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Ophthalmological findings in children with unicoronal craniosynostosis
Romain Touzé1,2, Giovanna Paternoster3, Eric Arnaud3,4
1Service d'ophtalmologie, 37072Hôpital Universitaire Necker - Enfants Malades, Paris, France.
Insights
Unicoronal craniosynostosis (UCS) in children often leads to significant ophthalmic issues like amblyopia and strabismus. Early and regular eye exams are crucial for timely diagnosis and preventing vision loss in these patients.
Area of Science:
- Ophthalmology
- Pediatrics
- Craniofacial Surgery
Background:
- Unicoronal craniosynostosis (UCS) is a common non-syndromic craniosynostosis with a high incidence of ophthalmic complications.
- Children with UCS face elevated risks of amblyopia, aniso-astigmatism, and strabismus, necessitating vigilant visual monitoring.
- Ophthalmic manifestations in UCS can significantly impact visual development and require specialized follow-up.
Purpose of the Study:
- To characterize the specific clinical ophthalmologic findings in a cohort of children diagnosed with isolated unicoronal craniosynostosis.
- To identify the prevalence and types of visual abnormalities associated with UCS in pediatric patients.
- To underscore the importance of comprehensive ophthalmological assessments for children with UCS.
Main Methods:
- Retrospective analysis of pediatric patients with isolated UCS treated between 2015 and 2021.
- Inclusion criteria required a complete ophthalmological assessment, including visual acuity, refractive status, and oculomotor examination.
- Exclusion of patients with associated craniofacial disorders to focus on isolated UCS.
Main Results:
- The study included 28 children, with a median age of 62 months; 86% were girls, and 71% had right-sided UCS.
- Amblyopia or a history of amblyopia was present in 36% of children.
- Higher astigmatism was noted on the side contralateral to the UCS (0.97 D) compared to the ipsilateral side (0.56 D, p=0.03).
- Strabismus, predominantly esotropia with a vertical component, affected 71% of patients, with pseudo-superior oblique palsy in 65%.
Conclusions:
- Children with unicoronal craniosynostosis exhibit a high frequency of diverse visual impairments.
- A rigorous ophthalmological follow-up is essential for early detection and management of visual complications in UCS patients.
- Prompt diagnosis and intervention are key to preventing long-term visual deficits in children with UCS.
Introduction:
Among non-syndromic, single-suture craniosynostoses, unicoronal craniosynostosis (UCS) presents the highest rate of ophthalmic manifestations requiring a visual follow-up, due to the high risk of amblyopia. After birth or during childhood, children with UCS have a high risk to present an aniso-astigmatism and a strabismus. The aim of this study was to characterize clinical ophthalmologic findings associated with UCS in a paediatric cohort.
Methods:
This retrospective study included children admitted in our unit between 2015 and 2021, with isolated UCS treated in our institution and complete ophthalmological assessment comprising visual assessment, refractive status and oculomotor examination. Children with associated craniofacial disorders were excluded.
Results:
A total of 28 children met the inclusion criteria. Median age was 62 [13-192] months with a large proportion of girls (86%) and 71% of right-sided UCS. The mean best corrected visual acuity was 0.07 (±0.13) LogMAR, including 10 (36%) children with an amblyopia or history of amblyopia. Astigmatism was significantly higher on the contralateral side of the UCS than on the ipsilateral side, with a refractive cylinder error of 0.97 (±1.06) vs 0.56 (±0.68) diopters, respectively (p = 0.03). Strabismus was observed in 20 patients (71%) with a main pattern of esotropia with a vertical component. A pseudo-superior oblique palsy was found in 13 children (65%) with a median cyclodeviation of 8.7° [-5,4°-20.6°].
Conclusion:
Children with UCS experience a high rate of various visual manifestations. This study highlights their need for a strict ophthalmological follow-up, in order to early diagnose and prevent visual complications.
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