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Fulminant Central Nervous System Nocardiosis in a Patient Treated With Alemtuzumab for Relapsing-Remitting Multiple Sclerosis

2016; American Medical Association; Volume: 73; Issue: 6 Linguagem: Inglês

10.1001/jamaneurol.2016.0146

2168-6157

Horst Penkert, Claire Delbridge, Nina Wantia, Benedikt Wiestler, Thomas Korn,

Fungal Infections and Studies

A woman in her late 40s w ith relapsing-remitting m u l t i p l e s c l e r o s i s ( M S ) w a s a d m itt e d b e c a u s e o f reduced ambulation and personality change in the previous 3 weeks.Her diagnosis of MS was made in 2007, and she was initially t re ate d w it h i nte r fe ro n beta-1a and switched to natalizumab because of ongoing disease activity.After 80 infusions, natalizumab was discontinued because of repeated relapses and seroconversion to positivity for JC virus.Her first cycle of alemtuzumab was given in February 2015 (Expanded Disability Status Sc ale score of 4.5), 18 weeks after the cessation of natalizumab treatment.Her medical history was remarkable for hypothyroidism, rec urrent infec tions, and an anorectic disorder (body mass index [calculated as weight in kilograms divided by height in meters squared] on admission, 14).On admission in June 2015, she had a tetraspastic syndrome and was wheelchair-bound.Her temperature was 37.2°C and her white blood cell count was 9100/μL (to convert to ×10 9 /L, multiply by 0.001) with an essentially depleted mononuclear fraction (Table ).Cerebrospinal fluid analysis showed 2 leukoc ytes per mic roliter.M a g n e t i c r e s o n a n c e i m a g i n g s h o w e d m u l t i p l e ring-enhancing lesions (Figure).Despite broad screening, no infectious agent was identified in the patient's blood or cerebrospinal fluid (including Toxoplasma gondii, mycobacterial species, Pneumocystis jiroveci, fungi, or human immunodeficiency virus), and a search for infectious foc i in the systemic compartment remained inconclusive.Because the patient's condition deteriorated within 2 days despite broad-spectrum antibiotic treatment, an intracranial biopsy specimen was obtained.Culture of intracranial abscess material resulted in identification of Nocardia farcinica (Figure).The patient's antibiotic treatment was revised to imipenem, co-trimoxazole, and amikacin plus dexamethasone.She required transient noradrenaline infusion and was mechanically ventilated for 10 days before she improved and could be discharged for rehabilitation.On follow-up, she was ambulatory, her peripheral blood mononuclear cell count had partly recovered (Table ), and number and size of Nocardia-associated intracerebral lesions were markedly reduced on the magnetic resonance image (Figure).Her intravenous antibiotic therapy was stopped after 8 weeks of treatment and oral administration of co-trimoxazole and amoxicillin-clavulanic acid was continued to maintain remission.