The second principle was involved management complexity hierarchy (MCH). In terms of MCH, the anomalies were arranged from the simple-to-treat to the more difficult-to-treat cases and finally the most technically complex cases. According to MCH, cases that present during adolescence and require immediate interventions because of menstrual retention were allocated to subclass d in all the major classes. A descriptive example can be seen in the arrangement of class I as uterine aplasia/hypoplasia with normal vagina currently requiring no treatment. Uterovaginal aplasia or Mayer–Rokitansky–Kuster–Hauser syndrome requires the creation of a neovagina. Isolated cervical aplasia requires endoscopic canalization or uterovaginal anastomosis. Isolated vaginal aplasia requires the creation of a neovagina and uteroneovaginal anastomosis (Fig. 1). The same principles were applied for the arrangement of other major classes.
Another descriptive example of arrangement within the context of AH and MCH is the arrangement of anomalies resulting from failures of fusion (class III) of the paired Müllerian ducts, which result in the duplication of a part or all the Müllerian duct derivatives. In terms of AH, this major class included, first affection of the uterine fundus, resulting in a bicornuate uterus, followed by extension to the uterine corpus, resulting in what was previously known as uterus didelphys unicollis or (duplicated uterine body with a single cervix), and so on until duplication of the entire genital tract. The MCH is also well evident as we started with simple anomalies that usually require no treatment and finally we included complex anomalies that require urgent and difficult surgeries (Fig. 3).
The differences from the general outline of the AFS can be summarized in five main points. The first is the merging of AFS class III (didelphys) and class IV (bicornuate) into a single class in the treatment focused classfication (TFC); we called it class III or lateral fusion defects. Although this incorporation seems to be very different from the general characteristics of a well-recognized classification, there are strong justifications as well as many advantages. Both a bicornuate and a didelphys uterus arise when midline fusion of the Müllerian ducts is arrested 30. Therefore, the embryological and etiological backgrounds of the didelphys and the bicornuate uteri are common. In addition, didelphys and bicornuate uteri share a common clinical presentation as well as similar obstetric performance 31,32. Finally, both are candidates for either no treatment versus the requirement of only a cervical cerclage operation or, on rare occasions, Strassmann’s operation 33. Therefore, merging of two major classes with the same etiology, clinical/obstetrical performance, and treatment options is not only justifiable but also global, abbreviated and accurate.
The second difference is in the expansion of each major class to include all related subclasses with a special focus on those with obstruction of the menstrual outflow 34,35. These subclasses are invariably organized as an extension (subclass D) of the five major classes (Table 1 and Box 1). Such anomalies require urgent surgical interventions for the treatment of pain and to prevent complications related to prolonged menstrual retention.
The third difference is that the arcuate uterus was included in the septate class on the basis of the very same principles of incorporation of the didelphys and bicornuate uteri. This has been a subject of extensive discussion in the previous literature 1,36, and we did not categorize the arcuate uterus into a separate major class 2. In this class, we included the presence of a special variety of septate with fundal depression. Septate uterus with fundal depression is present, but if unreported, it may be misdiagnosed as bicornuate uterus with the possibility of depriving these cases from the merits of hysteroscopic metroplasty. We reported this hybrid septate variety in 17 of 339 cases of pure septate uteri (El Saman AM, Shahin AY, Nasr A, Tawfik RM, Saadeldeen HS, Othman ER, Habib DM, Abdel-Aleem MA; unpublished data). These hybrid septate/bicornuate cases may be candidates for hysteroscopic metroplasty under laparoscopic monitoring as they are susceptible to perforation; thus, they were allocated to subclasses of a septate uterus (Fig. 4).
The fourth difference is in the creation of class IV: failures of communication or longitudinal fusion defects (Fig. 5). These groups of disorders are specific to adolescence as they present mainly during this period of life. This important class has not been mentioned in the AFS classification most probably because of its somewhat different embryogenesis. However, as mentioned in the brief embryological backgrounds presented earlier in this article, experimental and observational studies identified the sinovaginal bulbs as being derived from the caudal aspects of the Wolffian ducts and the Müllerian ducts. Therefore, the Müllerian ducts are implicated in part in the embryogenesis of these groups of defects. Furthermore, the identification and incorporation of this important category is important as corrective surgeries can be performed, which should be carried out in a timely manner to avoid the adverse effects of prolonged menstrual retention, which may affect reproductive potential 2,14,15.
The final difference was renaming of the last AFS major class and including diethylstilbosterol-related anomalies as a subclass of TFC class VI (Table 1). In addition, we also included other anomalies such as other tubal anomalies (accessory tube, accessory ostia, diverticulum, elongation, etc.).
Furthermore, we kept class VI open ended to include other undescribed rare entities and complex associations (Table 1; Fig. 6). This was done because with cumulative reporting and advancements in diagnostic modalities, new classes or subclasses are unlikely to be discovered, but sporadic cases may be found and can be allocated into the appropriate subclass in the TFC (Figs 2 and 4).
One of the well-known characteristics of Müllerian anomalies is the possibility of association with other anomalies, especially those of the urinary and other systems. Each major class of the treatment focused classification included two letters indicating the possibility of such associations: I/C, where I indicates isolated genital anomalies irrespective of their complexity and C indicates the presence of combined genital and extragenital anomalies. Extragenital malformations include urinary, gastrointestinal, skeletal, or any other associated anomalies.
Some tubal anomalies can be corrected, but others may not. Assisted reproductive technology represents an efficient tool for uncorrectable isolated tubal anomalies such as aplasia, hypoplasia, and non canalization.
In cases of isolated uterine aplasia and/or hypoplasia with an anatomically and functionally normal vagina, there is no current treatment. Gestational carrier is an option in some societies.
Patients with vaginal aplasia are offered nonsurgical use of successive dilators or the creation of a surgical space to be covered with a graft, and a mold to maintain the graft, with variable rates of success and wide range of complications 37–45. Recently, a novel treatment option was introduced by El Saman and colleagues, balloon vaginoplasty (BV), where traction is applied from the abdominal side on a silicon-coated balloon catheter, creating a natural neovagina over 1 week of time. With BV, there is a unique possibility of manipulating both the depths and the widths of the neovaginas using different schedules of catheter traction and balloon distention. The procedure has shown a high success rate, with the possibility of early onset of initiation of coital activities, and minimal complications 2–5,46–49.
Endoscopically monitored canalization of isolated cervical aplasia is a recent procedure in which a special inserter can be used to pass a silicon drain attached to its caudal end across the uterine fundus to the vaginal pouch, which is then fixed securely for three months 3. Other methods include uterovestibular anastomosis or vaginoplasty and uteroneovaginal anastomosis. However, the logical management plan should involve the creation of a neovagina using any of the aforementioned techniques, followed by uteroneovaginal anastomosis 2. Because BV represents the fastest way to create a functional neovagina, it is the first choice of treatment in patients with isolated vaginal aplasia 3,50.
We reported one case with a communicating hypoplastic hemiuterus leading to a normal vagina and a noncommunicating well-developed functioning horn. The patients had presented with primary amenorrhea and cyclic pain. We were able to perform a procedure to establish communication of the functioning horn with the existing vagina. Normal menstruation was established after surgery.
The didelphys uterus has extraordinary capacity, intercourse is often possible in both vaginas, and simultaneous pregnancies in each uterus can occur 57–59. Cervical unification is technically difficult and can result in cervical stenosis or incompetence 33,55.
The management of a nonobstructing longitudinal septum is simple and appears to be associated with improvement in fecundability. Excision of the septum allows the simultaneous insemination of both hemiuteri during a single act of coitus. Both cold knife/scissor excision and diathermy excision have been reported to yield similar outcomes 40,60.
The obstructed unilateral vagina is a clear indication for resection of the vaginal septum, followed by a laparoscopic treatment of associated endometriosis and/or adhesions 40,61.
The arcuate uterus is the most commonly observed uterine anomaly detected by HSG 62,63. It is a clinically benign anomaly; adverse obstetric outcomes are rare and it may not affect reproductive outcomes 63,64. Women with recurrent spontaneous abortions or histories of preterm delivery are considered candidates for 65,66 hysteroscopic metroplasty, which is a state-of-the-art treatment plan 3,52,60,67,68.
The depth of the groove and the length of the uterine septum in the adult uterus depend on the length of the incompletely fused Müllerian ducts in the fetus. Failure of resorption in the remaining part results in the down prolongation of the dividing septum. On laparoscopy, a shallow fundal depression not correlating with the depth of the internal division may be seen. Thus, it is important to consider the results of other investigations (MRI, US, and HSG) with the laparoscopic procedure. Symptomatic patients with this disorder are candidates for hysteroscopic metroplasty but there is a greater risk of perforation. Laparoscopy helps reduce the risk of uterine perforation and aids the diagnosis of associated pelvic pathology 10,52.
Excision of the vaginal septum is carried out either by cold knife/scissor division or diathermy resection, followed by hysteroscopic metroplasty 60.
An obstructed unilateral vagina is a clear indication for resection of the vaginal septum 40,61,69. This is followed by definitive resection of the cervicouterine extension of the septum 60.
This important class represents a group of defects that are not classified by the AFS system 1,2. Appropriate timely diagnosis and management will help prevent irreversible damage of the pelvic organs. Fortunately, this group of defects is amenable to complete surgical correction.
Early diagnosis of an imperforate hymen and timely optimized interventions are very important 25,70–73. Premenarchal asymptomatic girls are best treated conservatively until puberty, except in those presenting with mucolpos or hydrocolpos 74–76. An imperforate hymen is treated by two perpendicular or X-shaped hymenotomy incisions, followed by complete excision. However, procedures that spare the integrity of the hymen are strongly preferred by patients and families in many societies 38,77–79.
The management of a TVS is technically difficult; because most of these septa are located in the superior vagina, management should be in a tertiary-care center with a qualified surgical team. A TVS may be found concurrently with an imperforate hymen 24,84. Patients presenting with acute pain can be treated with laparoscopic drainage, providing pain relief without compromising the success of definitive surgery that can be performed at a later date 85.
Excision of the septum should be performed under transabdominal or transrectal ultrasound guidance. The widest possible excision should be made with a careful approximation of edges 86. Patients with cyclical hematuria require reconstruction using a transvaginal and transabdominal approach 87.
Segmental atresia usually requires reconstructive surgery to cover the raw area created from the excision of long atretic segments 88,89. In extreme cases of segmental vaginal atresia, retropubic BV may be required 3,48. Other techniques used for the creation of neovaginas could be used for the treatment of this rare condition. We have described a successful pull-down BV technique for patients presenting with segmental atresia of the middle or lower one-third in association with upper hematocolpos.
In this class, treatment is individualized according to the presentation of the cases. In addition, in all classes/subclasses, it is important to pay attention to any associated extragenital anomalies or complications of long-standing obstructions.
The current proposal represents a model for a logical and updatable classification of Müllerian duct anomalies. It has the advantages of being uniform, being based on embryologic backgrounds, and following anatomical, management complexity hierarchies, and may be potentially user friendly.
There are no conflicts of interest.
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