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Case Report: Extra-Articular Diffuse Tenosynovial Giant Cell Tumor of the Temporomandibular Joint.

ABSTRACT: Diffuse tenosynovial giant cell tumor (D-TSGCT) is a benign but locally destructive tumor of synovium that may involve joints, tendon sheaths, and bursae. Its occurrence in the temporomandibular joint (TMJ) is extremely rare. The authors reported a case of 48-year-old man with an extra-articular D-TSGCT in the TMJ with medial cranial fossa extension. computed tomography (CT) and magnetic resonance imaging (MRI) features are described. The lesion was a cystic-solid mass centered at the temporal bone without involvement of the condylar head, and its solid component presented high-density on CT and hypointensity on MRI. No signs of recurrence or metastasis was observed during 12-months of follow-up. The present report suggested the potential characteristics of radiologic imaging of D-TSGCT in TMJ.

SUBMITTER: Yang X

PROVIDER: S-EPMC7953063 | biostudies-literature | 2021

REPOSITORIES: biostudies-literature

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Case Report: Extra-Articular Diffuse Tenosynovial Giant Cell Tumor of the Temporomandibular Joint.

Yang Xibiao X Yao Li L Yu Tianping T Du Xiaoli X Yue Qiang Q

Frontiers in oncology 20210226

Diffuse tenosynovial giant cell tumor (D-TSGCT) is a benign but locally destructive tumor of synovium that may involve joints, tendon sheaths, and bursae. Its occurrence in the temporomandibular joint (TMJ) is extremely rare. The authors reported a case of 48-year-old man with an extra-articular D-TSGCT in the TMJ with medial cranial fossa extension. computed tomography (CT) and magnetic resonance imaging (MRI) features are described. The lesion was a cystic-solid mass centered at the temporal b ...[more]

PMID: 33718242

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Project description:BackgroundTenosynovial giant cell tumor (TGCT) is a rare, locally aggressive neoplasm arising from the synovium of joints, bursae, and tendon sheaths affecting small and large joints. It represents a wide spectrum ranging from minimally symptomatic to massively debilitating. Most findings to date are mainly from small, retrospective case series, and thus the morbidity and actual impact of this rare disease remain to be elucidated. This study prospectively explores the management of TGCT in tertiary sarcoma centers.MethodsThe TGCT Observational Platform Project registry was a multinational, multicenter, prospective observational study involving 12 tertiary sarcoma centers in 7 European countries, and 2 US sites. This study enrolled for 2 years all consecutive ≥ 18 years old patients, with histologically diagnosed primary or recurrent cases of diffuse-type TGCT. Patient demographic and clinical characteristics were collected at baseline and every 6 months for 24 months. Quality of life questionnaires (PROMIS-PF and EQ-5D) were also administered at the same time-points. Here we report baseline patient characteristics.Results166 patients were enrolled between November 2016 and March 2019. Baseline characteristics were: mean age 44 years (mean age at disease onset: 39 years), 139/166 (83.7%) had prior treatment, 71/166 patients (42.8%) had ≥ 1 recurrence after treatment of their primary tumor, 76/136 (55.9%) visited a medical specialist ≥ 5 times, 66/116 (56.9%) missed work in the 24 months prior to baseline, and 17/166 (11.6%) changed employment status or retired prematurely due to disease burden. Prior treatment consisted of surgery (i.e., arthroscopic, open synovectomy) (128/166; 77.1%) and systemic treatments (52/166; 31.3%) with imatinib (19/52; 36.5%) or pexidartinib (27/52; 51.9%). Treatment strategies at baseline visits consisted mainly of watchful waiting (81/166; 48.8%), surgery (41/166; 24.7%), or targeted systemic therapy (37/166; 22.3%). Patients indicated for treatment reported more impairment compared to patients indicated for watchful waiting: worst stiffness NRS 5.16/3.44, worst pain NRS 6.13/5.03, PROMIS-PF 39.48/43.85, and EQ-5D VAS 66.54/71.85.ConclusionThis study confirms that diffuse-type TGCT can highly impact quality of life. A prospective observational registry in rare disease is feasible and can be a tool to collect curated-population reflective data in orphan diseases. Name of registry: Tenosynovial Giant Cell Tumors (TGCT) Observational Platform Project (TOPP).Trial registration numberNCT02948088. Date of registration: 10 October 2016. URL of Trial registry record: https://clinicaltrials.gov/ct2/show/NCT02948088?term=NCT02948088&draw=2 .

Project description:Temporomandibular dysfunction involving anterior disc derangement with or without reduction, secondary to posterior ligament insufficiency is typically managed conservatively with success in a majority of patients. When conservative management fails, the next step in the continuum of care is unclear. Platelet-rich plasma injection combined with a 3-week immobilization period may be effective in treating posterior ligament insufficiency following a period of physical therapy. The result of this case was exceptionally successful, with the patient reporting 100% improvement 6 months post-injection. Prior to this case, we predicted a 20% success rate based on her inability to maintain the effects of conservative management over the long term.A 33-year-old white woman presented with temporomandibular dysfunction, which responded to an initial course of physical therapy aimed at restoring the mechanics of her temporomandibular joint, exercise management, and education on self-management strategies. She returned 20 months later and responded well to another course of physical therapy. Despite improvement in pain, range of motion, and mechanics, she continued to present with a reduction click at the end range of opening. The crisp and loud nature of the reduction click indicated a viable posterior ligament and reduction of the anteriorly displaced disc. She opted for platelet-rich plasma injection, provided by a chronic pain specialist with the assistance of a physical therapist. She was immobilized for 3 weeks, followed by a weaning period with reduced posterior support for an additional 5 weeks. Follow-up appointments with a physical therapist occurred at 3 weeks, 8 weeks, and 6 months post-injection.At 6 months, she reported 100% improvement. Objectively, there was no indication that the disc condyle relationship was disrupted. At 6 months post-platelet-rich plasma injection, preceded by a period of conservative physical therapy management, and followed with appropriate physical therapy follow-up, this individual had complete resolution of her anterior disc derangement with reduction.

Dataset Information

Case Report: Extra-Articular Diffuse Tenosynovial Giant Cell Tumor of the Temporomandibular Joint.

Publications

Case Report: Extra-Articular Diffuse Tenosynovial Giant Cell Tumor of the Temporomandibular Joint.

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