Pyoderma gangrenosum in refractory celiac disease: a case report

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• 作者：Silvia Sedda (1)
  Roberta Caruso (1)
  Irene Marafini (1)
  Elena Campione (1)
  Augusto Orlandi (1) (2)
  Francesco Pallone (1)
  Giovanni Monteleone (1)
  
• 关键词：Gluten ; Cutaneous ulcers ; Celiac disease ; Pyoderma gangrenosum
• 刊名：BMC Gastroenterology
• 出版年：2013
• 出版时间：December 2013
• 年：2013
• 卷：13
• 期：1
• 全文大小：223 KB
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  13. The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-230X/13/162/prepub
  
• 作者单位：Silvia Sedda (1)
  Roberta Caruso (1)
  Irene Marafini (1)
  Elena Campione (1)
  Augusto Orlandi (1) (2)
  Francesco Pallone (1)
  Giovanni Monteleone (1)
  
  1. Department of Systems Medicine, University of Rome 鈥淭or Vergata鈥? Via Montpellier, 1, 00133, Rome, Italy
  2. Department of Biomedicine and Prevention, Anatomic Pathology Unit, University of Rome 鈥淭or Vergata鈥? Rome, Italy
  
• ISSN：1471-230X

文摘

Background Pyoderma gangrenosum is an inflammatory neutrophilic dermatosis characterized by painful cutaneous ulcerations and often associated with systemic inflammatory and neoplastic diseases. Here we report the first case of pyoderma gangrenosum in a patient with refractory celiac disease. Case presentation A 52-year-old woman with a previously diagnosed refractory celiac disease resistant to steroids and immunosuppressive drugs presented to our hospital for a rapidly growing, painful inflammatory skin lesion of the left leg. Physical examination revealed a painful lesion with focal ulceration, necrosis and pus discharge with active inflammatory borders at the external part of the left leg. Histological evaluation of a skin biopsy and analysis of inflammatory cytokines and matrix-degrading proteases in lesional skin samples confirmed the clinical suspicion of pyoderma gangrenosum. Treatment with oral prednisone was rapidly followed by a complete healing of the skin lesion but no improvement of symptoms/signs of malabsorption. Conclusion Treatment of the patient with systemic steroids healed the skin lesion without improving the underlying refractory celiac disease. This observation raises the possibility that refractory celiac disease and pyoderma gangrenosum may be immunologically different.