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Kaposiform haemangioendothelioma of the spine associated with fixed hyperlordotic deformity and Kasabach–Merritt Syndrome: a case report and literature review
Skeletal Radiology ( IF 2.1 ) Pub Date : 2022-08-10 , DOI: 10.1007/s00256-022-04152-z
Leanne H Q Chin 1 , Kevin K F Fung 2 , Joyce P K Chan 2 , Amanda N C Kan 3 , M K Yuen 2
Affiliation  

Kaposiform haemangioendothelioma (KHE) is a rare childhood disease classified by the International Society for the Study of Vascular Anomalies (ISSVA) as a locally aggressive vascular tumor. It has been reported to affect any site, with a predilection for the extremities and trunk. Although it typically manifests as an enlarging cutaneous or soft tissue lesion, less than 10% of cases have no skin involvement, with the retroperitoneum being the most frequently involved extracutaneous site. Approximately twenty cases of KHE with bony involvement have been reported in the literature to date, with only five of those cases involving the spine specifically.

We present a, rare case of KHE who presented with progressive fixed hyperlordotic deformity, multiple non-specific spinal lesions, and abnormal blood tests, posing a clinical and radiological diagnostic challenge. Additionally, we conducted a thorough review of the literature to compare and contrast the various multimodality imaging manifestations of KHE involving the spine.



中文翻译:

与固定性脊柱前凸畸形和 Kasabach-Merritt 综合征相关的脊柱卡波西样血管内皮瘤:病例报告和文献复习

卡波西样血管内皮瘤 (KHE) 是一种罕见的儿童疾病,被国际血管异常研究学会 (ISSVA) 归类为局部侵袭性血管肿瘤。据报道,它会影响任何部位,偏爱四肢和躯干。虽然它通常表现为扩大的皮肤或软组织病变,但只有不到 10% 的病例没有皮肤受累,腹膜后是最常受累的皮肤外部位。迄今为止,文献中已经报道了大约 20 例涉及骨的 KHE,其中只有 5 例特别涉及脊柱。

我们提出了一例罕见的 KHE 病例,该病例表现为进行性固定性脊柱前凸过度畸形、多处非特异性脊柱病变和异常血液检查,给临床和放射学诊断带来了挑战。此外,我们对文献进行了全面审查,以比较和对比涉及脊柱的 KHE 的各种多模态成像表现。

更新日期:2022-08-10
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