Vaginal Agenesis is a rare condition occurring in the general population. However, the diagnosis of this condition has an important psychological and emotional impact for patients. The most important stages in the management of vaginal agenesis are correct diagnosis, research for associated malformations, and psychological support in conjunction with treatment. The psychological effect of diagnosis and treatment should not be underestimated [5].
According to the American College of Obstetricians and Gynecologists (ACOG) the treatment for Vaginal Agenesis aims to restore anatomy and functionality of the vaginal canal. It can be surgical or conservative. However, conservative treatment through vaginal dilation is the first choice due to the good results and low rate of complications [5].
In 1938, Frank described the first conservative treatment for vaginal dilation using Pyrex® tubes of gradually increasing sizes (0.8 cm, 1.5 cm, and 2.0 cm in diameter). This was used to force the mucous membrane into the vaginal introitus region. No incisions were required for this procedure [4]. The main criticism of this therapeutic modality is that it requires a special dedication from the patients, as only with the exercises with the dilators is it possible to create a vaginal canal that enables sexual intercourse. Maintaining a vaginal prosthesis is sometimes necessary to keep the vaginal canal patent, as well as performing exercises in the absence of regular sexual practice.
Decades after Frank's first description, several studies reported favorable results using his method [8–11]. In 1981, Ingram suggested that the failures in the technique used by Frank were due to tiredness of the hands and fingers during the procedure, the need to use the embarrassing position, and the inability to perform other productive activities during the procedure [10]. In an attempt to overcome these limitations, Ingram proposed a modification of Frank's original method. In Ingram's method, the patient's weight is used to replace manual and digital effort. The specially designed bicycle seat bench was used to facilitate perineal mold pressure [12].
In addition, corroborating the results of the successful experiment by Ingram [12], Roberts et al. reported a 91% success rate using the Ingram method in their study of 51 patients with MRKHS [13]. When well advised and emotionally prepared, almost all patients (90-96%) will achieve a satisfactory anatomical and functional result with vaginal dilation [5].
A recent study carried out at the same reference center of the present project, compared, in terms of anatomical, functional, and sexual aspects, two types of treatment for women with vaginal agenesis: progressive dilation (using the Frank method) or surgical neovaginoplasty (using the modified Abbé-McIndoe technique with oxidized cellulose). According to that study, both treatments had satisfactory efficacy and positive outcomes for patients with vaginal agenesis concerning anatomical, functional, and sexual aspects. Those data reinforce the reliability of the results the present study, which indicates that dilation treatment can remain the first-line therapy for vaginal agenesis [14].
The literature lacks more consistent and robust studies comparing the different surgical techniques and even comparing these with vaginal dilation. However, so far, no surgical technique has surpassed the success rate of non-surgical treatment; a fact that, together with the benefit of being a safer technique, places vaginal dilation as the first line in the treatment of vaginal agenesis [5, 15–22]. In this context, the present study offers and develops and conservative treatment for each patient through vaginal dilation with 3D personalized molds.
In 1984, Charles Hull founded the world's first 3D printing company, also known as Additive Manufacturing (AM) or Rapid Prototyping. The AM is a set of production technologies used in the synthesis of a given physical object by adding layers to form a part based on data generated by computer-aided design systems (CAD - Computer Aided Design). These technologies are widely used to quickly prototype products and tools for commercial purposes. Over time, it has also been integrated into other areas, such as the health area, since these tools have enabled the assistance of health professionals in diagnosis, surgical planning and in the synthesis of orthotics, prostheses for the rehabilitation of patients [23].
The choice of devices made using a 3D printer was based on the possibility of offering an individualized treatment for each patient, at a low cost and with a low rate of complications. The devices can be made with the most diverse formats and materials, which allows them to be adapted to the needs of each patient. Of the sixteen patients treated, fourteen reached the vaginal length considered for treatment, representing an 86% success rate. The only two patients who did not achieve a CVT of 6 cm or greater were still starting treatment (only 2 and 3 months ago).
According to the results of this present study, as well as in the literature [5, 15–22], conservative treatment for vaginal agenesis remains an excellent choice, with good efficacy and few complications, through personalized vaginal molds, made with 3D printing. These results highlight the good applicability of the devices, bringing a cost-effective and easily reproducible option for the treatment of vaginal agenesis, making this a promising and accessible tool. Therefore, it would be a fruitful option even to be taken to more distant locations in the country; training professionals to apply the treatment and shortening the distances so that more patients could benefit, thus eliminating the bias of distance and regularity in performing the exercises.
4.1. Strengths and limitations
The main relevance of this research is the possibility of offering an individualized treatment option that is recommended in the scientific community with ethics, efficacy, and safety for a patient in an international reference center for the treatment of vaginal agenesis. In addition, it is the first study to analyze reproductible 3D molds with the conservative treatment and improve sexual function in women with vaginal agenesis. The homogeneous patient sample, standardized procedures, and prospective model are also strong points.
Another positive impact of this project was an effective response with the guidance of perineal massage exercises with the cast in patients with smaller vaginal introitus. This strategy can be used before the beginning of the dilation itself, aiming to reduce the chance of inadvertent dilation of the urethra.
The present study was not a randomized trial because of ethical issues. The main limitation of this trial was the small sample size. However, vaginal agenesis is a rare disease, whose incidence is 1:4000 female births. The seriousness and scientific effort of this study are not diminished because of the difficulty including more patients.
The main difficulties encountered in this study attendance were the lack of motivation, the lack of privacy in the patient home and the distance from the city of origin to the hospital. Another obstacle was the attendance to outpatient returns, as many patients lived in different cities and some even in other districts, the regularity in the performance of the exercises, depending on the personal motivation of each patient, home privacy and emotional situation during treatment.
Considering the statements above, the authors believe that the study's strengths overcome its limitations. As vaginal agenesis is a rare disease that affects young women and involves the sensitive issues of sexuality and self-esteem, disclosure of well-structured trials can contribute to gaining knowledge so that an increasing number of women can benefit from the results of the studies.