Uterine Angiomyolipoma: A Case Report and Review of Literature
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REVIEW ARTICLE
Uterine Angiomyolipoma: A Case Report and Review of Literature Nikita J. Mulchandani1
•
R. Vimala1
Received: 19 April 2020 / Revised: 7 June 2020 / Accepted: 12 June 2020 Ó Association of Gynecologic Oncologists of India 2020
Abstract Background Angiomyolipoma (AML), a benign mesenchymal neoplasm, typically identified in the kidney, is composed of a mixture of mature adipocytes, smooth muscle and blood vessels. While extrarenal angiomyolipomas have been reported, uterine angiomyolipomas are unusual with 27 reported cases in the literature and none from India. Methods and results We report a case of a 59-year-old woman without associated history of tuberous sclerosis who was referred to us with a diagnosis of left ovarian teratoma. Immunohistochemistry of the tumor showed cells positive for smooth muscle actin (SMA), desmin, estrogen receptor (ER), progesterone receptor (PR), CD34 and negativity for melanocytes markers which was in contrast to renal and extrarenal angiomyolipomas. Conclusion This is the first case, to the best of our knowledge, of AML arising in uterine corpus, to be reported from India. We also review the previously reported cases of uterine AML which may help us enhance our understanding of the underpinnings of pathology behing these neoplasms. Keywords Uterine angiomyolipoma Immunohistochemistry Estrogen receptor Progesterone receptor
Introduction
Case Report
Angiomyolipoma (AML) is a benign tumor composed of varying proportions of blood vessels, fat and epithelioid or spindled smooth muscle cells [1]. While an AML arising in the kidney is a common occurrence, only few cases have been reported in the female genital tract including the uterus. AMLs constitute 0.06% of all the benign uterine tumors [2, 3]. Also, unlike renal AMLs, uterine AMLs are usually not associated with tuberous sclerosis (TS) [4]. Given the rarity of the presentation of AML in the uterus, this entity has been reclassified and redefined from being reported as hamartoma by McKeithen et al. [5] to now being proposed to be acknowledged as angiolipoleiomyoma as suggested by Jabripoor et al. [6] From India, to the best of our knowledge, we report the first case of an angiomyolipoma arising in the uterine corpus.
A 59-year-old woman was referred to us with the diagnosis of a left ovarian teratoma. The magnetic resonance imaging (MRI) reports revealed a solid, 6.3 9 5.8 9 3.2 cm mass arising in the left ovary and extending anterior to the uterine body (Fig. 1a). The lesion was well defined with fatty and non-fatty components. A total abdominal hysterectomy with bilateral salpingooophorectomy was performed and was sent for frozen section diagnosis. Grossly, both the ovaries and fallopian tubes were unremarkable. However, a well-circumscribed, thinly encapsulated nodule measuring 7 9 5 9 3cm was identified arising from the myometrium in the fundus and extending up to the left adnexae (Fig. 1b). Cut surface of the nodule was firm and lobular and showed gray tan to gray yellow areas. A diagnosis of a benign
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