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一名2岁女童的双侧膝关节滑膜软骨瘤病:首例报告及文献综述

Bilateral Knee Synovial Chondromatosis in a 2-Year-Old Girl: First Reported Case and Literature Review.

作者信息

Barbosa Ana Paula Fernandes, Barbosa Quintino Paulyana Fernandes, Motta Camara Hermann Fernandes, Dos Anjos Pedro Henrique Laurindo, Guimaraes Arthur Castro, Lopes Neto Eloisio Barbosa, Leite Duarte Maria Eugenia

机构信息

Department of Pathology, Centro de Estudos Superiores de Maceió, Maceió, AL, Brazil.

Department of Pathology, Universidade Federal de Alagoas, Maceió, AL, Brazil.

出版信息

Am J Case Rep. 2025 Mar 19;26:e945921. doi: 10.12659/AJCR.945921.

Abstract

BACKGROUND Synovial chondromatosis (SC) is a rare monoarticular benign condition characterized by the development of intra- or periarticular cartilage-like nodules or loose bodies. Typically affecting middle-aged men, SC is exceptionally rare in female pediatric patients. Symptoms include diffuse pain, swelling, and limited joint movement. The loose bodies are typically numerous, small, rounded, and uniform in size. Histologically, they consist of hyaline cartilage-like nodules containing clusters of chondrocytes. Bilateral SC in the knee of young children is even rarer and has not been previously documented in the medical literature. CASE REPORT A 2-year-old girl presented with recent pain in her right knee without preceding trauma. Physical examination revealed joint effusion, a limping gait, and limited knee flexion, with no signs of local inflammation. Radiographs showed normal joint structures, and laboratory test results, except for an elevated CRP, were normal. Joint aspiration ruled out septic arthritis. The worsening of symptoms within a relatively short period prompted further investigation, leading to the identification of numerous cartilaginous nodules within the joint during exploratory arthrotomy. Microscopic examination revealed unossified cartilage-like nodules containing disorganized chondrocytes. Four months after surgery, she was diagnosed with SC in the contralateral knee. CONCLUSIONS Given the rarity of synovial chondromatosis in young children, and the even rarer bilateral presentation in a large joint, this case report provides an opportunity to explore the challenges in diagnosing and managing such conditions in pediatric patients. To the best of our knowledge, this is the youngest reported case of bilateral SC of the knee in a pediatric patient.

摘要

背景

滑膜软骨瘤病(SC)是一种罕见的单关节良性疾病,其特征是关节内或关节周围出现软骨样结节或游离体。滑膜软骨瘤病通常影响中年男性,在女性儿科患者中极为罕见。症状包括弥漫性疼痛、肿胀和关节活动受限。游离体通常数量众多、体积小、呈圆形且大小均匀。组织学上,它们由含有软骨细胞簇的透明软骨样结节组成。幼儿双侧膝关节滑膜软骨瘤病更为罕见,医学文献中此前尚无记载。病例报告:一名2岁女童近期出现右膝疼痛,无先前外伤史。体格检查发现关节积液、跛行步态和膝关节屈曲受限,无局部炎症迹象。X线片显示关节结构正常,实验室检查结果除C反应蛋白升高外均正常。关节穿刺排除了化脓性关节炎。症状在相对较短时间内恶化促使进一步检查,在探查性关节切开术中发现关节内有大量软骨结节。显微镜检查显示未骨化的软骨样结节,含有排列紊乱的软骨细胞。术后4个月,她对侧膝关节被诊断为滑膜软骨瘤病。结论:鉴于幼儿滑膜软骨瘤病罕见,且在大关节中双侧发病更为罕见,本病例报告为探讨儿科患者此类疾病诊断和管理中的挑战提供了机会。据我们所知,这是儿科患者中报道的最年轻的双侧膝关节滑膜软骨瘤病病例。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9539/11932548/16f8e00c7e69/amjcaserep-26-e945921-g001.jpg

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