Hysteroscopy SIG Most Difficult Case: Cystic Adenomyosis

Adenomyosis is defined as the presence of endometrial glands and stoma invading the myometrium, along with different degrees of hyperplasia and hypertrophy of the smooth muscle fibers surrounding the lesion. The presence of ectopic endometrial gland and stroma in an ectopic location is similar to what is seen in patients diagnosed with endometriosis.

The reported prevalence of adenomyosis varies between 5-70% depending on the different criteria that are used to establish the diagnosis (1). The etiology of adenomyosis remains unknown and, similar to endometriosis, different theories have been postulated aiming the explain the origin of the disease. Two main theories have gained acceptance explaining the origin of adenomyosis. One suggests the involvement of tissue injury and repair mechanism claiming that adenomyosis results from invagination of the endometrial basalis into the underlying myometrium. Another theory supports that adenomyotic lesions result from metaplasia of displaced embryonic pluripotent Müllerian remnants or differentiation of adult stem cells.(2)

The most common symptoms of patient with adenomyosis are heavy menstrual bleeding (40-50%) and dysmenorrhea (15-30%). It is important to note that up to 33% of women with adenomyosis are asymptomatic.(3) The presence of adenomyosis seems to contribute to infertility by affecting the endometrial receptivity (4).

The initial diagnosis of this disorder is usually performed with 2D ultrasonography. There are some ultrasonographic findings considered suggestive of adenomyosis that were published in 2015 by the international Morphological Uterus Sono-graphic Assessment (MUSA) group (5). This sonographic criteria include asymmetrical thickening of the uterine walls, the presence of cysts inside the myometrium, hyperechoic islands, fan-shaped shadowing, echogenic subendometrial lines and buds, translesional vascularity, irregular junctional zone and interrupted junctional zone (5).

It is honest to say that hysteroscopy plays a limited role in the evaluation and management of the patient with adenomyosis. However, there are some hysteroscopic patterns including irregular endometrial surface with endometrial defects, areas of hypervascularization, strawberry pattern or cystic hemorrhagic lesions that are commonly associated with adenomyosis.

Cystic adenomyosis is a type of adenomyosis characterized by the presence of cystic areas of different size and shape composed by endometrial glands and stroma within the myometrium. These adenomyotic cysts are usually small (1 to 2 cm diameter), although larger cysts have been reported, and contain dark fluid that have no communication with the uterine cavity. In a review of cystic adenomyosis, Brosens et al described three types of cystic adenomyosis, based on the location of the cyst and the complexity of the lesion (6). Depending on the age of onset, it can be divided into adult of juvenile type. Adult cystic adenomyosis occurs mostly in women in their 40’s-50’s. The cystic structures can be found in any location of the uterine wall. When located close to the endometrial cavity, it is possible to open the cystic lesion and evacuate the retained dark fluid.

We present a case of a 41-year-old nulligravid woman with history of infertility and dysmenorrhea. The 2D Ultrasound scan revealed a cystic anechoic structure of 1,5 cm diameter located in the anterior wall of the uterus, in direct contact with the uterine cavity and covered by endometrium (Figure 1). A diagnostic in office hysteroscopy was performed identifying an area of hypervascularization located in the anterior uterine wall protruding into the uterine cavity. The patient was subsequently scheduled for an operative hysteroscopy aiming to drain the cystic lesion and fulgurate the wall of the cystic walls. The procedure was subsequently performed using normal saline as distension media with a 16 French diameter bipolar mini hystero-resectoscope (TONTARRA Medizintechnik GmbH) with a bipolar loop. Using ultrasound guide the cystic area was localized. An incision was made over the endometrium and myometrium covering the adenomyotic cyst until the cyst reached and opened (Figure 2). Dark-brown fluid contained inside the cyst was drained into the uterine cavity. Once all the content of the cyst was evacuated, fulguration of the ectopic endometrium was performed in order to prevent recurrence of the cyst. Immediately after the surgery the patient reported great improvement of her pelvic pain and dysmenorrhea.

Hysteroscopic opening and drainage of cystic adenomyotic lesions is a feasible minimally invasive approach that can be performed in selected cases of patients diagnosed with cystic adenomyotic lesions located in close proximity to the uterine cavity. The myometrium over the cyst can be opened with cold scissors or using electrosurgery. Some authors recommend the use of an ablative technique in patients with adenomyotic cysts localized deep in the myometrium. The resection of adenomyotic cysts usually results in a defect over the endometrial surface. Further studies are needed to investigate the reproductive outcomes of patients undergoing hysteroscopic management of cystic adenomyosis.

References:

1. 1. Munro MG. Classification and Reporting Systems for Adenomyosis. J Minim Invasive Gynecol. 2020 Feb;27(2):296-308.
   2. García-Solares J, Donnez J, Donnez O, Dolmans MM. Pathogenesis of uterine adenomyosis: invagination or metaplasia? Fertil Steril. 2018 Mar;109(3):371-9.
   3. Abbott JA. Adenomyosis and Abnormal Uterine Bleeding (AUB-A)-Pathogenesis, diagnosis, and management. Best Pract Res Clin Obstet Gynaecol. 2017 Apr;40:68-81.
   4. Vercellini P, Consonni D, Barbara G, Buggio L, Frattaruolo MP, Somigliana E. Adenomyosis and reproductive performance after surgery for rectovaginal and colorectal endometriosis: a systematic review and meta-analysis. Reprod Biomed Online. 2014 Jun;28(6):704-13.
   5. Van den Bosch T, Dueholm M, Leone FPG, Valentin L, Rasmussen CK, Votino A, et al. Terms, definitions and measurements to describe sonographic features of myometrium and uterine masses: a consensus opinion from the Morphological Uterus Sonographic Assessment (MUSA) group. Ultrasound in Obstetrics & Gynecology. 2015;46(3):284-98.
   6. Brosens I, Gordts S, Habiba M, Benagiano G. Uterine Cystic Adenomyosis: A Disease of Younger Women. J Pediatr Adolesc Gynecol. 2015 Dec;28(6):420-6.

Figure 1.

2D ultrasound revealing the presence of an anechoic cystic structure of 1.5 cm in the anterior uterine wall in close proximity with the uterine cavity.

Figure 2.

Resectoscopic excision of the cystic adenomyoma lesion using the 15 F mini-resectoscope. The cystic wall was resected (Upper left image), then the dark fluid content of the cystic structure was drained (Upper right image), once the cyst was completely drained (Lower left image) the wall of the cyst were subsequently fulgorized to prevent cyst recurrence (Lower right image).