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BACKGROUND: Pigmented villonodular synovitis (PVNS) is a rare benign proliferative disorder of the synovium. It rarely occurs in adolescents, particularly in immature patients with bilateral manifestation. CASE PRESENTATION: We present a case of atypical and bilateral PVNS of wrist in a 14-year-old boy. Initially, the patient presented with left wrist pain and swelling without the history of trauma. Physical examination revealed an obvious lesion in the dorsal part of left wrist. Radiographs, computed tomography (CT) and magnetic resonance imaging (MRI) showed multiple abnormal signal shadows and arthroedema in the left wrist. Arthroscopy operation was performed, and histologic examination suggested the diagnosis of PVNS. Only 10 months later, the patient presented with the similar symptoms and signs in the right wrist. But MIR and histologic examination were atypical. In this article, we also review and summarize 26 studies on 30 adolescent patients with PVNS. CONCLUSIONS: This study provides an example of atypical and bilateral PVNS in adolescents.
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PURPOSE: Esophageal carcinoma (EC) is one of the most aggressive type cancers and dysregulation of retinoid X receptor α (RXRα) involves various tumors. However, the relationship of RXRα with the clinicopathological factors of EC, particularly prognostic characteristics, remains unclear. This present study was to evaluate the effect of RXRα expression in the development of EC. METHODS: The mRNA and protein expression level of RXRα in EC and normal esophageal tissues using reverse transcription-polymerase chain reaction (RT-PCR) and Western blot, respectively. The subcellular localization was detected by immunohistochemistry (IHC) analysis. The clinicopathological parameters were included age, sex, tumor size, differentiation, TNM stages and lymph node metastasis. Kaplan-Meier method and Cox's regression analyses were performed to evaluate the prognosis of 60 patients with EC. RESULTS: RXRα was elevated in EC tissues comparing with normal esophageal tissues at both mRNA and protein levels. The overexpression level of RXRα was closely associated to the tumor differentiation, TNM stage and lymph node metastasis of patients with EC. In addition, EC patients with RXRα high expression had significantly lower disease-free survival (DFS) and overall survival (OS). Multivariate analysis showed RXRα expression as an independent predictor for the DFS and OS rate of patients with EC. CONCLUSIONS: Our results showed that overexpression of RXRα was correlated with unfavorable prognosis, suggesting that RXRα may serve as a potential targeted therapeutic marker in the treatment of EC.