Management of temporomandibular joint diseases: a rare case report of coexisting calcium pyrophosphate crystal deposition and synovial chondromatosis

BMC Oral Health

Case Report

Toshihiro Kawano¹  Hiroyuki Nakamura¹  Edward Hosea Ntege²  Masaru Higa³  Makoto Murahashi⁴  Nobuyuki Maruyama⁴  Yusuke Shimizu⁵ 

[1] Department of Oral and Maxillofacial Functional Rehabilitation, Graduate School of Medicine, University of the Ryukyus, 207 Uehara, Nakagami, 903-0215, Nishihara, Okinawa, Japan;Department of Oral and Maxillofacial Functional Rehabilitation, Graduate School of Medicine, University of the Ryukyus, 207 Uehara, Nakagami, 903-0215, Nishihara, Okinawa, Japan;Department of Plastic and Reconstructive Surgery, Graduate School of Medicine, University of the Ryukyus, 207 Uehara, Nakagami, 903-0215, Nishihara, Okinawa, Japan;Department of Oral and Maxillofacial Surgery, Chubu Tokushukai Hospital, 801 Higa, Kitanakagusuku, 901-2393, Nakagami-gun, Okinawa-ken, Japan;Department of Oral and Maxillofacial Surgery, Chubu Tokushukai Hospital, 801 Higa, Kitanakagusuku, 901-2393, Nakagami-gun, Okinawa-ken, Japan;Department of Oral and Maxillofacial Functional Rehabilitation, Graduate School of Medicine, University of the Ryukyus, 207 Uehara, Nakagami, 903-0215, Nishihara, Okinawa, Japan;Department of Plastic and Reconstructive Surgery, Graduate School of Medicine, University of the Ryukyus, 207 Uehara, Nakagami, 903-0215, Nishihara, Okinawa, Japan;

关键词: Synovial chondromatosis;    Calcium pyrophosphate;    Pseudogout;    Temporomandibular joint;    Crystal arthropathy;    Inorganic pyrophosphate;    ANKH;    Case report;   

DOI  :  10.1186/s12903-022-02695-0

 received in 2022-08-10, accepted in 2022-12-22,  发布年份 2022

来源: Springer

【 摘 要 】

BackgroundThe coexistence of calcium pyrophosphate dihydrate crystal deposition (CPP) and synovial chondromatosis (SC) in the temporomandibular joint (TMJ) is rarely reported. CPP disease (CPPD) is complex arthritis synonymous with excessive pyrophosphate production and variable aberrations in mineral and organic phase metabolism of the joint cartilage, leading to local inundated CPP and crystal deposition of partially deciphered predispositions. Meanwhile, SC is a rare benign synovial joint proliferative disease of unclear etiology and has a low risk of malignant transformation. However, SC manifests severe joint disability and dysfunction because of connective tissue metaplasia of the synovial membrane, which forms cartilaginous nodules with or without calcifications or ossifications. These nodules often detach and form intra-articular loose bodies and very rarely within extraarticular spaces.Case presentationWe report the case of a 61-year-old man to expand the body of literature on these unusual coexisting arthropathies of the TMJ. The patient presented to our hospital in 2020 with complaints of pain in the right TMJ and trismus for over 6 months. Radiographic assessments of the TMJ provided a preoperative provisional diagnosis of SC. However, the histopathology of the open biopsy revealed tumor-like lesions comprising several deposits of rhomboid and rod-shaped crystals that displayed positive birefringence in polarized light, confirming a coexistence of CPPD. A second-stage operation was performed for the complete removal of the loose bodies and chalk-like lesions including synovectomy. No evidence of recurrence was recorded after a follow-up of nearly 1.5 years.ConclusionsIsolated CPPD and SC of the TMJ are prevalent in the literature however, monoarticular coexistence of these diseases is rare, due to the lack of consistency in the diagnostic criteria in clinical practice. Moreover, optimal treatment depends on several considerations. This report delineated the molecular etiopathology and underscored the need for continued deciphering of the causal mechanisms of coexisting CPPD and SC of the TMJ. In addition, the importance of confirmatory testing for accurate diagnosis, and appropriate management of these diseases were discussed.

【 授权许可】

CC BY   
© The Author(s) 2022

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