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Genotype Associated With Visual Prognosis in Patients With Congenital Ectopia Lentis Following Lens Surgery: A Prospective Cohort Study.
Jia, Wan-Nan; Chen, Ze-Xu; Wang, Ya-Lei; Shen, Xin; Chen, Xin-Yao; Chen, Tian-Hui; Sun, Yang; Liu, Yan; Song, Ling-Hao; Huo, Qiu-Yi; Jiang, Yong-Xiang.
Afiliação
  • Jia WN; From the Eye Institute and Department of Ophthalmology (W.N.J., Z.X.C., Y.L.W., X.S., X.Y.C., T.H.C., Y.S., Y.L., L.H.S., Q.Y.H., and Y.X.J.), Eye & ENT Hospital, Fudan University, Shanghai China; NHC Key Laboratory of Myopia (Fudan University), Key Laboratory of Myopia (W.N.J., Z.X.C., Y.L.W.,
  • Chen ZX; From the Eye Institute and Department of Ophthalmology (W.N.J., Z.X.C., Y.L.W., X.S., X.Y.C., T.H.C., Y.S., Y.L., L.H.S., Q.Y.H., and Y.X.J.), Eye & ENT Hospital, Fudan University, Shanghai China; NHC Key Laboratory of Myopia (Fudan University), Key Laboratory of Myopia (W.N.J., Z.X.C., Y.L.W.,
  • Wang YL; From the Eye Institute and Department of Ophthalmology (W.N.J., Z.X.C., Y.L.W., X.S., X.Y.C., T.H.C., Y.S., Y.L., L.H.S., Q.Y.H., and Y.X.J.), Eye & ENT Hospital, Fudan University, Shanghai China; NHC Key Laboratory of Myopia (Fudan University), Key Laboratory of Myopia (W.N.J., Z.X.C., Y.L.W.,
  • Shen X; From the Eye Institute and Department of Ophthalmology (W.N.J., Z.X.C., Y.L.W., X.S., X.Y.C., T.H.C., Y.S., Y.L., L.H.S., Q.Y.H., and Y.X.J.), Eye & ENT Hospital, Fudan University, Shanghai China; NHC Key Laboratory of Myopia (Fudan University), Key Laboratory of Myopia (W.N.J., Z.X.C., Y.L.W.,
  • Chen XY; From the Eye Institute and Department of Ophthalmology (W.N.J., Z.X.C., Y.L.W., X.S., X.Y.C., T.H.C., Y.S., Y.L., L.H.S., Q.Y.H., and Y.X.J.), Eye & ENT Hospital, Fudan University, Shanghai China; NHC Key Laboratory of Myopia (Fudan University), Key Laboratory of Myopia (W.N.J., Z.X.C., Y.L.W.,
  • Chen TH; From the Eye Institute and Department of Ophthalmology (W.N.J., Z.X.C., Y.L.W., X.S., X.Y.C., T.H.C., Y.S., Y.L., L.H.S., Q.Y.H., and Y.X.J.), Eye & ENT Hospital, Fudan University, Shanghai China; NHC Key Laboratory of Myopia (Fudan University), Key Laboratory of Myopia (W.N.J., Z.X.C., Y.L.W.,
  • Sun Y; From the Eye Institute and Department of Ophthalmology (W.N.J., Z.X.C., Y.L.W., X.S., X.Y.C., T.H.C., Y.S., Y.L., L.H.S., Q.Y.H., and Y.X.J.), Eye & ENT Hospital, Fudan University, Shanghai China; NHC Key Laboratory of Myopia (Fudan University), Key Laboratory of Myopia (W.N.J., Z.X.C., Y.L.W.,
  • Liu Y; From the Eye Institute and Department of Ophthalmology (W.N.J., Z.X.C., Y.L.W., X.S., X.Y.C., T.H.C., Y.S., Y.L., L.H.S., Q.Y.H., and Y.X.J.), Eye & ENT Hospital, Fudan University, Shanghai China; NHC Key Laboratory of Myopia (Fudan University), Key Laboratory of Myopia (W.N.J., Z.X.C., Y.L.W.,
  • Song LH; From the Eye Institute and Department of Ophthalmology (W.N.J., Z.X.C., Y.L.W., X.S., X.Y.C., T.H.C., Y.S., Y.L., L.H.S., Q.Y.H., and Y.X.J.), Eye & ENT Hospital, Fudan University, Shanghai China; NHC Key Laboratory of Myopia (Fudan University), Key Laboratory of Myopia (W.N.J., Z.X.C., Y.L.W.,
  • Huo QY; From the Eye Institute and Department of Ophthalmology (W.N.J., Z.X.C., Y.L.W., X.S., X.Y.C., T.H.C., Y.S., Y.L., L.H.S., Q.Y.H., and Y.X.J.), Eye & ENT Hospital, Fudan University, Shanghai China; NHC Key Laboratory of Myopia (Fudan University), Key Laboratory of Myopia (W.N.J., Z.X.C., Y.L.W.,
  • Jiang YX; From the Eye Institute and Department of Ophthalmology (W.N.J., Z.X.C., Y.L.W., X.S., X.Y.C., T.H.C., Y.S., Y.L., L.H.S., Q.Y.H., and Y.X.J.), Eye & ENT Hospital, Fudan University, Shanghai China; NHC Key Laboratory of Myopia (Fudan University), Key Laboratory of Myopia (W.N.J., Z.X.C., Y.L.W.,
Am J Ophthalmol ; 268: 285-295, 2024 Aug 08.
Article em En | MEDLINE | ID: mdl-39121940
ABSTRACT

PURPOSE:

To investigate the relationship between visual prognosis and genotype in patients undergoing lens surgery for congenital ectopia lentis (EL).

DESIGN:

Prospective clinical cohort study.

METHODS:

Patients with congenital EL who underwent lens removal and intraocular lens implantation received panel-based next-generation sequencing. Patients were grouped into children and adolescents/adults based on the age at surgery. The visual prognosis, including best-corrected visual acuity (BCVA) and amblyopia, was stratified into short-term and medium to long-term.

RESULTS:

This study included 329 probands with congenital EL, with a median age at lens surgery of 7.00 years (interquartile range [IQR] = 5.00, 12.50 years). Children with the non-FBN1 mutation exhibited inferior medium to long-term postoperative BCVA (0.26 [IQR 0.14, 0.33] vs 0.15 [IQR 0.10, 0.22], P = .034) and a higher prevalence of amblyopia (44.4% vs 16.8%, P = .012) compared to those with FBN1 mutation. Multivariable analysis showed that genotype (FBN1 vs non-FBN1 mutation) was significantly associated with medium to long-term postoperative BCVA (b = -0.128, 95% CI -0.214 to -0.042, P = .004) and amblyopia (OR = 0.20, 95% CI 0.05-0.78, P = .020) in children. Further classification of FBN1 genotype did not yield significant correlations with visual prognosis. However, no significant correlation was observed between genotype and short-term visual prognosis in the children. Children with less severe EL (OR = 0.13, 95% CI 0.02-0.85, P = .033) had lower risks of amblyopia in the short-term follow-up. For adolescent and adult patients with congenital EL, those with poor preoperative BCVA and long axial length should be informed of suboptimal visual prognosis.

CONCLUSIONS:

Genotype significantly influences the medium to long-term visual prognosis in children with congenital EL. Genotype, along with preoperative BCVA, may assist in establishing reasonable expectations for patients regarding their visual outcomes after the lens surgery.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Idioma: En Revista: Am J Ophthalmol Ano de publicação: 2024 Tipo de documento: Article
Texto completo
Imprimir
XML
PubMed Links
Buscar no Google

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Idioma: En Revista: Am J Ophthalmol Ano de publicação: 2024 Tipo de documento: Article