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一名10岁男孩的支气管乳头状涎腺瘤

Bronchial Sialadenoma Papilliferum in a 10-Year-Old Boy.

作者信息

Guo Shunhua, Schmitt Alessandra Ceolin, Lewis James S, Lo Ying-Chun, Rumilla Ande M, Tazelaar Henry D

机构信息

Department of Pathology and Laboratory Medicine, Indiana University School of Medicine, Indianapolis, IN, USA.

Department of Laboratory Medicine and Pathology, Mayo Clinic, Scottsdale, AZ, USA.

出版信息

Int J Surg Pathol. 2025 May;33(3):765-771. doi: 10.1177/10668969241283482. Epub 2024 Oct 3.

Abstract

Sialadenoma papilliferum (SP) is a rare salivary gland tumor mostly reported in the oral cavity. Here we describe a bronchial SP in the left upper lobe bronchus of a 10-year-old boy. At bronchoscopy, a well-circumscribed polypoid lesion protruding into the bronchial lumen was identified. The tumor was excised, but eventually, the patient had to undergo a sleeve resection after 2 recurrences. Pathology revealed a papillocystic lesion with exophytic and endophytic components. The cells lining the exophytic surface and papillary structures were columnar and squamous, and the cells lining endophytic cystic and papillary structures were cuboidal to columnar, all of which were diffusely reactive with antibodies to SOX10 protein. The presence of basal cells was demonstrated by p63 immunoreactivity. The cells failed to immunohistochemically express BRAF V600E. Fluorescence in situ hybridization analysis revealed no or gene rearrangement. The patient is alive 24 years after resection with no additional recurrences. Bronchial SP needs to be recognized and distinguished from other benign and malignant salivary gland and pulmonary neoplasms so that patients can receive appropriate treatment and follow-up.

摘要

乳头状唾液腺瘤(SP)是一种罕见的涎腺肿瘤,大多报道于口腔。本文描述了一名10岁男孩左上叶支气管的支气管SP。在支气管镜检查时,发现一个边界清晰的息肉样病变突入支气管腔。肿瘤被切除,但最终,患者在两次复发后不得不接受袖状切除术。病理显示为一个具有外生性和内生性成分的乳头状囊性病变。外生性表面和乳头状结构内衬的细胞为柱状和鳞状,内生性囊性和乳头状结构内衬的细胞为立方状至柱状,所有这些细胞均与SOX10蛋白抗体呈弥漫性反应。p63免疫反应性证实了基底细胞的存在。这些细胞在免疫组织化学上未表达BRAF V600E基因。荧光原位杂交分析未发现 或 基因重排。患者在切除术后24年仍存活,无额外复发。支气管SP需要被识别并与其他良性和恶性涎腺及肺部肿瘤相鉴别,以便患者能够接受适当的治疗和随访。

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