Atypical polypoid adenomyoma of the uterus. A case report and a review of the literature

Atypical polypoid adenomyoma (APA) is a rare, benign lesion. The tumor occurs in nulliparous women aged 22-48 years (average 33 years) and it has been suggested as being related to prolonged estrogenic stimulation. We describe a case of a 72-year-old woman who presented at our hospital with persistent, worsening urinary incontinence and pelvic pain. Physical examination and pelvic ultrasound disclosed uterine enlargement, a mass in the endometrial cavity and multiple small myomas. Total hysterectomy with bilateral salpingo-oophorectomy was performed. The histological diagnosis for the mass of the endometrial cavity was atypical polypoid adenomyoma. APA should be distinguished from endometrial carcinoma and other malignant uterine neoplasms such as adenofibroma, adenosarcoma and malignant mixed mullerian tumor. The immunohistochemical panel which usually includes alpha smooth muscle actin, desmin, Ki67 and recently CD I 0 is often helpful in establishing the diagnosis. The treatment may vary depending on the patient's age, her desire to preserve fertility, and the severity of her symptoms.

Uterus; Atypical polypoid adenomyoma; Endometrial stromal cells; Immunohistochemistry

[1] Mazur M.T.: “Atypical polypoid adenomyomas of the endometrium”. Am. J. Surg. Pathol., 1981, 5, 473.

[2] Young R.H., Treger T., Scully R.E.: “Atypical polypoid adenomyomas of the uterus. A report of 27 cases”. Am. J. Clin. Pathol., 1986, 86, 139.

[3] Clement P.B., Young R.H.: “Atypical polypoid adenomyoma of the uterus associated with Turner's syndrome. A report of three cases, including a review of “estrogen-associated” endometrial neoplasms and neoplasms associated with Turner's syndrome”. Int. J. Gynecol. Pathol., 1987, 6, 104.

[4] Lee K.R.: “Atypical polypoid adenomyoma of the endometrium associated with adenomyomatosis and adenocarcinoma”. Gynecol. Oncol., 1993, 51, 416.

[5] Savelli L., De Iaco P., Santini D., Rosati F., Ghi T., Pignotti E., Bovicelli L.: “Histopathologic features and risk factors for benignity, hyperplasia and cancer in endometrial polyps”. Am. J. Obstet. Gynecol., 2003, 188, 927.

[6] Fukunaga M., Endo Y., Ushigome S., Ishikawa E.: “Atypical polypoid adenomyomas of the uterus”. Histopathology, 1995, 27, 35.

[7] Kuwashima Y., Uehara T., Kurosumi M., Kishi K., Ohno H., Maruyama M., Takayama S.: “Atypical polypoid adenomyoma of the uterus in a very old woman: a report of a case with immunohistochemical characterization of its stromal components and proliferative status”. Eur. J. Gynaecol. Oncol., 1995, 16, 115.

[8] Heatley M.K.: “Atypical polypoid adenomyoma: A systematic review of the English literature”. Histopathology, 2006, 48, 609.

[9] Rollason T.P., Redman C.W.: “Atypical polypoid adenomyoma -clinical, histological and immunohistochemical findings”. Eur. J Gynecol. Oncol., 1988, 9, 444.

[10] Longacre T.A., Chung M.H., Rouse R.V., Hendrickson M.R.: “Atypical polypoid adenomyofibromas (atypical polypoid adenomyomas) of the uterus: a clinicopathologic study of 55 cases”. Am. J. Surg. Pathol., 1996, 201, 1.

[11] Wong A.Y., Chan K.S., Law W.L., Tang L.C.: “Pregnancy outcome of a patient with atypical polypoid adenomyoma”. Fertil. Steril., 2007, 88, 1438.e7.

[12] Sugiyama T., Ohta S., Nishida T., Okura N., Tanabe K., Yakushiji M.: “Two cases of endometrial adenocarcinoma arising from atypical polypoid adenomyoma”. Gynecol. Oncol., 1998, 71, 141.

[13] Mittal K.R., Peng X.C., Wallach R.C., Demopoulos R.I.: “Coexistent atypical polypoid adenomyoma and endometrial adenocarcinoma”. Hum. Pathol., 1995, 26, 574.

[14] Soslow R.A., Chung M.H., Rouse R.V., Hendrickson M.R., Longacre T.A.: “Atypical polypoid adenofibroma/APA versus welldiffentiated endometrial carcinoma with prominent stromal matrix: an immunohistochemical study”. Int. J. Gynecol. Pathol., 1996, 15, 209.

[15] Di Palma S., Santini D., Martinelli G.: “Atypical polypoid adenomyoma of the uterus. An immunohistochemical study of a case”. Tumori, 1989, 75, 292.

[16] McCluggage W.G., Sumathi V.P., Maxwell P.: “CD10 is a sensitive and diagnostically useful immunohistochemical marker of normal endometrial stroma and of endometrial stromal neoplasms”. Histopathology, 2001, 39, 273.

[17] Srodon M., Klein W.M., Kurman R.J.: “CD10 imunostaining does not distinguish endometrial carcinoma invading myometrium from carcinoma involving adenomyosis”. Am. J. Surg. Pathol., 2003, 27, 786.

[18] Ohishi Y., Kaku T., Kobayashi H., Aishima S., Umekita Y., Wake N. et al.: “CD10 immunostaining distinguishes atypical polypoid adenomyofibroma (atypical polypoid adenomyoma) from endometrial carcinoma invading the myometrium”. Hum. Pathol., 2008, 39, 1446.

[19] Nascimento A.F., Hirsch M.S., Cviko A., Quade B.J., Nucci M.R.: “The role of CD10 staining in distinguishing invasive endometrial adenocarcinoma from adenocarcinoma involving adenomyosis”. Mod. Pathol., 2003, 16, 22.

[20] Ota S., Catasus L., Matias-Guiu X., Bussaglia E., Lagarda H., Pons C. et al.: “Molecular pathology of atypical polypoid adenomyoma of the uterus”. Hum. Pathol., 2003, 34, 784.

[21] Vilos G.A., Ettler H.C.: “Atypical polypoid adenomyoma and hysteroscopic endometrial ablation”. J. Obstet. Gynaecol. Can., 2003, 25, 760.