Severe polyneuropathy complicating active Crohn’s disease: rapid response to Infliximab

Treatment with a chimeric antitumour necrosis factor (TNF) antibody (Infliximab) has been shown to be highly efficient for patients with steroid refractory Crohn’s disease (CD). However, the mechanism of action remains largely unknown. Recently, a favourable response to Infliximab treatment was demonstrated in some diseases complicating active CD such as acute idiopathic pancreatitis. We report a case of a middle aged female with CD that developed an aggressive form of polyneuritis resistant to corticosteroids. A 55 year old White female, weighting 68 kg, presented with exacerbation of CD (CD activity index (CDAI) >450) associated with an aggressive form of polyneuritis involving the right arm and leg with arthralgias, myalgias, and functional impotence. She had been suffering from refractory severe CD involving the ileum and right colon for 10 years and she was taking oral corticosteroids for two years continually with signs and symptoms of chronic corticosteroid abuse. Immunosuppressive therapy with azathioprine was rapidly stopped for gastric intolerance. Neuropathy was characterised by arthralgias, myalgias, and functional impotence of the right arm and leg. Severe muscle atrophy of the right arm was evident and deambulation was very difficult. Therapy with folate, vitamin B12, and vitamin B6 was ineffective. A magnetic resonance scan of the cranium and spinal cord excluded central, optic nerve, and spinal cord demyelinating lesions. Electromyography showed demyelinating neuropathy involving the right and left esternal popliteus ischiatic nerve, a mixed (motor and sensitive) neuropathy involving the right and left radial nerve, and an axonal neuropathy involving the right ulnar and medial nerve. Other conditions such as polyarteritis nodosa, Wegener’s granulomatosis, primary mixed cryoglobulinaemia, rheumatoid arthritis, systemic lupus erythematous, or sarcoidosis had been excluded. An infusion of 5 mg/kg Infliximab (Remicade; Schering-Plough SpA) was given at weeks 0, 2, and 6. Five repeated 5 mg/kg Infliximab infusions at eight week intervals were administered. Infliximab was well tolerated and no side effects were recorded. Arthralgias, myalgias, and functional impotence of the right arm and leg progressively improved after the first Infliximab infusion. Muscle atrophy of the right hand improved dramatically two weeks later. Electromyography performed at week 22 after the start of therapy was normal. CDAI score is <150 at this time. Sign and symptoms of chronic corticosteroid therapy rapidly disappeared. In conclusion, Infliximab may be a suitable therapeutic option in patients with rare extraintestinal manifestations of CD such as severe polyneuritis not responding to conventional therapy.