An Epidemiologic Study of Craniosynostosis: Risk Indicators for the Occurrence of Craniosynostosis in Colorado

Alderman, Beth W.; Lammer, Edward J.; Joshua, Sandra C; Cordero, José F.; Ouimette, David; Wilson, Marvin C.; Ferguson, Stanley W.

doi:10.1093/oxfordjournals.aje.a114983

Cited by 57 publications

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“…Most previous studies identified advanced maternal age as a risk factor for craniosynostosis [15,26,29,30] while a few [2,18], like ours, failed to show this association. This could have been due to the average young maternal age in our study and that the mothers did not make use of fertility assistance.…”

Section: Discussioncontrasting

confidence: 58%

“…It mostly occurs sporadically; the incidence of familial sagittal synostosis is in 2-6% of craniosynostosis cases, and only 8-14% of infants born with coronal synostosis are carrying an autosomal dominant disorder [2]. Several risk factors have been proposed for craniosynostosis such as white maternal race [14], smoking during pregnancy, maternal medical problems such as thyroid disease [11,12], postterm gestational age and advanced maternal age [11,15,16,17]; however, some studies do not support advanced maternal age as being an independent risk factor [16,18]. The relationship between paternal age and occurrence of craniosynostosis has not been shown [15].…”

Section: Introductionmentioning

confidence: 99%

“…Several risk factors have been proposed for craniosynostosis such as white maternal race [14], smoking during pregnancy, maternal medical problems such as thyroid disease [11,12], postterm gestational age and advanced maternal age [11,15,16,17]; however, some studies do not support advanced maternal age as being an independent risk factor [16,18]. The relationship between paternal age and occurrence of craniosynostosis has not been shown [15]. Most of the previous studies have reported craniosynostosis, especially sagittal and metopic synostosis, to occur more often among boys [19,20,21,22,23].…”

Section: Introductionmentioning

confidence: 99%

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Risk Factors Associated with Craniosynostosis: A Case Control Study

et al. 2012

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Background: Craniosynostosis is a relatively common congenital abnormality. The underlying etiology and most probable risk factors of nonsyndromic craniosynostosis are unknown. We conducted a study to identify the risk factors for craniosynostosis. Materials and Methods: In this case-control project, 70 children with craniosynostosis (syndromic or nonsyndromic) hospitalized at the Children's Hospital Medical Center from September 2010 to 2011 were studied for the potential risk factors. At the same time, 70 age- and sex-matched children hospitalized with other congenital anomalies were reviewed. Information from case and control groups was obtained via personal interviews with parents of patients and a prepared checklist was filled out for each child. Results: The mean (±SE) age at admission time in the case-control group was 13 ± 1.95 (1.5-96) months, and almost half of them were boys (n = 37 or 53%). The most frequent sutures involved in craniosynostosis were coronal (n = 30 or 42.9%) and then metopic sutures (n = 16 or 22.9%), multiple (n = 12 or 17.1%), sagittal (n = 11 or 15.7%) and lambdoid (n = 1 or 1.4%). In the case group, maternal diabetes mellitus and thyroid disease were found in 8 (11.6%) and 6 (8.6%) patients, respectively. The most frequent medication used by mothers (n = 66 or 94%) in the case group during pregnancy were vitamins (including iron supplements, omega 3, folic acid and multivitamins). Conclusion: A positive family history of craniosynostosis [odds ratio (OR) 19.01 and 95% confidence interval (CI) 2.24-160.7] and using clomiphene citrate for infertility (OR 12.71 and 95% CI 1.42-113.6) were the strongest independent risk factors for craniosynostosis. More comprehensive studies with a larger sample size are required to confirm the role of environmental factors in order to decrease the occurrence of craniosynostosis.

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Section: Discussioncontrasting

confidence: 58%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Risk Factors Associated with Craniosynostosis: A Case Control Study

et al. 2012

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“…At the same time, environmental factors such as maternal smoking, 10 nitrosatable drug use, 4,14 vitamin intake, 2 and hormonal factors such as hyperthyroidism 9 have also been shown to play roles in premature cranial suture closure, as well as advanced maternal age 11 and factors influencing fetal head constraint such as macrosomia, breech presentations, and multiple births. 1,8,11,21 Thus, demographic variabilities of craniosynostosis may be due to higher proportions of predisposed populations or higher rates of environmental exposures and maternal-fetal comorbidities. Further studies are needed to differentiate between the predisposing causes that lead to the racial and regional disproportions of craniosynostosis.…”

Section: Discussionmentioning

confidence: 99%

Differences in surgical outcomes for patients with craniosynostosis in the US: impact of socioeconomic variables and race

Shweikeh

Foulad

Nuño

et al. 2016

Journal of Neurosurgery: Pediatrics

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OBJECT Craniosynostosis is often treated with neurosurgical intervention. The aim of this study was to report and analyze the clinical and socioeconomic characteristics of patients with craniosynostosis and to present current national trends. METHODS Using the Kids’ Inpatient Database for the years 2000, 2003, 2006, and 2009, the authors identified patients with craniosynostosis using International Classification of Diseases, Ninth Revision, Clinical Modification diagnosis codes and their associated procedure codes. Clinical features, demographics, inpatient procedures, outcomes, and charges were collected and analyzed. RESULTS Of the 3415 patients identified, 65.8% were White, 21.4% were Hispanic, and 3.2% were Black. More than 96% were treated at urban teaching hospitals and 54.2% in southern or western regions. White patients were younger (mean 6.1 months) as compared with Blacks (mean 10.9 months) and Hispanics (mean 9.1 months; p < 0.0001) at the time of surgery. A higher fraction of Whites had private insurance (70.3%) compared with nonwhites (34.0%–41.6%; p < 0.001). Approximately 12.2% were nonelective admissions, more so among Blacks (16.9%). Mean hospital length of stay (LOS) was 3.5 days with no significant differences among races. Following surgical treatment, 12.1% of patients developed complications, most commonly pulmonary/respiratory (4.8%), wound infection (4.4%), and hydrocephalus (1.4%). The mean overall hospital charges were significantly lower for Whites than nonwhites ($34,527 vs $44,890–$48,543, respectively; p < 0.0001). CONCLUSIONS The findings of this national study suggest a higher prevalence of craniosynostosis in Hispanics. The higher predisposition among males was less evident in Hispanics and Blacks. There was a significant percentage of nonelective admissions, more commonly among Blacks. Additionally, Hispanics and Blacks were more likely to receive surgery at an older age, past the current recommendation of the optimum age for surgical intervention. These findings are likely associated with a lack of early detection. Although mean LOS and rate of complications did not significantly differ among different races, nonwhites had, on average, higher hospital charges of $10,000–$14,000. This discrepancy may be due to differences in type of insurance, craniosynostosis type, rates of comorbidities, and delay in treatment. Although there are several limitations to this analysis, the study reports on relevant disparities regarding a costly neurosurgical intervention, and ways to diminish these disparities should be further explored.

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“…Studies suggest various risk factors for CS, such as smoking, advanced maternal age, in utero constraint, male sex, and Caucasian race-ethnicity [13][14][15][16], but its actual causes remain largely unknown. This is particularly true of non-syndromic CS, i.e., cases for which no genetic cause has been identified, which comprise about 90 % of all cases.…”

Section: Introductionmentioning

confidence: 99%

Craniosynostosis: The Potential Contribution of Thyroid-Related Mechanisms

2015

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Craniosynostosis, the premature fusion of one or more cranial sutures, leads to abnormal craniofacial form and function. Its causes remain largely unknown. One of the strongest clues regarding non-syndromic craniosynostosis etiology is its association with thyroid-related disorders, which is based on a mix of epidemiologic and experimental findings. This paper reviews evidence for the contribution of thyroidrelated mechanisms to craniosynostosis and makes recommendations for how to improve this knowledge.

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An Epidemiologic Study of Craniosynostosis: Risk Indicators for the Occurrence of Craniosynostosis in Colorado

Cited by 57 publications

References 0 publications

Risk Factors Associated with Craniosynostosis: A Case Control Study

Risk Factors Associated with Craniosynostosis: A Case Control Study

Differences in surgical outcomes for patients with craniosynostosis in the US: impact of socioeconomic variables and race

Craniosynostosis: The Potential Contribution of Thyroid-Related Mechanisms

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