PMID- 36147445 OWN - NLM STAT- PubMed-not-MEDLINE LR - 20220924 IS - 1687-8450 (Print) IS - 1687-8469 (Electronic) IS - 1687-8450 (Linking) VI - 2022 DP - 2022 TI - Unicystic Mucoepidermoid Carcinoma: A Pitfall for Clinical and Pathologic Diagnosis. PG - 2676367 LID - 10.1155/2022/2676367 [doi] LID - 2676367 AB - Unicystic mucoepidermoid carcinoma (UC-MEC) is a rare MEC variant, and its diagnosis is frequently problematic. This study is aimed at summarizing its clinicopathologic characteristics, treatment, and prognosis and proposing key points to avoid missed diagnosis and misdiagnosis in clinical and pathological conditions. This retrospective study included 30 UC-MEC cases, and the clinical findings were collected from the clinical medical records. Radiographic features, histologic behaviors, MAML2 rearrangement by fluorescence in situ hybridization (FISH), and follow-up data were analyzed. Moreover, glandular odontogenic cyst (GOC) and cytadenoma (CA) were used as controls. In the UC-MEC group, 19 patients were female (63%), and 11 were male (37%). The mean patient age was 39.5 (range, 7-72 years). The affected locations included the jaw (8 maxillary, 3 mandibular) and salivary glands (7 parotid, 11 palates, and 1 floor of the mouth). The chief complaint was swelling; the lesions were all cystic, among which 66.7% were well circumscribed and 33.3% poorly defined. Microscopic examination showed two UC-MEC histologic subtypes. Type A presented as a single cyst with mural thickening (8/30, 27%) lined predominantly by epidermoid cells with interspersed intermediate and mucinous cells, and type B (22/30, 73%) showed infiltrative tumor islands in the cystic wall or the surrounding tissue. FISH analysis suggested that approximately 66.7% of UC-MEC harbored a MAML2 rearrangement. During the median follow-up period of 42 months (range, 6-120 months), all type A patients and 68% of type B patients who underwent complete surgical resection lived without relapse. Seven cases with type B cancer that underwent curettage initially had local recurrence. Clinicians and pathologists hardly recognize UC-MEC owing to its cystic architecture. Specific epidermoid, mucous, and intermediate tumor cells, and MAML2 fusion testing, are essential to avoid potential diagnostic pitfalls. Prompting and completing resection surgery with negative margins would have a favorable prognosis. CI - Copyright (c) 2022 Xi Wang et al. FAU - Wang, Xi AU - Wang X AD - Department of Oral Pathology, Peking University School and Hospital of Stomatology & National Center of Stomatology & National Clinical Research Center for Oral Diseases & National Engineering Research Center of Oral Biomaterials and Digital Medical Devices, Beijing 100081, China. AD - Research Unit of Precision Pathologic Diagnosis in Tumors of the Oral and Maxillofacial Regions, Chinese Academy of Medical Sciences (2019RU034), China. FAU - Li, Wei AU - Li W AD - Department of Oral Pathology, Peking University School and Hospital of Stomatology & National Center of Stomatology & National Clinical Research Center for Oral Diseases & National Engineering Research Center of Oral Biomaterials and Digital Medical Devices, Beijing 100081, China. AD - Research Unit of Precision Pathologic Diagnosis in Tumors of the Oral and Maxillofacial Regions, Chinese Academy of Medical Sciences (2019RU034), China. FAU - Feng, Yanrui AU - Feng Y AD - Central Laboratory, Peking University School and Hospital of Stomatology & National Center of Stomatology & National Clinical Research Center for Oral Diseases & National Engineering Research Center of Oral Biomaterials and Digital Medical Devices, Beijing 100081, China. FAU - Liu, Lingchao AU - Liu L AUID- ORCID: 0000-0003-0455-2657 AD - Department of Pathology, School of Basic Medical Sciences, Third Hospital, Peking University Health Science Center, Beijing, China. FAU - He, Huiying AU - He H AUID- ORCID: 0000-0001-6237-2190 AD - Department of Pathology, School of Basic Medical Sciences, Third Hospital, Peking University Health Science Center, Beijing, China. FAU - Li, Binbin AU - Li B AUID- ORCID: 0000-0003-0521-2945 AD - Department of Oral Pathology, Peking University School and Hospital of Stomatology & National Center of Stomatology & National Clinical Research Center for Oral Diseases & National Engineering Research Center of Oral Biomaterials and Digital Medical Devices, Beijing 100081, China. AD - Research Unit of Precision Pathologic Diagnosis in Tumors of the Oral and Maxillofacial Regions, Chinese Academy of Medical Sciences (2019RU034), China. LA - eng PT - Journal Article DEP - 20220913 PL - Egypt TA - J Oncol JT - Journal of oncology JID - 101496537 PMC - PMC9489395 COIS- The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. EDAT- 2022/09/24 06:00 MHDA- 2022/09/24 06:01 PMCR- 2022/09/13 CRDT- 2022/09/23 02:39 PHST- 2022/07/08 00:00 [received] PHST- 2022/08/30 00:00 [accepted] PHST- 2022/09/23 02:39 [entrez] PHST- 2022/09/24 06:00 [pubmed] PHST- 2022/09/24 06:01 [medline] PHST- 2022/09/13 00:00 [pmc-release] AID - 10.1155/2022/2676367 [doi] PST - epublish SO - J Oncol. 2022 Sep 13;2022:2676367. doi: 10.1155/2022/2676367. eCollection 2022.