Being considered as a common benign uterine disorder, Adenomyosis (AD) is defined as the presence of endometrial gland and stroma within the myometrium in more than 2.5 mm of depth, which is surrounded by reactive fibrosis, hyperplastic and hypertrophic smooth muscle (1). AD is mostly diagnosed in multiparous women in their 30s or 40s; although, it can occur in nulligravid women, and for cultural and emotional issues, these women are unwilling to hysterectomy. Because it is mostly determined by pathologic examination after hysterectomy, the prevalence of AD is different, ranging between 10% to 66% (2). Based on histological evaluations, AD is classified into the localized (focal) or diffuse; when the whole myometrium or more than half of it, is involved. Its main symptoms include secondary progressive dysmenorrhea, menorrhagia, chronic pelvic pain, and infertility (3). Management of AD comprises long-term hormonal therapy, intrauterine device, gonadotropin-releasing hormone (GnRH) agonists, analgesic, and finally, hysterectomy (4). Hysterectomy is the most common choice, especially in those women with complete family planning; however, the patients' desire for future fertility makes us perform conservative surgeries, which results in uterine preservation. This study aims to describe the management of diffuse AD in a 36-year-old virgin female by triple flap technique and levonorgestrel intrauterine device (LNG-IUD) insertion.

 

A 36-year-old virgin female was referred to Imam Khomeini hospital complex in 2020, due to heavy and intermittent vaginal bleeding ten months before our visit. Her past medical history consisted of diabetes mellitus and hypertension, using Amlodipine, Losartan, Metformin, and Glibenclamide. She received oral contraceptive pills, progesterone, and five months of GnRH agonist for the symptoms, which were not useful, and the symptoms were refractory to the conservative medication. Hysterectomy was recommended due to abnormal bleeding unresponsive to medical management, but since she was virgin and nulligravid and willing to future pregnancy, she was referred to our institution for uterine-preserving conservative surgery.
On the physical examination, uterine mass, equivalent to 20-22 weeks of gestation, was obvious. The laboratory tests showed a hemoglobin level of 6.6 g/dL and a platelet level of 444*10³ /mcL. On the abdominal ultrasound study, the uterine measured 200*180 mm with an endometrial thickness of 4mm, and a posterior uterine mass without obscure boundary was noted. The magnetic resonance imaging (MRI) was in favor of the diffuse posterior AD. MRI was performed with a pelvic coil in 3 tesla MR scanner (GE.healthcare SIGNA) the patient had fasting for about 6 hours intramuscular injection of hyoscine was done 15 minutes before MRI. Routine pelvic MRI protocol (sagittal axial and coronal T2 and axial T1 Images) DWI with b value 0.400,800 and post-contrast images in three plans was done. T2 weighted images showed diffuse enlargement of posterior myometrium of the uterus (low T2 with small bright foci), which appears inhomogeneous on both T1w and T2w images without discrete mass that represents the typical appearance of diffuse adenomyosis and endometrium was thin (Figure 1a, b). On post-contrast T1 FS images, diffuse heterogeneous enhancement was noted without any separate mass (Figure 1c). Axial Diffusion-weighted imaging at b = 800 and ADC map showed no restriction. The ADC value was 1.29 × 10−3 mm2/s (Figure 1d, e). Both adnexa, cervix, and other pelvic organs were normal on MRI.

Figure 1. Pelvic MRI in a 36 year-old female, showing abnormally enlarged uterus a) sagittal T2 weighted fast spin-echo Images and b) axial T2 weighted Images shows uterus is diffusely enlarged with the fundus at the level of L4 vertebral body. The junctional zone is thick, containing multiple small high signal foci typical for diffuse Adenomyosis. Endometrial thickness was 3mm without intracavitary mass or distortion. c) T1 weighted fat-suppressed images show diffuse heterogeneous enhancement. d and e) DWI and ADC map shows no restriction.

 
Adenomyomectomy with triple flap technique was performed, as previously described by Osada in 2011 (5). The surgery was performed following the transverse suprapubic incision and placing the rubber tube around the proximal od cervix. The AD was visible after bisection of the uterus from the serosa surface of the fundus in midline and in the sagittal plane and the AD tissue was resected from the surrounding myometrium by grasping by Martin Forceps. The uterus was then reconstructed by the triple flap technique. An LNG-IUD (Mirena, Bayer Co., Germany) was applied intraoperatively in uterine cavity for the prevention of future AD recurrence. She was administered with four units of packed red blood cells intraoperatively. During the postoperative period, no signs and symptoms of fever and hemorrhage were noted. In the 6-month follow-up visit, the uterine size was decreased significantly (the uterine size was equivalent to 12 weeks of gestation), the hemoglobin level was alleviated (hemoglobin level of 12.5 g/dL), and menstrual cycles was normal without any abnormal bleeding.