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The Korean Journal of Pathology 2008; 42: 123-4

Adenomyoma of Endocervical Type in a First-Trimester Gravid Uterine Corpus - A Brief Report -

Hyunchul Kim∙Hoiseon Jeong Uterine adenomyoma is a that is composed of endometrial glands and smooth Sangho Lee∙Young Seok Lee muscle, and this is found in the uterine corpus; however, the glandular component is occa- Bong Kyung Shin∙Insun Kim sionally of mucin-secreting endocervical type. We describe here a case of uterine adenomy- oma of the endocervical type, and this presented in a 38-year-old pregnant woman. Department of Pathology, College of Medicine, Korea University, Seoul, Korea

Received : December 11, 2006 Accepted : October 29, 2007

Corresponding Author Insun Kim, M.D. Department of Pathology, Korea University Guro Hospital, 80 Guro-dong, Guro-gu, Seoul 152-703, Korea Tel: 02-2626-1472 Fax: 02-2626-1482 E-mail: [email protected] Key Words : Adenomyoma; Cervix uteri; Myometrium

Uterine adenomyoma is a neoplasm that’s composed of glan- the largest measured 6.5 cm in diameter. There was an implan- dular and components, usually found in the uter- tation site in the fundic portion of the endometrium. Addition- ine corpus, and only rarely seen in the uterine cervix.1 A similar ally, a relatively well demarcated lesion, 1.8 cm in size and con- tumor composed of endocervical glands and smooth muscle, sisting of small cystic spaces was found in the myometrium of designated as ‘adenomyoma of the endocervical type’ has been the uterine corpus, and it protruded into the endometrial cavity reported in the uterine corpus.2 Here we report on a case of ade- like an endometrial polyp (Fig. 1). Histologically, the cystic mass nomyoma of the uterine corpus, and the adenomyoma was entire- was composed of variable-sized glandular components with inter- ly lined with endocervical type glandular epithelium. Further vening myomatous smooth muscle bundles (Fig. 2A, C). The this tumor was associated with multiple and ade- lining epithelium of the glandular components was mucin- nomyosis in a pregnant woman. secreting tall columnar epithelium of the endocervical type and it showed a positive reaction for periodic acid Schiff stain (Fig. 2B). The epithelial cells showed neither cytological atypia nor CASE REPORT increased mitotic figures. was observed in the adja- cent myometrium. A 38-year-old woman (gravida 5, para 2) was referred to the Department of Gynecology of Korea University Medical Cen- ter; she was at a gestational age of 6 weeks. Ultrasound exami- DISCUSSION nation showed enlargement of her with numerous large masses within the uterine corpus. Total hysterectomy was per- Uterine adenomyoma is not rare, but it is uncommon benign formed because her pregnancy could not be maintained. The lesion of the uterine corpus and cervix. The histogenesis of uter- removed uterus showed numerous intramural leiomyomas, and ine adenomyoma is not yet clearly understood; however, the rela-

123 124 Hyunchul Kim∙Hoiseon Jeong∙Sangho Lee, et al.

B

A C

Fig. 1. A portion of the uterus shows an area of mucus-containing Fig. 2. (A) Low magnification view shows variable-sized, cystic cystic lesion in the myometrium with attached endometrial poly- glands with intervening myomatous smooth muscle bundles. (B) poid mass (arrowheads). There are also an implantation site The gland shows mucinous endocervical type epithelium with (arrow) and several myomas. PAS-positive material. (C) The mucinous gland is intimately abut- ted by smooth muscle. tionship between adenomyoma and adenomyosis has been sug- gested in the medical literature.3,4 It is not definite if they are but they can show variable changes in their appearances. This or circumscribed variants of adenomyosis. Adenomy- case is one of the various examples of adenomyoma, and we feel oma is composed of endometrial glands and neoplastic changed it is interesting enough to be shared by this report. smooth muscle, in contrast to the endometrial glands and stro- ma that are separated by normal myometrium in adenomyosis. These tumors may occur in submucosal, intramural and sub- REFERENCES serosal locations, and they macroscopically appear as solid, solid and cystic, or cystic tumors.1 One recent study4 reported that 7 1. Gilks CB, Young RH, Clement PB, Hart WR, Scully RE. Adenomy- and 9 cases of 26 uterine adenomyomas had concomitant ade- omas of the uterine cervix of endocervical type: a report of ten cases nomyosis and leiomyomas, respectively. Although there has been of a benign cervical tumor that may be confused with adenoma no documented relationship between adenomyoma and preg- malignum [corrected]. Mod Pathol 1996; 9: 220-4. nancy, the hormonal change of pregnancy might exaggerate this 2. Gilks CB, Clement PB, Hart WR, Young RH. Uterine adenomyomas lesion. Ten cases of adenomyoma of the endocervical type have excluding atypical polypoid adenomyomas and adenomyomas of been described in the uterine cervix by Gilks et al.2 and there endocervical type: a clinicopathologic study of 30 cases of an under- has been one additional case by Kuwabara et al.5 Minor foci of emphasized lesion that may cause diagnostic problems with brief the endocervical type glands were found in 2 of the 30 uterine consideration of adenomyomas of other female genital tract sites. adenomyomas reported by Gilks et al.1 The tumor of our case Int J Gynecol Pathol 2000; 19: 195-205. was centered within the myometrium with a portion of it pro- 3. Silverberg SG. Adenomyomatosis of endometrium and endocervix- truding into the endometrial cavity in a polyp-like manner, and a hamartoma. Am J Clin Pathol 1975; 64: 192-9. it showed a predominantly cystic configuration. Because muci- 4. Tahlan A, Nanda A, Mohan H. Uterine adenomyoma: a clinico- nous change of the endometrium is not an uncommon metaplas- pathologic review of 26 cases and a review of the literature. Int J tic process, adenomyomas of the endocervical type in the uterine Gynecol Pathol 2006; 25: 361-5 corpus may be the possible result of the metaplastic change of 5. Kuwabara H, Ohno M, Moriwaki S. Uterine adenomyoma of endo- adenomyomas with endometrial type gland. cervical type. Pathol Int 1999; 49: 1019-21. Adenomyomas are sometimes overlooked by the pathologists,