TY - JOUR
T1 - Sialadenoma Papilliferum of the Bronchus
T2 - An Unrecognized Bronchial Counterpart of the Salivary Gland Tumor with Frequent BRAF V600E Mutations
AU - Nakaguro, Masato
AU - Mino-Kenudson, Mari
AU - Urano, Makoto
AU - Ogawa, Ikuko
AU - Honda, Yumi
AU - Hirai, Hideaki
AU - Tanigawa, Maki
AU - Sukeda, Aoi
AU - Kajiwara, Naohiro
AU - Ohira, Tatsuo
AU - Ikeda, Norihiko
AU - Mikami, Yoshiki
AU - Tada, Yuichiro
AU - Ikeda, Jun Ichiro
AU - Matsubayashi, Jun
AU - Faquin, William C.
AU - Sadow, Peter M.
AU - Nagao, Toshitaka
N1 - Publisher Copyright:
© 2021 Wolters Kluwer Health, Inc. All rights reserved.
PY - 2021/5
Y1 - 2021/5
N2 - Sialadenoma papilliferum (SP) is a rare benign tumor of the salivary glands, and only 3 unequivocal cases of SP arising in the bronchus have been reported. We herein describe the histomorphologic and molecular features of 4 bronchial SP cases and discuss the differential diagnosis of this entity and the relationship with its clinicopathologic mimics, in particular, glandular papilloma and mixed squamous cell and glandular papilloma (GP/MP). We encountered 2 male and 2 female patients with bronchial SP (mean: 66.8 y old). All 4 tumors arose in the central bronchus and were characterized by a combination of surface exophytic endobronchial papillary proliferation and a submucosal multicystic component with complex architecture. The neoplastic epithelium consisted predominantly of nonciliated stratified columnar cells with ciliated, squamous, and mucinous cells present focally. While 2 tumors (50%) harbored a BRAF V600E mutation by molecular and immunohistochemical analysis, similar to GP/MP, no KRAS, HRAS, AKT1, or PIK3CA mutations were detected in any of the cases. Two patients were treated with limited resection, while 2 patients underwent lobectomy based on the diagnosis of adenocarcinoma or possible squamous cell carcinoma in situ in the preoperative biopsy. All survived without recurrence or metastasis for 23 to 122 months after treatment. SP can develop in the central bronchus as the bronchial counterpart of the salivary gland tumor and should be considered in the differential diagnosis of endobronchial tumors. In addition, some histologic resemblance and frequent BRAF V600E mutation raise the possibility of SP and GP/MP being on the same disease spectrum.
AB - Sialadenoma papilliferum (SP) is a rare benign tumor of the salivary glands, and only 3 unequivocal cases of SP arising in the bronchus have been reported. We herein describe the histomorphologic and molecular features of 4 bronchial SP cases and discuss the differential diagnosis of this entity and the relationship with its clinicopathologic mimics, in particular, glandular papilloma and mixed squamous cell and glandular papilloma (GP/MP). We encountered 2 male and 2 female patients with bronchial SP (mean: 66.8 y old). All 4 tumors arose in the central bronchus and were characterized by a combination of surface exophytic endobronchial papillary proliferation and a submucosal multicystic component with complex architecture. The neoplastic epithelium consisted predominantly of nonciliated stratified columnar cells with ciliated, squamous, and mucinous cells present focally. While 2 tumors (50%) harbored a BRAF V600E mutation by molecular and immunohistochemical analysis, similar to GP/MP, no KRAS, HRAS, AKT1, or PIK3CA mutations were detected in any of the cases. Two patients were treated with limited resection, while 2 patients underwent lobectomy based on the diagnosis of adenocarcinoma or possible squamous cell carcinoma in situ in the preoperative biopsy. All survived without recurrence or metastasis for 23 to 122 months after treatment. SP can develop in the central bronchus as the bronchial counterpart of the salivary gland tumor and should be considered in the differential diagnosis of endobronchial tumors. In addition, some histologic resemblance and frequent BRAF V600E mutation raise the possibility of SP and GP/MP being on the same disease spectrum.
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U2 - 10.1097/PAS.0000000000001657
DO - 10.1097/PAS.0000000000001657
M3 - Article
C2 - 33443864
AN - SCOPUS:85104209669
SN - 0147-5185
VL - 45
SP - 662
EP - 671
JO - American Journal of Surgical Pathology
JF - American Journal of Surgical Pathology
IS - 5
ER -