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What is the Optimal Age for Cranial Vault Remodeling in Syndromic Craniosynostosis? Insights from the Johns Hopkins Experience
Alan F. Utria, BA1, Gerhard S. Mundinger, MD1, Joy Zhou, Undergraduate Student2, Ali Ghasemzadeh, BS1, Robin Yang, MD DDS3, Amir H. Dorafshar, MBChB1.
1Johns Hopkins School of Medicine, Baltimore, MD, USA, 2Johns Hopkins University, Baltimore, MD, USA, 3University of Maryland School of Medicine, Baltimore, MD, USA.
Optimal timing of cranial vault remodeling in syndromic patients with craniosynostosis is controversial. The purpose of this study was to gain insight into the impact of age at repair on relapse rates through the Johns Hopkins experience.
Retrospective chart review was performed for 58 patients surgically treated for syndromic craniosynostosis at Johns Hopkins Hospital between 1990-2013. Patient demographics, suture involvement, age at surgery, syndrome, surgical management, hospital course, and complications were recorded. Surgical procedures were assigned a Whitaker category based on need for reoperation as follows: Category I signified no additional surgery required, Category II signified soft tissue and lesser bone contouring revisions required, Category III signified major alternative osteotomies or bone grafting required, and Category IV signified the need for major surgical revision essentially duplicating the original surgery. Multivariable logistic regression analysis was performed to determine the relationship between age at surgery and need for reoperation as categorized by the Whitaker scale, and to assign odds ratios (OR) for need for surgical revision by operative timepoint. Covariates included suture involvement, syndrome, race and gender.
58 patients undergoing a total of 71 cranial vault remodeling procedures for syndromic craniosynostosis were identified. Average follow up was 6 years (SD 5 years). Patient demographics were as follows: 32 male (55%), 26 female, 39(45%) Caucasian, 10 Black, and 9 of another race. Syndromes were comprised of Crouzons (n=14), Aperts (n=12), other (n =12), Pfiffers (n=10), undiagnosed (n=6), and Seathre-Chotzen (n=4). Average number of sutures involved was 2.4 (range 1 to 5) as follows: right coronal (n=37), left coronal (n=38), sagittal (n=18), metopic (n=11), left lambdoid (n=7), and right lambdoid (n=6). Whitaker category for the 71 procedures was as follows: 31 in category I, 10 in category II, 3 in category III, and 27 in category IV. Multivariable logistic regression analysis for the effect of age on reoperation revealed a greater odds of major reoperation (category IV) in patients with less than 3 months of age (OR 5.6, p=0.015, 95% CI: 1.4-24.7) and 3-6 months of age (OR 4.3, p=0.03, 95% CI: 1.2-16.1), and a greater odds of no reoperation necessary (category I) in patients 6-9 months of age (OR 7.0, p=0.006, 95% CI: 1.7-27.9). Patients older than 12 months of age had a greater odds (OR 8.4, p=0.011, 95% CI: 1.6-43.2) of requiring minor operative revisions (category II).
Timing of surgery is an important factor to consider when planning vault remodeling in syndromic craniosynostosis. We found that operating before 6 months of age had greater odds of requiring a complete revision, and that patients undergoing remodeling after 12 months of age were more likely to require minor revisions. In our experience, the ideal operative window that demonstrated the greatest odds of requiring no additional surgery was 6-9 months of age.
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