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Pain improvement in Camurati-Engelmann disease after anti-TNFα therapy
  1. Sónia Moreira,
  2. Bernardo Cunha,
  3. Nelson Pedro Jesus,
  4. Lèlita Santos
  1. Department of Internal Medicine, Centro Hospitalar e Universitário de Coimbra, Coimbra, Portugal
  1. Correspondence to Dr Sónia Moreira, soniabastosmoreira{at}


Camurati-Engelmann disease (CED) is a rare disorder included in the group of craniotubular hyperostosis diseases. Corticosteroids are used for pain management in CED, but in refractory or corticosteroid-non-tolerant patients, pain management is limited. We report the case of a woman with CED diagnosed in early infancy whose initial complaints included persistent bone pain associated with progressive functional disability. She was treated with steroids but over time became dependent on higher doses with only mild pain relief. In her third decade, she was diagnosed with ulcerative colitis (UC) and was treated with mesalazine, azathioprine and prednisolone. Due to recurrent exacerbations of UC, treatment was changed to infliximab, an antitumour necrosis factor-alpha (TNFα). Remission of UC was achieved and CED-associated pain also improved with infliximab. This is the first report showing a possible role of anti-TNFα in pain management in CED with unsatisfactory response to steroids.

  • biological agents
  • musculoskeletal syndromes
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  • Contributors SM and BC conceived the case report. SM was a major contributor in writing the manuscript. NPJ treated the patient and also interpreted the patient data. NPJ and LS were involved in the review. All authors read and approved the final manuscript.

  • Competing interests None declared.

  • Patient consent Obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.

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