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Age at craniosynostosis surgery and its impact on ophthalmologic diagnoses: A single-center retrospective review.

Age at Craniosynostosis Surgery and Its Impact on Ophthalmologic Diagnoses: A Single-Center Retrospective Review Sir: B et al.1 present a study they believe supports the hypothesis that timing of primary craniofacial repair does not impact ophthalmic outcomes in children with single-suture craniosynostosis. We believe the data do not support the conclusion, as the cohort studied had relatively late intervention. Craniofacial surgery was performed at a median age of 10 months (interquartile range, 7 to 12.7 months). Endoscopic repair, unlike cranial vault expansion, is ideally performed by 2 to 4 months of age to yield optimal results.2–4 One cannot conclude that “the timing of craniofacial repair does not impact ophthalmic outcomes” based on a study that does not include patients with earlier repair performed endoscopically. The authors themselves found that with each month of age at the time of craniofacial surgery, the odds of having a preoperative diagnosis of an ophthalmic abnormality increased by 6 percent. V-pattern strabismus, typically seen in patients with craniosynostosis,2–5 begins to present in the first year of life, does not disappear, and may worsen, particularly in the presence of anisometropia and astigmatism (associated with unicoronal synostosis)6 and with the development of amblyopia, which increased over time in the study by Bennett et al. as well. As would be predicted, the authors demonstrated a large increase in these disorders after cranial vault expansion. The authors report that 24.4 percent of their patients demonstrated an ophthalmic disorder after cranial vault expansion, 9.9 percent demonstrated strabismus (5.8 percent requiring strabismus surgery), 8.7 percent had amblyopia, and 14 percent were diagnosed with astigmatism. Postoperative ophthalmic disorders particularly impacted patients with unicoronal synostosis (OR, 7.1; p < 0.001) and metopic synostosis (OR, 5.6; p < 0.001). The odds ratio of having strabismus was 17.25 (p = 0.001) for patients with unicoronal synostosis following cranial vault expansion, an association noted by others.2–5 This does not imply that cranial vault surgery caused the strabismus. Although the procedure itself may result in posterior slippage of the trochlea with secondary superior oblique palsy and V-pattern strabismus,7 the passage of time itself is a likely culprit. Strabismus tends to worsen over time, particularly in the presence of anisometropic astigmatism so common in patients with unicoronal synostosis, particularly if amblyopia and suppression begin to occur. This type of strabismus is not comparable to the kind of transient intermittent strabismus of infancy noted by Dr. Fearon in his commentary on the article by Bennett et al. In our most recent retrospective study on 94 patients treated for unicoronal synostosis with either endoscopic strip craniectomy (n = 60) performed typically before 4 month of age, or fronto-orbital advancement (n = 34) performed close to 1 year of age, we found after 3 to 5 years of follow-up, 32 percent of patients treated by endoscopic strip craniectomy (and orthosis) and 65 percent of those treated by frontoorbital advancement developed clinically significant strabismus requiring surgical repair (p = 0.002).4 This confirmed our earlier reports demonstrating that treatment by fronto-orbital advancement rather than endoscopic strip craniectomy was associated with more severe strabismus, and the need for strabismus surgical repair.2,3 It is noteworthy that those treated endoscopically had a lower mean operative time (42 ± 12 minutes versus 215 ± 50 minutes), length of stay (1.1 days versus 4.0 days), estimated blood loss (26 ± 10 cc versus 275 ± 116 cc), and blood transfusion rate (0 percent versus 88 percent).4 Thus, the claim that increasing age at surgery is not associated with increasing risk of ophthalmic disorders is a conclusion based on appraisal of a population treated too late to demonstrate the potential benefits of truly early intervention. By close to 1 year of age, the “cat is out of the bag,” as strabismus, anisoastigmatism, and amblyopia have begun to evolve in a significant portion of this population. DOI: 10.1097/PRS.0000000000007111