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腮腺嗜酸性细胞癌作为Birt-Hogg-Dube综合征的一种表现。

Oncocytic carcinoma of the parotid gland as a manifestation of Birt-Hogg-Dube syndrome.

作者信息

Chen Eric L, Siu Jason, Bastawrous Sarah, Truong Camtu D, Wu Lei

机构信息

Department of Radiology, University of Washington School of Medicine, 1959 NE Pacific St., Seattle, WA 98195, USA.

Department of Laboratory Medicine and Pathology, University of Washington School of Medicine, 1959 NE Pacific St., Seattle WA, 98195, USA.

出版信息

Radiol Case Rep. 2023 Feb 7;18(4):1536-1543. doi: 10.1016/j.radcr.2023.01.056. eCollection 2023 Apr.

DOI:10.1016/j.radcr.2023.01.056
PMID:36815148
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9939550/
Abstract

Renal oncocytomas are commonly reported in association with Birt-Hogg-Dube (BHS) syndrome, while BHD-associated oncocytomas of the parotid gland are rare. To date, there have been only 11 cases of BHD-associated parotid gland oncocytoma, without a reported case of malignant transformation. We present the first reported case of oncocytic carcinoma of the parotid gland associated with BHD, with radiologic and histologic correlation. This case establishes that BHD-associated parotid oncocytic lesions, previously identified only as benign oncocytomas in the literature to date, can undergo malignant transformation, and should potentially be regarded with a higher index of suspicion and lower threshold for aggressive management.

摘要

肾嗜酸细胞瘤通常与Birt-Hogg-Dube(BHS)综合征相关,而与BHD相关的腮腺嗜酸细胞瘤则较为罕见。迄今为止,仅有11例与BHD相关的腮腺嗜酸细胞瘤病例,尚无恶变病例的报道。我们报告首例与BHD相关的腮腺嗜酸细胞癌病例,并进行影像学和组织学对照分析。该病例证实,此前文献中仅认定为良性嗜酸细胞瘤的BHD相关腮腺嗜酸细胞性病变可发生恶变,因此对其应提高怀疑指数并降低积极治疗的阈值。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/4f315e2cdafd/gr6.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/6aec2acbd3e3/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/f16957dd4c92/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/6e6d25ac0e34/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/d68680fffc4b/gr4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/9138a133fddb/gr5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/4f315e2cdafd/gr6.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/6aec2acbd3e3/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/f16957dd4c92/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/6e6d25ac0e34/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/d68680fffc4b/gr4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/9138a133fddb/gr5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0914/9939550/4f315e2cdafd/gr6.jpg

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本文引用的文献

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Ann Dermatol. 2022 Feb;34(1):66-71. doi: 10.5021/ad.2022.34.1.66. Epub 2022 Jan 27.
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Birt-Hogg-Dubé Syndrome and Hereditary Leiomyomatosis and Renal Cell Carcinoma Syndrome: An Effective Multidisciplinary Approach to Hereditary Renal Cancer Predisposing Syndromes.Birt-Hogg-Dubé综合征与遗传性平滑肌瘤病和肾细胞癌综合征:遗传性肾癌易感综合征的有效多学科诊疗方法
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Oncocytic Lesions of Salivary Glands with Morphological and Immunohistochemical Findings.
唾液腺嗜酸性细胞病变的形态学及免疫组化研究结果
Sisli Etfal Hastan Tip Bul. 2020 Mar 24;54(1):88-93. doi: 10.14744/SEMB.2018.04935. eCollection 2020.
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FLCN alteration drives metabolic reprogramming towards nucleotide synthesis and cyst formation in salivary gland.FLCN 改变驱动唾液腺核苷酸合成和囊泡形成的代谢重编程。
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The key radiologic and cytomorphologic features of oncocytic and oncocytoid lesions of the salivary gland.唾液腺嗜酸性细胞及嗜酸性细胞样病变的关键放射学和细胞形态学特征。
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Parotid Oncocytoma as a Manifestation of Birt-Hogg-Dubé Syndrome.腮腺嗜酸性细胞瘤作为Birt-Hogg-Dubé综合征的一种表现
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