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Editorial. Head shape at 36 months with and without helmeting

Matthew D. Smyth

InJournal of Neurosurgery: Pediatrics Volume 21 (2018): Issue 3 (Mar 2018)

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Head shape at age 36 months among children with and without a history of positional skull deformation

Brent R. Collett, Brian G. Leroux, Erin R. Wallace, Emily Gallagher, Jason Shao, and Matthew L. Speltz

OBJECTIVE

In this study, the authors examined head shape through age 36 months for children with and without a history of positional plagiocephaly and/or brachycephaly (PPB).

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Infants with PPB (cases) were identified through a craniofacial clinic at the time of diagnosis. Infants without diagnosed PPB were identified through a participant registry. Clinician ratings of 3D cranial images were used to confirm the presence or absence of PPB. The cohort included 235 case infants (diagnosed PPB, confirmed with 3D imaging), 167 unaffected controls (no diagnosed PPB, no deformation detected), and 70 affected controls (no diagnosed PPB, discernible skull deformation). Participants were seen in infancy (age 7 months, on average) and again at ages 18 and 36 months. At each visit, automated 3D measures of skull deformation quantified posterior flattening and generated an absolute asymmetry score. The authors also used automated 2D measures to approximate overall asymmetry (approximate oblique cranial length ratio) and calculate the cephalic index. They used linear regression to compare cases to unaffected controls and to compare affected versus unaffected controls on all measures. They also calculated the proportion of children in each group with “persistent PPB,” defined as one or more head shape measures above the 95th percentile relative to unaffected controls at 36 months.

RESULTS

Head shape became more rounded and symmetric for children with and without PPB, particularly between infancy and age 18 months. However, children with PPB continued to show greater skull deformation and asymmetry than unaffected controls at age 36 months. These differences were large in magnitude, ranging from 1 to 2 standard deviations (SDs), and in most (85.6%) of the cases, there was evidence of persistent PPB at 36 months. Similarly, although differences were more modest (i.e., 0.26–0.94 SD), affected controls continued to exhibit skull deformation on most measures relative to unaffected controls and approximately 30% had persistent PPB. Within the case group, head shape at 36 months was similar for untreated patients with PPB and for those who received helmet treatment and for patients with and without a history of torticollis.

CONCLUSIONS

孩子们,虽然头形状继续改善with a history of skull deformation in infancy continue to exhibit measureable cranial flattening and asymmetry through age 36 months.

InJournal of Neurosurgery: Pediatrics Volume 21 (2018): Issue 3 (Mar 2018)

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Language, learning, and memory in children with and without single-suture craniosynostosis

Kathleen A. Kapp-Simon, Erin Wallace, Brent R. Collett, Mary Michaeleen Cradock, Canice E. Crerand, and Matthew L. Speltz

OBJECTIVE

The language and memory functions of children with and without single-suture craniosynostosis (SSC) were compared at school age (mean 7.45 years, standard deviation [SD] 0.54 years). The children in this cohort were originally recruited in infancy and prior to cranial surgery for those with SSC.

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Individual evaluations of 179 school-aged children with SSC and 183 controls were conducted (70% of the original cohort) using standardized measures of language, learning, and memory. Parents participated in an interview about specialized education interventions and school progress. Parents and teachers completed questionnaires about language development.

RESULTS

Children with SSC (cases) obtained lower scores than controls on all measures. The adjusted differences in language, learning, and memory scores were modest, with SD ranging from 0.0 to −0.4 (p values ranged from 0.001 to 0.99). Censored normal regression was used to account for intervention services received prior to the school-age evaluation; this increased case-control differences (SD range 0.1 to −0.5, p value range 0.001 to 0.50). Mean scores for cases in each SSC diagnostic group were lower than those for controls, with the greatest differences observed among children with unilateral coronal craniosynostosis.

CONCLUSIONS

Children with SSC continue to show poorer performance than controls on language, learning, and memory tasks at early elementary school age, even when controlling for known confounders, although mean differences are small. Multidisciplinary care, including direct psychological assessment, for children with SSC should extend through school age with a specific focus on language and conceptual learning, as these are areas of potential risk. Future research is needed to investigate language, memory, and learning for this population during the middle to high school years.

InJournal of Neurosurgery: Pediatrics Volume 17 (2016): Issue 5 (May 2016)

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Structural brain differences in school-age children with and without single-suture craniosynostosis

Kristina Aldridge, Brent R. Collett, Erin R. Wallace, Craig Birgfeld, Jordan R. Austin, Regina Yeh, Madison Feil, Kathleen A. Kapp-Simon, Elizabeth H. Aylward, Michael L. Cunningham, and Matthew L. Speltz

OBJECTIVE

Single-suture颅缝早闭(SSC), premature fusion of a cranial suture, is characterized by dysmorphology of the craniofacial skeleton. Evidence to suggest that children with SSC are at an elevated risk of mild to moderate developmental delays and neurocognitive deficits is mounting, but the associations among premature suture fusion, neuroanatomy, and neurocognition are unexplained. The goals of this study were to determine 1) whether differences in the brain are present in young children with the 2 most common forms of SSC (sagittal and metopic) several years following surgical correction, and 2) whether the pattern of differences varies by affected suture (sagittal or metopic). Examination of differences in the brains of children with SSC several years after surgery may illuminate the growth trajectory of the brain after the potential constraint of the dysmorphic cranium has been relieved.

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The authors compared quantitative measures of the brain acquired from MR images obtained from children with sagittal or metopic craniosynostosis (n = 36) at 7 years of age to those obtained from a group of unaffected controls (n = 27) at the same age. The authors measured the volumes of the whole brain, cerebral cortex, cerebral white matter, cerebral cortex by lobe, and ventricles. Additionally, they measured the midsagittal area of the corpus callosum and its segments and of the cerebellar vermis and its component lobules. Measurements obtained from children with SSC and controls were compared using linear regression models.

RESULTS

No volume measures of the cerebrum or of the whole brain differed significantly between patients with SSC and controls (p > 0.05). However, ventricle volume was significantly increased in patients with SSC (p = 0.001), particularly in those with sagittal craniosynostosis (p < 0.001). In contrast, the area of the corpus callosum was significantly reduced in patients with metopic synostosis (p = 0.04), particularly in the posterior segments (p = 0.004). Similarly, the area of lobules VI–VII of the cerebellar vermis was reduced in patients with SSC (p = 0.03), with those with metopic craniosynostosis showing the greatest reduction (p = 0.01).

CONCLUSIONS

The lack of differences in overall brain size or regional differences in the size of the lobes of the cerebrum in children with metopic and sagittal synostosis suggests that the elevated risk of neurodevelopmental deficits is not likely to be associated with differences in the cerebral cortex. Instead, this study showed localized differences between sagittal and metopic craniosynostosis cases as compared with controls in the ventricles and in the midsagittal structures of the corpus callosum and the cerebellum. It remains to be tested whether these structural differences are associated with the increased risk for developmental delay and neurocognitive deficits in children with SSC.

InJournal of Neurosurgery: Pediatrics Volume 19 (2017): Issue 4 (Apr 2017)

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Presurgical and postsurgical assessment of the neurodevelopment of infants with single-suture craniosynostosis: comparison with controls

Jacqueline R. Starr, Kathleen A. Kapp-Simon, Yona Keich Cloonan, Brent R. Collett, Mary Michaeleen Cradock, Lauren Buono, Michael L. Cunningham, and Matthew L. Speltz

Object

Although most infants with single-suture craniosynostosis (SSC) appear to have neurodevelopmental test scores in the average range, SSC has been associated with cognitive and motor delays during infancy. Whether and when surgery improves such deficits are not yet known. The authors aimed to compare the pre- and postsurgical neurodevelopmental status of patients with SSC with those of control infants without craniosynostosis.

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The authors conducted a large, multicenter, longitudinal study of 168 infants with craniosynostosis and 115 controls without synostosis who were of similar age, race, sex, and socioeconomic status. The authors assessed participants by using the Bayley Scales of Infant Development, Second Edition (BSID-II) and the Preschool Language Scale, Third Edition (PLS-3) at enrollment, before patients' intracranial surgery, and when participants were 18 months of age (after surgery for patients).

Results

After adjusting for potential confounding factors in linear regression analyses, the authors found a tendency for patients to perform similarly to or slightly worse than controls on neurodevelopmental examinations at both visits. After surgery, the patients' mean scores were 0.6 to three points lower than those of controls on the five BSID-II and PLS-3 scales (p = 0.02–0.07). Compared with controls, patients had 2.3 and 1.9 times the adjusted odds of scoring in the delayed range on either BSID-II scale (Mental Development Index and Psychomotor Development Index) for the first and second visits, respectively (p = 0.001 and p = 0.015, respectively). The patients' mean adjusted test scores were nearly unrelated to the timing of their surgery.

Conclusions

These findings support recommendations for neurodevelopmental screening in infants with SSC. Longer follow-up, as is being conducted with the patients in the present study, will be critical for identifying the potential longer-term correlates of SSC and its surgical correction.

InJournal of Neurosurgery: Pediatrics Volume 107: Issue 2 (Aug 2007)

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School-age anthropometric and patient-reported outcomes after open or endoscopic repair of sagittal craniosynostosis

Annahita R. Fotouhi, Kamlesh B. Patel, Gary B. Skolnick, Corinne M. Merrill, Katherine M. Hofmann, Esperanza Mantilla-Rivas, Brent R. Collett, Virginia D. Allhusen, Sybill D. Naidoo, Gary F. Rogers, Robert F. Keating, Matthew D. Smyth, and Suresh N. Magge

OBJECTIVE

几项研究已经围手术期parame相比ters and early postoperative morphology between endoscope-assisted strip craniectomy with orthotic therapy (endoscopic repair) and cranial vault remodeling (open repair). To extend these results, the authors evaluated school-age anthropometric outcomes after these techniques across three institutions.

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School-aged children (age range 4–18 years) with previously corrected isolated sagittal craniosynostosis were enrolled. Upon inclusion, 3D photographs and patient-reported outcomes were obtained, and the cephalic index and head circumference z-scores were calculated. Analyses of covariance models controlling for baseline differences and a priori covariates were performed.

RESULTS

Eighty-one participants (median [range] age 7 [4–15] years) were included. The mean (95% CI) school-age cephalic index was significantly higher in the endoscopic cohort, though within the normal range for both groups (endoscopic 78% [77%–79%] vs open 76% [74%–77%], p = 0.027). The mean change in the cephalic index from preoperation to school age was significantly greater in the endoscopic group (9% [7%–11%] vs open 3% [1%–5%], p < 0.001). Compared to preoperative measurements, mean school-age head circumference z-scores decreased significantly more in the open cohort (−1.6 [−2.2 to −1.0] vs endoscopic −0.3 [−0.8 to −0.2], p = 0.002). Patient-reported levels of stigma were within the normal limits for both groups.

CONCLUSIONS

Endoscopic and open repair techniques effectively normalize school-age anthropometric outcomes. However, endoscopic repair produces a clinically meaningful and significantly greater improvement in the school-age cephalic index, with maintenance of head growth. These findings demonstrate the importance of early referral by pediatricians and inform treatment decisions.

InJournal of Neurosurgery: Pediatrics Publish Before Print

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