A 58-year-old man visited our hospital with trismus and anorexia in the past 1 month (Fig. 1A, B). Multicentric Castleman disease was suspected due to cervical and axillary lymphadenopathy on CT, a high inflammatory response and IL-6 level elevation. Lymph node biopsy revealed mantle zone development, interfollicular expansion, extensive plasma cell infiltration and cluster of differentiation 3-positive (CD3+) T cell accumulation, leading to the diagnosis of plasmacytic Castleman disease. CT findings could not explain the cause of trismus (Fig. 1C). PET-CT revealed increased fluorodeoxyglucose uptake in the right temporomandibular joint, implicating temporomandibular joint arthritis as the cause of trismus (Fig. 1D, arrow). Trismus rapidly improved after prednisolone and IL-6 inhibitor administration. Similar to RA, Castleman disease is a lymphoproliferative disease with IL-6–related systemic inflammation. Temporomandibular joint arthritis is an articular symptom of RA, but arthritis in Castleman disease has rarely been reported. Activation of CD3+ cells by IL-6 is a potential mechanism of arthritis [1]. CD3+ T cells were clearly observed in the lymph nodes in our patient, which indicates arthritis. We report a case of trismus associated with temporomandibular arthritis, which we consider to be highly representative of the pathophysiology of Castleman disease.
Trismus and its imaging findings. (A, B) The patient’s mouth opening was restricted. (C, D) A CT showed no evidence of trismus, but fluorodeoxyglucose (FDG)-PET/CT showed an accumulation of FDG in the left temporomandibular joint
Data availability statement
Data are available upon reasonable request by any qualified researchers who engage in rigorous, independent scientific research, and will be provided following review and approval of a research proposal and Statistical Analysis Plan (SAP) and execution of a Data Sharing Agreement (DSA). All data relevant to the study are included in the article.
Funding
No specific funding was received from any bodies in the public, commercial or not-for-profit sectors to carry out the work described in this article.
Disclosure statement: The authors have declared no conflicts of interest.
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