Cerebral blood flow in children with syndromic craniosynostosis: cohort arterial spin labeling studies

Author:

Doerga Priya N.1,Lequin Maarten H.2,Dremmen Marjolein H. G.3,den Ottelander Bianca K.1,Mauff Katya A. L.4,Wagner Matthias W.567,Hernandez-Tamames Juan A.3,Versnel Sarah L.1,Joosten Koen F. M.8,van Veelen Marie-Lise C.9,Tasker Robert C.10,Mathijssen Irene M. J.1

Affiliation:

1. Department of Plastic and Reconstructive Surgery and Hand Surgery, Dutch Craniofacial Center,

2. Department of Radiology, University Medical Center Utrecht, The Netherlands;

3. Department of Radiology,

4. Department of Biostatistics,

5. Department of Radiology and Radiological Science, Section of Pediatric Neuroradiology, Division of Pediatric Radiology, The Johns Hopkins University School of Medicine, Baltimore, Maryland;

6. Institute of Diagnostic and Interventional Radiology, University Hospital Zurich, University of Zurich, Switzerland;

7. Department of Diagnostic Imaging, Division of Neuroradiology, The Hospital for Sick Children, Toronto, ON, Canada; and

8. Pediatric Intensive Care Unit, and

9. Department of Neurosurgery, Sophia Children’s Hospital, Erasmus MC, University Medical Center Rotterdam;

10. Departments of Neurology and Anesthesiology (Pediatrics), Harvard Medical School and Boston Children’s Hospital, Boston, Massachusetts

Abstract

OBJECTIVEIn comparison with the general population, children with syndromic craniosynostosis (sCS) have abnormal cerebral venous anatomy and are more likely to develop intracranial hypertension. To date, little is known about the postnatal development change in cerebral blood flow (CBF) in sCS. The aim of this study was to determine CBF in patients with sCS, and compare findings with control subjects.METHODSA prospective cohort study of patients with sCS using MRI and arterial spin labeling (ASL) determined regional CBF patterns in comparison with a convenience sample of control subjects with identical MRI/ASL assessments in whom the imaging showed no cerebral/neurological pathology. Patients with SCS and control subjects were stratified into four age categories and compared using CBF measurements from four brain lobes, the cerebellum, supratentorial cortex, and white matter. In a subgroup of patients with sCS the authors also compared longitudinal pre- to postoperative CBF changes.RESULTSSeventy-six patients with sCS (35 female [46.1%] and 41 male [53.9%]), with a mean age of 4.5 years (range 0.2–19.2 years), were compared with 86 control subjects (38 female [44.2%] and 48 male [55.8%]), with a mean age of 6.4 years (range 0.1–17.8 years). Untreated sCS patients < 1 year old had lower CBF than control subjects. In older age categories, CBF normalized to values observed in controls. Graphical analyses of CBF by age showed that the normally expected peak in CBF during childhood, noted at 4 years of age in control subjects, occurred at 5–6 years of age in patients with sCS. Patients with longitudinal pre- to postoperative CBF measurements showed significant increases in CBF after surgery.CONCLUSIONSUntreated patients with sCS < 1 year old have lower CBF than control subjects. Following vault expansion, and with age, CBF in these patients normalizes to that of control subjects, but the usual physiological peak in CBF in childhood occurs later than expected.

Publisher

Journal of Neurosurgery Publishing Group (JNSPG)

Subject

General Medicine

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