57 Cerebral Blood Flow | Trigonocephaly Supplemental table 4. Perfusion per brain region from the structural atlas in mL/100g/min. Trigonocephaly Control Brain Region mean sd se lower.ci upper.ci mean sd se lower.ci upper.ci Caudate 63.25 14.04 1.65 59.95 66.55 56.20 15.61 2.76 50.57 61.83 Cerebellum 73.96 15.67 1.85 70.28 77.64 66.47 21.88 3.87 58.58 74.36 Frontal lobe 72.97 16.77 1.98 69.03 76.91 70.51 19.97 3.53 63.31 77.71 Insula 79.93 15.96 1.88 76.18 83.68 73.34 21.95 3.88 65.43 81.25 Occipital lobe 80.32 20.95 2.47 75.40 85.25 71.60 18.20 3.22 65.04 78.16 Parietal lobe 77.49 18.37 2.16 73.18 81.81 72.94 17.35 3.07 66.68 79.20 Putamen 78.93 15.20 1.79 75.36 82.50 70.88 25.23 4.46 61.79 79.98 Temporal lobe 71.90 15.91 1.88 68.16 75.64 69.93 19.87 3.51 62.77 77.10 Thalamus 93.24 25.25 2.98 87.30 99.17 80.03 28.15 4.98 69.88 90.18 DISCUSSION The aim of this study was to investigate cerebral blood flow in the frontal lobe of patients with trigonocephaly as compared to age-matched control subjects. Our findings suggest that there is no significant difference in cerebral blood flow in trigonocephaly patients as compared to healthy control subjects. This is consistent with our hypothesis that there are no abnormalities in frontal lobe perfusion in trigonocephaly patients aged 0 to 18 months. This aligns with previous findings of very low risk to develop raised intracranial pressure in the first 18 months of life in trigonocephaly patients and that trigonocephaly patients have normal intracranial volume compared to control subjects without cerebral pathology.2, 3 Theoretically, it would be possible to have a normal cerebral blood flow but a raised intracranial pressure, which may be compensated by reduced cerebrovascular resistance. However, as trigonocephaly patients rarely have increased intracranial pressure, we do not expect that this is the reason for a normal cerebral blood flow in our cohort. The functional indication for surgery of trigonocephaly has been debated among craniofacial surgeons for a few years. Whether premature closure of the metopic suture restricts brain development mechanically and whether craniofacial surgery has a positive effect have never been proven. Our current finding of equal cerebral blood flow in trigonocephaly patients before surgery and controls implies that metopic synostosis does not impair perfusion in the forebrain up to 18 months of age and, thus, rejects the hypothesis of mechanical restriction of brain development. As with the theory that trigonocephaly is mainly an inborn brain disorder, recent studies have shown that some genetic mutations found in patients with trigonocephaly overlap with 3
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