The Changing Epidemiologic Spectrum of Single-Suture Synostoses

Selber, Jesse C.; Reid, Russell R.; Chike-Obi, Chuma J.; Sutton, Leslie N.; Zackai, Elaine H.; McDonald‐McGinn, Donna M.; Sonnad, Seema S.; Whitáker, Linton A.; Bartlett, Scott P.

doi:10.1097/prs.0b013e31817d548c

Cited by 126 publications

(65 citation statements)

References 20 publications

(12 reference statements)

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“…The most common of these is sagittal synostosis (one in 5,000 births), with the least common being lambdoid synostosis (one in 200,000 births) [1]. Incidence trends suggest increasing incidence rates of metopic synostosis relative to other forms of the single-sutural conditions [3].…”

Section: Introductionmentioning

confidence: 99%

Neurodevelopmental functioning of infants with untreated single-suture craniosynostosis during early infancy

et al. 2012

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Section: Introductionmentioning

confidence: 99%

Neurodevelopmental functioning of infants with untreated single-suture craniosynostosis during early infancy

et al. 2012

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“…If only one suture is involved, it is termed a simple craniosynostosis and if more than one suture is fused, it is known as compound craniosynostosis [2,3]. The sagittal and lambdoid sutures are the most and the least common sutures involved in nonsyndromic craniosynostosis, respectively [1,9,10,11,12,13]. …”

Section: Introductionmentioning

confidence: 99%

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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“…In large historical clinical series, metopic craniosynostosis accounts for 3–4% percent of all single suture craniosynostosis, but recently there has been a marked epidemological increase in its prevalance to as high as 28% 1–3 of all surgically treated synostosis cases. The etiopathogenesis of this shift is unknown but it has been speculated that the reasons are multifactorial, including an increasing proportion of syndromic patients, and advanced paternal age 4 . However, some practitioners are concerned that this phenomenon may be secondary to over-diagnosis and concomitant decision to offer operation on benign metopic ridge (BMR), which is present in 10–25% of infants as a variant of normal 5 .…”

Section: Introductionmentioning

confidence: 99%

Comparison of an unsupervised machine learning algorithm and surgeon diagnosis in the clinical differentiation of metopic craniosynostosis and benign metopic ridge

Cho

Hallac

Effendi³

et al. 2018

Sci Rep

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Metopic suture closure can manifest as a benign metopic ridge (BMR), a variant of normal, to “true” metopic craniosynostosis (MCS), which is associated with severe trigonocephaly. Currently, there is no gold standard for how much associated orbitofrontal dysmorphology should trigger surgical intervention. In our study, we used three-dimensional (3D) curvature analysis to separate the phenotypes along the spectrum, and to compare surgeons’ thresholds for operation. Three-dimensional curvature analyses on 43 subject patients revealed that the mean curvature of mid-forehead vertical ridge was higher for patients who underwent operation than those who did not undergo operation by 1.3 m−1 (p < 0.0001). In addition, these patients had more retruded supraorbital areas by −16.1 m−1 (p < 0.0001). K-means clustering classified patients into two different severity groups, and with the exception of 2 patients, the algorithm’s classification of deformity completely agreed with the surgeons’ decisions to offer either conservative or operative therapy (i.e. 96% agreement). The described methods are effective in classifying severity of deformity and in our experience closely approximate surgeon therapeutic decision making. These methods offer the possibility to consistently determine when surgical intervention may be beneficial and to avoid unnecessary surgeries on children with benign metopic ridge and associated minimal orbitofrontal deformity.

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The Changing Epidemiologic Spectrum of Single-Suture Synostoses

Abstract: Metopic synostosis is on the rise. Changing demographic bases and increasing proportions of syndromic patients may be clues to the etiology of this epidemiologic event.

Cited by 126 publications

References 20 publications

Neurodevelopmental functioning of infants with untreated single-suture craniosynostosis during early infancy

Neurodevelopmental functioning of infants with untreated single-suture craniosynostosis during early infancy

Risk Factors Associated with Craniosynostosis: A Case Control Study

Comparison of an unsupervised machine learning algorithm and surgeon diagnosis in the clinical differentiation of metopic craniosynostosis and benign metopic ridge

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