Subtalar joint pigmented villonodular synovitis misdiagnosed at the first visit:A case report

BACKGROUND Pigmented villonodular synovitis(PVNS)is an uncommon disease that usually occurs in large joints,and involvement of the subtalar joint is rare.The lack of comprehensive knowledge of subtalar joint PVNS could lead to misdiagnosis.CASE SUMMARY We present a 64-year-old woman who,at her first visit,complained of discomfort in the right ankle when she walked.Based on the physical signs and X-ray report,the physician failed to make the suspected diagnosis of PVNS.Eighteen months later,the patient returned with a complaint of a mass in her right lateral malleolus with intermittent blunt pain.The X-ray presented an osteophyte formation and soft tissue calcification at the margin of the subtalar joint.The laboratory tests were normal,whereas magnetic resonance imaging(MRI)showed a low-intensity area on both T1-and T2-weighted images.A suspected diagnosis of PVNS was made and later confirmed by postoperative pathology.Subsequently,the patient received radiotherapy with 32 Gy in 16 fractions.At 6 mo postoperatively,the patient only complained of discomfort after walking three blocks.The American Orthopedic Foot and Ankle Society Ankle-Hindfoot score was 97.CONCLUSION MRI is necessary for the diagnosis of PVNS.Early soft tissue calcification and painless joint swelling should be of concern.

Pigmented Villonodular Tenosynovitis of the Temporomandibular Joint—A Case Report

Background: Pigmented villonodular synovitis is a rare condition typically affecting the major joints and the involvement of the temporomandibular joint is rare. Although, it is histologically benign, it can be locally aggressive, capable of extensive bone destruction. The disease has aggressive behavior and a high incidence of recurrence (40% - 60%). Case report: A 35 years old male presented with history of unilateral hearing loss and otalgia of six month duration. On examination, there was a soft bulge in the superior wall of external auditory canal. Contrast enhanced CT scan showed soft tissue density occupying the right external auditory canal and bulging into the middle ear cavity, displacing the intact ossicular chain medially with evidence of destruction of the posterior wall of the external auditory canal, glenoid fossa, mastoid bone and the scutum. The squamous part of the right temporal bone showed evidence of lateral bowing with discontinuity. Surgical resection was done through post aural approach. Histopathological examination revealed pigmented villonodular tenosynovitis which stained positive for CD 68. Post operatively patient was evaluated clinically and with radiological modalities which showed healthy mastoid cavity and no increase in post operative granulations on magnetic resonance imaging. Conclusion: Pigmented villonodular tenosynovitis of temporomandibular joint is a rare entity. Due to the aggressive behavior of the lesion, radical or aggressive surgical resection should be done. Long term follow up with radiological modality should be done due to the high recurrence rate.

Effect of synoviectomy combined with postoperative radiotherapy on pigmented villonodular synovitis in knee:A meta-analysis

Objective:To evaluate the efficacy of synovectomy combined with postoperative radiotherapy in the treatment of pigmented villonodular synovitis compared with only synovectomy in knee. Methods:The related references were searched from SinoMed,CNKI,Wanfang,VIP,PubMed,Embase,Cochrane Library and Web of Science from date of databases establishment to October 2020,and the studies of synovectomy combined with postoperative radiotherapy were screened. According to the type of research,the corresponding evaluation methods were selected for quality evaluation of the literature. RevMan 5.3 software was used for meta-analysis. The primary outcome was recurrence,while the secondary outcomes included complications,excellent and good rate of function of knee,range of motion of knee,Lysholm score,and IKDC score of knee etcetera. Results:A total of 11 articles were included in meta-analysis,with 510 cases,280 in the combined group and 230 in the control group. The results of meta-analysis showed that:(1)The recurrence rate in the combination group was significantly lower than that in the control group,[OR=0.33,95%CI(0.20,0.55),P<0.000 1];(2)The complications in the combined group were higher than that in the control group[OR=2.70,95%CI(1.16,6.31),P=0.02];(3)The excellent and good rate of the combined group was significantly better than that of the control group[OR=2.86,95%CI(1.23,6.64),P=0.01],and it also may be the result of different evaluation criteria of each study;(4)There was no significant difference range of motion[MD=2.96,95%CI(-0.54,6.47),P=0.10],Lysholm score[MD=3.22,95%CI(-0.51,6.96),P=0.09],or IKDC score[MD=2.88,95%CI(-1.07,6.82),P=0.15]in knee between the two groups. Conclusion:The meta-analysis shows that synovectomy combined with radiotherapy can reduce the recurrence rate of pigmented villonodular synovitis in knee,but compared with only operation,there are higher complications. However,the above conclusions need to be verified by long-term follow-up of more high-quality clinical studies.