Clinical features of syndromic microphthalmia in two novel RARB variants

Here we describe unrelated Japanese patients with distinct novel heterozygous retinoic acid receptor beta (RARB) gene variants underlying syndromic microphthalmia-12: case 1 with a frameshift variant, c.1205_1206del, had bilateral microphthalmia, corneal opacity, anterior segment dysgenesis, widespread multiorgan anomalies, hypotonia and cognitive impairment; case 2 with a missense variant, c.844G>T had Peters anomaly, extreme microphthalmia, spasticity, profound psychomotor delay and refractory epilepsy. These findings highlight the need for RARB testing.

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Publication Details

Journal
Human Genome Variation
Published
2026-04-06
DOI
https://doi.org/10.1038/s41439-026-00345-3
Primary Topic
Ocular Disorders and Treatments
Type
article
Field-Weighted Citation Impact
0.00
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Clinical features of syndromic microphthalmia in two novel RARB variants

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Human Genome Variation
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Clinical features of syndromic microphthalmia in two novel RARB variants

Rika Kosaki, Yoshito Koyanagi, Tomoyo Yoshida, Hazuki Morikawa-Anzai, Keiko Matsubara, Maki Fukami, Yuichi Abe, Makiko Tominaga, Sachiko Nishina
article en

Abstract

Here we describe unrelated Japanese patients with distinct novel heterozygous retinoic acid receptor beta (RARB) gene variants underlying syndromic microphthalmia-12: case 1 with a frameshift variant, c.1205_1206del, had bilateral microphthalmia, corneal opacity, anterior segment dysgenesis, widespread multiorgan anomalies, hypotonia and cognitive impairment; case 2 with a missense variant, c.844G>T had Peters anomaly, extreme microphthalmia, spasticity, profound psychomotor delay and refractory epilepsy. These findings highlight the need for RARB testing.

Human Genome Variation
Kyushu University (JP), SHOWA Medical University Northern Yokohama Hospital (JP), National Center For Child Health and Development (JP)
Openalex Percentile: Top 19%
Ocular Disorders and Treatments
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