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New-Onset Craniosynostosis Following Posterior Vault Distraction Osteogenesis
Youssef Tahiri, MD, CM, MSc, FRCSC, J. Thomas Paliga, Valery Shubinets, MD, Scott Barteltt, MD, FACS, Jesse A. Taylor, MD. University of Pennsylvania, Philadelphia, PA, USA.
New-Onset Craniosynostosis Following Posterior Vault Distraction Osteogenesis Tahiri Y, Paliga JP, Shubinets V, Bartlett SP, Taylor JA ABSTRACT Background: The aim of this study is to document the incidence of new-onset craniosynostosis (NOC) following posterior vault distraction osteogenesis (PVDO), to determine risk factors for the development of NOC, and to deduce the cranial ramifications of NOC. Methods: An IRB-approved retrospective review of all patients who underwent PVDO at the Children’s Hospital of Philadelphia was performed. Demographics, peri-operative data, and pre-operative and post-operative 3D-CT scans were analyzed. Suture patency pre- and post-operatively were recorded. Results: 30 patients underwent PVDO for suspected increased intracranial pressure and/or severe turribrachicephaly from 2008 to 2013. 24 patients had syndromic diagnoses. The average age at the time of PVDO was 2.03 years. Distraction distances range from 19 to 40 mm, with an average of 28.7mm. Among the 19 patients who had a patent lambdoid sutures prior to PVDO, new-onset lambdoid fusion was seen in 17 patients following PVDO (89.5%), while the suture remained open in two patients (10.5%). New-onset lambdoid fusion was not significantly associated with age at distraction (p=0.28), gender (p=0.47), length of distraction (p=0.93), or diagnosis (p=0.61). Similarly, new-onset sagittal synostosis was not associated with age at distraction (p=0.06), gender (p=0.64), length of distraction (p=0.83), or diagnosis (p=0.25). None of the patients who developed NOC had characteristic head shape changes such as mastoid bulges or scaphocephaly. Conclusion: New-onset lambdoid and sagittal synostosis occur frequently following PVDO. While the diagnosis of NOC is obvious radiographically, the clinical importance of the diagnosis morphometrically, neurodevelopmentally, and in cranial growth have yet to be fully investigated. Table 1: Demographics and diagnoses | | | | | N | % | Total Number of patients | 30 | | | Non-syndromic | 6 | 20 | | Apert | 6 | 20 | | Crouzon | 6 | 20 | | Saethre Chotzen | 6 | 20 | | Muenke | 3 | 10 | | Pfeiffer | 2 | 6.7 | | Others (Vacterl) | 1 | 3.3 | Gender | | | | Female | 17 | 56.6 | | Male | 13 | 43.3 | Mean age at PVDO | 2.03 years | | Previous Surgeries | | | | No | 20 | 66.6 | | Yes (FOA) | 10 | 33.3 | Indications for Surgery | | | | | Concern for IH | 13 | 43.3 | | Turribrachycephaly | 12 | 40 | | Concern for IH and Turribrachycephaly | 5 | 16.7 | Average distraction distance | 28.7mm | | Tab le 2: Lambdoid and Sagittal cranial suture patency status prior to and following PVDO | | | | | Lambdoid (N) | Sagittal (N) | Prior to PVDO | | | | Open | 19 | 17 | | Closed | 11 | 13 | After PVDO | | | | Open | 2 | 10 | | Closed | 28 | 20 | Table 3: Variables tested for correlation with the development of NOC | | | | P-value | Risk Factors | NOC - Lambdoid | NOC - Sagittal | Age | 0.28 | 0.06 | Gender | 0.47 | 0.64 | Length of Distraction | 0.93 | 0.83 | Diagnosis (syndromic vs. non-syndromic) | 0.61 | 0.25 |
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