|Year : 2020 | Volume
| Issue : 1 | Page : 39-43
Customized hollow surgical stent for congenital vaginal agenesis in early adolescent female with MRKH syndrome: a case report
Manu Rathee1, Meenakshi Chauhan2, Prachi Jain3, Akanksha Shetye4
1 Senior Professor and Head, Department of Prosthodontics, Post Graduate Institute of Dental Sciences, Pt. B.D Sharma University of Health Sciences, Rohtak, Haryana, India
2 Professor, Department of Obstetrics and Gynaecology, Post Graduate Institute of Medical Sciences, Pt. B.D Sharma University of Health Sciences, Rohtak, Haryana, India
3 Senior Resident, Department of Prosthodontics, Post Graduate Institute of Dental Sciences, Pt. B.D Sharma University of Health Sciences, Rohtak, Haryana, India
4 Post Graduate Student, Department of Prosthodontics, Post Graduate Institute of Dental Sciences, Pt. B.D Sharma University of Health Sciences, Rohtak, Haryana, India
|Date of Submission||12-Sep-2019|
|Date of Acceptance||17-Jun-2020|
|Date of Web Publication||17-Sep-2020|
Dr. Prachi Jain
Post Graduate Institute of Dental Sciences, Pandit Bhagwat Dayal Sharma University of Health Sciences, Rohtak, 124001, Haryana
Source of Support: None, Conflict of Interest: None
Mayer-Rokitansky-Kuster-Hauser syndrome (MRKH) is a congenital malformation resulting in the absent or small uterus and variable degrees of vaginal hypoplasia of its upper portion. One of the treatment goals for these patients is the creation of an artificial vagina either conservatively or surgically. Use of long-term prosthetic vaginal stents prevents the possible contraction of the reconstructed vagina, maintain vaginal width and depth and avoid vaginal stenosis. Prefabricated stents of standard sizes have widely been utilized to assist surgical creation of a neovagina. However, a customized stent has the evident advantage of the possibility of adjusting the size as per individual patient requirements. In the present clinical report, an early adolescent female patient with MRKH syndrome was surgically managed with insertion of a customized vaginal stent that offers the versatility of design configuration to suit the various clinical situations.
Keywords: Amniotic graft, customized stent, Mayer–Rokitansky–Kuster–Hauser syndrome, vaginal agenesis
|How to cite this article:|
Rathee M, Chauhan M, Jain P, Shetye A. Customized hollow surgical stent for congenital vaginal agenesis in early adolescent female with MRKH syndrome: a case report. Nigerian J Plast Surg 2020;16:39-43
|How to cite this URL:|
Rathee M, Chauhan M, Jain P, Shetye A. Customized hollow surgical stent for congenital vaginal agenesis in early adolescent female with MRKH syndrome: a case report. Nigerian J Plast Surg [serial online] 2020 [cited 2023 Mar 30];16:39-43. Available from: https://www.njps.org/text.asp?2020/16/1/39/295256
| Introduction|| |
Vaginal agenesis is a rare condition having devastating consequences on fertility and sexual function with an incidence ranging from 1 in 4,000 to 1 in 10,000 females. The most common aetiology of vaginal agenesis is Mayer–Rokitansky–Kuster–Hauser syndrome (MRKH) which is characterized by a congenital absence of uterus and vagina compromising the normal functions of the genital tract, thereby causing psychological trauma.
Vaginal agenesis is one of the important causes of primary amenorrhea and can be detected by diagnosis performed during puberty. Following diagnosis, the patients can be treated by both surgical and non-surgical approaches. The surgical procedure commonly preferred is McIndoe’s vaginoplasty which involves the creation of a space between bladder and rectum, followed by insertion of a mould covered with split-thickness skin graft into that neovaginal space, and use of postoperative vaginal dilation to avoid stenosis. Autologous graft like buccal mucosa has also been used with success. Allograft like amnion has been used to line the neovagina, which can reduce the morbidity of the graft donor site.
This clinical report presents a case of MRKH syndrome managed surgically using McIndoe’s vaginoplasty surgical technique assisted by a vaginal stent customized as per the required design and size.
| Case report|| |
A 19-year-old young female patient with a diagnosis of MRKH syndrome was referred to the Department of Prosthodontics, from the Department of Obstetrics and Gynaecology for the fabrication of vaginal stent. The chief complaint of the patient was cyclic abdominal pain since 5-6 years. There was a history of primary amenorrhea. The patient and the accompanying mother was in utmost distress and was consented for immediate surgical treatment.
On general examination, the patient appeared normal and of average intellectuality. Slight clinodactyly was observed on bilateral hands. Gynaecological examination showed that her secondary sexual characteristics were of Tanner stage 4. External genitalia was normal for age with the presence of vaginal dimple. The ultrasonography (USG) revealed a left ectopic kidney.
Surgical treatment was scheduled after detailed counselling regarding the surgical procedure, its purpose, outcome and its possible complications. The dimensions of the stent were determined preoperatively by the gynaecologist based on the thickness of the intervening tissue between the perineum and pelvic peritoneum which was determined by magnetic resonance imaging (MRI) and was thereafter fabricated by the prosthodontist accordingly. The benefits of vaginal stent postoperatively were explained to the patient and her mother. Written consent was taken before commencement of treatment.
| Fabrication of vaginal stent|| |
As per the required dimensions of the gynaecologist, a vaginal stent was fabricated measuring 9 × 3 × 2.5 cm. A hollow heat-cured acrylic stent was planned. A hollow metal cylinder was used and was cut as per the required dimensions [Figure 1]. Upon the external surface of the metal cylinder, modelling wax (API Rolex Modelling Wax) was shaped according to the necessary width and then smoothened [Figure 1]. The wax mould was then invested [Figure 2]. The final packing was done with heat cure acrylic resin (DPI Heat cure) and the final vaginal stent was obtained [Figure 3]. The final stent was adjusted in dimensions to a conical shape. Thereafter, finishing and polishing of the stent were done to provide a smooth surface.
|Figure 1 Modelling wax adapted to the metal cylinder, (A) length dimensions (B) Width dimensions|
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|Figure 3 Heat cured acrylic vaginal stent (A) Final length (B) Final width|
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| Surgical procedure|| |
Surgery was preceded with a strict note of overnight fasting. Abbe McIndoe vaginoplasty procedure was planned. A single intravenous dose of 1000 mg cefotaxime was given 30 minutes before the operation for antimicrobial prophylaxis. Under spinal anaesthesia, the patient was laid in the lithotomy position. The bladder was catheterized with Foley’s catheter. An incision was given over both the sides of median raphe on the vaginal dimples. A potential space was created between the urethra and bladder anteriorly and rectum posteriorly by blunt dissection. The dissection continued until a neovagina was obtained [Figure 4]. Haemostasis was achieved. The specially designed vaginal stent was covered with amnion graft and was later inserted into the neovagina [Figure 5A and 5B]. The amnion was obtained from a women who delivered within six hours, had intact membranes and whose viral markers for Hepatitis B, C and HIV were negative. The labia minora were sutured with two transverse vulvar sutures to keep the stent in position and retained in the neovagina.
|Figure 5 (A) Heat cured acrylic vaginal stent covered with amnion (B) McIndoe vaginoplasty with vaginal stent sutured in neovagina|
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Postoperatively after six hours, the patient was followed by a soft diet. The perineal area was regularly checked for any bleeding, discharge, the position of mould, and any cutting through of the vulvar sutures.
| Follow up|| |
On seventh postoperative day, the labial stitches were cut and the stent removed. The amnion was taken up well and a vagina of approximately 8 cm was formed. There were no complications except for a minimal fibrous tissue formation which was managed by changing the stent dimension size and further adjusted through trimming the stent by a few millimetres. The patient continued wearing the stent day and night until the next follow up which was scheduled after one month. The follow up showed a well created resultant neovagina [Figure 6]. Compliance for the use of the stent was satisfactory. The patient and her parents were satisfied with the treatment outcome and follow-ups were maintained regularly.
| Discussion|| |
Mayer-Rokitansky-Kuster-Hauser (MRKH) syndrome refers to the congenital aplasia or severe hypoplasia of the structures that are derived from the Mullerian ducts, including the upper vagina, uterus, and fallopian tubes. Young women diagnosed with MRKH syndrome are phenotypically normal women with karyotype 46, XX who suffer from extreme anxiety and very high psychological distress.
Various techniques have been used to create a neovagina as a part of the treatment through a non-surgical or surgical approach. Non-surgical methods include vaginal dilation which is recommended as the first line of treatment in vaginal agenesis. It involves insertion into the vaginal dimple, vaginal moulds of specific length and width which is gradually increased. These moulds apply local pressure, hence, increase the space between rectum and bladder. However, it is a time-consuming process and distressing to the patient as it acts as a constant reminder of her abnormality. Surgical vaginal reconstruction includes Williams vulvovaginoplasty, pressure method (Vecchietti procedure), neovagina creation procedures within the rectovesical space and lined with different types of tissue, such as skin (McIndoe Reed), peritoneum (Davydov), intestine or perhaps in the future tissue-engineered vaginal mucosa.
Several authors have documented good results after operations using McIndoe’s method. Satisfactory or good sexual relationships have been described in 83% to 100% of patients in studies, which include a total of more than 1000 patients. The McIndoe procedure is one of the most often performed surgical procedure which requires the surgical creation of a space in between the rectum and the bladder, placement of a mould covered with a split-thickness skin graft into space, and use of vaginal dilators postoperatively. The use of skin graft involves a plastic surgeon and is a time-consuming procedure. Thus material like amnion has been used. Amnion is readily available and has been found to give good results. Over recent years, new treatment options have been emphasized. Due to the difficulty for women with MRKHS to accept infertility, surrogacy is also an alternative method in many countries.
Surgical vaginal stents have also been described for postoperative maintenance after McIndoe vaginoplasty., The use of stent maintains the desired width and depth of vagina postoperatively and avoids shrinkage and stricture of neovagina during healing. Vaginal stents can be prefabricated or customized. They are used with increasing diameters during the healing period postsurgery. Several prefabricated stents of various designs are available. However, these are hard, costly, and only available in specific sizes and do not suit all the clinical situations. A customized stent, on the other hand, overcomes this disadvantage as they provide a provision of adjusting the size as per individual patient’s requirements, thus is suitable for variable clinical situations.
Customized vaginal stents can be of various materials like inflatable soft stents with or without suction, silicone-based or acrylic-based rigid stents. Silicone vaginal stents if not maintained properly can be prone to fungal infections and deterioration with time. Moreover, it is costly compared to acrylic resin and has the tendency to tear in long-term use and may require refabrication. In the present case report, the vaginal stent was fabricated using heat cure acrylic resin due to its several advantages of simple design, ease of fabrication, cost-effectiveness, rigidity, adequate strength, wear-resistant and less irritative for vaginoplasty surgeries.
These procedures are meant to improve the sexual functionality of the vagina and improve the quality of life of the women. However, concomitant psychological interventions and follow-up directions are further needed for management.
| Conclusion|| |
MRKH syndrome has to be carefully recognized and when seeking treatment options, it is necessary to consider interventions which majorly depend on patient compliance. McIndoe vaginoplasty is the most commonly used surgical method in recent years which include the creation of an artificial vagina followed by insertion of prosthetic vaginal stents to prevent vaginal stenosis and to maintain vaginal width and depth. Customised heat-cured acrylic resin vaginal stent fabricated in the present case offers various advantages over other materials used in the past as it is economical, has adequate strength, wear-resistant, is easy to prepare and cause less irritation. It offers size and configuration adjustability to best suit the surgeon and the patient.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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