A case of calcium pyrophosphate dehydrate crystal deposition with synovial chondromatosis and calcification of the disc in the temporomandibular joint

Katayama, Yoshiko; Kurita, Kenichi; Fukuda, Kota; Kondo, Norihiro; Sugita, Yoshihiko; Maeda, Hideaki

doi:10.5794/jjoms.58.366

Cited by 1 publication

(1 citation statement)

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“…It may also be misdiagnosed because its symptoms are similar to those of TMJ disorders. The mean incubation period is long, with TMJ SC having an average onset time of more than 1 year [10]; some studies have reported onset times of about 20 years [11,12]. Similarly, in our cases, it took 11 years for patient 1 and 5 years for patient 2 to be Histopathological findings, immunohistochemical staining findings, and Gl sequence analysis (Figures 9 and 10a,b) were the same as those in patient 1.…”

Section: Discussionsupporting

confidence: 75%

Two Cases of Temporomandibular Synovial Chondromatosis Associated with Gli1 Gene Mutation

Fukutani

Toratani

Kanda

et al. 2022

IJERPH

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Synovial chondromatosis (SC) is a rare benign disease involving multifocal generation of ectopic cartilage in the synovial tissue. Herein, we report two cases of SC in the temporomandibular joint: a 38-year-old woman (patient 1) and 39-year-old woman (patient 2). Both patients had trismus, jaw joint noises, and jaw-opening pain in the temporomandibular joint. Cone-beam computed tomography (CT) and magnetic resonance imaging (MRI) in patient 1 showed multiple calcified loose bodies around the right mandibular condyle. In addition, CT and MRI in patient 2 showed multiple calcified loose bodies around the left mandibular condyle and temporal bone perforation. Following establishing a diagnosis of SC, both patients underwent tumor resection via open surgery. In immunohistochemical examinations of the resected tissues, tumor cells showed intense nuclear staining with labeled anti-Gli1 antibody. Gene sequencing revealed that both patients had a homozygous mutation in the Gli1 gene (rs2228226 G>C). In conclusion, we suggest that the Gli1 gene (rs2228226 G>C) may be involved in the etiology of SC.

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Section: Discussionsupporting

confidence: 75%