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Neonatal ulcerative colitis associated with Familial Mediterranean fever: a case report

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Abstract

Neonatal inflammatory bowel disease (IBD) is a subclass of very early onset IBD that includes children younger than 1 month. It is characterized by more colonic involvement and monogenetic etiology, resistance to classical anti-inflammatory/immunomodulatory treatments and associated with colitis in first-degree family members. Herein we report a 3 month-old girl who was admitted with bloody diarrhea since 10 days of age. Her symptoms persist despite diet elimination. She was diagnosed with neonatal ulcerative colitis (UC) based on clinical, laboratory and histopathological examination. But, she was unresponsive to the immunosuppressive therapy. On the follow-up, she was hospitalized for the high fever two times. Genetic analysis revealed homozygote M694 V mutation. Bloody diarrhea and other clinical findings were improved after colchicine therapy. Neonatal UC associated with Familial Mediterranean fever is an extremely rare condition and to the best of our knowledge our case is the first case in literature. Early diagnosis autoinflammatory disease may prevent complications related to unnecessary immunosuppressive drug usage and the risk of development of amyloidosis associated with autoinflammatory disorders.

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Authors and Affiliations

  1. Department of Pediatric Gastroenterology Hepatology and Nutrition, Karadeniz Technical University, Faculty of Medicine, Trabzon, Turkey

    Elif Sag & Murat Cakir

  2. Department of Pediatric Rheumatology, Karadeniz Technical University, Faculty of Medicine, Trabzon, Turkey

    Ferhat Demir & Mukaddes Kalyoncu

  3. Department of Pathology, Karadeniz Technical University, Faculty of Medicine, Trabzon, Turkey

    Mustafa Emre Ercin

Authors
  1. Elif Sag
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  2. Ferhat Demir
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  3. Mustafa Emre Ercin
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  4. Mukaddes Kalyoncu
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  5. Murat Cakir
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Corresponding author

Correspondence to Murat Cakir.

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The authors have declared no conflict of interest.

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Informed consent was obtained from the parents of the case.

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Sag, E., Demir, F., Ercin, M.E. et al. Neonatal ulcerative colitis associated with Familial Mediterranean fever: a case report. Rheumatol Int 38, 137–140 (2018). https://doi.org/10.1007/s00296-017-3848-5

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  • DOI: https://doi.org/10.1007/s00296-017-3848-5

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