TY - JOUR
T1 - Salivary gland polymorphous adenocarcinoma
T2 - Clinicopathological features and gene alterations in 36 Japanese patients
AU - Fukumura, Masahiro
AU - Ishibashi, Kenichiro
AU - Nakaguro, Masato
AU - Nagao, Toshitaka
AU - Saida, Kosuke
AU - Urano, Makoto
AU - Tanigawa, Maki
AU - Hirai, Hideaki
AU - Yagyuu, Takahiro
AU - Kikuchi, Kentaro
AU - Yada, Naomi
AU - Sugita, Yoshihiko
AU - Miyabe, Megumi
AU - Hasegawa, Shogo
AU - Goto, Mitsuo
AU - Yamamoto, Hidetaka
AU - Ohuchi, Tomoyuki
AU - Kusafuka, Kimihide
AU - Ogawa, Ikuko
AU - Suzuki, Hiroaki
AU - Notohara, Kenji
AU - Shimoda, Masayuki
AU - Tada, Yuichiro
AU - Kirita, Tadaaki
AU - Takata, Takashi
AU - Morinaga, Shojiroh
AU - Maeda, Hatsuhiko
AU - Warnakulasuriya, Saman
AU - Miyabe, Satoru
AU - Nagao, Toru
N1 - Publisher Copyright:
© 2022 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.
PY - 2022/9
Y1 - 2022/9
N2 - Background: Polymorphous adenocarcinoma is a common intraoral minor salivary gland carcinoma in Western countries but is extremely rare in Japan. The current study aimed to characterize the clinicopathological features and status of molecular alterations of polymorphous adenocarcinoma-associated genes, such as PRKD1/2/3, ARID1A, and DDX3X, in a large cohort of Japanese patients with polymorphous adenocarcinoma. Methods: We examined the cases of 36 Japanese patients with salivary gland polymorphous adenocarcinoma and 26 cases involving histopathological mimics. To detect gene splits, fluorescence in situ hybridization was carried out for polymorphous adenocarcinoma-associated genes. Additionally, we applied a SNaPshot multiplex assay to identify PRKD1 hotspot mutations. Results: This study revealed the indolent clinical course of polymorphous adenocarcinoma with a high 10-year overall survival rate (92.9%), accompanied by occasional local recurrences and cervical lymph node metastasis (23.3%). Twenty cases (55.6%) of polymorphous adenocarcinoma (but none of the mimics) exhibited alterations in at least one polymorphous adenocarcinoma-associated gene. Rearrangement of polymorphous adenocarcinoma-associated genes and PRKD1 E710D were identified in 17 (47.2%) and 4 (11.1%) cases, respectively; one case showed coexisting PRKD3 split and PRKD1 E710D. In the multivariate analysis, high clinical stage (p = 0.0005), the presence of prominent nucleoli (p = 0.0003), and ARID1A split positivity (p = 0.004) were independent risk factors for disease-free survival. Conclusion: Japanese patients with polymorphous adenocarcinoma showed clinicopathological features similar to those reported in Western countries. This study disclosed that polymorphous adenocarcinoma-associated genetic alterations were common and specific findings in polymorphous adenocarcinomas. The diagnostic role and possible prognostic significance of polymorphous adenocarcinoma-associated genetic alterations in polymorphous adenocarcinomas were suggested.
AB - Background: Polymorphous adenocarcinoma is a common intraoral minor salivary gland carcinoma in Western countries but is extremely rare in Japan. The current study aimed to characterize the clinicopathological features and status of molecular alterations of polymorphous adenocarcinoma-associated genes, such as PRKD1/2/3, ARID1A, and DDX3X, in a large cohort of Japanese patients with polymorphous adenocarcinoma. Methods: We examined the cases of 36 Japanese patients with salivary gland polymorphous adenocarcinoma and 26 cases involving histopathological mimics. To detect gene splits, fluorescence in situ hybridization was carried out for polymorphous adenocarcinoma-associated genes. Additionally, we applied a SNaPshot multiplex assay to identify PRKD1 hotspot mutations. Results: This study revealed the indolent clinical course of polymorphous adenocarcinoma with a high 10-year overall survival rate (92.9%), accompanied by occasional local recurrences and cervical lymph node metastasis (23.3%). Twenty cases (55.6%) of polymorphous adenocarcinoma (but none of the mimics) exhibited alterations in at least one polymorphous adenocarcinoma-associated gene. Rearrangement of polymorphous adenocarcinoma-associated genes and PRKD1 E710D were identified in 17 (47.2%) and 4 (11.1%) cases, respectively; one case showed coexisting PRKD3 split and PRKD1 E710D. In the multivariate analysis, high clinical stage (p = 0.0005), the presence of prominent nucleoli (p = 0.0003), and ARID1A split positivity (p = 0.004) were independent risk factors for disease-free survival. Conclusion: Japanese patients with polymorphous adenocarcinoma showed clinicopathological features similar to those reported in Western countries. This study disclosed that polymorphous adenocarcinoma-associated genetic alterations were common and specific findings in polymorphous adenocarcinomas. The diagnostic role and possible prognostic significance of polymorphous adenocarcinoma-associated genetic alterations in polymorphous adenocarcinomas were suggested.
KW - cribriform adenocarcinoma of salivary gland
KW - gene fusion
KW - polymorphous adenocarcinoma
KW - prognosis
KW - salivary gland neoplasms
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U2 - 10.1111/jop.13336
DO - 10.1111/jop.13336
M3 - Article
C2 - 35880805
AN - SCOPUS:85135866563
SN - 0904-2512
VL - 51
SP - 710
EP - 720
JO - Journal of Oral Pathology and Medicine
JF - Journal of Oral Pathology and Medicine
IS - 8
ER -