Jordan W Swanson1, Wen Xu1, Gui-Shuang Ying2, Wei Pan2, Shih-Shan Lang3, Gregory G Heuer3, Scott P Bartlett1, Jesse A Taylor4. 1. Craniofacial Surgery Center and Division of Plastic Surgery, The Children's Hospital of Philadelphia and the University of Pennsylvania, Philadelphia, PA, USA. 2. Scheie Eye Institute and the Department of Ophthalmology, Perelman Center for Advanced Medicine and the University of Pennsylvania, Philadelphia, PA, USA. 3. Division of Neurosurgery, The Children's Hospital of Philadelphia and the University of Pennsylvania, Philadelphia, PA, USA. 4. Craniofacial Surgery Center and Division of Plastic Surgery, The Children's Hospital of Philadelphia and the University of Pennsylvania, Philadelphia, PA, USA. taylorj5@email.chop.edu.
Abstract
BACKGROUND: Better understanding the incidence and patterns of elevated intracranial pressure (ICP) in patients with craniosynostosis may facilitate more timely intervention to alter neurocognitive outcomes. Spectral-domain optical coherence tomography (OCT) of the retina can non-invasively diagnose elevated ICP, and has demonstrated high sensitivity and specificity among patients with craniosynostosis. This study sought to characterize patterns of elevated ICP among patients with craniosynostosis. METHODS: Quantitative retinal parameters were prospectively assessed in both eyes of patients with craniosynostosis using spectral-domain OCT. Based on retinal OCT thresholds associated with elevated ICP (> 15 mmHg), subjects were assigned an OCT diagnosis of elevated or non-elevated ICP which was analyzed relative to clinical characteristics and craniosynostosis patterns. RESULTS: Eighty subjects (aged 0.2-18 years) with craniosynostosis were enrolled; among these, 67 (84%) were nonsyndromic. OCT evaluation was performed at initial vault expansion in 56 (70%) patients. Among this subset, 27 (48%) patients had peri-papillary changes suggestive of elevated ICP, reflecting a 44% incidence in nonsyndromic and 83% in syndromic patients. The median age at initial vault expansion was higher among those with elevated ICP (11.1 months) than those without (7.8 months; p = 0.04.) Multi-suture synostosis was associated with changes consistent with elevated ICP in 9 (75%) patients compared with 18 (41%) with single suture synostosis (p = 0.05). CONCLUSIONS: OCT of the retina produces a potentially sensitive indicator of ICP in craniosynostosis patients. Elevated ICP may be associated with number of involved sutures and older patient presentation, and refining appropriate "cutoffs" will be important as the technology becomes more widespread.
BACKGROUND: Better understanding the incidence and patterns of elevated intracranial pressure (ICP) in patients with craniosynostosis may facilitate more timely intervention to alter neurocognitive outcomes. Spectral-domain optical coherence tomography (OCT) of the retina can non-invasively diagnose elevated ICP, and has demonstrated high sensitivity and specificity among patients with craniosynostosis. This study sought to characterize patterns of elevated ICP among patients with craniosynostosis. METHODS: Quantitative retinal parameters were prospectively assessed in both eyes of patients with craniosynostosis using spectral-domain OCT. Based on retinal OCT thresholds associated with elevated ICP (> 15 mmHg), subjects were assigned an OCT diagnosis of elevated or non-elevated ICP which was analyzed relative to clinical characteristics and craniosynostosis patterns. RESULTS: Eighty subjects (aged 0.2-18 years) with craniosynostosis were enrolled; among these, 67 (84%) were nonsyndromic. OCT evaluation was performed at initial vault expansion in 56 (70%) patients. Among this subset, 27 (48%) patients had peri-papillary changes suggestive of elevated ICP, reflecting a 44% incidence in nonsyndromic and 83% in syndromicpatients. The median age at initial vault expansion was higher among those with elevated ICP (11.1 months) than those without (7.8 months; p = 0.04.) Multi-suture synostosis was associated with changes consistent with elevated ICP in 9 (75%) patients compared with 18 (41%) with single suture synostosis (p = 0.05). CONCLUSIONS: OCT of the retina produces a potentially sensitive indicator of ICP in craniosynostosispatients. Elevated ICP may be associated with number of involved sutures and older patient presentation, and refining appropriate "cutoffs" will be important as the technology becomes more widespread.
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