PMID- 29184006
OWN - NLM
STAT- MEDLINE
DCOM- 20180705
LR  - 20191128
IS  - 1757-790X (Electronic)
IS  - 1757-790X (Linking)
VI  - 2017
DP  - 2017 Nov 28
TI  - Pain improvement in Camurati-Engelmann disease after anti-TNFalpha therapy.
LID - bcr-2017-221760 [pii]
LID - 10.1136/bcr-2017-221760 [doi]
LID - bcr2017221760
AB  - 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 (TNFalpha). Remission 
      of UC was achieved and CED-associated pain also improved with infliximab. This is 
      the first report showing a possible role of anti-TNFalpha in pain management in CED 
      with unsatisfactory response to steroids.
CI  - (c) BMJ Publishing Group Ltd (unless otherwise stated in the text of the article) 
      2017. All rights reserved. No commercial use is permitted unless otherwise 
      expressly granted.
FAU - Moreira, Sonia
AU  - Moreira S
AUID- ORCID: 0000-0003-3363-0589
AD  - Department of Internal Medicine, Centro Hospitalar e Universitario de Coimbra, 
      Coimbra, Portugal.
FAU - Cunha, Bernardo
AU  - Cunha B
AD  - Department of Internal Medicine, Centro Hospitalar e Universitario de Coimbra, 
      Coimbra, Portugal.
FAU - Jesus, Nelson Pedro
AU  - Jesus NP
AD  - Department of Internal Medicine, Centro Hospitalar e Universitario de Coimbra, 
      Coimbra, Portugal.
FAU - Santos, Lelita
AU  - Santos L
AD  - Department of Internal Medicine, Centro Hospitalar e Universitario de Coimbra, 
      Coimbra, Portugal.
LA  - eng
PT  - Case Reports
PT  - Journal Article
DEP - 20171128
PL  - England
TA  - BMJ Case Rep
JT  - BMJ case reports
JID - 101526291
RN  - 0 (Gastrointestinal Agents)
RN  - 0 (Tumor Necrosis Factor-alpha)
RN  - B72HH48FLU (Infliximab)
SB  - IM
MH  - Camurati-Engelmann Syndrome/complications/*drug therapy/genetics
MH  - Chronic Pain/*drug therapy/genetics
MH  - Colitis, Ulcerative/*drug therapy/genetics
MH  - Female
MH  - Gastrointestinal Agents/*therapeutic use
MH  - Humans
MH  - Infliximab/*therapeutic use
MH  - Middle Aged
MH  - Tumor Necrosis Factor-alpha/*antagonists & inhibitors
PMC - PMC5720334
OTO - NOTNLM
OT  - biological agents
OT  - musculoskeletal syndromes
COIS- Competing interests: None declared.
EDAT- 2017/12/01 06:00
MHDA- 2018/07/06 06:00
PMCR- 2019/11/28
CRDT- 2017/11/30 06:00
PHST- 2017/11/30 06:00 [entrez]
PHST- 2017/12/01 06:00 [pubmed]
PHST- 2018/07/06 06:00 [medline]
PHST- 2019/11/28 00:00 [pmc-release]
AID - bcr-2017-221760 [pii]
AID - 10.1136/bcr-2017-221760 [doi]
PST - epublish
SO  - BMJ Case Rep. 2017 Nov 28;2017:bcr2017221760. doi: 10.1136/bcr-2017-221760.