title: An unusual case of uterine cotyledonoid dissecting leiomyoma with adenomyosis.
abstract: BACKGROUND
  Cotyledonoid dissecting leiomyoma is a rare variant of uterine smooth muscle tumor with an unusual growth pattern that shows intramural dissection within uterine myometrium and often a placenta-like appearance in its extrauterine components.
  
  
  METHODS
  We present a unique case of cotyledonoid dissecting leiomyoma with adenomyosis. A 40-year-old Japanese female presented with prolonged menorrhagia and severe anemia. She had a pelvic mass followed-up for 6 years with a diagnosis of leiomyoma. However, increase in tumor size and cystic changes with hemorrhage were found by magnetic resonance imaging, and total abdominal hysterectomy with bilateral salpingectomy was performed. Macroscopically, the placenta-like exophytic mass protruding from the posterior uterine wall was composed of multiple nodules containing numerous hemorrhagic cysts. The mass showed continuity as a white multinodular dissecting mass infiltrating the posterolateral myometrium. Microscopically, both extra-and intrauterine portions of the mass were composed of nodules that contained swirled neoplastic smooth muscle cells with marked hyalinized degeneration, as observed in cotyledonoid dissecting leiomyomas of conventional type. In addition, numerous non-neoplastic glands of endometrial type surrounded by abundant endometrium-like stromal cells and non-neoplastic smooth muscle cells were found in the tumor, suggesting that it involved a part of concomitant adenomyosis originating from the nontumoral myometrium.
  
  
  CONCLUSIONS
  Thus far, over 30 cases of cotyledonoid dissecting leiomyoma have been reported, none of which have described the presence of adenomyosis within the tumor. The present case suggested that cotyledonoid dissecting leiomyoma might have a unique clinical presentation involving concomitant uterine adenomyosis. It is critical for pathologists, gynecologists, and radiologists to be cognizant of cotyledonoid dissecting leiomyoma variants for timely and appropriate diagnosis and treatment.
authors.affiliation: Department of Pathology, KKR Sapporo Medical Center, 6-3-40, Ichijyo, Hiragishi, Toyohira-ku, Sapporo, Hokkaido, 062-0931, Japan.
authors.forename: Ai
authors.initials: A
authors.lastname: Shimizu
authors.affiliation: Department of Gynecology, KKR Sapporo Medical Center, 6-3-40, Ichijyo, Hiragishi, Toyohira-ku, Sapporo, Hokkaido, 062-0931, Japan.
authors.forename: Hoshihito
authors.initials: H
authors.lastname: Tanaka
authors.affiliation: Department of Pathology, KKR Sapporo Medical Center, 6-3-40, Ichijyo, Hiragishi, Toyohira-ku, Sapporo, Hokkaido, 062-0931, Japan. sarikano@med.hokudai.ac.jp.
authors.forename: Sari
authors.identifier: http://orcid.org/0000-0002-2075-1122
authors.initials: S
authors.lastname: Iwasaki
authors.affiliation: Department of Gynecology, KKR Sapporo Medical Center, 6-3-40, Ichijyo, Hiragishi, Toyohira-ku, Sapporo, Hokkaido, 062-0931, Japan.
authors.forename: Yukio
authors.initials: Y
authors.lastname: Wakui
authors.affiliation: Department of Pathology, KKR Sapporo Medical Center, 6-3-40, Ichijyo, Hiragishi, Toyohira-ku, Sapporo, Hokkaido, 062-0931, Japan.|Department of Physical Therapy, Faculty of Human Science, Hokkaido Bunkyo University, 196-1, 5-chome, Koganechuo, Eniwa, 061-1449, Japan.
authors.forename: Hitoshi
authors.initials: H
authors.lastname: Ikeda
authors.affiliation: Department of Pathology, KKR Sapporo Medical Center, 6-3-40, Ichijyo, Hiragishi, Toyohira-ku, Sapporo, Hokkaido, 062-0931, Japan.
authors.forename: Akira
authors.initials: A
authors.lastname: Suzuki
mesh_terms: D062788:Adenomyosis / Q000175:diagnosis* / Q000000981:diagnostic imaging / Q000473:pathology; D000328:Adult; D004717:Endometrium / Q000473:pathology; D005260:Female; D006801:Humans; D007044:Hysterectomy; D007889:Leiomyoma / Q000175:diagnosis* / Q000000981:diagnostic imaging / Q000473:pathology; D008279:Magnetic Resonance Imaging; D008595:Menorrhagia; D018235:Smooth Muscle Tumor / Q000175:diagnosis* / Q000000981:diagnostic imaging / Q000473:pathology; D014594:Uterine Neoplasms / Q000175:diagnosis* / Q000000981:diagnostic imaging / Q000473:pathology; D014599:Uterus / Q000473:pathology
keywords: Adenomyoma; Adenomyosis; Cotyledonoid dissecting leiomyoma; Leiomyoma of the uterus