Abstract
Angiomyolipoma (AML) is a mesenchymal tumor commonly found in the kidneys. Extra-renal AML is uncommon and especially rare in the nasal cavity. To our knowledge, fewer than five cases of nasal AML are reported in the United States. We present a case of nasal AML in a 65-year-old man with a history of rhinosinusitis and obstruction of the left nasal cavity. The lesion comprised of admixed spindled smooth muscle cells, mature adipose tissue, and thick-walled blood vessels. Smooth muscle differentiation of the spindled cells was confirmed by expression of smooth muscle actin. Surprisingly, melanocytic markers, such as HMB45, were negative. The histologic features and immunoprofile suggest that nasal AML is pathologically distinct from neoplastic AMLs/perivascular epithelioid cell tumors (PEComas) that typically occur in the kidney. We propose that nasal AML is a hamartomatous lesion rather than neoplastic. Although AML is rare in the nasal cavity, it should be considered in the differential diagnosis of clinically benign nasal masses.
Similar content being viewed by others
References
Hornick JL, Fletcher CDM. PEComa: what do we know so far? Histopathology. 2006;48:75–82.
McCullough DL, Scott R, Seybold HM. Renal angiomyolipoma (hamartoma): review of the literature and report of 7 cases. J Urol. 1971;105:32–44.
Tsui WM, Colombari R, Portmann BC, Bonetti F, Thung SN, Ferrell LD, et al. Hepatic angiomyolipoma: a clinicopathologic study of 30 cases and delineation of unusual morphologic variants. Am J Surg Pathol. 1999;23:34–48.
Val-Bernal JF, Mira C. Cutaneous angiomyolipoma. J Cutan Pathol. 1996;23:364–8.
Shimizu M, Manabe T, Tazelaar HD, Hirokawa M, Moriya T, Ito J, et al. Intramyocardial angiomyolipoma. Am J Surg Pathol. 1994;18:1164–9.
Ide F, Shimoyama T, Horie N. Angiomyolipomatous hamartoma of the tongue. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1998;85:581–4.
Ameen S-A, Salina H, Zahedi F-D, Primuharsa-Putra S-H-A, Masir N. Nasal angiomyolipoma (AML) mimicking juvenile nasopharyngeal angiofibroma. Iran J Otorhinolaryngol. 2019;31:191–5.
Aleem MA, Fatima A, Kumudachalam P, Priyadarshini R. Nasal angiomyolipoma: report of two cases of an extremely rare entity. Indian J Pathol Microbiol. 2017;60:393–5.
Cibas ES, Goss GA, Kulke MH, Demetri GD, Fletcher CD. Malignant epithelioid angiomyolipoma (‘sarcoma ex angiomyolipoma’) of the kidney: a case report and review of the literature. Am J Surg Pathol. 2001;25:121–6.
Mao J-X, Teng F, Liu C, Yuan H, Sun K-Y, Zou Y, et al. Two case reports and literature review for hepatic epithelioid angiomyolipoma: pitfall of misdiagnosis. World J Clin Cases. 2019;7:972–83.
Garoufalia Z, Machairas N, Kostakis ID, Liakea A, Tsaparas P, Liapis G, et al. Malignant potential of epithelioid angiomyolipomas of the liver: a case report and comprehensive review of the literature. Mol Clin Oncol. 2018;9:226–30.
Flum AS, Hamoui N, Said MA, Yang XJ, Casalino DD, McGuire BB, et al. Update on the diagnosis and management of renal angiomyolipoma. J Urol. 2016;195:834–46.
Iwata AJ, Friedmann DR, Kaplan J, Wang BY, Lebowitz RA. A man with recurrent right-sided epistaxis. Angiomyolipoma (AML) of the nasal cavity. JAMA Otolaryngol Head Neck Surg. 2013;139:1067–8.
Watanabe K, Suzuki T. Mucocutaneous angiomyolipoma. A report of 2 cases arising in the nasal cavity. Arch Pathol Lab Med. 1999;123:789–92.
Taneja R, Singh DV. Giant renal angiomyolipoma: unusual cause of huge abdominal mass. J Clin Imaging Sci. 2013;3:56.
Pandey V, Khatib Y, Gupte P, Pandey R, Khare MS. Monotypic angiomyolipoma of the nasal cavity: an extremely rare cause of nasal mass with recurrent epistaxis. Indian J Pathol Microbiol. 2020;63:106–8.
Banerjee SS, Eyden B, Trenholm PW, Sheikh MY, Wakamatsu K, Ancans J, et al. Monotypic angiomyolipoma of the nasal cavity: a heretofore undescribed occurrence. Int J Surg Pathol. 2001;9:309–15.
Wenig BM. Recently described sinonasal tract lesions/neoplasms: considerations for the new world health organization book. Head Neck Pathol. 2014;8:33–41.
Willén R, Gad A, Willén H. Lipomatous lesions of the uterus. Virchows Arch A Pathol Anat Histol. 1978;377:351–61.
Ahn SH, Lee YJ, Kim CH, Chang JH. Angiomyolipoma of the nasal cavity resected with preoperative angio-embolization. Korean J Otorhinolaryngol-Head and Neck Surg. 2013;56:528–31.
Ibáñez Rodríguez JA, Segura Sánchez J, Gómez Galán MJ, García-Eloy Carrasco C. Nasal angiomyolipoma. Acta Otorrinolaringol Esp. 2014;65:205–7.
Moreira MD, Lessa MM, Lima CMF de, Lessa HA, Fonseca Júnior LE. Angiomyolipoma of the nasal cavity. Braz J Otorhinolaryngol. 2011;77:269.
Erkiliç S, Koçer NE, Mumbuç S, Kanlikama M. Nasal angiomyolipoma. Acta Otolaryngol. 2005;125:446–8.
Go JH. Angiomyolipoma of the nasal cavity. Korean J Pathol. 2005;39(4):284–6.
Tardío JC, Martín-Fragueiro LM. Angiomyolipoma of the nasal cavity. Histopathology. 2002;41:174–5.
Gatalica Z, Lowry LD, Petersen RO. Angiomyolipoma of the nasal cavity: case report and review of the literature. Head Neck. 1994;16:278–81.
Güezmes A, Mazorra F, García-Mantilla F, Eizaguirre MJ, Ondiviela R. Nasal angiomyolipoma. Acta Otorrinolaringol Esp. 1990;41:341–2.
Dawlatly EE, Anim JT, el-Hassan AY. Angiomyolipoma of the nasal cavity. J Laryngol Otol. 1988;102:1156–8.
Author information
Authors and Affiliations
Corresponding author
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Wang, L., Leng, B. & Liu, L. Angiomyolipoma of the Nasal Cavity: A Distinct Entity. A Case Report and Literature Review. Head and Neck Pathol 15, 709–714 (2021). https://doi.org/10.1007/s12105-020-01224-1
Received:
Revised:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12105-020-01224-1