Hysteroscopic diagnosis and excision of myometrial cystic adenomyosis

The present cases illustrate the possibility of hysteroscopy for diagnosis and resection or ablation of intramural cystic adenomyosis. Although diagnostic hysteroscopy does not reveal the pathognomonic signs of adenomyosis, some studies suggests that an irregular endometrium with endometrial defects, altered vascularization, and cystic hemorrhagic lesion are possibly associated with adenomyosis [9].

With the use of MRI imaging and 3-D ultrasound adenomyosis, junctional zone hyperplasia and adenomyotic cysts can now be detected in younger patients during their reproductive years. The use of 3-D vaginal ultrasound for the diagnosis of a junctional zone abnormality or adenomyotic pathology has been extensively described by Exacoustos et al. [14] and can now be implemented in patients seeking fertility treatment. As patients are postponing their desire to conceive, we can expect an increasing incidence of adenomyotic pathology in patients with fertility problems.

Brosens et al. [2] suggested the acronym MUSCLE for the classification of the cystic adenomyosis: M = myometrial location (intramural, submucous, subserous), U = uterine site (midline, paramedian, lateral); S = structure (cystic, mixed, polypoid), C = contents (clear, hemorrhagic), L = level (fundus, body, cervix), and E = endometrial or inner lining (endometrium, metaplastic). The first patient can be classified as M(2)U(2)S(1)C(2)L(2)E(1) and the second as M(1)U(2)S(1)C(2)L(2)E(1). To distinguish the condition from other intramural cysts, histologic diagnosis is necessary to identify the endometrial inner layering and the presence of outer myometrium.

In cases of submucous cystic adenomyotic lesions or an adenomyoma bulging into the uterine cavity, direct hysteroscopic access is possible. Using 5-Fr scissors during hysteroscopy allows a clear dissection of the myometrial wall of the cyst from the surrounding myometrium. Instead of dissection with scissors, an ablative technique that destroys the inner cystic wall is a possible treatment option. We believe that this ablative approach is preferable for those cysts localized deeper in the intramural portion. In contrast to the healing process after hysteroscopic myomectomy, where a normal uterine cavity is expected, resection or ablation of adenomyotic cysts bulging into the uterine cavity results in a visible defect of the myometrium.

In cases in which intramural cystic structures are present, ultrasound guidance is mandatory for localization of the cystic structure. The spirotome offers the possibility of penetrating the cyst and leaving behind a visible channel allowing hysteroscopic entrance into the cystic structure for an ablative procedure using bipolar current.

It is currently unclear whether cases of intramural cystic adenomyosis, which do not communicate with the endometrial cavity, can be adequately treated with an ablative procedure or should be managed by enlarging the cyst opening to allow communication with the endometrial cavity.

The hysteroscopic technique has the advantage of leaving the outer myometrium intact and avoiding an abdominal scar. Because the incidence of adenomyosis is increasing with age and women are postponing their childbearing, increases of incidence of adenomyotic pathology in those patients referred for fertility treatment can be expected. Therefore, uterine exploration should not only focus on a careful inspection of the endometrial cavity but also entail an evaluation of the uterine wall with special attention to the junctional zone. This can routinely be performed in an office setting using 3-D vaginal ultrasound and office mini-hysteroscopy.

Cystic adenomyosis represents a specific entity of adenomyosis and has to be distinguished from other intramural cystic structures. The diagnosis necessitates a histologic examination showing endometrial epithelial lining of the inner cystic wall with surrounding outer myometrium. The differential diagnosis in adolescents includes the congenital anomaly of a hematometra in a non-communicating horn; therefore, hysteroscopy is indicated to observe both tubal openings and the presence of a normal endometrial cavity [15]. In younger women, Acien et al. [16] have described comparable cystic structures as accessory cavitated uterine masses (ACUMs). The lesions were primarily isolated cysts located at the insertion of the round ligament without infringement on the uterine cavity. He suggested that these lesions might arise from persistence of ductal Müllerian tissue near the round ligament. At present, there is no consensus regarding the most appropriate way to treat these lesions and the need of treatment for infertility patients. Thalluri and Tremelen [17] reported an impaired clinical pregnancy rate in patients with adenomyosis (23.6 versus 44.6 % in controls) undergoing IVF treatment using an antagonist ovarian stimulation protocol. A lower clinical pregnancy rate and higher spontaneous abortion rate are also reported in the meta-analysis of Vercellini et al. [18]. A conservative fertility-sparing technique is preferable. A laparoscopic cystectomy for a limited number of patients has been described in the Japanese literature [4, 5, 19, 20]. Takeuchi [4] described the use of laparoscopic cystectomy in nine cases with a reduction of pain postoperatively; two of three patients who desired to conceive became pregnant afterwards; however, the time between intervention and conception was 2 and 7 years. Also, Kriplani [21] reported on the laparoscopic treatment of four patients who experienced a reduction of pain postoperatively. Akar [22] described the use of robotics for performing a resection of cystic adenomyosis. Reduction of pain has been described after simple transvaginal aspiration of the fluid content [23].

If larger cystic adenomyotic structures localized in the outer intramural third are present, a laparoscopic approach is preferable. Although the cyst is unencapsulated (unlike the myoma), complete removal is possible, in contrast to focal or diffuse adenomyosis. Of the reported adenomyotic cysts [2], only 13 % had a diameter greater than 50 mm and 40 % were smaller than 25 mm. In these smaller structures, laparoscopic identification can be difficult. In these cases, laparoscopic access can cause significant trauma to the myometrial wall. Most laparoscopic resections have been performed for severe dysmenorrhea and/or dysfunctional uterine bleeding. Fertility was not a major factor in these patients. For patients desirous of pregnancy, a procedure with the highest fertility preservation and minimal trauma must be chosen. As in cases of a small adenomyoma, our experience showed that when smaller cystic structures were present in the inner third of the intramural part or submucosally, a minimally invasive hysteroscopic approach is possible. Some of the lesions are directly recognizable at hysteroscopy because they bulge into the endometrial cavity, thus favoring a minimally invasive dissection. For lesions localized deeper in the intramural portion, the spirotome can be introduced under ultrasound guidance; it creates a channel and provides hysteroscopic access to the cystic structure. Treatment by resection or ablation can then be performed.

Giana et al. [11] described the use of a bipolar resectoscope for the treatment of a cystic adenomyosis in a 46-year-old woman. In a report by Kumar [24], an intramural cyst was inadvertently opened during an ablative procedure for dysfunctional uterine bleeding. After exclusion of the possibility of uterine perforation, the procedure was continued. Histology confirmed the diagnosis of an adenomyotic cyst.