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 coul...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.展开更多
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 locall...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.展开更多
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 we...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.展开更多
背景:色素沉着绒毛结节性滑膜炎在病因、临床表现、诊断与治疗等研究领域依然存在较大争议,对色素沉着绒毛结节性滑膜炎进行文献计量学及可视化研究可以理清该领域研究发展脉络,为未来的研究指明方向。目的:分析色素沉着绒毛结节性滑膜...背景:色素沉着绒毛结节性滑膜炎在病因、临床表现、诊断与治疗等研究领域依然存在较大争议,对色素沉着绒毛结节性滑膜炎进行文献计量学及可视化研究可以理清该领域研究发展脉络,为未来的研究指明方向。目的:分析色素沉着绒毛结节性滑膜炎全球研究现状、热点及趋势。方法:选用Web of Science及中国知网数据库检索1995-2023年所有与色素沉着绒毛结节性滑膜炎相关的文献出版物,用Citespace和Bibliometrics软件对所有文献进行聚类、共现和突现词分析。Web of Science核心合集数据库采用主题词加自由词进行检索,中国知网数据库通过主题词进行检索。最终纳入986篇英文文献和599篇中文文献。结果与结论:①美国在该领域研究占有绝对领导地位,共发文数量、H指数和被引次数均排名第一。中国总发文量排名第4,H指数排名第12,发文质量与国际合作方面仍有待加强。②中国知网数据库相关研究聚类分析显示排名前5的聚类为放射治疗、软组织肉瘤、类风湿性关节炎、磁共振影像和诊断。③持续突现至2023年的关键词有集落刺激因子1、腱鞘巨细胞瘤、个案报道、染色体易位、放射治疗、表达和激酶。④依据关键词及共被引文献分析发现,关于色素沉着绒毛结节性滑膜炎临床特征的研究、集落刺激因子1抑制剂新药开发,以及集落刺激因子1抑制剂在治疗过程中的应用是目前的研究热点。⑤结合主题演化及现有研究热点分析,未来在明确该病病因、发病机制及临床特征的基础上,提高色素沉着绒毛结节性滑膜炎诊断准确率、增强治疗的精准性、降低治疗后复发率将是需要重点关注的问题。展开更多
文摘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.
文摘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.
基金General Project of National Natural Science Foundation of China(No.81873327)Youth Project of National Natural Science Foundation of China(No.81904226)。
文摘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.
文摘背景:色素沉着绒毛结节性滑膜炎在病因、临床表现、诊断与治疗等研究领域依然存在较大争议,对色素沉着绒毛结节性滑膜炎进行文献计量学及可视化研究可以理清该领域研究发展脉络,为未来的研究指明方向。目的:分析色素沉着绒毛结节性滑膜炎全球研究现状、热点及趋势。方法:选用Web of Science及中国知网数据库检索1995-2023年所有与色素沉着绒毛结节性滑膜炎相关的文献出版物,用Citespace和Bibliometrics软件对所有文献进行聚类、共现和突现词分析。Web of Science核心合集数据库采用主题词加自由词进行检索,中国知网数据库通过主题词进行检索。最终纳入986篇英文文献和599篇中文文献。结果与结论:①美国在该领域研究占有绝对领导地位,共发文数量、H指数和被引次数均排名第一。中国总发文量排名第4,H指数排名第12,发文质量与国际合作方面仍有待加强。②中国知网数据库相关研究聚类分析显示排名前5的聚类为放射治疗、软组织肉瘤、类风湿性关节炎、磁共振影像和诊断。③持续突现至2023年的关键词有集落刺激因子1、腱鞘巨细胞瘤、个案报道、染色体易位、放射治疗、表达和激酶。④依据关键词及共被引文献分析发现,关于色素沉着绒毛结节性滑膜炎临床特征的研究、集落刺激因子1抑制剂新药开发,以及集落刺激因子1抑制剂在治疗过程中的应用是目前的研究热点。⑤结合主题演化及现有研究热点分析,未来在明确该病病因、发病机制及临床特征的基础上,提高色素沉着绒毛结节性滑膜炎诊断准确率、增强治疗的精准性、降低治疗后复发率将是需要重点关注的问题。