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Post suturectomy site re-ossification (PSRO) in a mouse model of Saethre-Chotzen syndrome

Jack E. Brooker Michael Bykowski Ethan Hurley Gregory M. Cooper Joseph E. Losee
University of Pittsburgh
2018-01-30

Presenter: Jack E. Brooker

Affidavit:
This work has neither been presented nor published. Michael Bykowski was instrumental in the conception and surgical work in this study and Jack E. Brooker performed all analytical work and prepared the abstract.

Director Name: Vu Nguyen

Author Category: Fellow Plastic Surgery
Presentation Category: Basic Science Research
Abstract Category: Craniomaxillofacial

Background: Craniosynostosis (CS) frequently requires surgical suturectomy. PSRO can require reoperation, associated with more severe complications than first-round suturectomy. PSRO is well-recognized with rates of 5-13% for single suture CS and much higher (11-65%) for syndromic CS.

Methods: TWIST+/- mice carry a heterozygotic mutation for twist basic helix-loop-helix transcription factor 1 and are a model for Saethre-Chotzen syndrome. Individuals underwent coronal suturectomy. Care was taken not to tear the dura. C57BL/6J (WT) acted as controls. TWIST heterozygotes were identified though evaluation of their calvaria per previous methods published by our team.
At 2 weeks, 4 weeks, 8 weeks and 12 weeks postoperatively, mice underwent micro-CT evaluation of their calvarial defect and were euthanized. CTs were analyzed using osiriX (Špixmeo SARL, Switzerland) to calculate and the percentage re-ossification.

Results: Mean suture re-ossification at 12 weeks was 55.1% for WT mice (n=10) and 38.7% for TWIST mice (n=6) (p<0.05). At 2 weeks, re-ossification was greater in WT mice (45.6%, n=3) than TWIST mice (35.1%, n=2) though the difference was not significant. PSRO was greater at 4 weeks among TWIST mice (16.3%, n=2) vs WT mice (7.6%, n=2) though again this was non-significant.

Conclusion: Early time points have low sample sizes. However, among the 12-week group which has a larger sample size, WT mice show significantly greater bone regeneration than the model of Saethre-Chotzen syndrome used in this study. Additional samples at earlier time points are being analyzed. We will perform histology on re-ossified sutures.

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