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PURPOSE To describe "neonatal-onset congenital ectropion uveae (N-CEU)" as a distinct clinical entity of newborn glaucoma (NG), and study its significance towards the severity and outcome of NG. DESIGN Prospective clinical cohort study METHODS: Setting: Tertiary care Postgraduate Teaching Institute PATIENT POPULATION: Consecutive patients of NG who presented between 1st July 2016 to 30th September 2017, with a minimum post-operative follow up of 1 year. Babies with any ocular anomaly apart from CEU were excluded. N-CEU were compared with those with neonatal-onset primary congenital glaucoma(N-PCG) INTERVENTION PROCEDURES: All babies underwent goniotomy or trabeculotomy with trabeculectomy depending upon corneal clarity. MAIN OUTCOME MEASURES Clinical features at presentation and outcome after one year of surgery (defined as good or satisfactory if intraocular pressure (IOP) was ≤16.0 mm Hg under anaesthesia without or with topical medications, respectively, and poor if the baby required additional surgery). RESULTS Twenty eyes of 10 patients with N-CEU were compared with 16 eyes of 9 patients with N-PCG. N-CEU babies had significantly worse corneal clarity(mean grade 2.0±0.7 vs.1.4±0.8; p=0.026), and poorer outcome compared to N-PCG. 7 of 16(43.7%) eyes with N-PCG had a cornea clear enough at presentation for a goniotomy, compared to only 2 of the 20(10%) eyes with N-CEU(P=0.026). 13 of 16(81.2%) N-PCG eyes had a good or satisfactory outcome, compared to 6 of 20 (30%) N-CEU eyes(p=0.001). CONCLUSIONS Neonatal-onset CEU appears to be distinct from the unilateral CEU in older patients described in literature, and may be considered a poorer prognosis phenotype of neonatal-onset glaucoma.