value of P < .05 was considered statistically significant. Two-tailed statistical tests were chosen to reduce the risk of type I errors.
Results
Of the 18 women approached, 16 agreed to participate ( Figure 3 ). Two women were reluctant to discuss this sensitive issue, but both did start with the dilation program. The characteristics of the participants are displayed in Table 2 .
| Number | Diagnosis | Age at start dilation, y | Partner at start dilation | Status | Total duration of dilation treatment, mo | Sexual activity | ||
|---|---|---|---|---|---|---|---|---|
| Before (T0) | After (T1) | Follow-up (T2) | ||||||
| 1 | MRKH | 15 | Yes | Finished dilation | 12.0 | No | No a | No |
| 2 | MRKH | 17 | Yes | Finished dilation | 1.5 | No | Yes | Yes |
| 3 | MRKH | 17 | Yes | Finished dilation | 8.0 | Yes | No | Yes |
| 4 | Denis-Drash syndrome | 23 | Yes | Finished dilation | 5.5 | Yes | Yes | Yes |
| 5 | MRKH | 18 | Yes | Finished dilation | 7.0 | No | Yes | Yes |
| 6 | MRKH | 18 | No | Finished dilation | 9.0 | No | No | No |
| 7 | CAIS | 22 | No | Finished dilation | 3.0 | No | Yes | No |
| 8 | MRKH | 16 | Yes | Finished dilation | 6.0 | Yes | Yes | TBC |
| 9 | CAIS | 18 | Yes | Finished dilation | 3.0 | No | Yes | TBC |
| 10 | MRKH | 23 | Yes | Finished dilation | 3.0 | Yes | Yes a | TBC |
| 11 | MRKH | 18 | No | Dilation in progress | TBC b | Yes | TBC | TBC |
| 12 | MRKH | 17 | Yes | Dilation in progress | TBC | Yes | TBC | TBC |
| 13 | MRKH | 17 | No | Dilation in progress | TBC | No | TBC | TBC |
| 14 | MRKH | 15 | No | Stopped dilation plus Vecchietti | 9.0 | Yes | Yes | No |
| 15 | MRKH | 15 | No | Stopped dilation plus Vecchietti | 5.0 | No | No | Yes |
| 16 | 5αRD | 19 | Yes | Stopped dilation plus sigmoid vaginoplasty | 3.0 | Yes | Yes | TBC |
a Did not fill in (all) questionnaires
b She had already dilated before during 3 months in another hospital but was unhappy about the lack of psychological counseling during treatment.
Attitudes toward vaginal dilator use before and after dilation treatment
Table 3 summarizes the benefits and disadvantages of dilation treatment as mentioned by the participants. Before the start, they demonstrated good knowledge on the purpose of using vaginal dilators (ie, to have normal/pain-free intercourse) and to possibly avoid surgery and scars (ie, implying visibility of the condition). After treatment, women included also advantages of connectedness to the body and feeling better as a woman, but the majority reported that dilator use was an unpleasant reminder of abnormality.
| Variable | Number of participants |
|---|---|
| Before treatment | |
| Benefits of dilation | |
| Being able to have normal/pain-free sex, whenever I want | 13/13 |
| Being in control (of the pain) | 3/13 |
| Better than surgery (eg, no scars, no possible loss of sensation) | 3/13 |
| Disadvantages of dilation | |
| Feel abnormal | 2/13 |
| Pain/discomfort | 2/13 |
| Afraid of association of sex with dilation | 1/13 |
| After treatment a | |
| Benefits of dilation | |
| Enough vaginal length to have sex | 8/13 |
| Being more connected with your own body through dilation | 3/10 |
| You achieve something yourself | 3/10 |
| Feel better as woman | 1/10 |
| Disadvantages of dilation | |
| Unpleasant reminder of something preferably forgotten (eg, “It [dilation] reminds me of being abnormal”) | 8/10 |
| Chore/boring | 4/10 |
| Discomfort/pain | 4/10 |
| Fear of pushing too hard or hurting myself | 2/10 |
| Too slow progress | 2/10 |
| Privacy/discretion needed | 2/10 |
| Disadvantages of not dilating | |
| Lose any progress already made and feeling guilty of not doing it | 3/10 |
| Will not be able to have “normal” sex | 1/10 |
| Have to have surgery | 1/10 |
a Not including the 3 women who are currently still in the program.
Vaginal dilation frequency, vaginal size, and total duration of therapy
Ten women (77%) successfully completed the program, and 9 women (69%) achieved a value within the normal range (6.5–12.5 cm) ( Table 4 ). All women started to dilate as recommended (15-20 minutes daily, 7 days a week), but after 1-3 months the reported frequency decreased significantly, ranging from 3-4 days a week to once a week in 7 of 10 women, both in women with and without a partner (63% vs 100%, P = .30).
| Variable | Vaginal length before dilation, cm | Vaginal length after dilation, cm | Total duration of dilation treatment, mo |
|---|---|---|---|
| Dilation only (n = 10) | 2.7 ± 1.2 | 7.7 ± 0.8 | 5.8 ± 3.3 |
| Frequency of dilation | |||
| ≤4 days per week (n = 5) | 2.7 ± 1.3 | 7.2 ± 0.4 | 6.7 ± 4.4 |
| >4 days per week (n = 5) | 2.7 ± 1.3 | 8.2 ± 0.8 | 4.9 ± 1.8 |
| Intercourse during treatment a | |||
| Yes (n = 4) | 2.1 ± 0.9 | 7.6 ± 0.3 | 5.9 ± 2.2 |
| No (n = 5) | 2.8 ± 1.2 | 7.5 ± 0.9 | 6.3 ± 4.3 |
| Dilation plus surgery (n = 3) | |||
| ≤ 4 days per week (n = 3) | 3.5 ± 1.3 | 4.8 ± 0.7 | 5.7 ± 3.1 |
Reported motives were as follows: time and effort constraints (eg, no dilation during certain time periods such as examinations (n = 5), privacy reasons (ie, shared student housing) (n = 2), and less perceived vaginal increase (n = 1). Those who dilated frequently (more than 4 days a week) had a significantly larger end vaginal length ( P = .034) and a nonsignificant shorter total duration of dilation therapy ( P = .459) ( Table 4 ). There was no significant correlation between the start or end vaginal length and duration of treatment (R s = –0.44, P = .204 and R s = –0.14, P = .69, respectively) or between the start and end vaginal length (R s = 0.06, P = .86). Although not significant, women with MRKH (n = 12) had on average a smaller start vaginal length than women with 46, XY disorders of sex development (n = 4) (2.5 ± 1.0 cm vs 3.6 ± 1.5 cm, P = .159).
Three participants (numbers 14-16) stopped with dilator therapy because of little perceived progress (n = 3), pain (n = 2), and negative confrontation with the condition (n = 3). At the stop they had gained significantly less vaginal length ( P = .009) ( Table 4 ), although they had a comparable start vaginal length and dilated during a comparable period but with a lower frequency, especially toward the end (less than once a week). A decrease in vaginal length could even be observed ( Figure 4 ). These women were on average younger (16.3 ± 2.3 years vs 18.4 ± 2.6 years), although not significantly ( P = .244). After surgery, they had a significant longer vaginal length (10.3 ± 2.1 cm) compared with the women after dilation therapy only (7.7 ± 0.8 cm, P = .029).
