Adalimumab is a frequently prescribed TNFα inhibitor for treatment of rheumatoid arthritis. We report on a patient who probably developed a Miller-Fisher syndrome after the second injection of adalimuma

Kurmann, Patric

So, Alexander

Van Linthoudt, Daniel

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CASE REPORTMiller–Fisher syndrome in a patient with rheumatoidarthritis treated with adalimumabPatric Thierry Kurmann & Daniel Van Linthoudt &Alexander Kai-Lik SoReceived: 29 July 2008 /Accepted: 10 September 2008 /Published online: 26 September 2008# Clinical Rheumatology 2008Abstract Adalimumab is a frequently prescribed TNFαinhibitor for treatment of rheumatoid arthritis. We report ona patient who probably developed a Miller–Fisher syn-drome after the second injection of adalimumab.Keywords Adalimumab .Miller–Fisher .Rheumatoid arthritisIntroductionRheumatoid arthritis (RA) is a common chronic arthropathyaffecting ≈1% of the adult population worldwide. Tumornecrosis factor alpha (TNFα) inhibitors have been increas-ingly used as DMARD therapy in conjunction withconventional drugs, in particular methotrexate. Adalimumabis a widely prescribed TNFα inhibitor. We report on a femalepatient with RA who developed Miller–Fisher syndrome [1]after the second injection of adalimumab.Case presentationThe patient, a 77-year-old female with seropositive erosiveRA, had previously received hydroxychloroquine, goldsalts, penicillamine, sulfasalazine, and cyclosporine withoutsuccess. Methotrexate had to be discontinued because ofdrug-induced pneumonitis. As a result, she was maintainedon azathioprine (75 mg/day=1 mg/kg) and prednisone (5 mg/day). In 2006, she underwent a cervical laminectomy C4–C5and C6–C7with plate from C4 to C6 because of a myelopathysecondary to cervical osteoarthritis. At surgery, there was nolocal rheumatoid pannus. Her medical history included type IIdiabetes mellitus, hypertension, hyperlipidemia and ischemicheart disease.In 2007, she was started on adalimumab after a RA flare.Twenty-four hours after the second injection of 40 mg ofadalimumab, she reported dizziness and loss of cutaneoussensation that progressed proximally in the lower limbs andshe was hospitalized. On admission, the general examina-tion was unremarkable except for mild fever. The neuro-logical examination showed ataxia of all four limbs anddisappearance of the deep tendon reflexes in the lowerlimbs. The next day, there was a complete loss ofproprioception, loss of balance and impossibility to walk.One week later, she developed a horizontal nystagmus withimpossibility to lift the upper eyelids [1, 2].Laboratory tests showed a raised ESR (40 mm in the firsthour) and CRP (69 mg/l). Autoantibodies including anti-ganglioside antibodies (GQ1b) were negative. The searchfor an infection was negative.Radiological investigations included a cerebral and athoraco-abdo-pelvic computed tomography, and a cervicalto lumbar spine MRI; they were all normal. The electro-myogram showed a decreased evocated sensitive responseto peripheral nerve stimulation on the upper limbs andabsence of this response on the lower limbs. The motorrecording was normal. There was no muscle involvement.A diagnosis of a Miller–Fisher syndrome [1, 2] was made.Treatment consisted of high-dose steroids (75 mg/day=1 mg/kg), gabapentine, and an increased dose of azathio-prine (75 mg tad). Physical therapy was initiated. TheClin Rheumatol (2009) 28:93–94DOI 10.1007/s10067-008-1017-7P. T. Kurmann :D. Van Linthoudt (*)Department of Rheumatology, Hôpital Neuchâtelois,CH-2300 La Chaux-de-Fonds, Switzerlande-mail: Daniel.vanlinthoudt@ne.chP. T. Kurmann :D. Van Linthoudt :A. K.-L. SoDepartment of Rheumatology and Rehabilitation,Centre Hospitalier Universitaire Vaudois,Lausanne, Switzerlandbiological signs of inflammation disappeared quickly butthe clinical evolution was marked by a slow improvementover one year of balance, proprioception, and reflexes.DiscussionThe Miller–Fisher syndrome [1, 2] is an autoimmunepolyneuropathy characterized by an ascending muscleweakness of the limbs, as in the Guillain–Barré syndrome,with loss of sensation, areflexia, ataxia, and opthalmople-gia. Although neurological symptoms have been reportedwith adalimumab [3], this case reports, to our knowledge,on the first Miller–Fisher syndrome induced by treatmentwith adalimumab. Immunological mechanisms have beenincriminated in the development of this syndrome, thoughin our case, anti-ganglioside antibodies were absent. Thelumbar puncture showed an isolated increased proteincontent (1,000 mg/l) but the patient underwent a cervicalsurgery a few weeks before.Neurological complications have been described withseveral TNFα inhibitors. The use of lenercept in multiplesclerosis produced worsening of the disease causing thepremature interruption of the study [4]. Similarly, 17 patientstreated with etanercept [3] and two patients treated withinfliximab [3] developed neurological symptoms. Theselatter included confusion, ataxia, dysesthesia, paresthesia,facial paralysis, optical neuritis, hemiparesia, transversemyelitis and an ascending neuropathy resembling theGuillain–Barré syndrome. All these cases were proven by aretrial of the drug. The risk was estimated for etanercept (31cases for 100,000 patients) and infliximab (41 cases for100,000) [3]. No case has been described with adalimumabuntil now. Therefore, a Miller–Fisher syndrome secondary tothe use of a TNFα inhibitor seems to be a class effect, whichcan be, as in our case, rapidly and severely progressive.Disclosures None.References1. Fisher M (1956) An unusual variant of acute idiopathic polyneuritis(syndrome of ophtalmoplegia ataxia and areflexia). N Eng J Med255:57–652. Lo YL (2007) Clinical and immunological spectrum of the MillerFisher syndrome. Muscle Nerve 36:915–273. Mohan N, Edwards ET, Cupps TR et al (2001) Demyelinationoccurring during anti-tumor necrosis factor alpha therapy forinflammatory arthritides. Arthritis Rheum 44:2862–28694. Van Oosten BW, Barkhof F, Truyen L et al (1996) Increased MRIactivity and immune activation in two multiple sclerosis patientstreated with the monoclonal anti-tumor necrosis factor antibodycA2. Neurology 47:1531–153494 Clin Rheumatol (2009) 28:93–94

Miller-Fisher syndrome in a patient with rheumatoid arthritis treated with adalimumab

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Miller-Fisher syndrome in a patient with rheumatoid arthritis treated with adalimumab

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