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      Diagnostic pitfall of a rare variant of angiomyolipoma, epithelioid angiomyolipoma - a case report

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          Abstract

          Angiomyolipoma of the kidney is a common benign mesenchymal neoplasm of kidney. A rare variant, epithelioid angiomyolipoma, however, may show malignant behavior. We report a case of epithelioid angiomyolipoma in a patient not having tuberous sclerosis which was initially misdiagnosed as renal cell carcinoma. A 39-year-old woman presented with a history of flank pain. Ultrasonography revealed a left renal mass. Contrast-enhanced computed tomography (CECT) abdomen revealed mass involving hilum of the kidney. On core biopsy a possibility of renal cell carcinoma was suggested. The patient underwent radical nephrectomy. After immunohistochemical analysis, a final diagnosis of epithelioid angiomyolipoma was made. Renal epithelioid angiomyolipoma without adipocytic component is extremely rare. It is pivotal to keep a possibility of epithelioid angiomyolipoma whenever an epithelioid renal tumor is encountered showing marked pleomorphism and mitosis. The use of melanocytic markers and specific markers of renal cell carcinoma will aid the diagnosis.

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          Update on the Diagnosis and Management of Renal Angiomyolipoma.

          Andrew Flum, Nabeel Hamoui, Mohammed A Said … (2016)
          Advances in minimally invasive therapies and novel targeted chemotherapeutics have provided a breadth of options for the management of renal masses. Management of renal angiomyolipoma has not been reviewed in a comprehensive fashion in more than a decade. We provide an updated review of the current diagnosis and management strategies for renal angiomyolipoma.
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            Renal angiomyolipoma: clinicopathologic study of 194 cases with emphasis on the epithelioid histology and tuberous sclerosis association.

            I. Lopez Paz, R. Lane, Linda Sercia … (2009)
            The majority of renal angiomyolipoma (AML) is sporadic and occasionally it occurs as part of tuberous sclerosis complex (TSC). Epithelioid AML (EAML), an uncommon variant, is considered potentially malignant based on anecdotal case reports. The prognostic significance of epithelioid component in an otherwise typical AML is uncertain. We studied 194 AMLs for the clinicopathologic features of epithelioid and TSC-associated AMLs. Epithelioid component was present in 15 cases (7.7%) with an average amount of 51% (range: 10% to 100%). Histologically, the epithelioid tumor cells were categorized into small, intermediate, and large cell type based on the cell size. Worrisome histologic features were seen in many EAMLs, including coagulative tumor necrosis in 27% (4/15), nuclear atypia in 93% (14/15), mitosis in 47% (7/15), and atypical mitosis in 1 case. All 15 EAML patients had a mean follow-up time of 5.1 years and none had local recurrence or distant metastasis. Sixteen (8.2%) AMLs occurred in patients with definitive TSC. Three histologic features, namely microscopic AML foci, epithelioid component, and epithelial cysts, were present in 10 (62.5%), 4 (25%), and 44% (7/16), respectively, of TSC-associated AMLs, compared with 11 (6.2%), 11 (6.2%), and 6 (3.4%), respectively, in non-TSC-associated AMLs (P value all <0.01). In summary, all 15 cases of EAMLs in our study had benign clinical outcomes despite adverse pathologic features. Epithelioid component, epithelial cysts, and microscopic AML foci are strongly associated with TSC and the presence of all 3 features should raise strong suspicion for TSC.
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              Epithelioid angiomyolipoma of the kidney: pathological features and clinical outcome in a series of consecutively resected tumors.

              Wenlei He, John Cheville, Peter M. Sadow … (2013)
              The 2004 World Health Organization classification of tumors defines epithelioid angiomyolipoma of kidney as a potentially malignant mesenchymal neoplasm with reported metastasis in approximately one-third of the cases. However, this conclusion was based primarily on individual case reports and small retrospective series. More recently reported larger series have shown varying results. We reviewed 437 consecutive renal angiomyolipomas with primary resection at three tertiary-care institutions with high nephrectomy volumes. Only tumors showing >80% epithelioid histology were included in this study. Tumors resected elsewhere and reviewed in consultation were not included. Twenty of these 437 (4.6%) were classified as epithelioid angiomyolipoma. The female to male ratio was 11:9, mean age 49.7 (range, 30-80) years, and mean tumor size 8.7 (range, 1-25) cm. Microscopic tumor necrosis was present in 10 (50%) tumors and mitotic activity (range, <1-5/10 high power fields) in 8 (40%); atypical mitoses were seen in only 1 (5%) tumor. Pleomorphic ganglion-like or multinucleated giant cells were seen in 18 (90%) tumors. With a mean follow-up of 82.5 (range, 1-356) months, seventeen patients were alive with no-evidence-of-disease at the time of last follow-up; two patients died of unrelated causes with no-evidence-of-disease, and one patient (5%) developed distant metastases. Our data, based on consecutively resected angiomyolipomas with long clinical follow-up, suggests that epithelioid angiomyolipomas constitute a small proportion of all angiomyolipomas, and the rate of aggressive behavior among epithelioid angiomyolipomas, even when showing morphologic features previously reported to portend aggressive clinical behavior, is very low.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Pan Afr Med J
                Journal ID (iso-abbrev): Pan Afr Med J
                Journal ID (publisher-id): PAMJ
                Title: The Pan African Medical Journal
                Publisher: The African Field Epidemiology Network
                ISSN (Electronic): 1937-8688
                Publication date (Electronic): 02 November 2020
                Publication date Collection: 2020
                Volume: 37
                Electronic Location Identifier: 210
                Affiliations
                [1 ]Department of Pathology and Laboratory Medicine, All India Institute of Medical Sciences, Rishikesh, India,
                [2 ]Department of Urology, All India Institute of Medical Sciences, Rishikesh, India
                Author notes
                Corresponding author: Rishabh Sahai, Department of Pathology and Laboratory Medicine, All India Institute of Medical Sciences, Rishikesh, India. rishabh.sahai90@ 123456gmail.com
                Article
                Publisher ID: PAMJ-37-210
                DOI: 10.11604/pamj.2020.37.210.26269
                PMC ID: 7813654
                PubMed ID: 33505578
                SO-VID: da9db902-d17e-4fc5-bd22-2c5f7f17d020
                Copyright © Copyright: Michael Leonard Anthony et al.
                License:

                The Pan African Medical Journal (ISSN: 1937-8688). This is an Open Access article distributed under the terms of the Creative Commons Attribution International 4.0 License ( https://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 26 September 2020
                Date accepted : 13 October 2020
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Medicine
                Keywords: epithelioid cells,perivascular,pecoma,renal cell carcinoma
                Data availability:
                ScienceOpen disciplines: Medicine
                Keywords: epithelioid cells, perivascular, pecoma, renal cell carcinoma

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