PA: Neovagina methoden (>behandelmogelijkheid bij patiënten met PA/DSD)
Uitgangsvraag
Welke methoden van neovagina kunnen worden toegepast bij patiënten met agenesie van de vagina?
Aanbeveling
Gebruik bij voorkeur de methode Frank of de vaginale coitus methode voor het creëren van een neovagina bij patiënten met vaginale agenesie. Verwijs patiënten naar een ervaren en toegewijde hulpverlener voor de medische en psychosociale begeleiding.
Bespreek met patiënte dat er meerdere methodes zijn voor het creëren van een vagina bij vaginale agenesie.
Verwijs patiënten bij wie de conservatieve methode niet succesvol of niet mogelijk is naar een centrum met expertise in chirurgische methodes voor neovagina.
Overwegingen
Voor- en nadelen van de interventie en de kwaliteit van het bewijs
Voor de cruciale uitkomstmaat seksueel functioneren wordt geen klinisch relevant verschil gezien tussen de chirurgische interventie en de non-invasieve methodes (methode Frank en vaginale coitus). De algemene bewijskracht van de evidentie is dan ook zeer laag.
De vaginale lengte die bereikt wordt is mogelijk langer bij chirurgische interventie door middel van vaginale coitus dan bij methode Frank. Gezien de gelijke uitkomsten voor seksueel functioneren en het kleine verschil in vaginale lengte in de studies die een significant verschil rapporteren (maximaal 1,7 cm) wordt dit verschil in lengte niet als klinisch relevant geacht.
Voor de uitkomstmaat vaginale wijdte is alleen zeer lage graad evidence aanwezig. In de literatuur is er geen klinisch relevant verschil gevonden tussen de verschillende methodes.
Het complicatierisico is mogelijk hoger bij chirurgische interventies vergeleken met de methode Frank. Beschreven incidentie complicaties bij de chirurgische interventies verschilt, de complicaties die beschreven worden zijn ernstig.
Waarden en voorkeuren van patiënten (en evt. hun verzorgers)
Het belangrijkste doel van het creëren van een neovagina bij vaginale agenesie is het mogelijk maken van penetratieve vaginale seks. Het moment om deze behandeling te starten zal daarom altijd zijn wanneer patiënte hiertoe de wens uitspreekt. Er is medisch gezien geen noodzaak voor het creëren van een vagina als patiënte dit niet wil.
Voor patiënten kunnen verschillende uitkomstmaten belangrijk zijn. De uitkomstmaat seksueel functioneren is voor patiënten de best te beoordelen uitkomstmaat en vaak ook de belangrijkste. De afweging welke eventuele bijwerkingen van behandeling hierbij acceptabel zijn voor patiënte kan per patiënt verschillen.
De methode Frank zal de patiënte, met begeleiding door de arts, thuis uitvoeren. Intrinsieke motivatie van de patiënte is hierbij van belang, aangezien er dagelijks met pelottes gewerkt moet worden. De vaginale coitus doet patiënte samen met haar partner, die dan ook betrokken dient te worden in het proces.
Patiënten komen initieel vaak met verzoek voor operatie met de gedachte dan snel resultaat te behalen. Echter ook de operatie vergt veelal nabehandeling met pelotte therapie.
Kosten (middelenbeslag)
Er zijn geen data beschikbaar over de precieze kosteneffectiviteit. Zowel bij de conservatieve als de chirurgische behandeling is de aanschaf van pelottes nodig. Als eerst een operatie wordt uitgevoerd, is de chirurgische behandeling uiteraard duurder.
Ook zijn bij zowel de conservatieve methode als bij de chirurgische interventies meerdere poliklinische controles nodig. Alleen bij de kleine subgroep waarbij conservatieve therapie uiteindelijk niet succesvol is en alsnog chirurgische behandeling nodig is met nazorg zullen de kosten hoger zijn dan directe chirurgische therapie. Aangezien er geen goede voorspellende factoren zijn voor het niet slagen van de conservatieve therapie is de werkgroep van mening dat dit geen argument kan zijn om direct over te gaan tot chirurgische interventie.
Aanvaardbaarheid, haalbaarheid en implementatie
Conservatieve therapie is, met goede medische (en indien nodig psychische) begeleiding voor veel patiënten veilig en haalbaar. De psychische impact hiervan moet niet onderschat worden. Daarbij is begeleiding door een toegewijde en ervaren hulpverlener belangrijk. De vaginale coitus methode is uiteraard niet voor alle patiënten mogelijk.
De werkgroep is van mening dat met de beschikbare evidence er niet primair meerwaarde is van de chirurgische interventie. Waarschijnlijk zal dit slechts voor een klein aantal patiënten nodig zal zijn. Om voldoende kwaliteit te waarborgen van de interventie is het belangrijk dat deze zorg gecentreerd wordt. De zorg valt onder verzekerde zorg.
Rationale van de aanbeveling: weging van argumenten voor en tegen de interventies
Alleen op de cruciale uitkomstmaat complicaties wordt een mogelijk verschil beschreven, meest gunstig voor intercourse methode, gevolgd door methode Frank. Voor de uitkomstmaat seksueel functioneren is geen verschil beschreven. Voor de uitkomstmaat vaginale lengte is er een mogelijk verschil ten gunste van de chirurgische behandeling, dit wordt echter niet als klinisch relevant geacht.
Gezien het grote verschil in invasiviteit, kosten (hier niet apart onderzocht), het complicatierisico en nadelige effecten is de werkgroep van mening dat de chirurgische interventie niet geschikt is als eerstelijns therapie. Ook dient mee te worden gewogen dat postoperatief intensieve nazorg (met pelotte therapie) nodig is.
Onderbouwing
Achtergrond
Patiënten met agenesie van de vagina, meestal adolescenten, worden gecounseld over de conservatieve methode voor creëren van een neovagina. Daarnaast wordt besproken dat er verschillende methodes zijn om operatief een neovagina te creëren. Patiënten en soms ook hun ouders melden zich vaak initieel met vraag voor een operatieve ingreep. Er is behoefte aan een overzicht met voor- en nadelen, voor goede counseling; bovendien, is er een voorkeur van behandeling?
Conclusies / Summary of Findings
All GRADE scores started at a low GRADE because all included studies were non-randomized, observational, comparative studies.
Comparison 1) Surgery versus Frank dilation
1. Sexual functioning
The level of evidence regarding the outcome measure sexual functioning was downgraded by one level because of conflicting results (inconsistency).
|
Very low GRADE |
The evidence is very uncertain about the effect of surgical intervention on sexual functioning when compared with Frank dilation in patients with vaginal agenesis.
Callens (2012), Carrard (2012), Cheikhelard (2018), Herlin (2018), Kang (2020), Morcel (2013), and Willemsen (2015) |
2. Anatomical outcome
2.1 Vaginal length
The level of evidence regarding the outcome measure vaginal length was not further downgraded.
|
Low GRADE |
Surgical intervention may increase vaginal length when compared with Frank dilation in patients with vaginal agenesis.
Callens (2012), Cheikhelard (2018), Herlin (2018), Kang (2020), and Willemsen (2015) |
2.2 Vaginal width
The level of evidence regarding the outcome measure vaginal width was downgraded by one level because of number of included participants (n=146) (imprecision).
|
Very low GRADE |
The evidence is very uncertain about the effect of surgical intervention on vaginal width when compared with Frank dilation in patients with vaginal agenesis.
Cheikhelard (2018) |
3. Complications
The level of evidence regarding the outcome measure complications was not further downgraded.
|
Low GRADE |
Surgical intervention may increase the risk of complications compared with Frank dilation in patients with vaginal agenesis.
Callens (2012), Carrard (2012), Herlin (2018), and Willemsen (2015) |
4. Undesirable effects
The level of evidence regarding the outcome measure undesirable effects was downgraded by one level because of number of included participants (n=59) (imprecision).
|
Very low GRADE |
The evidence is very uncertain about the effect of surgical intervention on undesirable effects when compared with Frank dilation in patients with vaginal agenesis.
Carrard (2012) |
Comparison 2) Surgery versus intercourse dilation
1. Sexual functioning
The level of evidence regarding the outcome measure sexual functioning was downgraded by one level because of conflicting results (inconsistency).
|
Very low GRADE |
The evidence is very uncertain about the effect of surgical intervention on sexual functioning when compared with intercourse dilation in patients with vaginal agenesis.
Callens (2012) and Cheikhelard (2018) |
2. Anatomical outcome
2.1 Vaginal length
The level of evidence regarding the outcome measure complications was not further downgraded.
|
Low GRADE |
Surgical intervention may not result in a greater increase of vaginal length when compared with intercourse dilation in patients with vaginal agenesis.
Callens (2012), Cheikhelard (2018), Herlin (2018), Kang (2020), and Willemsen (2015) |
2.2 Vaginal width
The level of evidence regarding the outcome measure vaginal width was downgraded by one level because of number of included participants (n=146) (imprecision).
|
Very low GRADE |
The evidence is very uncertain about the effect of surgical intervention on vaginal width when compared with intercourse dilation in patients with vaginal agenesis.
Cheikhelard (2018) |
3. Complications
The level of evidence regarding the outcome measure complications was not further downgraded.
|
Low GRADE |
Surgical intervention may increase the risk of complications compared with intercourse dilation in patients with vaginal agenesis.
Callens (2012), Carrard (2012), Herlin (2018), and Willemsen (2015) |
4. Undesirable effects
The level of evidence regarding the outcome measure vaginal width was downgraded by one level because of number of included participants (n=59) (imprecision).
|
Very low GRADE |
The evidence is very uncertain about the effect of surgical intervention on vaginal width when compared with intercourse dilation in patients with vaginal agenesis.
Carrard (2012) |
Comparison 3) Frank dilation versus intercourse dilation
1. Sexual functioning
The level of evidence regarding the outcome measure sexual functioning was downgraded by one level because of conflicting results (inconsistency).
|
Very low GRADE |
The evidence is very uncertain about the effect of Frank dilation on sexual functioning when compared with intercourse dilation in patients with vaginal agenesis.
Callens (2012) and Cheikhelard (2018) |
2. Anatomical outcome
2.1 Vaginal length
The level of evidence regarding the outcome measure complications was not further downgraded.
|
Low GRADE |
Intercourse dilation may increase vaginal length when compared with Frank dilation in patients with vaginal agenesis.
Callens (2012), Cheikhelard (2018), Herlin (2018), Kang (2020), and Willemsen (2015) |
2.2 Vaginal width
The level of evidence regarding the outcome measure vaginal width was downgraded by one level because of number of included participants (n=146) (imprecision).
|
Very low GRADE |
The evidence is very uncertain about the effect of Frank dilation on vaginal width when compared with intercourse dilation in patients with vaginal agenesis.
Cheikhelard (2018) |
3. Complications
The level of evidence regarding the outcome measure complications was downgraded by one level because of number of included participants (imprecision).
|
Very low GRADE |
The evidence is very uncertain about the effect of Frank dilation on complications when compared with intercourse dilation in patients with vaginal agenesis.
Callens (2012) and Cheikhelard (2018) |
4. Undesirable effects
No evidence was found regarding the effect of Frank dilation on outcome undesirable effects when compared with intercourse dilation in patients with vaginal agenesis.
Samenvatting literatuur
Description of studies
Callens (2012) performed a prospective cohort study to compare surgery (total n=15), including Baldwin vaginoplasty (n=2), McIndoe vaginoplasty (n=11), and Vecchietti vaginoplasty (n=2) to Frank dilation (n=8). Cross-sectional data was gathered from women with no treatment (n=3) and intercourse dilation (n=9). Women with MRKH syndrome or CAIS were included, at least 2 years after vaginal agenesis management. Women were excluded if they were less than 18 years old or more than 60 years old, if they had been diagnosed less than 6 months before inclusion, or if they had gonadal dysgenesis. Median (range) age was: 29 (20–42) in the surgery group, 24 (18–36) in the Frank dilation group, 28 (20–48) in the intercourse dilation group, and 19 (18–19) in the group with no treatment. Mean (range) years of follow up was 6 (2–23) in the surgery group, and 5 (0.5–17) in the Frank dilation group. Data from last follow-up were analyzed. Outcome measures were FSFI score, vaginal length (cm), and complications.
Carrard (2012) used a prospective cohort to compare Sigmoid vaginoplasty (n=48) to Frank dilation (n=11). Women with MRKH were included. Women were excluded if they had been operated less than 6 months before inclusion. Mean age at time of the study was 24 years (not specified for groups. Time (range) since surgery was 6 years (10 months-17.8 years). Follow up was not mandatory and was performed 6 months after surgery and then repeated yearly. Mean length of follow up was not reported. Data from last follow up were analyzed. Outcome measures were FSFI score, complications, and complaints/undesirable effects.
Cheikhelard (2018) performed a multicenter, comparative observational study. Sigmoid vaginoplasty (n=57), Davidov vaginoplasty (n=8), Dupuytren vaginoplasty (n=8), Vecchietti vaginoplasty (n=6), and McIndoe vaginoplasty (n=5) were all included in the surgery group (total: n=84) and were compared to dilation therapy (n=26) and intercourse dilation (n=20). Women with MRKH-syndrome of 18 years or older, and vaginal agenesis management of at least one year ago were included. Women were excluded if they had a severe chronic disease. Mean age (SD) at inclusion was 27.1 (5.2) for the surgery group, 27.8 (6.6) for the intercourse group, and significantly lower in the dilation group 23.8 (4.7) (p=0.01). Mean age (SD) at management was 18.6 (2.4) for the surgery group and 17.8 (3.41) for the dilation group (not significantly different). Data was collected at one timepoint, at least one year after intervention. Outcome measures were FSFI score, vaginal length (cm), vaginal width (cm), and complications.
A retrospective cohort described by Herlin (2018) compares McIndoe vaginoplasty (n=54), Baldwin vaginoplasty (n=4), Williams vaginoplasty (n=3), and Davidov vaginoplasty (n=2) with Frank dilation (n=60), intercourse (d’Alberton) (n=20) and no treatment (n=29). Women with MRKH syndrome were included. Women were excluded if they had any other diagnosis than congenital absence of uterus, congenital absence of cervix, atresia of uterus hypoplasia of uterus, or congenital absence of vagina. Median age (IQR) at start of treatment was 19.7 (17.8-21.1) for McIndoe vaginoplasty, 17.9 (17.2–20.1) for Frank dilation, and 18.1 (17.0–20.6) for intercourse dilation. Other intervention groups were not analyzed, due to the small number of participants. Median years (IQR) of follow up was 5.6 (0.9–10.3) in the McIndoe group, 2.1 (0.9–3.9) in the Frank dilation group, and 3.9 (2.4–4.5) in the intercourse group. Outcomes were measured at every follow-up appointment (not mandatory), and data from last follow-up was analyzed. Outcome measures were vaginal depth (cm) and complications.
Kang (2020) performed a cross-sectional study comparing surgery (total: n=45) including Davidov vaginoplasty (n=17) and vaginoplasty with a biomaterial graft (n=28), to Frank dilation (n=88). Women with MRKH syndrome were included, if they had been treated to create a neovagina before, and if they were between 18 and 40 years old. Women were excluded if they had vaginal atresia caused by other factors, such as complete androgen insensitivity syndrome or congenital cervical atresia, if they had any severe chronic medical disease. Mean (SD) age at inclusion was 26.9 (4.56) in the surgery group, and 26.4 (4.40) in the dilation group (p=0.512). Data were collected at one timepoint after dilation or surgery (no specific timepoint was used). Outcome measures were FSFI score and vaginal length (cm).
Morcel (2013) performed a cross-sectional study to compare surgery (total n=20) including Sigmoid vaginoplasty (n=12) and Davydov vaginoplasty (n=8) to Frank dilation (n=20). Women with MRKH syndrome were included. Mean (SD) age at inclusion was: 26.2 (12.6) in Frank dilation group, and 25.0 (8.9) in the surgery group (p=0.7). Mean (SD) age at procedure was: 18.9 (5.1) in the Frank dilation group and 18.6 (2.5) in the surgery group (p=0.8). There was no follow up. Data were collected at one timepoint at least 6 months after vaginal agenesis management. Outcome measure was FSFI score.
A retrospective cohort performed by Willemsen (2015) compared Davydov vaginoplasty (n=68, including n=34 after unsuccessful Frank dilation) with Frank dilation (n=75, including n=5 combined with intercourse) and intercourse only (n=17). Women with MRKH syndrome were included. Mean (range) age at surgery was 23 (16-45). Mean age at inclusion was not mentioned. Length of follow up was defined as last clinical follow up or death and was not mandatory. Mean (range) length of follow up was: 35 (1-128) in the intercourse only group, 45 (1-320) in the Frank dilation group (combined with intercourse: 40 (1-147)), 135 (0-444) in the Davydov group (with Frank dilation: 95 (1-300); with other surgery: 138 (8-361). Data from last follow-up were analyzed. Outcome measures were sexual satisfaction, vaginal depth (cm), and complications.
Results
Seven studies were included in analysis. Pooling of data was not possible since no risk ratios or odds ratios were reported. Three comparisons were made: 1) Surgery versus Frank dilation, 2) Surgery versus intercourse dilation, and 3) Frank dilation versus intercourse dilation.
Comparison 1) Surgery versus Frank dilation
Seven studies reported on surgery versus Frank dilation. In total, 326 women were included in the surgery group, including McIndoe, Baldwin, Williams, Davidov, Vecchieti, Sigmoid, and Dupuytren vaginoplasty, versus 288 women included in the Frank dilation group. Six of seven studies report on sexual functioning of the neovagina after surgery compared to Frank dilation, of which 5 report FSFI scores. Five studies reported on vaginal length in cm, and one study reported on vaginal width. Four studies reported on complications. One study reported on undesirable effects.
Critical outcome measures
1. Sexual functioning
Callens (2012) reported a mean ± SD FSFI score of 21.9 ± 9.5 for the surgery group (n=8, including Baldwin, McIndoe and Vecchietti vaginoplasty) versus 23.8 ± 10.5 for the Frank dilation group (n=6), favouring Frank dilation (not significant, p-value was not mentioned).
Carrard (2012) reported a mean ± SD FSFI score of 28.00 ± 3.1 for the surgery group (n=48, Sigmoid vaginoplasty) versus and 30.25 ± 6.2 for the Frank dilation group (n=11) (p=0.13), favouring Frank dilation (not significant).
Cheikhelard (2018) reported a median (range) FSFI score of 26 (2.8 to 34.8) in the surgery group (n=84, including Sigmoid, Davidov, Dupuytren, Vecchietti, and McIndoe vaginoplasty) versus 24.7 (2.6 to 34.4) in the Frank dilation group (n=26) (p=0.85), favouring surgery (not significant).
Kang (2020) reported a mean ± SD FSFI score of 23.79 ± 3.57 in the surgery group (n=45, including Davidov and biomaterial graft vaginoplasty) versus 24.49 ± 4.51 in the Frank dilation group (n=88) (p=0.507), favouring Frank dilation (not significant).
Morcel (2013) reported a mean ± SD FSFI score of 25.3 ± 8.0 in the surgery group (n=20, Sigmoid vaginoplasty) versus 25.3 ± 7.5 for the Frank dilation group (n=20) (p=1.0), which was equal.
Willemsen (2015) reported that 11% (n=7) of surgery patients were not sexually satisfied, due to deep dyspareunia. No FSFI score was reported.
Herlin (2018) did not report on sexual functioning.
Table 1: FSFI scores of included studies
|
Study |
Surgery mean ± SD cm |
Number of participants |
Frank dilation mean ± SD cm |
Number of participants |
p-value |
|
Callens (2012) |
21.9 ± 9.5 |
8 Baldwin, McIndoe and Vecchietti vaginoplasty |
23.8 ± 10.5 |
6 |
NS |
|
Carrard (2012) |
28.00 ± 3.1 |
48 Sigmoid vaginoplasty |
30.25 ± 6.2 |
11 |
0.13 |
|
Cheikhelard (2018) |
26 (2.8 to 34.8) median (IQR) |
84 Sigmoid, Davidov, Dupuytren, Vecchietti, and McIndoe vaginoplasty |
24.7 (2.6 to 34.4) |
26 |
0.85 |
|
Kang (2020) |
23.79 ± 3.57 |
45 Davidov and biomaterial graft vaginoplasty |
24.49 ± 4.51 |
88 |
0.507 |
|
Morcel (2013) |
25.3 ± 8.0 |
20 Sigmoid vaginoplasty |
25.3 ± 7.5 |
20 |
1.0 |
2. Anatomical outcomes:
2.1 Vaginal length
Callens (2012) reported a mean ± SD vaginal length of 9.1 ± 2.7 cm after surgery group (n=8, including Baldwin, McIndoe and Vecchietti vaginoplasty), versus 7.3 ± 1.3 cm after Frank dilation (n=6) (p=0.21), favouring surgery (not significant).
Cheikhelard (2018) reported a median (range) vaginal length of 11.3 (11-12) cm following Vecchietti vaginoplasty, 11 (6-15) cm following Sigmoid vaginoplasty, 12 (11-13) cm following McIndoe vaginoplasty, 8.5 (7-11) cm following Davidov vaginoplasty, 9.3 (8-12) cm following Dupuytren vaginoplasty. The median (range) vaginal length for surgery overall (n=84) was 11 (6-15) cm versus 9.3 (5.5-12) cm after Frank dilation (n=26) (p=0.039), favouring surgery (significant).
Herlin (2018) reported a mean (95% CI) vaginal length of 7.4 (6.8–8.1) cm after surgery (n=54, McIndoe vaginoplasty), versus 7.3 (6.7–7.9) cm after Frank dilation (n=60) (p=1.0), which was equal.
Kang (2020) reported a mean ± SD vaginal length of 8.1 ± 1.59 cm after surgery (n=45, Davidov or with graft), versus 6.5 ± 2.04 cm after Frank dilation (n=88) (p=0.005), favouring surgery (significant).
Willemsen (2015) reported a mean (range) vaginal length of 8.4 (3-13) cm after surgery (n=86, Davidov vaginoplasty), 8.4 (4-13) cm when combined with Frank dilation, and 7.4 (4-11) cm when combined with other surgery versus 6.7 (1-13) cm after Frank dilation (n=75) and 7.2 (4-10) cm after Frank dilation combined with intercourse (n=5), which was equal.
Carrard (2012) and Morcel (2013) did not report on vaginal length.
Table 2: Results of vaginal length (cm) of included studies
|
Study |
Surgery mean ± SD cm |
Number of participants |
Frank dilation Mean ± SD cm |
Number of participants |
p-value |
|
Callens (2012) |
9.1 ± 2.7 |
8 Baldwin, McIndoe and Vecchietti vaginoplasty |
7.3 ± 1.3 |
6 |
0.21 |
|
Cheikhelard (2018) |
11 (6-15) median (range) |
84 Sigmoid, Davidov, Dupuytren, Vecchietti, and McIndoe vaginoplasty |
9.3 (5.5-12) median (range) |
26 |
0.039 |
|
Herlin (2018) |
7.4 (6.8–8.1) mean (95% CI) |
54 McIndoe |
7.3 (6.7–7.9) mean (95% CI) |
60 |
1.0 |
|
Kang (2020) |
8.1 ± 1.59 |
45 Davidov and biomaterial graft vaginoplasty |
6.5 ± 2.04 |
88 |
0.005 |
|
Willemsen (2015) |
8.4 (3-13) mean (range) |
68 Davidov vaginoplasty |
6.7 (1-13) mean (range) |
75 |
Not reported |
2.2 Vaginal width
Only Cheikhelard (2018) reported a median (range) vaginal width of 3 (2-4) cm in all groups.
Callens (2012), Carrard (2012), Herlin (2018), Kang (2020), Morcel (2013), and Willemsen (2015) did not report on vaginal width.
3. Complications
Callens (2012) reported 3 (30%) complications in the surgery group (n=8, Baldwin, McIndoe, and Vecchietti). No complications were reported in the Frank dilation group. This favours Frank dilation (significance not reported).
Carrard (2012) reported 2 major complications in the surgery group (n=48, Sigmoid vaginoplasty). Two patients developed pelvic hematomas due to bleeding in the perineal cleavage. Also, 5 minor complications were observed, which were: rectal injury (n=2), left tubal abscess (n=1), and prolapse of neovagina (n=2). No comparison was made with Frank dilation.
Herlin (2018) reported 35 (65%) complications after surgery (n=54, McIndoe vaginoplasty), of which 19 (35%) were shrinkage/stenosis, 13 (24%) were granulation tissue, 11 (20%) were transplant defect, 12 (22%) were infection, 8 (15%) were bleeding, and 2 (94%) were psychiatric trauma. Following Frank dilation (n=60), 21 (35%) complications were observed, of which 15 (25%) were low compliance, 5 (8%) were urethral dilation, and 2 (3%) were vaginal prolapse. This favours Frank dilation (significance not reported).
Willemsen (2015) reported 9 events in 7 patients that underwent Davydov vaginoplasty, which were rectal perforation (n=4), liver necrosis (due to allergic reaction to narcotic gas) (n=1), adhesions between vagina and sigmoid (n=1), rectal prolapse (n=1), rectovaginal fistula (n=1), and bladder lesion (n=1). No comparison was made with Frank dilation.
Cheikhelard (2018), Kang (2020), and Morcel (2013) did not report on complications.
Important outcome measures
4. Undesirable effects
Only Carrard (2012) reported on undesirable effects. Vaginal discharge discomfort was reported in 23 patients (68%) after Sigmoid vaginoplasty (n=48), and in no patients after Frank dilation (n=11). Abdominal pain was observed in 22 patients (65%) following Sigmoid vaginoplasty, and in 1 patient (20%) following Frank dilation. Dyspareunia was observed in 24 patients (80%) after Sigmoid vaginoplasty, and in 2 patients (50%) after Frank dilation. This favours Frank dilations (significance was not reported).
Callens (2012), Cheikhelard (2018), Herlin (2018), Kang (2020), Morcel (2013), and Willemsen (2015) did not report on undesirable effects.
Comparison 2) Surgery versus intercourse dilation
Four studies reported on surgery versus intercourse dilation. In total, 213 women were included in the surgery group, including McIndoe, Baldwin, Williams, Davidov, Vecchieti, Sigmoid, and Dupuytren vaginoplasty, versus 66 women included in the intercourse dilation group. Two of four studies report on sexual functioning of the neovagina after surgery compared to intercourse dilation, of which all report FSFI scores. All studies reported on vaginal length in cm, and one study reported on vaginal width. All studies reported on complications. No study reported on comparison of undesirable effects between Frank dilation and intercourse dilation.
Critical outcome measures
1. Sexual functioning
Callens (2012) reported a mean ± SD FSFI score of 21.9 ± 9.5 for the surgery group (n=8, including Baldwin, McIndoe and Vecchietti vaginoplasty) versus 19.2 ± 4.9 for the intercourse group (n=2), favouring the surgery group (not significant, p-value was not mentioned).
Cheikhelard (2018) reported a median (range) FSFI score of 26 (2.8 to 34.8) in the surgery group (n=84, including Sigmoid, Davidov, Dupuytren, Vecchietti, and McIndoe vaginoplasty) versus 30.2 (7.8 to 34.8) in the intercourse group (n=20) (p=0.048), favouring the intercourse group (significant).
Carrard (2012), Herlin (2018), Kang (2020), Morcel (2013), and Willemsen (2015) did not report on sexual functioning of surgery versus intercourse dilation.
Table 3: results of sexual functioning of included studies
|
Study |
Surgery mean ± SD cm |
Number of participants |
Intercourse dilation mean ± SD cm |
Number of participants
|
p-value |
|
Callens (2012) |
21.9 ± 9.5 |
8 Baldwin, McIndoe and Vecchietti vaginoplasty |
19.2 ± 4.9 |
2 |
NS |
|
Cheikhelard (2018) |
26 (2.8 to 34.8) median (IQR) |
84 Sigmoid, Davidov, Dupuytren, Vecchietti, and McIndoe vaginoplasty |
30.2 (7.8 to 34.8) median (IQR) |
20 |
0.048 |
2. Anatomical outcomes:
2.1 Vaginal length
Callens (2012) reported a mean ± SD vaginal length of 9.1 ± 2.7 cm after surgery (n=8, Baldwin, McIndoe and Vecchietti vaginoplasty), versus 8.9 ± 2.6 cm after intercourse dilation (n=9), which was equal (not significant, p-value was not mentioned).
Cheikhelard (2018) reported a median (range) vaginal length of 11.3 (11-12) cm following Vecchietti vaginoplasty, 11 (6-15) cm following Sigmoid vaginoplasty, 12 (11-13) cm following McIndoe vaginoplasty, 8.5 (7-11) cm following Davidov vaginoplasty, 9.3 (8-12) cm following Dupuytren vaginoplasty. The median (range) vaginal length for surgery overall (n=84) was 11 (6-15) cm versus 11 (6-12.5) cm after intercourse dilation (n=20) (p=1.00), which was equal.
Herlin (2018) reported a mean (95% CI) vaginal length of 7.4 (6.8–8.1) cm after surgery (n=54, McIndoe), versus 8.7 (7.9–9.5) cm after intercourse dilation (n=20) (p=0.07), favouring the intercourse group (not significant).
Willemsen (2015) reported a mean (range) vaginal length of 8.4 (3-13) cm after surgery (n=68, Davidov vaginoplasty), when combined with Frank dilation 8.4 (4-13) cm, and when combined with other surgery 7.4 (4-11) cm versus 7.7 (4-10) cm after intercourse dilation (n=17), which was equal (significance was not reported).
Carrard (2012), Kang (2020), and Morcel (2013) did not report on vaginal length.
Table 4: Outcomes of vaginal length (cm) of included studies
|
Study |
Surgery mean ± SD cm |
Number of participants |
Intercourse dilation mean ± SD cm |
Number of participants |
p-value |
|
Callens (2012) |
9.1 ± 2.7 |
8 Baldwin, McIndoe and Vecchietti vaginoplasty |
8.9 ± 2.6 |
9 |
0.07 |
|
Cheikhelard (2018) |
11 (6-15) median (range) |
84 Sigmoid, Davidov, Dupuytren, Vecchietti, and McIndoe vaginoplasty |
11 (6-12.5) median (range) |
20 |
1.00 |
|
Herlin (2018) |
7.4 (6.8–8.1) mean (95% CI) |
54 McIndoe vaginoplasty |
8.7 (7.9–9.5) mean (95% CI) |
20 |
1 |
|
Willemsen (2015) |
8.4 (3-13) mean (range) |
68 Davidov |
7.7 (4-10) mean (range) |
17 |
Not reported |
2.2 Vaginal width
Only Cheikhelard (2018) reported a median (range) vaginal width of 3 (2-4) cm in all groups.
Callens (2012), Carrard (2012), Herlin (2018), Kang (2020), Morcel (2013) and Willemsen (2015) did not report on vaginal width.
3. Complications
Callens (2012) reported 3 (30%) complications in the surgery group (n=8, Baldwin, McIndoe, and Vecchietti). No complications were reported in the intercourse dilation group.
Carrard (2012) reported 2 major complications in the surgery group (n=48, Sigmoid vaginoplasty). Two patients developed pelvic hematomas due to bleeding in the perineal cleavage. Also, 5 minor complications were observed, which were: rectal injury (n=2), left tubal abscess (n=1), and prolapse of neovagina (n=2). Significance was not reported.
Herlin (2018) reported 35 (65%) complications after surgery (n=54, McIndoe vaginoplasty), of which 19 (35%) were shrinkage/stenosis, 13 (24%) were granulation tissue, 11 (20%) were transplant defect, 12 (22%) were infection, 8 (15%) were bleeding, and 2 (94%) were psychiatric trauma. One (5%) complication was observed after intercourse dilation (n=20), which was low compliance. The p-value of the Pearson Chi-square test was p<0.00001.
Willemsen (2015) reported 9 events in 7 patients that underwent Davydov vaginoplasty, which were rectal perforation (n=4), liver necrosis (due to allergic reaction to narcotic gas) (n=1), adhesions between vagina and sigmoid (n=1), rectal prolapse (n=1), rectovaginal fistula (n=1), and bladder lesion (n=1). Significance is not reported.
Cheikhelard (2018), Kang (2020), and Morcel (2013) did not report on complications.
Important outcome measures
4. Undesirable effects
No study reported on comparison of undesirable effects between Frank dilation and intercourse dilation.
Comparison 3) Frank dilation versus intercourse dilation
Four studies reported on Frank dilation versus intercourse dilation. In total, 169 women were included in the Frank dilation group, versus 66 women included in the intercourse dilation group. Two of four studies report on sexual functioning of the neovagina after surgery compared to intercourse dilation, of which all report FSFI scores. All studies reported on vaginal length in cm, and one study reported on vaginal width. Two studies reported on complications. No study reported on comparison of undesirable effects between Frank dilation and intercourse dilation.
Critical outcome measures
1. Sexual functioning
Callens (2012) reported a mean ± SD FSFI score of 23.8 ± 10.5 for the Frank dilation group (n=6) versus 19.2 ± 4.9 for the intercourse group (n=2), favouring the intercourse group (not significant, p-value is not mentioned).
Cheikhelard (2018) reported a median (range) FSFI score of 24.7 (2.6 to 34.4) in the Frank dilation group (n=26), versus 30.2 (7.8 to 34.8) in the intercourse group (n=20), favouring Frank dilation (p=0.048).
Carrard (2012), Herlin (2018), Kang (2020), Morcel (2013), and Willemsen (2015) did not report on sexual functioning of Frank dilation versus intercourse dilation.
Table 5: results of sexual functioning of included studies
|
Study |
Frank dilation mean ± SD cm |
Number of participants |
Intercourse dilation mean ± SD cm |
Number of participants
|
p-value |
|
Callens (2012) |
23.8 ± 10.5 |
6 |
19.2 ± 4.9 |
2 |
NS |
|
Cheikhelard (2018) |
24.7 (2.6 to 34.4) median (range) |
26 |
30.2 (7.8 to 34.8) median (IQR) |
20 |
0.048 |
2. Anatomical outcomes:
2.1 Vaginal length
Callens (2012) reported a mean ± SD vaginal length of 7.3 ± 1.3 cm after Frank dilation (n=6), versus 8.9 ± 2.6 cm after intercourse dilation (n=9), favouring intercourse dilation (p=0.21).
Cheikhelard (2018) reported a median (range) vaginal length of 9.3 (5.5-12) cm after Frank dilation (n=26) versus 11 (6-12.5) cm after intercourse dilation (n=20), favouring intercourse dilation (p=0.039).
Herlin (2018) reported a mean (95% CI) vaginal length of 7.3 (6.7–7.9) cm after Frank dilation (n=60), versus 8.7 (7.9–9.5) cm after intercourse dilation (n=20), favouring intercourse dilation (p=0.03).
Willemsen (2015) reported a mean (range) vaginal length of 6.7 (1-13) cm after Frank dilation (n=75) and 7.2 (4-10) cm after Frank dilation combined with intercourse versus 7.7 (4-10) cm after intercourse dilation (n=17), favouring intercourse dilation (significance was not reported).
Carrard (2012), Kang (2020), and Morcel (2013) did not report on vaginal length after intercourse dilation.
Table 6: Outcomes of vaginal length (cm) of included studies
|
Study |
Frank dilation mean ± SD cm |
Number of participants |
Intercourse dilation mean ± SD cm |
Number of participants |
p-value |
|
Callens (2012) |
7.3 ± 1.3 |
6 |
8.9 ± 2.6 |
9 |
0.07 |
|
Cheikhelard (2018) |
9.3 (5.5-12) median (range) |
26 |
11 (6-12.5) median (range) |
20 |
1.00 |
|
Herlin (2018) |
7.3 (6.7–7.9) mean (95% CI) |
60 |
8.7 (7.9–9.5) mean (95% CI) |
20 |
1 |
|
Willemsen (2015) |
6.7 (1-13) mean (range) |
75 |
7.7 (4-10) mean (range) |
17 |
Not reported |
2.2 Vaginal width
Only Cheikhelard (2018) reported a median (range) vaginal width of 3 (2-4) cm in all groups.
Herlin (2018), Kang (2020), Willemsen (2015), Callens (2012), Carrard (2012), and Morcel (2013) did not report on vaginal width.
3. Complications
Callens (2012) reported no complications in the Frank dilation group or intercourse dilation group.
Cheikhelard (2018) reported 21 (35%) complications following Frank dilation (n=26), of which 15 (25%) were low compliance, 5 (8%) were urethral dilation, and 2 (3%) were vaginal prolapse. One (5%) complication was observed after intercourse dilation (n=20), which was low compliance. The p-value of the Pearson Chi-square test was p<0.00001.
Carrard (2012), Herlin (2018), Kang (2020), Morcel (2013), and Willemsen (2015) did not report on complications, between Frank dilation and intercourse dilation.
Important outcome measures
4. Undesirable effects
No study reported on comparison of undesirable effects between Frank dilation and intercourse dilation.
Zoeken en selecteren
A systematic review of the literature was performed to answer the following question: What is the effectivity of surgical methods compared to non-surgical methods for creation of a neovagina in females with vaginal agenesis with a desire for a neovagina?
P: Females with vaginal agenesis, with a wish for a neovagina.
I: Surgical method of creating a neovagina.
C: Non-surgical method of creating a neovagina (e.g., Frank’s dilation or intercourse).
O: Functionality: sexual functioning, complications, complaints/adverse events (e.g., blood loss, mucus).
Relevant outcome measures
The guideline development group considered sexual functioning, vaginal length, vaginal width and complications as critical outcome measures for decision making; and complaints/adverse events as important outcome measures for decision making.
A priori, the working group did not define the outcome measures listed above but used the definitions used in the studies.
The working group did not define clinically (patient) important differences.
Search and select (Methods)
The databases Medline (via OVID) and Embase (via Embase.com) were searched with relevant search terms until July 21st, 2021. The detailed search strategy is depicted under the tab Methods. The systematic literature search resulted in 166 hits. Studies were selected based on the following criteria: systematic reviews, randomized controlled trials, or comparative observational studies on surgical vs non-surgical methods to create a neovagina in females with vaginal agenesis. Thirty-seven studies were initially selected based on title and abstract screening. After reading the full text, 30 studies were excluded (see Table of excluded studies) and seven studies were included.
Results
Seven studies were included in the analysis of the literature. Important study characteristics and results are summarized in the evidence tables. The assessment of the risk of bias is summarized in the risk of bias tables.
Referenties
- Callens N, De Cuypere G, Wolffenbuttel KP, Beerendonk CC, van der Zwan YG, van den Berg M, Monstrey S, Van Kuyk ME, De Sutter P; Belgian-Dutch Study Group on DSD, Dessens AB, Cools M. Long-term psychosexual and anatomical outcome after vaginal dilation or vaginoplasty: a comparative study. J Sex Med. 2012 Jul;9(7):1842-51.
- Carrard C, Chevret-Measson M, Lunel A, Raudrant D. Sexuality after sigmoid vaginoplasty in patients with Mayer-Rokitansky-Kster-Hauser syndrome. Fertil Steril. 2012 Mar;97(3):691-6. doi: 10.1016/j.fertnstert.2011.12.015. Epub 2012 Jan 14. PMID: 22245530.
- Cheikhelard A, Bidet M, Baptiste A, Viaud M, Fagot C, Khen-Dunlop N, Louis-Sylvestre C, Sarnacki S, Touraine P, Elie C, Aigrain Y, Polak M; French MRKH Study Group. Surgery is not superior to dilation for the management of vaginal agenesis in Mayer-Rokitansky-Kster-Hauser syndrome: a multicenter comparative observational study in 131 patients. Am J Obstet Gynecol. 2018 Sep;219(3):281.e1-281.e9.
- Herlin M, Bay Bj¿rn AM, J¿rgensen LK, Trolle B, Petersen MB. Treatment of vaginal agenesis in Mayer-Rokitansky-Kster-Hauser syndrome in Denmark: a nationwide comparative study of anatomical outcome and complications. Fertil Steril. 2018 Sep;110(4):746-753.
- Kang J, Chen N, Song S, Zhang Y, Ma C, Ma Y, Zhu L. Sexual function and quality of life after the creation of a neovagina in women with Mayer-Rokitansky-Kster-Hauser syndrome: comparison of vaginal dilation and surgical procedures. Fertil Steril. 2020 May;113(5):1024-1031.
- Morcel K, Lavou V, Jaffre F, Paniel BJ, Rouzier R. Sexual and functional results after creation of a neovagina in women with Mayer-Rokitansky-Kster-Hauser syndrome: a comparison of nonsurgical and surgical procedures. Eur J Obstet Gynecol Reprod Biol. 2013 Jul;169(2):317-20.
- Willemsen WN, Kluivers KB. Long-term results of vaginal construction with the use of Frank dilation and a peritoneal graft (Davydov procedure) in patients with Mayer-Rokitansky-Kster syndrome. Fertil Steril. 2015 Jan;103(1):220-7.e1.
Evidence tabellen
|
Study reference |
Study characteristics |
Patient characteristics |
Intervention (I) |
Comparison/control (C) |
Follow-up |
Outcome measures and effect size |
Comments |
|
Callens, 2012 |
Type of study: prospective (vaginoplasty + Frank dilation) and retrospective (no treatment) follow-up
Setting and country: University Hospital Ghent, Belgium; Erasmus Medical Center Rotterdam, the Netherlands; or Radboud University Nijmegen Medical Center, the Netherlands, February 2007 - January 2010
Funding and conflicts of interest: not reported |
Inclusion criteria: - women with MRKH syndrome, or CAIS
Exclusion criteria: - age <18 years and >60 years - recent diagnosis (<6 months) - gonadal dysgenesis (because of the heterogeneous clinical picture) - intellectual disability
N total at baseline: Intervention: n=15 Control: n=20
Important prognostic factors2: 1. Age median (range): Surgery: 29 (20–42) Frank dilation: 24 (18–36) Coital dilation: 28 (20–48) No treatment: 19 (18–19) No p-value reported
4. Stable relationship: n=21 (60%)
Groups comparable at baseline? Yes
|
Baldwin (n=2), McIndoe (n=11), Vecchietti (n=2) |
Frank dilation (n=8) Coital dilation (n=9) No treatment (n=3) |
Length of follow-up: Years: mean (range) Frank dilation: 5 (0.5–17) Surgery: 6 (2–23)
Loss-to-follow-up: Surgery N (%): 6 (40%) Frank dilation N (%): 2 (25%)
Reasons (surgery and Frank dilation; not specified): n=4 lacked the time; n=4 refused because of repeated and shameful examination of the genitalia in the past.
Coital dilation: n/a No treatment: n/a
Incomplete outcome data: Surgery: N (%): 7 (47%) Reasons (describe); not mentioned
Frank dilation: N (%): 2 (25%) Reasons (describe): not mentioned
Coital dilation: N (%): 7 (78%) Reasons (describe): not mentioned
No treatment: N (%) 2 (67%) Reasons (describe): not mentioned |
1.1 FSFI score: mean ± SD Surgery: 21.9 ± 9.5 (n=8) Frank dilation: 23.8 ± 10.5 (n=6) Coital dilation: 19.2 ± 4.9 (n=2) No treatment: 6.2 (n=1)
1.2 Anatomical outcomes Vaginal Length: cm mean ± SD Surgery: 9.1 ± 2.7 Frank dilation: 7.3 ± 1.3 Coital dilation: 8.9 ± 2.6 No treatment: 7.3 ± 3.7
Coital vs frank: p=0.21 Surgery vs coital: not significant Surgery vs frank: not significant
2. Complications: Surgery: n=3 Frank dilation: none Coital dilation: none No treatment: none
3. Undesirable effects: not reported |
Recommend dilation as first-line treatment.
“Both psychological and physical factors are predisposing for sexual difficulties. To optimize psycho- sexual comfort, the clinical management of women with vaginal hypoplasia needs to be multidisciplinary and individually tailored. With high success rates reported, vaginal dilation should remain the cornerstone of treatment.”
|
|
Carrard, 2012 |
Type of study: prospective study
Setting and country: the Hotel Dieu and Lyon Sud University Hospitals, France, August 1992 - July 2010
Funding and conflicts of interest: not reported |
Inclusion criteria: - women with MRKH syndrome
Exclusion criteria: - operated <6 months before study
N total at baseline: Intervention: n=48 Control: n=11
Important prognostic factors2: 1. Age (at time of study): mean: 24 years 2. Time since surgery: mean (range): 6 years (10 months-17.8 years) Groups comparable at baseline? Yes
|
Sigmoid vaginoplasty (n=48) |
Frank dilation (n=11) |
Length of follow-up: 6 months after surgery, then yearly Loss-to-follow-up: Surgery and Frank dilation: N=6 (10%) Reasons: not mentioned
Incomplete outcome data: Intervention: N (%): 13 (27%) Control: N (%) 6 (55%)
Reasons (for surgery and frank dilation; not specified): 6 (10%) lost to follow up, 13 (22%) did not answer questionnaire, of which one replied that the subject was too intimate to be discussed via a mailed questionnaire. |
1.1 FSFI score: mean ± SD Sigmoid: 28.00 ± 3.1 Frank dilation: 30.25 ± 6.2 P=0.13
1.2 Anatomical outcomes: not reported
2. Complications: 2 major, 5 minor
3. Undesirable effects: Vaginal discharge discomfort: N (%) Sigmoid: 23 (68%) Frank: 0 Abdominal pain Sigmoid: 22 (65%) Frank: 1 (20%) Dyspareunia Sigmoid: 24 (80%) Frank: 2 (50%) |
Recommend dilation as first-line treatment. But Sigmoid effective as well.
“Sigmoid vaginoplasty is an effective technique providing a nearly normal sexual function to patients with vaginal aplasia. Despite this, psychologic distress related to sexuality persists in most patients, demonstrating the need for a multidisciplinary support.” |
|
Cheikhelard, 2018 |
Type of study: multicenter comparative observational study
Setting and country: National Reference Center for Rare Gynecological Diseases, October 2012 -April 2015, France
Funding and conflicts of interest: Funded by a grant from the Programme Hospitalier de Recherche Clinique 2011 (Ministry of Health) and sponsored by the Assistance Publique-Hopitaux de Paris (Projet AP-HP: AOM11168-P110124). |
Inclusion criteria: - women with MRKH syndrome patients >18 y - VA management >1995 at least 1 year - French speaking
Exclusion criteria: - patients with severe chronic disease - illiterate patients
N total at baseline: Intervention: n=84 Control: n=60
Important prognostic factors2: 1. Age (at inclusion): mean ± SD surgery: 27.1 ± 5.2 intercourse: 27.8 ± 6.6 dilation: 23.8 ± 4.7 p=0.01 (not accounted for) 2. Age (at management) mean ± SD Surgery: 18.6 ± 2.4 Dilation: 17.8 ± 3.41 Not significant
Groups comparable at baseline? Yes |
Sigmoid vaginoplasty (n=57), Davidov vaginoplasty (n=8), Dupuytren vaginoplasty (n=8), Vecchietti vaginoplasty (n=6), McIndoe vaginoplasty (n=5) |
Dilation only (n=26), Intercourse (n=20) |
Length of follow-up: Not mentioned (mean no. sessions: 3)
Loss-to-follow-up: Total: n=74 (57%)
Incomplete outcome data: Intervention: N (%): 1 (1.19%) Reasons (describe): type of surgery could not be identified
Dilation: N (%): 1 (3.8%) Reasons (describe): not mentioned
Intercourse: N (%): 2 (10%) Reasons (describe): not mentioned |
1.1 FSFI score: median (range) Vecchietti: 30.2 (18.7-34.8) Sigmoid: 25.7 (2.8-34) McIndoe: 28.4 (8.4-34.8) Davidov: 23.0 (4.8-33.6) Dupuytren: 27.9 (15.3-34) Surgery overall: 26 (2.8-34.8) Dilation: 24.7 (2.6-34.4) Intercourse: 30.2 (7.8-34.8)
Surgery vs dilation: p=0.85 Surgery vs intercourse: p=0.048 Dilation vs intercourse: p=0.048
1.2 Anatomical outcomes Vaginal length: cm median (range) Vecchietti: 11.3 (11-12) Sigmoid: 11 (6-15) McIndoe: 12 (11-13) Davidov: 8.5 (7-11) Dupuytren: 9.3 (8-12) Surgery overall: 11 (6-15) Dilation: 9.3 (5.5-12) Intercourse: 11 (6-12.5)
Surgery vs dilation: p=0.039 Surgery vs intercourse: p=1.00 Dilation vs intercourse: p=0.039
Vaginal width: cm median (range) All groups: 3 (2-4)
2. Complications: Surgery: 34/84 (40%) Dilation: 7/26 (27%)
3. Undesirable effects: not reported |
‘’Surgery is not superior to therapeutic or intercourse dilation, bears complications, and should therefore be only a second-line treatment. Psychological counselling is mandatory at diagnosis and during therapeutic management. ‘’ |
|
Herlin, 2018 |
Type of study: retrospective cohort
Setting and country: Public hospitals, Denmark, January 1, 1994 - April 10, 2015
Funding and conflicts of interest: not reported |
Inclusion criteria: - women with MRKH syndrome
Exclusion criteria: - other diagnosis than: congenital absence of uterus, congenital absence of cervix, atresia of uterus hypoplasia of uterus, congenital absence of vagina.
N total at baseline: Intervention: n=59 Control: n=80 No treatment: n=29
Important prognostic factors2: 2. Age (at start of treatment) median (IQR) McIndoe: 19.7 (17.8-21.1) Frank dilation: 17.9 (17.2–20.1) Coital dilation: 18.1 (17.0–20.6) No p-value reported 3. Vaginal Depth cm (at referral): median (IQR) McIndoe: 2.0 (1.0-3.0) Frank dilation: 1.5 (1.0-2.5) Coital dilation: 3.75 (2.5-5.0) No p-value reported
Groups comparable at baseline? Yes |
McIndoe vaginoplasty (n=54), Baldwin vaginoplasty (n=4), Williams vaginoplasty (n=3), Davidov vaginoplasty (n=2) |
Frank-dilation (n=60), Coital dilation (d’Alberton) (n=20), No treatment (n=29) |
Length of follow-up: years median (IQR) McIndoe: 5.6 (0.9–10.3) Frank-dilation: 2.1 (0.9–3.9) Coital dilation: 3.9 (2.4–4.5)
Loss-to-follow-up: McIndoe: N (%): 21 (39%) Reasons (describe): not mentioned
Frank dilation: N (%): 8 (13%) Reasons (describe) not mentioned
Coital dilation: N (%): 0 Reasons (describe): n/a
Incomplete outcome data: not mentioned
|
1.1 FSFI score: not reported
1.2 Anatomical outcomes Vaginal depth: cm mean (95%CI) McIndoe: 7.4 (6.8–8.1) Frank-dilation: 7.3 (6.7–7.9) Coital dilation: 8.7 (7.9–9.5)
McIndoe vs Coital: p=0.07 McIndoe vs self-dilation: p=1.00 Coital vs self-dilation: p=0.03
2. Complication rate: n (%) McIndoe: 35 (65) Self-dilation: 21 (35) Coital dilation: 1 (5) p<0.00001
3. Undesirable effects: not reported |
Recommend dilation as first-line treatment, emphasize on coital dilation.
“Our findings support the current recommendations of dilation therapy as the first-line treatment of vaginal agenesis and emphasize the relevance of coital dilation in patients able to regularly engage in coital activity.” |
|
Kang, 2020 |
Type of study: cross-sectional study
Setting and country: Tertiary teaching hospital, Peking Union Medical College Hospital, January 2019 - June 2019
Funding and conflicts of interest: CAMS Initiative for Innovative Medicine [CAMS-2017- I2M-1–002] and National Science Foundation for Youth [81801401] |
Inclusion criteria: - women with MRKH syndrome - patients who had been treated with the goal of creating a vagina - patients who were >18 years and <40 years of age - patients who were Chinese speaking
Exclusion criteria: - patients presenting with vaginal atresia caused by other factors, such as complete androgen insensitivity syndrome or congenital cervical atresia - patients aged <18 or >40 years - surgical vaginoplasty other than peritoneal vaginoplasty or vaginoplasty with a biomaterial graft - patients who had MRKH syndrome combined with severe chronic medical disease.
N total at baseline: Surgery: n=45 Dilation: n=88
Important prognostic factors2: 1. Age (at inclusion) mean ± SD Surgery: 26.9 ± 4.56 Dilation: 26.4 ± 4.40 P=0.512 4. Sexual partner Surgery: 0: 19 (42.2%) 1: 25 (55.6%) >1: 1 (2.2%) Dilation: 0: 40 (45.5%) 1: 47 (53.4%) >1: 1 (1.1%) P=0.848
Groups comparable at baseline? Yes |
Davidov vaginoplasty (n=17) Vaginoplasty with a biomaterial graft (n=28)
|
Vaginal dilation (n=88) |
Length of follow-up: n/a
Loss-to-follow-up: n/a
Incomplete outcome data: Surgery: N (%): 20 (44%) Reasons (describe): not mentioned
Dilation: N (%): 42 (48%) Reasons (describe): not mentioned |
1.1 FSFI score: mean ± SD Dilation: 24.49 ± 4.51 Surgery: 23.79 ± 3.57 p=0.507
1.2 Anatomical outcomes Vaginal Length: cm mean ± SD Dilation: 6.5 ± 2.04 Surgery: 8.1 ± 1.59 p=0.005
2. Complications: not reported
3. Undesirable effects: not reported |
Recommend dilation as first-line treatment.
“Although the vaginal length was shorter in the dilation therapy group than in the surgical therapy group, sexual function and quality of life were similar between these two groups. Vaginal dilation should be proposed as the first-line therapy for MRKH patients.” |
|
Morcel, 2013 |
Type of study: cross-sectional study
Setting and country: French National Association of Women with MRKH syndrome, France
Funding and conflicts of interest: not reported |
Inclusion criteria: - women with MRKH syndrome
Exclusion criteria: - not reported
N total at baseline: Intervention: n= 20 Control: n=20
Important prognostic factors2: Age (at questionnaire) mean ± SD: not significant Frank dilation: 26.2 ± 12.6 Surgery: 25.0 ± 8.9 Age (at procedure): mean ± SD: not significant Frank dilation: 18.9 ± 5.1 Surgical: 18.6 ± 2.5
Groups comparable at baseline? Yes
|
Sigmoid vaginoplasty (n=12) Davydov (n=8) |
Frank dilation (n=20) |
Length of follow-up: n/a
Loss-to-follow-up: n/a
Incomplete outcome data: not reported |
FSFI score: mean ± SD Surgical: 25.3 ± 8.0 Frank dilation: 25.3 ± 7.5 p=1.0
Anatomical outcomes: not reported
Complications: not reported
3. Undesirable effects: not reported |
Recommend dilation as first-line treatment, since no difference is observed, and Frank dilation is less invasive than surgical procedures.
“Functional sexual outcomes after nonsurgical and surgical methods were similar. Therefore, the Frank’s method should be proposed as first line therapy because it is less invasive than surgical procedures. In the case of failure of this technique or of refusal by the patient, surgical reconstruction may then be offered.” |
|
Willemsen, 2015 |
Type of study: retrospective cohort
Setting and country: University hospital, Gynaecology Department of the Radboud University Medical Center, Nijmegen, the Netherlands, 1962-2012
Funding and conflicts of interest: not reported |
Inclusion criteria: - women with MRKH syndrome
Exclusion criteria: - none mentioned
N total at baseline: Surgery: n=68 Control: n=75 Intercourse only: n=17
Important prognostic factors2: Age: (at surgery): mean (range): 23 (16-45)
Groups comparable at baseline? Yes
|
Davydov vaginoplasty (n=68) - of which n=34 after unsuccessful Frank dilation |
Intercourse (n=17) Frank dilation (n=75) - of which n=5 combined with intercourse |
Length of follow-up: Months: mean (range) Last clinical follow up or death Intercourse: 35 (1-128) Franks dilation: 45 (1-320) - combined with intercourse: 40 (1-147) Davydov: 135 (0-444) - with Frank dilation: 95 (1-300) - with other surgery: 138 (8-361)
Loss-to-follow-up: n/a since end is last clinical follow-up
Incomplete outcome data: not mentioned |
Sexual functioning: Sexual satisfaction: No: n=7 (11%) of surgery patients
Anatomical outcomes Vaginal depth: cm mean (range) Intercourse: 7.7 (4-10) Franks dilation: 6.7 (1-13) - combined with intercourse: 7.2 (4-10) Davydov: 8.4 (3-13) - with Frank dilation: 8.4 (4-13) - with other surgery: 7.4 (4-11)
Complications: 9 events in 7 surgery patients
3. Undesirable effects: not reported |
“Long-term results of both Frank dilation and Davydov neovagina procedure in experienced hands were good. The use of a peritoneal graft may be regarded as a good alternative to other widely used neovagina techniques using a graft, such as split-thickness skin graft or sigmoid neovagina.”
|
Risk of bias table
|
Study reference
(first author, year of publication) |
Bias due to a non-representative or ill-defined sample of patients?
(unlikely/likely/unclear) |
Bias due to insufficiently long, or incomplete follow-up, or differences in follow-up between treatment groups? (unlikely/likely/unclear) |
Bias due to ill-defined or inadequately measured outcome?
(unlikely/likely/unclear) |
Bias due to inadequate adjustment for all important prognostic factors?
(unlikely/likely/unclear) |
|
Callens, 2012 |
Unlikely |
Likely Reason: Large difference between lost to follow up |
Unlikely |
Unlikely |
|
Carrard, 2012 |
Unlikely |
Likely Reason: Large difference between lost to follow up |
Unlikely |
Unlikely |
|
Cheikhelard, 2018 |
Unlikely |
Unclear Reason: Loss-to-follow-up is large. However, analysis is cross-sectional. Follow-up mentioned is not follow-up of this study but indicates follow up after intervention in the past. Not clear if that follow-up was mandatory. |
Unlikely |
Unclear |
|
Herlin, 2018 |
Unlikely |
Likely Reason from text: “’You could speculate that the patients with no follow-up after surgery did not experience any shrinkage, and this might have caused an underestimation of the outcome given the longer vaginal depth measured immediately after surgery. Moreover, some patients might have had the follow-up at their private gynaecologist/physician, from whom we could not obtain medical records” |
Unlikely |
Unlikely |
|
Kang, 2020 |
Unlikely |
Unlikely |
Unlikely |
Unlikely |
|
Morcel, 2013 |
Unlikely |
Unclear Reason: loss to follow up was not mentioned |
Unlikely |
Unlikely |
|
Willemsen, 2015 |
Unlikely |
Unclear Reason: No a priori follow up was determined. |
Unlikely |
Unlikely |
Table of excluded studies
|
Author, year |
Reason of exclusion |
|
Ben Hmid, 2012 |
In French; no control group included; comparison between Davidov and McIndoe |
|
Bhagavath, 2017 |
Wrong study design: no information on surgical vs non-surgical |
|
Bianchi, 2011 |
Wrong comparison: no control group included; comparison between two laparoscopic procedures (Davydov and Vecchietti) |
|
Callens, 2014 |
Wrong study population: includes transitioning males |
|
Cao, 2013 |
Wrong comparison: no control group included; comparison between: laparoscopic peritoneal vaginoplasty and laparoscopic sigmoid vaginoplasty |
|
Cezar, 2014 |
Wrong comparison: no control group included; only reporting on efficacy of Vecchietti method |
|
Choussein, 2017 |
Wrong study design: descriptive systematic review |
|
Dabaghi, 2019 |
Wrong outcome, wrong comparison |
|
Ding, 2015 |
Wrong comparison: no control group included; comparison between: SIS graft vaginoplasty and Davydov laparoscopic |
|
Duranteau, 2021 |
Wrong study population: includes also non-females |
|
Edmonds, 2012 |
Wrong comparison: no surgical intervention group: only self-dilation |
|
Fliegner, 2014 |
Wrong study design: no information on surgical vs non-surgical |
|
Georgas, 2018 |
Wrong study population: includes transitioning males |
|
Hayashida, 2015 |
Wrong outcome: no information on functionality, only on structural, biological aspects are compared between Frank dilation and McIndoe |
|
Kölle, 2019 |
Wrong article type: review; could be used as background article; describes all (dis)advantages for a lot of (non)surgical procedures |
|
Le, 2015 |
Retracted article: because of errors in the data that undermine the conclusions. |
|
Marin, 2012 |
Wrong outcome: no statistics used on outcome of interest, only described in text (no numbers); also: in Portuguese) |
|
McQuillan, 2014 |
Excluded: better systematic reviews available |
|
McQuillan, 2014 |
Excluded: better systematic reviews available (not primary outcome) |
|
Nakhal, 2012 |
Wrong article type: review; could be used as background article; describes all (dis)advantages for a lot of (non)surgical procedures |
|
Navarro, 2020 |
Wrong comparison: no control group included; comparison between: classical McIndoe and neovaginal PACIENA prosthesis |
|
Ng, 2020 |
Wrong comparison: no surgical intervention group: only self-dilation and dilation via intercourse |
|
Perez-Medina, 2013 |
Wrong comparison: no control group included; comparison between two laparoscopic procedures |
|
Roman, 2021 |
In Czech, no English article available |
|
Wang, 2021 |
Wrong comparison: no control group included; comparison between Vecchietti procedure and control group (no information on control group regarding dilation) |
|
Wu, 2016 |
Wrong comparison: no control group included; comparison between: pushing-technique and separating technique (both laparoscopic) |
|
Zhang, 2019 |
Wrong comparison: no control group included; comparison between: SIS graft and Interceed surgery |
|
Zhang, 2020 |
Wrong comparison: no control group included; only Wharton-Sheares-George technique |
|
Zhao, 2015 |
Wrong comparison: no control group included; comparison between: laparoscopic vaginoplasty using single peritoneal flap and Davydov's laparoscopic technique |
|
Zhao, 2021 |
Wrong comparison: no control group included; comparison between two laparoscopic procedures (Hebei I and Hebei II) |
Verantwoording
Beoordelingsdatum en geldigheid
Laatst beoordeeld : 09-11-2023
Inbreng patiëntenperspectief
Er werd aandacht besteed aan het patiëntenperspectief door het uitnodigen van Patiëntenfederatie Nederland voor de schriftelijke knelpunteninventarisatie en voor deelname aan de clusterwerkgroepen. De verkregen input is meegenomen bij het opstellen van de uitgangsvragen, de keuze voor de uitkomstmaten en bij het opstellen van de overwegingen. De conceptrichtlijn is voor commentaar voorgelegd aan Patiëntenfederatie Nederland en de eventueel aangeleverde commentaren worden bekeken en verwerkt.
Zoekverantwoording
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