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Nonsyndromic Single-Suture Metopic Craniosynostosis in the Fourth Dimension: An Evaluation of Complications, Revisions, and Long-Term Outcomes in 142 Patients Over 30 Years
J T. Paliga, BA, Jesse Goldstein, MD, Ari Wes, BA, Linton A. Whitaker, MD, Scott Bartlett, MD, Jesse A. Taylor, MD.
University of Pennsylvania and Children's Hospital of Philadelphia, Philadelphia, PA, USA.

PURPOSE: This study was designed to evaluate the long-term outcomes of patients with isolated metopic synostosis throughout the evolution of treatment at a single institution.
METHODS:An IRB approved retrospective chart review was performed on consecutive patients undergoing correction of metopic craniosynostosis from May 1978 to June 2012 at the Children’s Hospital of Philadelphia. Inclusion criteria required a diagnosis of metopic craniosynostosis, complete medical record and at least one year of follow-up. Patients were excluded if they had undergone previous interventions at other institutions or were diagnosed with syndromic or multisutural craniosynostosis. Demographic information, operative details, and post-operative course were collected. Outcomes were reported as Whitaker classification and post-operative clinical characteristics as assessed at the most recent clinical evaluation prior to any additional interventions, as well as the need for reoperation in patients with greater than 5 years of follow-up. Chi-squared and Fisher’s exact test were used to compare all categorical variables. Wilcoxon rank-sum and Kruskal-Wallis rank tests were used to compare all continuous variables.
RESULTS:From 1978 to 2012, 142 patients underwent surgical correction of isolated metopic craniosynostosis of which 113 met inclusion criteria. Average age of surgery was 0.83 (range: 0.3-4.7) years, and average follow-up was 6.0 (range: 1.0-17.8) years. There were ten surgical complications (8.8%). Three (2.6%) were considered major complications including one post-operative mortality in a patient with hypoplastic left heart syndrome and two reoperations for a post-operative hematoma and post-operative infection. Minor complications occurred in 7 (6.1%) patients and included hematoma (n=2), seroma (n=2), wound infection (n=1). Thirteen (21.0%) patients required more than one surgical intervention during the follow up period including complete revision in 2 (3.2%), removal of hardware in six (9.7%), bony recontouring in five (8.1%), and soft tissue augmentation in 1 (1.6%) patient. At follow-up, 63 (55.8%) patients were classified as Whitaker I, 6 (5.3%) were class II, 41 (36.3%) class III, and 3 (2.7%) class IV. Patients noted to have temporal hollowing (p=0.001) or lateral orbital retrusion (p<0.001) were associated with higher Whitaker classification, and this correlated with an increased number of interventions over time (p=0.0012). Patients were stratified based on length of follow up to determine outcome changes over time. Compared to patients with less than 5 years follow up (n=59), patients with greater than 5 years follow-up (n=54) were more likely to have temporal hollowing (OR 3.6, 95%CI 1.7-7.9, p=0.001), lateral orbital retrusion (OR 7.4, 95%CI 3.2-17.2, p<0.0001), and be classified as Whitaker III or IV (OR 6.2, 95%CI 2.6-14.5, p<0.0001).
CONCLUSION:These data represent the largest outcome evaluation of isolated metopic synostosis to date. We characterize the complication and reoperation rates in this population of patients and demonstrate a clear trend toward worsening outcomes over time. These data may change the way we counsel families as well as underscore the need to follow these patients to physical maturity.


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