PMID- 38298294
OWN - NLM
STAT- PubMed-not-MEDLINE
LR  - 20240202
IS  - 2168-8184 (Print)
IS  - 2168-8184 (Electronic)
IS  - 2168-8184 (Linking)
VI  - 16
IP  - 1
DP  - 2024 Jan
TI  - Sclerosing Odontogenic Carcinoma With a Prominent Clear Cell Component Mimicking 
      Odontogenic Clear Cell Carcinoma: An Extremely Rare Case With a Fatal Clinical 
      Outcome.
PG  - e51429
LID - 10.7759/cureus.51429 [doi]
LID - e51429
AB  - Sclerosing odontogenic carcinoma (SOC) is an exceedingly rare odontogenic 
      carcinoma known for its locally aggressive yet indolent behavior. There have been 
      no reports of metastasis to distant organs, except a single case involving lymph 
      node metastasis. This report details the case of a 49-year-old female who 
      presented with a well-demarcated radiolucent lesion in the mandible, accompanied 
      by root resorption and tooth displacement. Microscopically, the lesion exhibited 
      a distinctive composition, with two distinct components: cords of epithelium 
      embedded within an abundant collagenous stroma and solid nests of clear polygonal 
      cells surrounded by hyalinized stroma. Notably, the tumor exhibited direct 
      invasion into the submental muscles, accompanied by perineural and vascular 
      invasion, as well as cortical bone loss. Additionally, the clear cells contained 
      diastase-sensitive periodic acid-Schiff-positive granules. Immunohistochemically, 
      the tumor cells displayed positivity for cytokeratin 19 and p63 while testing 
      negative for myoepithelial markers. The Ki-67 index was measured at 23%. 
      Importantly, neitherEWSR1 nor MAML2 rearrangements were detected through 
      fluorescence in situ hybridization (FISH) analysis. Over several years, this 
      patient experienced three instances of local recurrence; notably, four years 
      after the initial surgery, fludeoxyglucose F18-positron emission tomography 
      (18FDG-PET)/CT scans confirmed the presence of pulmonary metastasis. This case 
      presents an unusual histological variation of SOC, marked by vascular invasion, 
      and is notably the first documented case of a fatal outcome in this context.
CI  - Copyright (c) 2024, Shen et al.
FAU - Shen, Fa-Chih
AU  - Shen FC
AD  - Department of Dentistry, Cathay General Hospital Sijhih Branch, New Taipei City, 
      TWN.
AD  - Department of Clinical Pathophysiology, Matsumoto Dental University, Shiojiri, 
      JPN.
FAU - Shimada, Katsumitsu
AU  - Shimada K
AD  - Department of Clinical Pathophysiology, Matsumoto Dental University, Shiojiri, 
      JPN.
FAU - Roy, Rita R
AU  - Roy RR
AD  - Department of Physiology, Matsumoto Dental University, Shiojiri, JPN.
FAU - Kitamura, Yutaka
AU  - Kitamura Y
AD  - Division of Oral and Maxillofacial Surgery, Center of Oral and Maxillofacial 
      Surgery and Dental Implant in Shinshu, Obuse City, JPN.
AD  - Department of Oral and Maxillofacial Surgery, Matsumoto Dental University, 
      Shiojiri, JPN.
FAU - Hasegawa, Hiromasa
AU  - Hasegawa H
AD  - Department of Laboratory Medicine, Shinshu University Hospital, Matsumoto, JPN.
AD  - Department of Clinical Pathophysiology, Matsumoto Dental University, Shiojiri, 
      JPN.
LA  - eng
PT  - Case Reports
DEP - 20240101
PL  - United States
TA  - Cureus
JT  - Cureus
JID - 101596737
PMC - PMC10828747
OTO - NOTNLM
OT  - clear cell morphology
OT  - ewsr1 fish
OT  - metastasis
OT  - recurrence
OT  - sclerosing odontogenic carcinoma
OT  - vascular invasion
COIS- The authors have declared that no competing interests exist.
EDAT- 2024/02/01 06:43
MHDA- 2024/02/01 06:44
PMCR- 2024/01/01
CRDT- 2024/02/01 04:06
PHST- 2024/01/01 00:00 [accepted]
PHST- 2024/02/01 06:44 [medline]
PHST- 2024/02/01 06:43 [pubmed]
PHST- 2024/02/01 04:06 [entrez]
PHST- 2024/01/01 00:00 [pmc-release]
AID - 10.7759/cureus.51429 [doi]
PST - epublish
SO  - Cureus. 2024 Jan 1;16(1):e51429. doi: 10.7759/cureus.51429. eCollection 2024 Jan.