Keywords
Mayer–Rokitansky–Küster–Hauser syndrome, vaginal agenesis, digitally fabricated vaginal stent, vaginoplasty
This article is included in the Datta Meghe Institute of Higher Education and Research collection.
A common congenital abnormality affecting the female genital system is vaginal agenesis. It may appear on its own as a developmental aberration or as a component of a larger collection of anomalies. This disorder is frequently linked to the MRKH syndrome (Mayer-Rokitansky-Küster-Hauser syndrome). A new vaginal canal must be made and positioned between the bladder and the rectum in order to treat vaginal agenesis. Long-term effectiveness depends on maintaining the width and depth of the surgically created vaginal region and avoiding restriction. In this article, the surgical management of nonsyndromic vaginal agenesis in a 42-year-old lady is described. Digital fabrication methods were used to create a personalised acrylic vaginal stent. This customised vaginal stent helped gain patency of vaginal canal. These stents can be used to prevent neovaginal stricture and shrinking as well as to preserve the width and depth of the vaginal canal.
Mayer–Rokitansky–Küster–Hauser syndrome, vaginal agenesis, digitally fabricated vaginal stent, vaginoplasty
A congenital defect that affects the female reproductive system, vaginal agenesis can happen alone or in combination with other congenital anomalies.1 According to estimates, it affects one in every 4,000 to 5,000 live female births worldwide.2 Mayer-Rokitansky-Küster-Hauser (MRKH) syndrome, which is characterised by the absence or partial presence of the uterus, is frequently connected to it. A tiny uterus and occasionally some ovarian tissue may be seen during ultrasound scans, which are frequently used to diagnose this illness. Due to the undeveloped uterus, women with MRKH syndrome may have issues including hematometra and hematosalpinx during menstruation.3 In addition, endometriosis is frequently seen. Although coexisting renal or skeletal abnormalities are possible, ovarian function usually isn’t impacted. Marriage, childbirth, and social connections are all significantly hampered by the lack of a functional vagina, which causes distress in both the affected person and their parents.4 Through a treatment known as vaginoplasty, surgical intervention—pioneered by McIndoe—has been used to manage this issue.5 Split-thickness skin grafts taken from the thighs or buttocks are used in this procedure. Instead, an autologous graft, like buccal mucosa, has proven effective in these situations. Some medical professionals have looked into lining the neovagina with allografts like amnion, which can lessen morbidity at the graft donor site.6
Postoperative stent use is advised to avoid problems including neovaginal stricture and shrinking as well as to preserve the width and depth of the vaginal canal. McIndoe’s operation is still one of the most often used methods for vaginoplasty despite the development of other methods that do not require protracted dilatation. Following a McIndoe vaginoplasty, a variety of vaginal stents are offered for postoperative care.7 This article describes the digital creation of a customised acrylic vaginal stent for a young patient who had vaginal agenesis surgery.
Due to primary amenorrhea, a 42-year-old female patient was referred to the prosthodontics division. Physical examination found that she had typical secondary sexual characteristics, but gynaecological evaluation suggested vaginal agenesis, prompting a suggestion for ultrasound imaging. The results of an abdominal ultrasound revealed regions with decreased echogenicity, which allowed the diagnosis of hematocolpos (blood buildup in the vagina) and hematosalpinx (blood buildup in the fallopian tubes). She had no ovaries, which was surprising. The patient and her parents received thorough counselling that covered the specifics of the surgical procedure, possible outcomes, and the advantages of using a postoperative stent. The patient referred to the prosthodontics division from the obstetrics and gynaecology division Three stents were fabricated of increasing sizes and the size of the stent was determined through a combination of physical examination and radiographic assessment. During the stent fabrication process, a carefully crafted hollow acrylic vaginal stent with dimensions of 3× 2 × 2.5 cm was created. These specific measurements were based on the tissue thickness between the perineum and pelvic peritoneum, as determined through magnetic resonance imaging (MRI) conducted by a gynecologist. To construct the first stent, Impression was made with impression compound (Figure 1) and the cast was poured (Figure 2) and was scanned with In EOS X 5 3 (Figure 3) to obtain an STL file and from that the first stent was digitally fabricated by 3D Printing by using resin material Creality 3D LD-002R LCD Resin 3D Printer (Figure 4).
For increasing the size of second stent (fabricated conventionally) an internal wax framework was fabricated conventionally, intentionally slightly larger in all dimensions compared to the first stent size by 1.5 cm. This framework had a conical shape, with one end having a larger diameter than the other. The wax surface was meticulously smoothed before the application of a 2-3 mm layer of chemically cured acrylic resin. Small central areas at both ends were left open to facilitate the drainage of tissue fluid and secretions. To make the stent hollow ice was used Once the acrylic resin had solidified, the ice was converted to water, resulting in the creation of a hollow structure. The stent was then carefully finished and polished with acrylic finishing burs in sequential order to achieve a smooth surface. The choice of acrylic as the casting material was made due to its simplicity, cost-effectiveness, adequate strength, resistance to wear, ease of maintenance, and ease of preparation for this specific application. Two additional holes were made at the handle of the stent to facilitate in placement and suturing of stent (Figure 5).
The patient underwent vaginoplasty under general anaesthesia once the stent was made by surgeons in the obstetrics and gynaecology department, which required draining hematocolpos and attaining hemostasis. To guarantee a good fit, the acrylic stent was slowly placed into the recently developed vaginal cavity. Amniotic membranes were wrapped around a chemically sterilised stent after being pre-rinsed with saline and combined with antibiotics. The labia minora were stitched with three stitches across the vulva to firmly hold the stent in place when it was placed within the artificial vagina. Following surgery, the patient had oral and intravenous infusions, antibiotics, and had to eat only certain things for 72 hours. To check for bleeding, discharge, stent location, and any potential problems with vulvar sutures, the perineal area was routinely monitored. The stent worked and was initially discomforting but later became acceptable to the patient.
The labia sutures were taken out a week after surgery, and the vaginal cast was carefully removed. For six months, the patient went to follow-up visits every two weeks. She received instruction on how to maintain the acrylic stent throughout this time, which she used overnight for the first three months before using it continuously for the next three months.
One significant congenital aberration in the female genital tract is the absence of vaginal development, which can occur either as a standalone developmental problem or as a component of a more complex set of defects. Since this disorder is linked to psychological, reproductive, and social concerns, it poses substantial obstacles for both the affected person and their parents. In some societies, it is regarded as culturally sensitive to treat genital anomalies surgically.8 The Mayer-Rokitansky-Küster-Hauser (MRKH) condition, which was initially identified in 1961, frequently results in vaginal agenesis. While maintaining a normal female genotype, phenotypic, and endocrine status, this syndrome frequently involves abnormalities in the renal and skeletal systems.8 After McIndoe vaginoplasty, surgical vaginal stents are commonly employed for postoperative maintenance. It is strongly recommended to use these postoperative stents to prevent neovaginal shrinkage and stricture. Preserving the width and depth of the neovagina is of paramount importance, and these stents also assist in controlling bleeding. While soft molds have been used historically, current recommendations favor replacing them with hard stents for improved outcomes.9–11 Creating a functional vagina in people with congenital vaginal absence or underdeveloped vaginas is not a universally approved standard technique. Utilising the current mucous membrane-lined canal for vaginal restoration is one of the treatment options for vaginal agenesis. To achieve a suitable appearance and function, an alternative to surgical surgery is to create a neovagina.12 This surgical procedure is typically carried out when the patient reaches an age at which they can tolerate wearing a vaginal stent for a minimum of six months. The surgical cavity can be lined with either an autologous graft from buccal mucosa or an allograft like amnion. The Abbe McIndoe procedure, initially introduced in 1888, remains the most effective and preferred method for this purpose.13–15 It is crucial for patients to adhere to wearing the stent, even if it proves inconvenient, as non-compliance can significantly contribute to treatment failure. In specific cases, a vacuum-assisted closure system has been utilized as an alternative to vaginal stents, eliminating the need for their use and enhancing the acceptance of grafts during vaginal reconstruction. Non-surgical methods involving prefabricated vaginal dilators have seen limited success, often causing discomfort and relying heavily on the patient’s motivation to use them effectively.16
Vaginal stents are commonly employed postoperatively to maintain the dimensions of the neovaginal structure, preventing contraction, shrinkage, and promoting hemostasis. A systematic review confirmed the benefits of dilators or stents in enhancing the well-being of women with a history of stenosis.
Various prefabricated or customized stent types have been described for maintaining neovaginal dimensions, including the ORFIT vaginal stent, tissue expanders, syringes, vacuum expandable molds, solid or hollow acrylic stents, silicone stents, and a novel silicone-coated acrylic stent combining both materials. The choice of acrylic resin for prosthetic vaginal stents in this case was due to its rigidity, essential for maintaining patency in a male patient with an artificially created natural vaginal tract, which is prone to wall collapse compared to vaginal agenesis cases. While silicone stents are also used, they may be susceptible to fungal infections and deterioration if not well-maintained. Over time, they can tear and require re-fabrication. In an effort to improve patient compliance, a hollow acrylic stent was fabricated to reduce the prosthesis’s weight. Several materials like wax, salt, sugar, caramel, and thermocol have been employed to create hollow prostheses. However, the novelty of this technique lies in using ice to create the hollow space. Wax residues can interfere with acrylic and silicone processing, while salt, sugar, caramel, and thermocol can lead to a rough inner prosthesis surface if not adequately cleaned, potentially causing infections. In contrast, ice is easily removable, less likely to cause issues, readily available, and cost-effective. Additionally, ice does not react with the prosthesis material, making it an easily manageable and ideal choice.9–11,17
Vaginal agenesis can be treated safely and effectively by using amnion as a graft during vaginoplasty to construct a neovagina. However, the patient’s dedication to conscientious stent usage during the postoperative period is the most important component in creating a functional neovagina after surgery. Acrylic moulds are a practical choice in this situation. Before the operation, any healthcare professional working with a prosthodontist can simply receive these moulds. This method offers a simple, quick, and affordable method for creating a vaginal stent with the main objective of minimising contracture and subsequently improving the outcome of the vaginoplasty process.
Written informed consent for publication of their clinical details and clinical images was obtained from the patient.
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Is the background of the case’s history and progression described in sufficient detail?
Partly
Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?
No
Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?
Partly
Is the case presented with sufficient detail to be useful for other practitioners?
Partly
Competing Interests: No competing interests were disclosed.
Reviewer Expertise: Gynaecology, Surgery, Neovagina, Mullerian Anomalies, Cervical Atresia, Vaginal Agenesis, MRKH syndrome, Vaginoplasty
Alongside their report, reviewers assign a status to the article:
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Version 1 27 Nov 23 |
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Provide sufficient details of any financial or non-financial competing interests to enable users to assess whether your comments might lead a reasonable person to question your impartiality. Consider the following examples, but note that this is not an exhaustive list:
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