Klippel-Trenaunay syndrome: a rare cause of disabling pain after a femoral fracture

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• 作者：Angela Notarnicola (1) angelanotarnicola@yahoo.it
  Vito Pesce (1)
  Giuseppe Maccagnano (1)
  Giovanni Vicenti (1)
  Biagio Moretti (1)
• 关键词：Klippel ; Trenaunay syndrome – ; Femoral fracture – ; Neuropathic pain – ; Hyperalgesia – ; Allodynia
• 刊名：Archives of Orthopaedic and Trauma Surgery
• 出版年：2012
• 出版时间：July 2012
• 年：2012
• 卷：132
• 期：7
• 页码：993-996
• 全文大小：251.4 KB
• 参考文献：1. Tsaridis E, Papasoulis E, Manidakis N, Koutroumpas I, Lykoudis S, Banos A, Sarikloglou S (2009) Management of a femoral diaphyseal fracture in a patient with Klippel-Trenaunay-Weber syndrome: a case report. Cases J 26(2):8852
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• 作者单位：1. Department of Neuroscience and Organs of Sense, Orthopedics Section, Faculty of Medicine and Surgery, General Hospital, University of Bari, Piazza Giulio Cesare 11, Bari, Italy
• 刊物类别：Medicine
• 刊物主题：Medicine & Public Health
  Orthopedics
  
• 出版者：Springer Berlin / Heidelberg
• ISSN：1434-3916

文摘

Klippel-Trenaunay syndrome (KTS) is characterized by a cutaneous vascular nevus of the involved extremity, as well as bone and soft tissue hypertrophy of the extremity and venous malformations. We present the case of a 52-year-old man with a femoral fracture and a history of haemangiomas, limb bone hypertrophy and varicosity. The patient was finally diagnosed with KTS and treated in mini-invasive surgery by endomedullary fracture nailing in general anaesthesia. Clinical management was particularly demanding, not only because of the need to monitor the risk of haemorrhage and thrombosis but also because of the onset of a rare picture of neuropathic pain with hyperalgesia and allodynia, never previously reported in patients affected by KTS, that required the administration of major opioid drugs. According to our knowledge, this is the second case of KTS managed for femoral fracture. Unlike the previous report in literature, in this case a severe disabling neuropathic pain complicated the clinical management.