Neurocognitive outcomes and associated clinical factors 5 years after surgery in children with craniosynostosis

Author:

Kim Joo Whan1,Kim Kyung Hyun1,Phi Ji Hoon12,Lee Ji Yeoun13,Koh Eun Jung1,Kim Byung Jun4,Chung Jee Hyeok4,Shin Min-Sup5,Kim Seung-Ki12

Affiliation:

1. Division of Pediatric Neurosurgery, Seoul National University Children’s Hospital, Seoul National University College of Medicine, Seoul;

2. Neuroscience Research Institute, Seoul National University Medical Research Center, Seoul National University College of Medicine, Seoul;

3. Neural Development and Anomaly Laboratory, Department of Anatomy and Cell Biology, Seoul National University College of Medicine, Seoul;

4. Division of Pediatric Plastic Surgery, Seoul National University Children’s Hospital, Seoul National University College of Medicine, Seoul; and

5. Division of Child and Adolescent Psychiatry, Seoul National University Children’s Hospital, Seoul National University College of Medicine, Seoul, Republic of Korea

Abstract

OBJECTIVE Craniosynostosis involves early closure of one or more sutures, which is known to limit normal cranium growth and interfere with normal brain development. Various surgical methods are used, ranging from minimally invasive strip craniectomy to more extensive whole-vault cranioplasty. This study aimed to evaluate neurocognitive outcomes 5 years after surgical treatment in children with craniosynostosis and to evaluate relevant clinical factors. METHODS After exclusion of genetically confirmed syndromic craniosynostosis patients, a retrospective review was conducted on 112 nonsyndromic craniosynostosis patients who underwent surgical treatment and follow-up neurocognitive assessment. Ninety-seven patients underwent strip craniectomy with postoperative orthotic helmet therapy, and 15 received other surgical treatment: 4 with distraction osteotomy and 11 with craniofacial reconstruction. Neurocognitive assessment using the Korean Wechsler Preschool and Primary Scale of Intelligence, Fourth Edition (K-WPPSI-IV), was performed 5 years postoperatively. Clinical factors were assessed regarding neurocognitive outcomes. RESULTS The mean age at surgery was significantly younger in the strip craniectomy group (strip craniectomy 4.6 months vs other surgical treatment 18.6 months, p < 0.01). Patients with 2 or more sutures involved were more likely to receive more extensive surgical treatment (16.5% in the strip craniectomy group vs 53.8% in the other group, p < 0.01). Four (3.5%) patients who showed evidence of increased intracranial pressure (ICP) also underwent more extensive surgical treatment. Multivariable linear regression revealed a significant correlation of age at neurocognitive testing (−3.18, 95% CI −5.95 to −0.40, p = 0.02), increased ICP (−34.73, 95% CI −51.04 to −18.41, p < 0.01), and the level of maternal education (6.11, 95% CI 1.01–11.20, p = 0.02) with the Full-Scale Intelligence Quotient (FSIQ). Age at surgery, involvement of 2 or more sutures, and type of operation demonstrated no correlation with FSIQ. Among the 97 patients who underwent strip craniectomy, the FSIQ ranged from mean ± SD 100.2 ± 10.2 (bicoronal) to 110.1 ± 12.7 (lambdoid), and there was no significant difference between the suture groups (p = 0.41). The 5 index scores were all within average ranges based on their age norms. CONCLUSIONS Age at neurocognitive assessment, increased ICP, and maternal education level showed significant correlations with the neurocognitive function of craniosynostosis patients. Although children with craniosynostosis exhibited favorable 5-year postoperative neurocognitive outcomes across various synostosis sutures, longer follow-up is needed to reveal the incidence of neurocognitive dysfunction in these patients.

Publisher

Journal of Neurosurgery Publishing Group (JNSPG)

Reference39 articles.

1. Craniofacial growth following experimental craniosynostosis and craniectomy in rabbits;Persson KM,1979

2. Cranial vault growth in craniosynostosis;Delashaw JB,1989

3. Mental development and learning disorders in children with single suture craniosynostosis;Kapp-Simon KA,1998

4. Prognosis for mental function in scaphocephaly;Arnaud E,1995

5. Speech, cognitive, and behavioral outcomes in nonsyndromic craniosynostosis;Becker DB,2005

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