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Isolated new onset 'atypical' optic neuritis in the NMO clinic: serum antibodies, prognoses and diagnoses at follow-up.
J Neurol 2016; 263(2):370-379JN

Abstract

Severe, recurrent or bilateral optic neuritis (ON) often falls within the neuromyelitis optica spectrum disorders (NMOSD), but the diagnosis can be particularly challenging and has important treatment implications. We report the features, course and outcomes of patients presenting with atypical ON when isolated at onset. We retrospectively analyzed 69 sequential patients referred to a single UK NMO center with isolated ON at onset. Aquaporin-4 antibody (AQP4-Ab) assessment was performed in all patients and IgG1 myelin-oligodenrocyte glycoprotein (MOG-Ab) in AQP4-Ab(neg) patients. 37 AQP4-Ab positive (AQP4-Ab(pos)) and 32 AQP4-Ab negative (AQP4-Ab(neg)) patients (8 with MOG-Ab) were identified. The AQP4-Ab(neg) group included heterogeneous diagnoses: multiple sclerosis (MS), NMO, relapsing isolated ON (RION), monophasic isolated ON and relapsing acute disseminated encephalomyelitis (ADEM)-like syndromes. Compared to AQP4-Ab(neg) patients, AQP4-Ab(pos) patients had a worse residual visual outcome from first attack (median VFSS 4 vs. 0, p = 0.010) and at last assessment (median VFSS 5 versus 2, p = 0.005). However, AQP4-Ab(neg) patients with RION also had poor visual outcome. Up to 35% of AQP4-Ab(neg) patients developed a LETM and two developed low positivity for AQP4-Ab over time. Eight AQP4-Ab(neg) patients (25%) were MOG-Ab positive, covering a range of phenotypes excluding MS; the first ON attack was often bilateral and most had relapsing disease with a poor final visual outcome [VFSS 4, range (0-6)]. In conlcusion, AQP4-Ab positivity is confirmed as a predictor of poor visual outcome but AQP4-Ab(neg) RION also had a poor visual outcome. Of those without AQP4-Ab, 25% had MOG-Ab and another 25% developed MS; thus, MOG-Ab is associated with AQP4-Ab(neg) non-MS ON.

Authors+Show Affiliations

Nuffield Department of Clinical Neurosciences, Level 3 West Wing, Oxford University Hospitals NHS Trust, University of Oxford, Headley Way, Oxford, OX3 9DU, UK. IRCCS C. Mondino, University of Pavia, Pavia, Italy.Neuroimmunology Group, Nuffield Department of Clinical Neurosciences, Level 6 West Wing, John Radcliffe Hospital, Headley Way, Oxford, OX3 9DU, UK.Nuffield Department of Clinical Neurosciences, Level 3 West Wing, Oxford University Hospitals NHS Trust, University of Oxford, Headley Way, Oxford, OX3 9DU, UK.Nuffield Department of Clinical Neurosciences, Level 3 West Wing, Oxford University Hospitals NHS Trust, University of Oxford, Headley Way, Oxford, OX3 9DU, UK.FMRIB Centre, John Radcliffe Hospital, Oxford University, Headington, Oxford, OX3 9DU, UK.Nuffield Department of Clinical Neurosciences, Level 3 West Wing, Oxford University Hospitals NHS Trust, University of Oxford, Headley Way, Oxford, OX3 9DU, UK. Centro Hospitalar do Porto, Hospital de Santo António, Serviço de Neurologia, Porto, Portugal.Nuffield Department of Clinical Neurosciences, Level 3 West Wing, Oxford University Hospitals NHS Trust, University of Oxford, Headley Way, Oxford, OX3 9DU, UK.Neuroimmunology Group, Nuffield Department of Clinical Neurosciences, Level 6 West Wing, John Radcliffe Hospital, Headley Way, Oxford, OX3 9DU, UK.Neuroimmunology Group, Nuffield Department of Clinical Neurosciences, Level 6 West Wing, John Radcliffe Hospital, Headley Way, Oxford, OX3 9DU, UK.Nuffield Department of Clinical Neurosciences, Level 3 West Wing, Oxford University Hospitals NHS Trust, University of Oxford, Headley Way, Oxford, OX3 9DU, UK.Nuffield Department of Clinical Neurosciences, Level 3 West Wing, Oxford University Hospitals NHS Trust, University of Oxford, Headley Way, Oxford, OX3 9DU, UK. jacqueline.palace@ndcn.ox.ac.uk.

Pub Type(s)

Journal Article
Research Support, Non-U.S. Gov't

Language

eng

PubMed ID

26668077

Citation

Piccolo, L, et al. "Isolated New Onset 'atypical' Optic Neuritis in the NMO Clinic: Serum Antibodies, Prognoses and Diagnoses at Follow-up." Journal of Neurology, vol. 263, no. 2, 2016, pp. 370-379.
Piccolo L, Woodhall M, Tackley G, et al. Isolated new onset 'atypical' optic neuritis in the NMO clinic: serum antibodies, prognoses and diagnoses at follow-up. J Neurol. 2016;263(2):370-379.
Piccolo, L., Woodhall, M., Tackley, G., Juryńczyk, M., Kong, Y., Domingos, J., ... Palace, J. (2016). Isolated new onset 'atypical' optic neuritis in the NMO clinic: serum antibodies, prognoses and diagnoses at follow-up. Journal of Neurology, 263(2), pp. 370-379. doi:10.1007/s00415-015-7983-1.
Piccolo L, et al. Isolated New Onset 'atypical' Optic Neuritis in the NMO Clinic: Serum Antibodies, Prognoses and Diagnoses at Follow-up. J Neurol. 2016;263(2):370-379. PubMed PMID: 26668077.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Isolated new onset 'atypical' optic neuritis in the NMO clinic: serum antibodies, prognoses and diagnoses at follow-up. AU - Piccolo,L, AU - Woodhall,M, AU - Tackley,G, AU - Juryńczyk,M, AU - Kong,Y, AU - Domingos,J, AU - Gore,R, AU - Vincent,A, AU - Waters,P, AU - Leite,M I, AU - Palace,J, Y1 - 2015/12/14/ PY - 2015/10/08/received PY - 2015/11/17/accepted PY - 2015/11/16/revised PY - 2015/12/16/entrez PY - 2015/12/17/pubmed PY - 2016/12/15/medline KW - AQP4-Ab KW - MOG-Ab KW - Neuromyelitis optica KW - Optic neuritis KW - Visual loss SP - 370 EP - 379 JF - Journal of neurology JO - J. Neurol. VL - 263 IS - 2 N2 - Severe, recurrent or bilateral optic neuritis (ON) often falls within the neuromyelitis optica spectrum disorders (NMOSD), but the diagnosis can be particularly challenging and has important treatment implications. We report the features, course and outcomes of patients presenting with atypical ON when isolated at onset. We retrospectively analyzed 69 sequential patients referred to a single UK NMO center with isolated ON at onset. Aquaporin-4 antibody (AQP4-Ab) assessment was performed in all patients and IgG1 myelin-oligodenrocyte glycoprotein (MOG-Ab) in AQP4-Ab(neg) patients. 37 AQP4-Ab positive (AQP4-Ab(pos)) and 32 AQP4-Ab negative (AQP4-Ab(neg)) patients (8 with MOG-Ab) were identified. The AQP4-Ab(neg) group included heterogeneous diagnoses: multiple sclerosis (MS), NMO, relapsing isolated ON (RION), monophasic isolated ON and relapsing acute disseminated encephalomyelitis (ADEM)-like syndromes. Compared to AQP4-Ab(neg) patients, AQP4-Ab(pos) patients had a worse residual visual outcome from first attack (median VFSS 4 vs. 0, p = 0.010) and at last assessment (median VFSS 5 versus 2, p = 0.005). However, AQP4-Ab(neg) patients with RION also had poor visual outcome. Up to 35% of AQP4-Ab(neg) patients developed a LETM and two developed low positivity for AQP4-Ab over time. Eight AQP4-Ab(neg) patients (25%) were MOG-Ab positive, covering a range of phenotypes excluding MS; the first ON attack was often bilateral and most had relapsing disease with a poor final visual outcome [VFSS 4, range (0-6)]. In conlcusion, AQP4-Ab positivity is confirmed as a predictor of poor visual outcome but AQP4-Ab(neg) RION also had a poor visual outcome. Of those without AQP4-Ab, 25% had MOG-Ab and another 25% developed MS; thus, MOG-Ab is associated with AQP4-Ab(neg) non-MS ON. SN - 1432-1459 UR - https://www.unboundmedicine.com/medline/citation/26668077/Isolated_new_onset_'atypical'_optic_neuritis_in_the_NMO_clinic:_serum_antibodies_prognoses_and_diagnoses_at_follow_up_ L2 - https://dx.doi.org/10.1007/s00415-015-7983-1 DB - PRIME DP - Unbound Medicine ER -