Ocular Phenotypes and Novel SLC45A2 Variants in Patients with Oculocutaneous Albinism Type 4

Objectives: SLC45A2-related oculocutaneous albinism type 4 (OCA4) is a genetically defined subtype of albinism; however, its ocular phenotype remains incompletely characterized. This study aimed to describe the systemic, ophthalmic, and genetic features of patients with genetically confirmed SLC45A2-related OCA4. Methods: Ninety patients with clinically diagnosed albinism were enrolled and underwent genetic testing. Patients with genetically confirmed SLC45A2-related OCA4 were included for further analysis. Demographic information, systemic pigmentation features, ophthalmic findings, and SLC45A2 variants were analyzed. Results: Five unrelated patients with genetically confirmed SLC45A2-related OCA4 were included, aged 5–33 years. Best-corrected visual acuity ranged from 0.1 to 1.0 logMAR, and spherical equivalent refractive error varied widely from −7.25 to +7.63 D. All patients exhibited iris transillumination defects and moderate-to-severe fundus hypopigmentation, both graded 2–3. Foveal hypoplasia was universally present, ranging from grade 1 to grade 4, with three patients showing grade 4 hypoplasia. Genetic analysis revealed nine disease-associated alleles representing eight distinct SLC45A2 variants: seven missense variants and one splice-altering variant. One patient carried a homozygous variant, and four carried compound heterozygous variants. Three novel variants were identified: c.1459C>G (p.Gln487Glu), c.137A>G (p.Glu46Gly), and c.561A>G. Conclusions: This study characterizes the ocular phenotype of SLC45A2-related OCA4, including iris transillumination defects, fundus hypopigmentation, and foveal hypoplasia of varying severity. Three novel SLC45A2 variants were identified, expanding the mutational spectrum of OCA4.

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Publication Details

Journal
Genes
Published
2026-08-17
DOI
https://doi.org/10.3390/genes17080963
Primary Topic
melanin and skin pigmentation
Type
article
Field-Weighted Citation Impact
0.00

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article

Ocular Phenotypes and Novel SLC45A2 Variants in Patients with Oculocutaneous Albinism Type 4

Chonglin Chen, Junyi Liu, Bingqi Wang, Xinping Yu et al.
Genes
melanin and skin pigmentation
article

Ocular Phenotypes and Novel SLC45A2 Variants in Patients with Oculocutaneous Albinism Type 4

Chonglin Chen, Junyi Liu, Bingqi Wang, Xinping Yu, Ye Zheng
article en

Abstract

Objectives: SLC45A2-related oculocutaneous albinism type 4 (OCA4) is a genetically defined subtype of albinism; however, its ocular phenotype remains incompletely characterized. This study aimed to describe the systemic, ophthalmic, and genetic features of patients with genetically confirmed SLC45A2-related OCA4. Methods: Ninety patients with clinically diagnosed albinism were enrolled and underwent genetic testing. Patients with genetically confirmed SLC45A2-related OCA4 were included for further analysis. Demographic information, systemic pigmentation features, ophthalmic findings, and SLC45A2 variants were analyzed. Results: Five unrelated patients with genetically confirmed SLC45A2-related OCA4 were included, aged 5–33 years. Best-corrected visual acuity ranged from 0.1 to 1.0 logMAR, and spherical equivalent refractive error varied widely from −7.25 to +7.63 D. All patients exhibited iris transillumination defects and moderate-to-severe fundus hypopigmentation, both graded 2–3. Foveal hypoplasia was universally present, ranging from grade 1 to grade 4, with three patients showing grade 4 hypoplasia. Genetic analysis revealed nine disease-associated alleles representing eight distinct SLC45A2 variants: seven missense variants and one splice-altering variant. One patient carried a homozygous variant, and four carried compound heterozygous variants. Three novel variants were identified: c.1459C>G (p.Gln487Glu), c.137A>G (p.Glu46Gly), and c.561A>G. Conclusions: This study characterizes the ocular phenotype of SLC45A2-related OCA4, including iris transillumination defects, fundus hypopigmentation, and foveal hypoplasia of varying severity. Three novel SLC45A2 variants were identified, expanding the mutational spectrum of OCA4.

GenesVol. 17(8)
Sun Yat-sen University (CN)
National Natural Science Foundation of China, Guangdong Science and Technology Department, State Key Laboratory of Ophthalmology
Openalex Percentile: Top 13%
melanin and skin pigmentation
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