Servelle-Martorell syndrome with extensive upper limb involvement: a case report

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• 作者：Raju Karuppal (1)
  Rajendran V Raman (1)
  Brijesh P Valsalan (1)
  TS Gopakumar (1)
  Chathoth Meethal Kumaran (1)
  Chembu Kara Vasu (1)
  
• 刊名：Journal of Medical Case Reports
• 出版年：2008
• 出版时间：December 2008
• 年：2008
• 卷：2
• 期：1
• 全文大小：390KB
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• 作者单位：Raju Karuppal (1)
  Rajendran V Raman (1)
  Brijesh P Valsalan (1)
  TS Gopakumar (1)
  Chathoth Meethal Kumaran (1)
  Chembu Kara Vasu (1)
  
  1. Medical College, Calicut, 673008, Kerala, India
  

文摘

Introduction Angio-osteohypotrophic syndrome is also known as Servelle-Martorell angiodysplasia. It is characterized by venous or, rarely, arterial malformations, which may result in limb hypertrophy and bony hypoplasia. Extensive involvement of the upper limb is a rare feature of Servelle-Martorell syndrome. Cases with minimal upper limb involvement have been described in the literature. Case presentation A young man presented with multiple separate swollen areas over the right upper limb and functional difficulty since birth. The arm muscles and muscles of the limb girdle were atrophic. The forearm and hand bones were hypoplastic and tender. Conclusion We report a case of Servelle-Martorell syndrome with extensive involvement of the entire upper limb and periscapular region. Servelle-Martorell syndrome is highlighted as one of the causes of angiodysplastic limb hypertrophy.