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Craniosynostosis

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2. Risk Factors for Preoperative Developmental Delay in Patients with Nonsyndromic Sagittal Craniosynostosis. (PubMed)

Risk Factors for Preoperative Developmental Delay in Patients with Nonsyndromic Sagittal Craniosynostosis. Some patients with isolated sagittal craniosynostosis have demonstrated mild neurodevelopmental delays. This study examined potential preoperative risk factors for developmental delay.Patients completed preoperative Bayley Scales of Infant and Toddler Development, Third Edition, and medical records were reviewed. Multivariate analyses of covariance and correlations were (...) correlations.Patients with nonsyndromic sagittal craniosynostosis that had delays in development had lower gestational age and birth weight, with more prenatal and birth complications. These factors can help identify patients who might be at risk for delay and need close monitoring.Risk, III.

2019 Plastic and reconstructive surgery

3. "The Cognitive Profile of Children with Non-syndromic Craniosynostosis." (PubMed)

"The Cognitive Profile of Children with Non-syndromic Craniosynostosis." Long-term neuropsychological and cognitive outcomes in patients with non-syndromic craniosynostosis have proven difficult to evaluate objectively due to methodological problems with published studies based on their small and biased samples of patients, wide age ranges, and testing with unacceptable psychometric properties. This study evaluated full-scale intelligence quotient (FSIQ) and its subscales in a cohort (...) with a small selection bias.Patients (aged 7-16 years) born with non-syndromic craniosynostosis and surgically treated were tested using the Wechsler Intelligence Scale for Children, Fourth Edition (WISC-IV). Ninety-one patients were invited, and 73 patients were tested.There was no difference in FSIQ between patients having undergone operations for sagittal synostosis or metopic synostosis and norms provided by the test. Patients operated on for sagittal synostosis showed a significantly higher perceptual

2019 Plastic and reconstructive surgery

4. Nonsyndromic craniosynostosis: novel coding variants. (PubMed)

Nonsyndromic craniosynostosis: novel coding variants. Craniosynostosis (CS), the premature fusion of one or more neurocranial sutures, is associated with approximately 200 syndromes; however, about 65-85% of patients present with no additional major birth defects.We conducted targeted next-generation sequencing of 60 known syndromic and other candidate genes in patients with sagittal nonsyndromic CS (sNCS, n = 40) and coronal nonsyndromic CS (cNCS, n = 19).We identified 18 previously published

2019 Pediatric Research

5. Reduced perioperative blood loss in children undergoing craniosynostosis surgery using prolonged tranexamic acid infusion: a randomised trial. (PubMed)

Reduced perioperative blood loss in children undergoing craniosynostosis surgery using prolonged tranexamic acid infusion: a randomised trial. Tranexamic acid (TXA) reduces intraoperative blood loss and transfusion during paediatric craniosynostosis surgery. Additional reduction of postoperative blood loss may further reduce exposure to allogeneic blood products. We studied the effect of combined intra- and postoperative TXA treatment on postoperative blood loss in children.Thirty children (...) admitted for craniosynostosis surgery were randomised to combined intra- and postoperative TXA treatment or placebo. The primary endpoint was postoperative blood loss. Secondary endpoints included total blood loss, transfusion requirements, and clot stability evaluated by tissue plasminogen activator-stimulated clot lysis assay.TXA reduced postoperative blood loss by 18 ml kg-1 (95% confidence interval 8.9) and total blood loss from a mean of 52 ml kg-1 (standard deviation [SD]; 20) ml kg-1 to 28 (14

2019 British Journal of Anaesthesia

6. Frustration and Emotional Regulation in Nonsyndromic Craniosynostosis: an fMRI Study. (PubMed)

Frustration and Emotional Regulation in Nonsyndromic Craniosynostosis: an fMRI Study. Nonsyndromic craniosynostosis (NSC) may manifest with complex behavioral, attentional, and emotional sequelae. We characterized higher-level brain connectivity in adolescent NSC patients in response to emotional frustration.Surgically corrected patients age >9 with NSC were age/gender/handedness matched to controls. Patients participated in a GoNoGo task, structured as 'win/lose/recovery' paradigms; 'Win

2019 Plastic and reconstructive surgery

7. Age at craniosynostosis surgery and its impact on ophthalmologic diagnoses-a single-center retrospective review. (PubMed)

Age at craniosynostosis surgery and its impact on ophthalmologic diagnoses-a single-center retrospective review. Ocular pathology in craniosynostosis is a persistent concern for patients and providers, and some surgeons feel that early surgical intervention for synostosis alleviates the progression of ophthalmologic abnormalities. In contradistinction, we hypothesize that operating early will have no bearing on post-operative ophthalmologic outcomes.Single-suture craniosynostosis patients who (...) underwent surgical correction between 1989 and 2015 were reviewed. Patients with multi-suture craniosynostosis, syndromic diagnoses, no pre-operative ophthalmology evaluation, and less than two years of follow-up were excluded. Logistic regression was used to determine odds of pre- and post-operative ophthalmologic abnormalities by age, while controlling for patient-level covariates.172 patients met inclusion criteria. The median age at surgery was 10 months (IQR 7-12.9 months). Increasing age

2019 Plastic and reconstructive surgery

8. An update of ophthalmic management in craniosynostosis. (PubMed)

An update of ophthalmic management in craniosynostosis. Craniosynostosis has a varied clinical spectrum, ranging from isolated single suture involvement to multisutural fusions. Syndromic and nonsyndromic patients require orchestrated and multidisciplinary care from birth to adulthood. Advances in our understanding of craniosynostosis over the last quarter-century have resulted in more systematic management of the problems associated with the syndromic and nonsyndromic forms of this condition

2019 JAAPOS - Journal of the American Association for Pediatric Ophthalmology and Strabismus

9. Impact of obstructive sleep apnea on optic nerve function in patients with craniosynostosis and recurrent intracranial hypertension. (PubMed)

Impact of obstructive sleep apnea on optic nerve function in patients with craniosynostosis and recurrent intracranial hypertension. Assessment of combined impact of intracranial pressure (ICH) and obstructive sleep apnea (OSA) on optic nerve function in children with craniosynostosis (CS).Retrospective cross-sectional study METHODS: Patients treated at Boston Children's Hospital for CS who had an ophthalmic examination that included pattern reversal (pr)VEP (2013-2014) and history of ICH based

2019 American Journal of Ophthalmology

10. Onset and resolution of Chiari malformations and hydrocephalus in syndromic craniosynostosis following posterior vault distraction. (PubMed)

Onset and resolution of Chiari malformations and hydrocephalus in syndromic craniosynostosis following posterior vault distraction. Patients with syndromic craniosynostosis have an increased incidence of progressive hydrocephalus and Chiari malformations (CM), with little data on relative benefit of various surgical interventions. The authors compare the incidence and resolution of CM and hydrocephalus between patients undergoing posterior vault distraction osteogenesis (PVDO) versus (...) conventional cranial vault remodeling (CVR).Patients with syndromic craniosynostosis who underwent cranial vault surgery from 2004 - 2016 at a single academic hospital with adequate radiographic assessments were reviewed. Demographics, interventions, the presence of a CM on radiographic studies and hydrocephalus requiring shunt placement were recorded. Mann-Whitney U and Fisher's exact tests were used as appropriate.49 patients underwent PVDO, and 23 patients underwent CVR during the study period. Median

2019 Plastic and reconstructive surgery

11. SMAD6 Genotype Predicts Neurodevelopment in Non-syndromic Craniosynostosis. (PubMed)

SMAD6 Genotype Predicts Neurodevelopment in Non-syndromic Craniosynostosis. De-novo or rare transmitted mutations in the SMAD6 gene affect non-syndromic midline synostosis (NSC) in 7% of patients. This study aimed to determine the neurocognitive sequelae of SMAD6 NSC.NSC patients >6 years of age, with SMAD6 mutations and non-SMAD6 NSC controls, were recruited. All patients completed a double-blinded neurodevelopmental battery (Wechsler Fundamentals, Wechsler Abbreviated Scale of Intelligence

2019 Plastic and reconstructive surgery

12. Age at Craniosynostosis Surgery and Its Impact on Ophthalmologic Diagnoses: A Single-Center Retrospective Review. (PubMed)

Age at Craniosynostosis Surgery and Its Impact on Ophthalmologic Diagnoses: A Single-Center Retrospective Review. Ocular abnormalities in craniosynostosis are a persistent concern for patients and providers, and some surgeons feel that early surgical intervention for synostosis alleviates the progression of ophthalmologic abnormalities. In contradistinction, the authors hypothesize that operating early will have no bearing on postoperative ophthalmologic outcomes.Single-suture craniosynostosis (...) patients who underwent surgical correction between 1989 and 2015 were reviewed. Patients with multisuture craniosynostosis, syndromic diagnoses, no preoperative ophthalmology evaluation, and less than 2 years of follow-up were excluded. Logistic regression was used to determine odds of preoperative and postoperative ophthalmologic abnormalities by age, while controlling for patient-level covariates.One hundred seventy-two patients met inclusion criteria. The median age at surgery was 10 months

2019 Plastic and reconstructive surgery

13. Serial Visual Evoked Potentials in Patients with Craniosynostosis and Invasive Intracranial Pressure Monitoring. (PubMed)

Serial Visual Evoked Potentials in Patients with Craniosynostosis and Invasive Intracranial Pressure Monitoring. This study aimed to detect the ability of pattern visual evoked potentials to detect visual pathway dysfunction in a cohort of patients with craniosynostosis who also had invasive intracranial pressure measurement. A retrospective review was conducted on craniosynostosis patients who had invasive intracranial pressure measurement and at least one pattern visual evoked potentials test (...) intracranial pressure (23.3 µV) (p = 0.03). The authors' results showed that serial pattern visual evoked potentials testing was able to detect visual pathway dysfunction resulting from raised intracranial pressure in five of seven craniosynostosis patients, and of these five patients, 80 percent had no evidence of papilledema, demonstrating the utility of serial pattern visual evoked potentials in follow-up of the visual function in craniosynostosis patients. CLINICAL QUESTION/LEVEL OF EVIDENCE

2019 Plastic and reconstructive surgery

14. "Quantification of head shape from three-dimensional photography for pre- and post-surgical evaluation of craniosynostosis". (PubMed)

"Quantification of head shape from three-dimensional photography for pre- and post-surgical evaluation of craniosynostosis". Evaluation of surgical treatment for craniosynostosis is typically based on subjective visual assessment or simple clinical metrics of cranial shape that are prone to inter-observer variability. 3D photography provides cheap and non-invasive information to assess surgical outcomes, but there are no clinical tools to analyze it. We aim to objectively and automatically (...) quantify head shape from 3D photography.We present an automatic method to quantify intuitive metrics of local head shape from 3D photography using a normative statistical head shape model built from 201 subjects. We use these metrics together with a machine learning classifier to distinguish between patients with (N=266) and without (N=201) craniosynostosis (age: 0-6 years). We also use our algorithms to quantify objectively local surgical head shape improvements on 18 patients with pre- and post

2019 Plastic and reconstructive surgery

15. Consolidation Time and Relapse: A Systematic Review of Outcomes in Internal Versus External Midface Distraction for Syndromic Craniosynostosis. (PubMed)

Consolidation Time and Relapse: A Systematic Review of Outcomes in Internal Versus External Midface Distraction for Syndromic Craniosynostosis. The choice between internal versus external distraction osteogenesis for midface advancement in patients with syndromic craniosynostosis is primarily based on surgeon preference and expertise. However, differences in outcomes between the two techniques have been sparingly compared. In this work, we performed a systematic review to compare outcomes

2019 Plastic and reconstructive surgery

16. Onset and Resolution of Chiari Malformations and Hydrocephalus in Syndromic Craniosynostosis following Posterior Vault Distraction. (PubMed)

Onset and Resolution of Chiari Malformations and Hydrocephalus in Syndromic Craniosynostosis following Posterior Vault Distraction. Patients with syndromic craniosynostosis have an increased incidence of progressive hydrocephalus and Chiari malformations, with few data on the relative benefit of various surgical interventions. The authors compare the incidence and resolution of Chiari malformations and hydrocephalus between patients undergoing posterior vault distraction osteogenesis (PVDO (...) ) and patients undergoing conventional cranial vault remodeling.Patients with syndromic craniosynostosis who underwent cranial vault surgery from 2004 to 2016 at a single academic hospital, with adequate radiographic assessments, were reviewed. Demographics, interventions, the presence of a Chiari malformation on radiographic studies and hydrocephalus requiring shunt placement were recorded. Mann-Whitney U and Fisher's exact tests were used as appropriate.Forty-nine patients underwent PVDO, and 23 patients

2019 Plastic and reconstructive surgery

17. Evaluating Trends in Headache and Revision Surgery following Cranial Vault Remodeling for Craniosynostosis. (PubMed)

Evaluating Trends in Headache and Revision Surgery following Cranial Vault Remodeling for Craniosynostosis. The recommended treatment for craniosynostosis, is cranial vault expansion to prevent increased intracranial pressure and optimize developmental outcomes. Some patients complain about postoperative headaches and occasionally require revision to treat increased intracranial pressure. This study examines whether specific factors are associated with an increased risk of postoperative (...) headaches or intracranial hypertension.This retrospective cohort included patients with craniosynostosis from 1995 and 2010. Primary outcomes included headaches and delayed intracranial hypertension. Logistic regression was used to evaluate the associations with clinical characteristics.The cohort included 383 patients, of whom 127 (33 percent) complained of headaches. The positive predictive value of a headache indicating intracranial hypertension was only 9.4 percent among all patients and 6.7 percent

2018 Plastic and reconstructive surgery

18. BBS9 gene in nonsyndromic craniosynostosis: Role of the primary cilium in the aberrant ossification of the suture osteogenic niche. (PubMed)

BBS9 gene in nonsyndromic craniosynostosis: Role of the primary cilium in the aberrant ossification of the suture osteogenic niche. Nonsyndromic craniosynostosis (NCS) is the premature ossification of skull sutures, without associated clinical features. Mutations in several genes account for a small number of NCS patients; thus, the molecular etiopathogenesis of NCS remains largely unclear. Our study aimed at characterizing the molecular signaling implicated in the aberrant ossification

2018 Bone

19. "Craniometric Analysis of Endoscopic Suturectomy for Bilateral Coronal Craniosynostosis." (PubMed)

"Craniometric Analysis of Endoscopic Suturectomy for Bilateral Coronal Craniosynostosis." Endoscopic suturectomy and helmeting represents a successful first-line surgical treatment for bilateral coronal craniosynostosis. Its effect on cranial morphology has not been previously described.Patients were identified who had bilateral coronal craniosynostosis treated with endoscopic suturectomy and postoperative helmeting at Boston Children's Hospital between 2005 and 2013 and who underwent (...) base length increased. The anterior cranial height-to-anterior cranial base length ratio significantly decreased with treatment (p = 0.128). Frontal bossing normalized with endoscopic suturectomy (craniosynostosis versus control: preoperatively, p = 0.001; postoperatively, p = 0.8). Cephalic indices also normalized with treatment (craniosynostosis versus control: preoperatively, p = 0.02; postoperatively, p = 0.13). No cases of hydrocephalus were observed.Endoscopic suturectomy and helmeting

2018 Plastic and reconstructive surgery

20. Incidence of Cranial Base Suture Fusion in Infants with Craniosynostosis. (PubMed)

Incidence of Cranial Base Suture Fusion in Infants with Craniosynostosis. Cranial base sutures are important drivers of both facial and cranial growth. The purpose of this study was to compare the incidence and location of cranial base suture fusion among three groups: nonaffected controls, patients with nonsyndromic craniosynostosis, and patients with syndromic craniosynostosis.Patients and computed tomographic scans were accrued from the authors' prospective craniofacial database. Computed (...) that of nonsyndromic patients (5.4 ± 3.1 months; p = 0.001) and control subjects (5.1 ± 3.2 months; p = 0.058). Syndromic craniosynostotic patients had over three times as many cranial base minor sutures fused (2.2 ± 2.5) as nonsyndromic craniosynostosis patients (0.7 ± 1.2; p < 0.001) and controls (0.4 ± 0.8; p = 0.002), whose rates of fusion were statistically equivalent (p = 0.342). Syndromic craniosynostosis patients had a greater frequency of cranial base suture fusion in the coronal branches, squamosal arch

2018 Plastic and reconstructive surgery

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