Experience with anti-TNF-α therapy for orofacial  granulomatosis.
BACKGROUND
Orofacial granulomatosis (OFG) can be challenging to treat and experience with anti-TNF-α therapy is limited. We report our experience with infliximab (IFX) and adalimumab (ADA) for OFG in 14 patients, the largest reported series to date.


METHODS
A review of patients receiving induction and maintenance IFX for OFG +/- Crohn's disease (CD) for active oral disease failing other therapies was performed. Clinical response defined by global physician assessment, aided by oral disease activity scores, was assessed at 2 months, 1 and 2 years. ADA was considered for patients failing IFX. Adverse events were recorded. Predictors of need for anti-TNF-α therapy were determined by comparison with OFG patients not requiring anti-TNF-α from our overall OFG database (n = 207).


RESULTS
Fourteen patients (9 men) were treated with IFX [OFG only (n = 7), OFG with CD (n = 7)]. Nine patients received concomitant immunosuppression. Median duration of treatment was 18 months. Shortterm response was achieved in 10/14 (71%) patients. Eight of 14 (57%) and 4/12 (33%) patients remained responsive at 1 and 2 years, respectively. Two patients who failed IFX responded to ADA. Factors predicting need for anti-TNF-α therapy were oral sulcal involvement, intestinal CD and a raised C-reactive protein (CRP). Oral sulcal involvement predicted response at 1 and 2 years. Intestinal CD did not predict response. The only significant adverse event was an IFX infusion reaction.


CONCLUSIONS
IFX provided good short-term response for most OFG patients; however, a significant proportion lost response long term. Adverse events were uncommon. Patients failing IFX may respond to ADA.
Department of Gastroenterology, Guy's and St. Thomas' NHS Foundation Trust and King's College London, London, UK.
Tim
T
Elliott
Helen
H
Campbell
Michael
M
Escudier
Tim
T
Poate
Carlo
C
Nunes
Miranda
M
Lomer
Alex
A
Mentzer
Pritash
P
Patel
Penelope
P
Shirlaw
Jonathon
J
Brostoff
Stephen
S
Challacombe
Jeremy
J
Sanderson
D000068879:Adalimumab; D000893:Anti-Inflammatory Agents / Q000627:therapeutic use*; D000911:Antibodies, Monoclonal / Q000627:therapeutic use*; D061067:Antibodies, Monoclonal, Humanized; D003424:Crohn Disease / Q000150:complications / Q000188:drug therapy / Q000276:immunology; D005260:Female; D051261:Granulomatosis, Orofacial / Q000150:complications / Q000188:drug therapy* / Q000276:immunology; D006801:Humans; D000069285:Infliximab; D008297:Male; D018709:Statistics, Nonparametric; D013997:Time Factors; D016896:Treatment Outcome; D014409:Tumor Necrosis Factor-alpha / Q000037:antagonists & inhibitors*
D000893:Anti-Inflammatory Agents; D000911:Antibodies, Monoclonal; D061067:Antibodies, Monoclonal, Humanized; D014409:Tumor Necrosis Factor-alpha; D000069285:Infliximab; D000068879:Adalimumab