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AB0859 Efficacy and Safety of Anti-tnfα Therapies in Chronic Recurrent Multifocal Osteomyelitis (CRMO): An Extensive Review of The Literature
  1. C. Campochiaro,
  2. L. Rovati,
  3. G. Cavalli,
  4. P. Aiello,
  5. A. Berti,
  6. M.G. Sabbadini,
  7. E. Baldissera,
  8. L. Dagna
  1. Unit of Internal Medicine and Clinical Immunology, IRCCS San Raffaele, Milan, Italy

Abstract

Background CRMO is an autoinflammatory disease characterized by relapsing and remitting episodes of pain related to the presence of foci of sterile bone inflammation [1]. TNFα levels are elevated in the serum of patients affected by CRMO and in bone-biopsy specimens [2]. NSAIDs and steroid afford symptomatic relief of pain and swelling in approximately 80% of patients [1]. Some recent reports have shown that anti-TNFα agents lead to clinical and radiological improvement in refractory cases of CRMO.

Objectives We provide a review of the literature concerning the efficacy and safety of anti-TNFα in CRMO.

Methods We searched the MEDLINE database and selected articles published in the English language with the headings “CRMO” and “anti-TNFα”. We only included unambiguous CRMO cases. We added a patient from our cohort.

Results A total of 13 patients met our inclusion criteria (see Table 1). Etanercept (ETN) was the anti-TNFα most commonly used (7 pts). In 6 cases ETN was used at a dosage of 0.8 mg/kg/week. In 5 cases the dosage was modified after 6 months to 0.8 mg/kg every 2 weeks. In 1 case the drug was used at a dosage of 0.4 mg/kg twice a week. In 2 cases MTX was added to ETN. Infliximab (IFX) was used in 4 pts. It was administered at a dose of 5 mg/kg in 3 pts. In 2 cases a loading dose similar to the one recommended for the treatment of RA was used. In our case, due to the recurrence of bone pain after 4 years with IFX 5 mg/kg every 8 weeks, we reduced the administration interval to 6 weeks. In another patient with worsening bone pain after an initial administration of IFX at a dosage of 5 mg/kg every 8 weeks, a loading dose of 6.7 mg/kg was used for three times every 2 weeks. The patient was then treated with an IFX 6.7 mg/kg every 4 weeks. In 1 pt, an initial regimen of IFX every 4 weeks was modified after 1 year to an 8-weeks regimen. 1 pt was instead treated with IFX at a dosage of 3 mg/kg every 8 weeks. IFX was used as a monotherapy in all cases. Adalimumab (ADA) was used in 2 pts. No data about ADA dose or administration interval were recorded in the published article. Of note, all patients underwent remission after starting an anti-TNFα therapy. In 3 cases a disease flare occurred during the anti-TNFα treatment. In 1 case the flare occurred after increasing the administration interval of ETA from one to two weeks. In the 2 remaining cases the disease flare occurred without any previous therapy modification. In all cases increasing the anti-TNFα agent dosage allowed to achieve remission of CRMO. No adverse events were recorded during the anti-TNFα therapy. No pt required a switch to a different anti-TNFα agent. Attempts to discontinue etanercept therapy were performed in 4 pts, but were followed by disease relapses within a month in 3 cases; in the remaining case, ETA was successfully discontinued after 4.5 years of treatment.

Conclusions Our experience and the review of the literature indicate that anti-TNFα agents represent an effective and possibly safe option in the treatment of refractory CRMO.

  1. Costa-Reis P. Chronic recurrent multifocal osteomyelitis. J Clin Immunol. 2013. 1) Hofmann SR. Chronic non-bacterial osteomyelitis is associated with impaired Sp1 signaling, reduced IL10 promoter phosphorylation, and reduced myeloid IL-10 expression. Clin Immunol. 2011

Disclosure of Interest None declared

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