The clinical challenge of a uterine cotyledonoid dissecting leiomyoma with adenomyosis: A case report

Abstract

Cotyledonoid dissecting leiomyoma (CDL) is a rare uterine tumor with unique clinical and histological features. We present a case of a 46-year-old woman with a 3-month history of left-flank pain radiating to the back. The patient had a history of infertility and a previous miscarriage. Ultrasound revealed a solid tissue mass suggestive of a degenerated fibroid. Laparoscopy identified subserosal leiomyoma and leiomyoma in the broad ligament. Histologically, CDL is characterized by disorganized smooth muscle with hyaline degeneration and no evidence of malignancy. Clinically, CDL can present with a variety of symptoms, including heavy menstrual bleeding, pelvic pain, and infertility. The coexistence of CDL and adenomyosis is exceedingly rare. This case highlights the importance of considering CDL in the differential diagnosis of pelvic mass, malignant neoplasms, and infertility, even with atypical symptoms. It also emphasizes the value of cooperation between clinicians and pathologists for accurate diagnosis and management of CDL. Adenomyosis in this case further complicated the diagnosis and highlighted the need for an index of suspicion for this rare condition.

Keywords: Adenomyosis; Case report; Cotyledonoid dissecting leiomyoma; Leiomyoma; Uterine tumor.

Fig. 1

The uterus is enlarged with a solid tissue mass measuring 5.4 cm × 5.2 cm seen at the posterior aspect of the uterus indicative of a degenerated fibroid tumor. The mass is not adherent to adjacent structures and does not infiltrate the cervix (compressing the cervical canal), and no free fluid is visualized behind the uterus.

Fig. 2

Many pieces measuring around 5. 9 cm × 5.6 cm × 2.6 cm, irregular in shape, firm in consistency, tan in color, and cut section revealing numerous hemorrhagic cysts.

Fig. 3

a. H&E, magnification X200. The sections display multiple nodules consisting of disturbed swirls of benign smooth muscle cells with marked hyalinized degeneration and hydropic changes, without atypia or atypical mitotic figures. Presence of cystic structures lined by cubodial to columnar epithelium with foci of siderophages and hemorrhage, surrounded by endometrial stroma. b. H&E, magnification X400. Hyalinized degeneration. c. CK7 immunohistochemical stain magnification X400. Positive CK7 membranous immunostaining is observed in the endometrial epithelium of the cysts. d. ER immunohistochemical stain magnification X400. Positive ER nuclear immunostaining is observed in the endometrial glands and stroma.