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From the Division of Pediatric Neurology and the Research Institute (B.B., E.U., J.K.), The Hospital for Sick Children, University of Toronto, Canada; Department of Pediatric Neurology (S.T.), Hospital de Pediatria Dr Garrahan, Buenos Aires, Argentina; Departments of Neurology (V.A.L., B.G.W., C.F.L., S.J.P.), Laboratory Medicine and Pathology (V.A.L., S.J.P.), and Immunology (V.A.L.), Mayo Clinic College of Medicine, Rochester, MN; and The Montreal Neurological Institute and McGill University (A.B.-O.), Montreal, Canada.
Address correspondence and reprint requests to Dr. Sean Pittock, Department of Neurology and Laboratory Medicine and Pathology, Mayo Clinic College of Medicine, Rochester, MN 55905 Pittock.Sean{at}mayo.edu
Objective: To determine seroprevalence of neuromyelitis optica (NMO)-IgG in childhood CNS inflammatory demyelinating disorders.
Methods: We analyzed demographic, clinical, and radiologic data in a blinded fashion and assessed serum NMO-IgG status for 87 children: 41 with relapsing-remitting multiple sclerosis (RRMS), 17 with NMO, 13 with monophasic/recurrent optic neuritis (ON), 13 with transverse myelitis, of whom 10 were longitudinally extensive on MRI spine (LETM), and another 3 with LETM in the context of acute disseminated encephalomyelitis (ADEM).
Results: Ten of the 87 children (11%) were seropositive. Eight of 17 with NMO (47%) were seropositive (7 of 9 with relapsing NMO [78%], 1 of 8 with monophasic NMO [12.5%]). Two other children were seropositive: 1 of 5 with recurrent ON and one child with recurrent LETM. No seropositive case was identified among 41 with RRMS (14% of whom had LETM at some point in their clinical course), 8 with monophasic ON, 9 with monophasic LETM, or 3 with LETM in the context of ADEM.
Conclusions: The similar frequency of neuromyelitis optica (NMO)-IgG in both childhood and adult cases of NMO, and its rarity in relapsing-remitting multiple sclerosis, supports the concept that these diseases have a similar pathogenesis in childhood and adulthood. It is noteworthy that none of nine children with monophasic longitudinally extensive transverse myelitis (LETM) was NMO-IgG-seropositive. Furthermore, LETM does not appear to be as predictive of an NMO spectrum disorder in children as it is in adults. Longitudinal studies of larger pediatric LETM cohorts are required to ascertain whether the absence of NMO-IgG is a negative predictor for relapse in this childhood entity.
GLOSSARY: ADEM = acute disseminated encephalomyelitis; EDSS = Expanded Disability Status Scale; LETM = longitudinally extensive transverse myelitis; NMO = neuromyelitis optica; ON = optic neuritis; RRMS = relapsing-remitting multiple sclerosis; TM = transverse myelitis.
e-Pub ahead of print on December 19, 2007, at www.neurology.org
*Both authors contributed equally to the study.
Supported in part by the Mayo Foundation, the Ralph Wilson Medical Research Foundation, The Wadsworth Foundation, and the Canadian Multiple Sclerosis Scientific Research Foundation, and the Don Paty Career Development Award from the Multiple Sclerosis Society of Canada (A. B.-O.).
Disclosure: The authors disclose that, in accordance with the Bayh-Dole Act of 1980 and Mayo Foundation policy, Drs. Lennon, Lucchinetti, and Weinshenker stand to receive royalties for the discovery related to AQP4 autoantigen. This intellectual property is licensed to a commercial entity for development of a simple antigen-specific assay to be made available worldwide for patient care. The test will not be exclusive to Mayo Clinic. To date the authors have received a total of <$10,000 in royalties. Mayo Clinic offers the test as an indirect immunofluorescence assay to aid the diagnosis of NMO, but the authors do not benefit personally from the performance of the test. Dr. Weinshenker has received consulting fees from Genentech.
Received April 4, 2007. Accepted in final form May 22, 2007.
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