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Int Urogynecol J

DOI 10.1007/s00192-013-2316-3

REVIEW ARTICLE

Systematic review of sexual function and satisfaction following


the management of vaginal agenesis
Sarah K. McQuillan & Sonia R. Grover

Received: 22 September 2013 / Accepted: 21 December 2013


# The International Urogynecological Association 2014

Abstract Historically, sexual satisfaction following the management of vaginal agenesis was assessed subjectively.
Standardized sexual function questionnaires are being used
more frequently as instruments to accurately and more objectively assess the subjective nature of sexual outcomes as part
of a more holistic approach to the care of women with vaginal
agenesis. Articles concerning the management of vaginal
agenesis were systematically reviewed, with specific focus
on those that discussed functional outcomes, sexual satisfaction and psychosomatic outcomes, and in particular attempted
to measure these outcomes. A total of 6,691 articles on vaginal
agenesis were identified, with 106 of these reporting sexual
satisfaction and psychosomatic outcomes. Only 1 randomized
control trial (RCT) was identified, the remaining articles being
made up of case series or case reports. Only 17 articles used
standardized objective assessment of sexual satisfaction.
While the bowel technique had the longest vaginal length at
12.87 cm, it had the most number of complaints of
dyspareunia (4.8 %), stenosis (10.5 %) and the lowest average
subjective sexual satisfaction. The Davydov method used
standardized sexual function assessments most frequently.
This technique had a higher average score than both the bowel
vaginoplasty technique in the only RCT and the Vecchietti
method in a prospective assessment. Overall, the management
S. K. McQuillan (*)
The Royal Childrens Hospital Melbourne, Australia, University of
Melbourne, Melbourne, Australia
e-mail: sarah.mcquillan@medportal.ca
S. R. Grover
Department of Paediatric and Adolescent Gynaecology, The Royal
Childrens Hospital, 50 Flemington Road, Parkville, Melbourne,
VIC, Australia 3052
S. R. Grover
Department of Paediatrics, University of Melbourne, Mercy Hospital
of Melbourne, Melbourne, VIC, Australia

of vaginal agenesis requires a multidisciplinary approach to


fully support these patients from initial diagnosis, through
management decision-making and long-term follow-up,
through transition to adulthood.
Keywords Management . Sexual satisfaction . Vaginal
agenesis

Introduction
Vaginal agenesis occurs in a number of different settings,
ranging from an isolated congenital anomaly, to a part of a
specific disorder of sexual development [1], to an iatrogenic complication of radiation or surgery, early or later in life.
Vaginal agenesis resulting from either MayerRokitansky
KusterHauser syndrome (MRKH) or androgen insensitivity syndrome (AIS) affects between 1 in 4,000 and 1 in
13,000 girls worldwide [2]. Currently, there are many
methods described in the literature for creating a neo-vagina,
from a conservative approach using pressure through dilation
[3] or surgically with traction [4] to more active surgical
approaches, which have previously been summarized in the
October 2013 edition of the International Urogynecology
Journal [5].
Historically, neo-vaginal creation success has been determined by measuring the vaginas length and at times also the
vaginal angle, with the implication that vaginal length and/or
angle might be a surrogate marker for successful sexual function [6]. However, the capacity of a vagina, although it may
imply that penetrative intercourse is possible, is unlikely to
adequately inform regarding the more important issues of
sexual function and satisfaction.
In the past, sexual satisfaction was assessed both objectively by measuring vaginal length and subjectively by simply
asking the patient if she was sexually active [7] and then also

Int Urogynecol J

sexually satisfied, either by answering yes or no [8, 9], not


defined [10, 11] or using a Likert scale up to 100 [12].
Additionally, asking her current partner whether they were
satisfied sexually was also considered an acceptable endpoint
in several articles [8, 1315].
Both the Female Sexual Function Index (FSFI) [16] and the
GolombokRust Inventory of Sexual Satisfaction (GRISS)
[17] have been validated in the general population as tools
for the assessment of sexual function and are increasingly
being used as an objective measure of sexual satisfaction in
women following neo-vagina creation as a way to standardize
the assessment of sexual function. The FSFI specifically looks
at six domains of sexual function, including desire, arousal,
orgasm, sexual satisfaction, lubrication, and pain, whereas the
GRISS is a questionnaire assessing seven domains of sexual
dysfunction including: anorgasmia, vaginismus, communication, frequency or avoidance of sexual contact, sensuality
(non-sexual touching), and satisfaction.
The purpose of this systematic review of sexual outcomes
of the various methods of vaginoplasty in MRKH and AIS is
to expand upon a previous review article summarizing objectively all available intraoperative and postoperative (both
short- and long-term) possible complications from the last
20 years [5]. Other conditions, where the vagina is absent, in
the context of multiple other anomalies (such as a cloacal
anomaly) or there is a hypoplastic vagina (secondary to surgery or radiotherapy), that require special consideration, have
been excluded because attempts at comparison between operative techniques is even more difficult. Conditions where there
is a uterus present also require careful consideration regarding
the possibility of allowing a fertility potential and thus this
potential would further complicate any assessment of sexual
function outcomes [18].
Thus, for the purposes of this review of sexual function,
satisfaction, and outcomes in women following neo-vaginal
creation, only MRKH and AIS patients will be included in the
analysis of sexual outcomes of the various treatment modes.

Materials and methods


A description of the methodology (including the full reference
list) used to identify all English language papers on vaginal
agenesis from 1992 onward in patients with MRKH and AIS
is described in our previous paper published in October 2013
in accordance with the PRISMA (Preferred Reporting Items
for Systematic Reviews and Meta-Analyses) checklist for
systematic reviews [5, 19]. The results of that previous literature search (including 162 articles) were then further restricted to articles referring specifically to sexual and psychosocial
outcomes. The two review authors (SM and SG) then independently assessed the articles included, specifically
discussing sexual outcomes and looking for the following

extracted data: patient population, vaginal length, sexual activity, timing of first intercourse, and sexual satisfaction (measured objectively or subjectively), including stenosis and
dyspareunia. The inclusion of papers looking at the quality
of life (QoL), depression, body image, and other factors that
might influence sexual satisfaction were reviewed for possible
inclusion in the discussion. For the studies included, the
assessed results reported included identification of the possibility of inherent author biases and patient attrition. Given the
potential of variation in the results reported, we attempted to
dichotomize data as much as possible for analysis. Data were
then synthesized and analysis was carried out using statistical
software. The limitations of the results were then summarized
in the discussion.

Results
A total of 6,691 articles were identified using the key search
words vaginal agenesis, Mllerian agenesis plus management up to June 2013. A total of 162 articles were
identified that fulfilled our previously defined selection
criteria and these were reviewed, accounting for 4,326 patients. Prior to 1992, laparotomy was the standard of care for
neo-vaginal creation, with the first articles pertaining to laparoscopic neo-vaginal creation only being introduced in 1992
[20, 21]. Of these articles identified in the previous systematic
review, 106 articles contained information regarding the sexual or psychosocial outcome (as mood may have an impact on
sexual function and satisfaction) of treatment. However, only
17 articles looked objectively at sexual outcome using the
FSFI [1227] with 2 articles comparing the FSFI outcomes
with regard to surgical techniques [22, 23] and a further 2
articles using the GRISS [24, 25] for sexual satisfaction assessment (Fig. 1). Also, 1 article on 150 sigmoid vaginoplasty
patients reported significantly higher FSFI scores than in a
standard control population; however, no objective FSFI number was reported [26].
The greatest lengths achieved with a neo-vagina were
created using the surgical bowel vaginoplasty method, with
an average vaginal length of 12.87 cm (confidence interval
[CI] 12.7612.98 cm) [5]. In contrast, the conservative dilation method resulted in the shortest average vaginal length of
6.65 cm (CI 5.268.04 cm) [5]. Statistical significance between the various neo-vaginal techniques was not calculated
owing to the heterogeneity between the groups in terms of preoperative use of dilation, surgical techniques, and vaginal
lengths prior to any intervention. Overall, all vaginas were
reported to be at least two finger-breadths wide (Table 1).
Of the articles reporting engagement in sexual activity, the
dissection vaginoplasty method had the highest percentage of
patients asked (254 out of 275) who reported sexual activity
post-treatment. However, these results were skewed by the

Int Urogynecol J
Fig. 1 Summary of articles
included and excluded pertaining
to sexual outcomes following the
management of vaginal agenesis.
Literature review of the
management of vaginal agenesis

6691
articles
1569
non
english

1769
prior
to 1992

310 8 non
MRKH/AIS
Previo us
surgery

83
Case
reports

162
included

28 Dilation
19 Vecchietti

10 Sheares/
Williams/
Creatsas

13 Davydov
(peritoneal)
12 Full
thickness
flaps

12
3 FSFI

15
3 FSFI

inclusion of Creatsas data, which reported a coitus rate of


100 % in all 200 patients who underwent surgery [27]. The
lowest coitus percentage of those patients questioned was
found in the full-thickness vaginoplasty group at 69.2 % (72
out of 104) followed by the split-thickness group (79.7 %, 169
out of 212). In terms of intention-to-treat analysis, the splitthickness vaginoplasty group was the least likely to engage in
coitus postoperatively, with only 169 patients out of the conglomerate of 688 patients.
The timing of first penetrative intercourse ranged from an
average of just 49 days following the Vecchietti procedure to
upward of 7 months (range 2 to 25 months) with the Creatsas
method. However, even with a delay in first penetrative sexual
activity, Creatsas et al. report that there is no need for postoperative dilation [27].
With regard to reports on dyspareunia and the need for
lubricants, Brucker et al., using a modified Vecchietti procedure, reported that none of the patients required lubricants, nor
did any of their cohort of 101 patients experience sustained

6
1 FSFI

12
5 FSFI

7
0 FSFI

50 graft
technique

45 bowel
technique

38 split
thickness

22
3 FSFI

32
5 FSFI

pain during intercourse. However, there was 1 patient with


deep dyspareunia and 7 complaints of superficial dyspareunia.
This report does not include any formal standardized assessment of sexual function or satisfaction for their patient population [28]. Interestingly, many articles report vaginal stenosis
in their patients, although there does not seem to be any
correlation between stenosis and dyspareunia, with many
more patients having stenosis than complaints of dyspareunia.
The largest number of complaints of dyspareunia occurred
with the bowel vaginoplasty cohort (4.8 %, 45 out of 945),
which also had a 10.5 % rate of stenosis complaints (n=99).
Regarding subjective sexual satisfaction scores, the bowel
vaginoplasty neo-vaginal patients had the lowest overall score
(82.6 %). In contrast, the full-thickness flap method reported
the highest subjective sexual satisfaction at 97.8 % (69 out of
72) in patients currently sexually active; however, they had the
fewest number of cases included overall and the least number
of respondents engaging in sexual activity post-treatment
(69.2 %).

Int Urogynecol J
Table 1 Summary of sexual outcomes following the creation of a neovagina in MayerRokitanskyKusterHauser syndrome (MRKH) or androgen insensitivity syndrome (AIS) patients. The summary results were

derived by pooling the data from the articles included. While not an ideal
form of statistical analysis, there was no heterogeneity between articles,
making a formal meta-analysis impossible

Analysis

Dilation

Vecchietti
Davydov (including Sheares
(including
both open and
both open and laparoscopy)
laparoscopic)

Full
thickness
flap

Split
thickness
flap

Bowel vaginoplasty
(including both
open and
laparoscopic)

Total number of patients


Long-term complications
Length (cm)a
Stenosis (absolute number)
Vaginal discharge (absolute
number)
Sexual results

802

934

500

289

166

688

945

6.65
0
0

7.87
4
1

8.86
2
3

11.49
5
0

9.04
9
0

8.84
46
9

12.87
99
71

3
80
5 monthse
92
25.7

24
83.8
49.8 days
83
29.8

0
81.5
Not available
96.7
27.6

3
92.4
7 monthse
84.1
29.9

2
70.9
Not available
96.2
Not available

31
45
75.8
86.3
4 to 6 months 4.3 months
90.7
82.6
28.2
27.8f

Dyspareunia (absolute number)


Penetrative sexual activity (%)b
Time to sexual activity
Sexual satisfaction (%)c
Female Sexual Function Index
(maximum score 36)d
a

All lengths were calculated as weighted averages including all articles mentioning vaginal length with all widths over two finger-breadths when width
was reported (previously reported in the accompanying review)
b
Penetrative sexual activity denominator is based on the number of patients who were asked about intercourse (no standardization of patients asked) and
is presented as a percentage, as the majority of articles grouped their sexually active patients and presented the data as percentages within the individual
articles
c

Sexual satisfaction not defined in the some studies and ranged from asking the patient directly, asking the partner regarding their satisfaction, using
different reference ranges from scores out of 10 to out of 100 to Likert scales with no standardization; thus, the results were pooled for simplicity

All FSFI are weighted average

Only 1 study mentioned time to intercourse

Including Erman Akar et al. [61], where 11 of the 20 women who answered the FSFI scored over 25

The FSFI was first introduced as an assessment of sexual


satisfaction in the vaginal agenesis population in 2003 [29].
Since that time and especially since 2010, the FSFI score has
seen increased use. The highest sexual function score was in
the population who had undergone a split thickness neovaginal method using oxidized cellulose. In one article, the
FSFI score was 32.5; however, the general population studies
on FSFI report scores of only 30.5 [16]. However, the sample
was very small (n=8) and intention-to-treat (ITT) was not
employed, with only 6 of the 7 patients sexually active
responding [30]. The Davydov peritoneal method used FSFI
assessment most frequently to report sexual function in 5 of
the 12 articles discussing sexual outcomes [10, 22, 23, 3142].
The lowest reported average FSFI score (21.4) was in 6
patients who underwent the Davydov procedure; however,
this is the only assessment of sexual function that used
intention-to-treat analysis and administered the questionnaire
to all patients (including patients who may not recently or ever
have engaged in penetrative sexual activity) [31]. The overall
lowest weighted average FSFI score was in the dilation cohort
(FSFI=25.65 [CI 24.8326.47]). In the articles looking at the

individual FSFI components, lubrication was the most commonly cited reduced score.
The only randomized control trial (RCT) looking at neovaginal creation revealed a trend toward a higher FSFI score
using the Davydov method compared with a sigmoid bowel
vaginoplasty [22]. Bianchi et al. looked at FSFI scores prospectively between the laparoscopic Davydov and Vecchietti
techniques, again revealing a trend toward a higher score
using the Davydov method (FSFI=31.8), which also correlated with a significantly longer vagina at 12 months (8.5
1.1 cm) [23].

Discussion
The premise behind creating a vagina in MRKH and AIS
patients is to normalize the genital anatomy and allow for
functional and potentially satisfactory sexual activity.
Ironically, many articles in the neo-vagina literature associate
sexual outcome with vaginal length, the proponents of surgical neo-vaginal creation justifying their technique on the basis

Int Urogynecol J

of achieving a good vaginal length. The bowel vaginoplasty


literature with the longest overall average vaginal length,
nevertheless, has the lowest overall subjective sexual satisfaction scores.
The initial approach of assessing the outcome of
vaginoplasty using vaginal length as a surrogate measure is
clearly limited, despite the work by Liao and Minto demonstrating some correlation between vaginal length and satisfaction. Liao et al. looked at both emotional and sexual wellbeing in women diagnosed with MRKH. Overall, the consensus from the prospective review of the 56 women with MRKH
was that 89 % had been sexually active and 70 % were
currently sexually active (n=39). However, the sampled
MRKH cohort from the UK had significantly decreased mental health scores, higher mean anxiety levels, and decreased
mean sexual function on all subscales of the FSFI tool (FSFI=
23.4 vs 30.5, p<0.001) compared with a standardized population of women [43]. Within the population of women with
AIS, Minto et al. surveyed 66 women using the GRISS
questionnaire [25]. This population showed similar findings
to the MRKH patients in terms of sexual function, particularly
in having difficulties in the areas of sexual frequency, vaginal
penetration, and communication [25]. Both articles make
reference to vaginal length being inversely associated with
increased sexual dissatisfaction.
However, the question that remains to be answered is what
vaginal length is necessary for sexual satisfaction? In a group
of 50 premenopausal women undergoing various gynecological procedures, the mean vaginal length (examined under
anesthesia) was 9.6 cm, with a large range of values independent of age, parity, ethnicity or sexual activity [44]. In a larger
study, looking at 505 women with prolapse, the average
vagina length was 9.1 1.2 cm in sexually active women
versus 8.91.3 cm in non-sexually active women, the statistical difference being explained by age in the multivariate
analysis [45]. Masters and Johnson (1966) found that the
length of the vagina changed depending on the state of arousal
from 78 cm when unstimulated to 1112 cm during the
sexually aroused state [46].
The simple approach of asking the woman or her partner
about participation in sexual activity and whether this is
satisfactory does yield some useful information. Some techniques used result in a relatively low rate of sexual activity,
suggesting that there might be either significant problems with
the technique, or potentially more importantly, the psychological aspect of the care might not be being fully undertaken and
nourished. It is hard to be certain, but the studies on skin
grafting do not mention the role or place of the psychologist
or counselor with this approach, suggesting that the recognition of the psychological aspects or the comfort in discussing
sexual activity might be omitted from care. This is illustrated
by Klingeles survey of patients who underwent a McIndoe
procedure, where 28 % of those who responded were unsure

of the reason why they did not develop a vagina at the time of
surgical correction [47]. On the other hand, the 100 % sexual
activity reported with the Creatsas technique also raises concerns of possible bias in patient selection, as 100 % of women
in the general population are not sexually active for a range of
different reasons [48].
Callens et al. were the first to systematically look at sexual
quality of life after both surgical vaginoplasty and dilatation,
and then compare the intervention group with a control Dutch
population. Twenty-six percent of the cohort (n=35) had a
vaginal length less than 6.6 cm [49], irrespective of the treatment they had received, which is more than two standard
deviations below the published mean value for neo-vaginal
length taken using the transsexual population [50]. While the
mean vaginal lengths of both groups were not statistically
significantly different, there was a trend toward greater reported body and genital dissatisfaction in the women who
underwent surgery, according to both a semi-structured interview and the female sexual distress scale, revised (FSDS-R).
The surgical intervention group also had a lower FSFI score
(23.9) compared with the dilation treatment group (24.6),
although this difference was not statistically significant.
However, there was a statistically significant increase in lubrication problems in the post-surgery cohort (11 out of 15
underwent the McIndoe technique [p=0.025]). Nevertheless,
the FSFI score did further increase in the combined subgroup
of patients who were sexually active (27.6). While the numbers are small, the study does suggest that with surgical
methods, there might be a trend toward more problems with
lubrication postoperatively, with no difference in vaginal
length or FSFI scores.
The impact of the diagnosis of MRKH and AIS, and the
subsequent impact on quality of life, self-esteem, body image,
as well as associated infertility, will all have a bearing on the
capacity to have positive interpersonal relationships and thus
enjoyment of sexual encounters. A prospective survey of
infertile couples (both newly diagnosed and those undergoing
fertility treatment) compared with a cohort of fertile cohabiting couples showed that female infertility patients scored
significantly lower on the FSFI subscales of arousal, orgasm,
sexual satisfaction, and lubrication compared with the control
group of fertile patients [18]. Fertility, relationship status, and
the ability to participate in penetrative sexual activity all
complicate the assessment of sexual satisfaction. Only 37 %
of postoperative colonovaginoplasty participants from Italy
(n=6) and Bangladesh (n=34) reported being married in a
survey of psychosexual satisfaction conducted 6 years posttreatment, 40 % being sexually active overall [51]. Distress
associated with known infertility in MRKH/AIS patients was
noted in 26 out of 44 women in the survey by Mbus and
Kreienberg, with 5 women attributing their diagnosis of
MRKH to the dissolution of their relationships [52]. The
inability to carry a pregnancy has been reported to be the most

Int Urogynecol J

distressing aspect of the diagnosis of vaginal agenesis in 79 %


of women from an Australian survey [24]. This Australian
survey consolidates the idea that infertility can contribute to
decreased sexual function in vaginal agenesis patients.
Of the studies that report sexual satisfaction, only 11 studies assessed these additional important contributing factors
[24, 25, 43, 49, 5157]. Kimberley et al. reported that time
since diagnosis correlated more positively with both quality of
life and sexual satisfaction, with 12 out of 13 women reporting
sexual satisfaction on the GRISS assessment more than 5 years
since diagnosis [24]. Expanding upon the work of Mbus and
Kreienberg, 50 % of sexually active women complained of
dyspareunia initially; however, 85 % were sexually satisfied
when questioned at a later (not defined) date [52]. Overall,
QoL scores (using the World Health Organization Quality of
LifeBREF (WHOQoL BREF) [58]) and sexual satisfaction
(as assessed using the GRISS) were strongly correlated (p<
0.001) in a survey of 20 MRKH post-treatment patients (i.e.,
dilation [n=16], Sheares [n=3], McIndoe [n=1]) [24].
Finally, the comparison of the FSFI scores of women who
have used dilators reveals lower overall scores. In the initial
pilot work by Liao, the possibility that the use of dilators
might contribute to negative body image is raised, with barriers to dilation being privacy, time, discomfort, and the dilators themselves not being female-friendly [59]. This negative
effect is not reported in the women who have undergone
operative procedures and require the use of a vaginal mold
for 3 to 6 months postoperativelyalthough this may simply
reflect the fact that this has not been explored. Some of the
difference may reflect studies carried out in women from
different countries where cultural factors may influence their
outcome. This is illustrated by a long-term follow-up review
in Sweden with 4 of the 8 patients contacted refusing participation, as their partners were unaware of their diagnosis and
previous McIndoe procedure [60].

Conclusion
In line with the more objective systematic review of the
operative outcomes in the vaginal agenesis literature [5] in
the setting of MRKH and AIS, this review of sexual satisfaction and functional outcomes of the management of vaginal
agenesis has failed to identify the best practice for a treatment
recommendation. Nevertheless important points to highlight
include the need for decision-making to be part of a wellestablished multidisciplinary team approach with adequate
psychosocial support for the women. Support needs to be
available from the time of the initial diagnosis to posttreatment, and continued follow-up if necessary is required
to assist with the multifaceted emotions these young women
experience which may change over time. These young women
require long-term support as they make the transition out of a

pediatric and adolescent gynecology setting into the adult


gynecology domain.

Conflicts of interest None.

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