TY - JOUR
T1 - Assessment of pediatric optic neuritis visual acuity outcomes at 6 months
AU - Writing Committee for the Pediatric Eye Disease Investigator Group (PEDIG)
AU - Pineles, Stacy L.
AU - Repka, Michael X.
AU - Liu, Grant T.
AU - Waldman, Amy T.
AU - Borchert, Mark S.
AU - Khanna, Sangeeta
AU - Heidary, Gena
AU - Graves, Jennifer S.
AU - Shah, Veeral S.
AU - Kupersmith, Mark J.
AU - Kraker, Raymond T.
AU - Wallace, David K.
AU - Cotter, Susan A.
AU - Holmes, Jonathan M.
AU - Chang, Melinda Y.
AU - Contractor, Dilshad
AU - Zolfaghari, Emily J.
AU - Vyas, Aarti
AU - Yuen, Tiffany
AU - Paysse, Evelyn A.
AU - Romany, Gihan
AU - Peragallo, Jason H.
AU - Brower, Judy L.
AU - Raghuram, Aparna
AU - AlWattar, Bilal
AU - Chinn, Ryan
AU - Kothari, Srishti
AU - Michael Siatkowski, R.
AU - Collinge, Janine E.
AU - Lim, Maria E.
AU - Brewer, Alisha N.
AU - Doughty, Annette M.
AU - Icks, Sonny W.
AU - Almeida, Shannon
AU - de Alba Campomanes, Alejandra
AU - Banwait, Premilla
AU - Hajkazemshirazi, Leila
AU - Bastea-Forte, Yizhuo
AU - Arjona, Jennifer K.
AU - Chen, Jeremy
AU - Cooper, Karen
AU - Ghadban, Rafif
AU - Chung, Sophia M.
AU - Cruz, Oscar A.
AU - Christenson, Traci A.
AU - Breeding, Lisa L.
AU - Govreau, Dawn M.
AU - Wallis, Beth A.
AU - Geddie, Brooke E.
AU - Diener-West, Marie
N1 - Publisher Copyright:
© 2020 American Medical Association. All rights reserved.
Copyright:
Copyright 2020 Elsevier B.V., All rights reserved.
PY - 2020
Y1 - 2020
N2 - IMPORTANCE Optic neuritis (ON) in children is uncommon. There are limited prospective data for visual acuity (VA) outcomes, associated diseases, and neuroimaging findings. Prospective data from a large sample would be useful for counseling families on treatment decisions and prognosis. OBJECTIVE To prospectively study children with a first episode of ON, describe VA after 6 months, and ascertain the network's (Pediatric Eye Disease Investigator Group and Neuro-Ophthalmology Research Disease Investigator Consortium) ability to enroll pediatric patients with ON prospectively. DESIGN, SETTING, AND PARTICIPANTS This nonrandomized cohort studywas conducted from September 20, 2016, to July 20, 2018, at 23 sites in the United States and Canada in pediatric ophthalmology or neuro-ophthalmology clinics. A total of 44 children (aged 3-15 years) presented with a first episode of ON (visual loss, pain on eye movements, or both) within 2 weeks of symptom onset and at least 1 of the following in the affected eye: a distance high-contrast VA (HCVA) deficit of at least 0.2 logMAR below age-based norms, diminished color vision, abnormal visual field, or optic disc swelling. Exclusion criteria included preexisting ocular abnormalities or a previous episode of ON. MAIN OUTCOMES AND MEASURES Primary outcomeswere monocular HCVA and low-contrast VA at 6 months. Secondary outcomes were neuroimaging, associated diagnoses, and antibodies for neuromyelitis optica andmyelin oligodendrocyte glycoprotein. RESULTS A total of 44 children (mean age [SD], 10.2 [3.5] years; 26 boys [59%]; 23 White individuals [52%]; 54 eyes) were enrolled in the study. Sixteen patients (36%) had bilateral ON. Magnetic resonance imaging revealed white matter lesions in 23 children (52%). Of these children, 8 hadmyelin oligodendrocyte glycoprotein-associated demyelination (18%), 7 had acute disseminated encephalomyelitis (16%), 5 had multiple sclerosis (11%), and 3 had neuromyelitis optica (7%). The baseline mean HCVA was 0.95 logMAR (20/200), which improved by a mean 0.76 logMAR (95%CI, 0.54-0.99; range, -0.70 to 1.80) to 0.12 logMAR (20/25) at 6 months. The baseline mean distance low-contrast VA was 1.49 logMAR (20/640) and improved by a mean 0.72 logMAR (95%CI, 0.54-0.89; range, -0.20 to 1.50) to 0.73 logMAR (20/100) at 6 months. Baseline HCVA was worse in younger participants (aged <10 years) with associated neurologic autoimmune diagnoses, white matter lesions, and in those of non-White race and non-Hispanic ethnicity. The data did not suggest a statistically significant association between baseline factors and improvement in HCVA. CONCLUSIONS AND RELEVANCE The study network did not reach its targeted enrollment of 100 pediatric patients with ON over 2 years. This indicates that future treatment trials may need to use different inclusion criteria or plan a longer enrollment period to account for the rarity of the disease. Despite poor VA at presentation, most children had marked improvement by 6 months. Associated neurologic autoimmune diagnoses were common. These findings can be used to counsel families about the disease.
AB - IMPORTANCE Optic neuritis (ON) in children is uncommon. There are limited prospective data for visual acuity (VA) outcomes, associated diseases, and neuroimaging findings. Prospective data from a large sample would be useful for counseling families on treatment decisions and prognosis. OBJECTIVE To prospectively study children with a first episode of ON, describe VA after 6 months, and ascertain the network's (Pediatric Eye Disease Investigator Group and Neuro-Ophthalmology Research Disease Investigator Consortium) ability to enroll pediatric patients with ON prospectively. DESIGN, SETTING, AND PARTICIPANTS This nonrandomized cohort studywas conducted from September 20, 2016, to July 20, 2018, at 23 sites in the United States and Canada in pediatric ophthalmology or neuro-ophthalmology clinics. A total of 44 children (aged 3-15 years) presented with a first episode of ON (visual loss, pain on eye movements, or both) within 2 weeks of symptom onset and at least 1 of the following in the affected eye: a distance high-contrast VA (HCVA) deficit of at least 0.2 logMAR below age-based norms, diminished color vision, abnormal visual field, or optic disc swelling. Exclusion criteria included preexisting ocular abnormalities or a previous episode of ON. MAIN OUTCOMES AND MEASURES Primary outcomeswere monocular HCVA and low-contrast VA at 6 months. Secondary outcomes were neuroimaging, associated diagnoses, and antibodies for neuromyelitis optica andmyelin oligodendrocyte glycoprotein. RESULTS A total of 44 children (mean age [SD], 10.2 [3.5] years; 26 boys [59%]; 23 White individuals [52%]; 54 eyes) were enrolled in the study. Sixteen patients (36%) had bilateral ON. Magnetic resonance imaging revealed white matter lesions in 23 children (52%). Of these children, 8 hadmyelin oligodendrocyte glycoprotein-associated demyelination (18%), 7 had acute disseminated encephalomyelitis (16%), 5 had multiple sclerosis (11%), and 3 had neuromyelitis optica (7%). The baseline mean HCVA was 0.95 logMAR (20/200), which improved by a mean 0.76 logMAR (95%CI, 0.54-0.99; range, -0.70 to 1.80) to 0.12 logMAR (20/25) at 6 months. The baseline mean distance low-contrast VA was 1.49 logMAR (20/640) and improved by a mean 0.72 logMAR (95%CI, 0.54-0.89; range, -0.20 to 1.50) to 0.73 logMAR (20/100) at 6 months. Baseline HCVA was worse in younger participants (aged <10 years) with associated neurologic autoimmune diagnoses, white matter lesions, and in those of non-White race and non-Hispanic ethnicity. The data did not suggest a statistically significant association between baseline factors and improvement in HCVA. CONCLUSIONS AND RELEVANCE The study network did not reach its targeted enrollment of 100 pediatric patients with ON over 2 years. This indicates that future treatment trials may need to use different inclusion criteria or plan a longer enrollment period to account for the rarity of the disease. Despite poor VA at presentation, most children had marked improvement by 6 months. Associated neurologic autoimmune diagnoses were common. These findings can be used to counsel families about the disease.
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U2 - 10.1001/jamaophthalmol.2020.4231
DO - 10.1001/jamaophthalmol.2020.4231
M3 - Article
C2 - 33057592
AN - SCOPUS:85094201338
JO - JAMA Ophthalmology
JF - JAMA Ophthalmology
SN - 2168-6165
ER -