Prenatal Ultrasound Parameters of Twins With Sagittal Suture Craniosynostosis Question Mechanical Constraint as the Leading Cause
Open accessAbstract
Background: Multiple pregnancy is a known predisposing factor for sagittal suture synostosis, and intrauterine constraint has been suggested as a potential cause. This study evaluates prenatal images of single and multiple pregnancies of children with sagittal suture synostosis to assess the possible pathogenesis. Materials and Methods: Prenatal ultrasound data of scaphocephaly twins treated at Erasmus MC was retrieved retrospectively. The head circumference, cephalic index (CI), and biparietal diameter (BPD) were analyzed. Data were compared with the nonaffected sibling, singletons with scaphocephaly, and to general fetal growth reference values. Results: Data of 8 twin pregnancies with 10 cases of scaphocephaly was gathered. For the head circumference no difference was found between affected fetuses and reference values throughout pregnancy. For the BPD a reduced growth was found, compared with control values. The growth curve for the CI of the affected twins is lower from the start and declines over time. When comparing affected fetuses to nonaffected fetuses, the same differences for the BPD and CI were found as compared with controls. No differences in growth parameters were found between the nonaffected siblings and reference values, nor between the affected scaphocephaly twins and singletons with scaphocephaly. Conclusions: Scaphocephaly in twin pregnancy develops early on in pregnancy, similar to singleton pregnancy, making the theory of constraint as a cause less likely. The development of scaphocephaly in twins is likely to be attributed to the same disturbed developmental processes in suture development as in singletons, and not to mechanical constraint.
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Cinca et al. (2022) proposed that mechanical factors such as crowding or abnormal positioning of twins may be a significant factor in the development of sagittal suture craniosynostosis alongside genetic causes.
“The authors propose that mechanical factors, such as crowding or abnormal positioning of the twins, may be a significant factor in the development of SSC, alongside genetic causes [38].”
From Developments in diagnostic and surgical techniques in children with sagittal suture craniosynostosis: a systematic review spanning the last 30 years (2025) · cites this paper as [38]
The Discussion concludes that scaphocephaly in twins is likely due to the same disturbed suture-development processes seen in singletons, “and not to mechanical constraint.” Although the Abstract notes that constraint had previously been suggested as a possible cause, the authors' findings argue against it rather than propose it as a significant factor alongside genetic causes. (LLM, conf=0.91)
Checked by a reasoning model — may be wrong.
Experimental — accuracy not yet measuredCinca et al. proposed that mechanical constraints in twin pregnancies may contribute to sagittal suture fusion.
“More recently Cinca et al. proposed that mechanical constraints in twin pregnancies may contribute to sagittal suture fusion, highlighting the multifactorial nature of this condition [38].”
From Developments in diagnostic and surgical techniques in children with sagittal suture craniosynostosis: a systematic review spanning the last 30 years (2025) · cites this paper as [38]
The Abstract describes mechanical constraint as an existing suggestion that Cinca et al. set out to test, not a proposal they make. Its conclusion says scaphocephaly in twins is likely due to disturbed suture-development processes, not mechanical constraint; the Discussion likewise calls reduced womb space an improbable influence at the apparent onset of synostosis. Page numbers are unavailable in our copy. (LLM, conf=0.88)
Checked by a reasoning model — may be wrong.
Experimental — accuracy not yet measuredCornelissen et al. and other authors highlighted the potential of prenatal ultrasound, with BPD deviations suggesting improved accuracy using 3D imaging.
“Cornelissen et al. and other authors highlighted the potential of prenatal ultrasound, with BPD deviations suggesting improved accuracy using 3D imaging.”
From Developments in diagnostic and surgical techniques in children with sagittal suture craniosynostosis: a systematic review spanning the last 30 years (2025) · cites this paper as [38]
The Biometrics results (page unavailable) report that affected twins had slower BPD growth and a smaller BPD later in pregnancy, supporting the point that prenatal ultrasound can reveal deviations. The paper does not report that 3D imaging improves accuracy, and it is not a study by Cornelissen et al. (LLM, conf=0.87)
Experimental — accuracy not yet measuredUltrasound is non-invasive and widely available, but CI alone is unreliable, and the feasibility of advanced imaging in routine screening requires further study.
“While ultrasound is non-invasive and widely available, CI alone is unreliable, and the feasibility of advanced imaging in routine screening requires further study [36, 38].”
From Developments in diagnostic and surgical techniques in children with sagittal suture craniosynostosis: a systematic review spanning the last 30 years (2025) · cites this paper as [38]
The Analysis section (page unavailable) says pregnancy ultrasound measurements are used in daily practice, and the Discussion notes that measurements are user-dependent. The paper does not establish that CI alone is unreliable or discuss the feasibility of advanced imaging in routine screening; it also does not address ultrasound's non-invasiveness. (LLM, conf=0.76)
Experimental — accuracy not yet measured
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Cites
Practice guidelines for performance of the routine mid-trimester fetal ultrasound scan
Salomon, L. J., Alfirevic, Z., Berghella, V., Bilardo, C., Hernandez‐Andrade, E., Johnsen, S. L., Kalache, K., Leung, K. ‐., Malinger, G., Munoz, H., et al. (2011) · Ultrasound in Obstetrics & Gynecology · doi:10.1002/uog.8831 · Cited by 1,006 · see citing works
Paladini, D., Malinger, G., Monteagudo, A., Timor Tritsch, I., Toi, A., & Pilu, G. (2007) · Ultrasound in Obstetrics and Gynecology · doi:10.1002/uog.3909 · Cited by 577 · see citing works
Verburg, B. O., Steegers, E. A. P., de Ridder, M. A. J., Snijders, R. J. M., Smith, E. A., Albert Hofman, D., Moll, H. A., Jaddoe, V. W. V., & Witteman, J. C. M. (2008) · Ultrasound in Obstetrics and Gynecology · doi:10.1002/uog.5225 · Cited by 505 · see citing works
Fetal biometry at 14–40 weeks' gestation
Snijders, R. J. M., & Nicolaides, K. H. (1994) · Ultrasound in Obstetrics and Gynecology · doi:10.1046/j.1469-0705.1994.04010034.x · Cited by 501 · see citing works
Sutural biology and the correlates of craniosynostosis
Cohen, M. M. (1993) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320470507 · Cited by 394 · see citing works
Charts of fetal size: 2. Head measurements*
Chitty, L. S., Altman, D. G., Henderson, A., & Campbell, S. C. (1994) · BJOG An International Journal of Obstetrics & Gynaecology · doi:10.1111/j.1471-0528.1994.tb13007.x · Cited by 323 · see citing works
Evolution in the frequency of nonsyndromic craniosynostosis: Clinical article
Di Rocco, F., Arnaud, E., & Renier, D. (2009) · Journal of Neurosurgery: Pediatrics · doi:10.3171/2009.3.peds08355 · Cited by 253 · see citing works
Increase of prevalence of craniosynostosis
Cornelissen, M., Ottelander, B. D., Rizopoulos, D., van der Hulst, R., Mink van der Molen, A., van der Horst, C., Delye, H., van Veelen, M.-L., Bonsel, G., & Mathijssen, I. (2016) · Journal of Cranio-Maxillofacial Surgery · doi:10.1016/j.jcms.2016.07.007 · Cited by 226 · see citing works
Self-citationVu, H. L., Panchal, J., Parker, E. E., Levine, N. S., & Francel, P. C. (2001) · Journal of Craniofacial Surgery · doi:10.1097/00001665-200111000-00005 · Cited by 222 · see citing works
Fetal Size and Dating: Charts Recommended for Clinical Obstetric Practice
Loughna, P., Chitty, L. S., Evans, T., & Chudleigh, T. (2009) · Ultrasound · doi:10.1179/174313409x448543 · Cited by 209 · see citing works
Chitty, L. S., & Altman, D. G. (1994) · Ultrasound · doi:10.1177/1742271x9400200404 · Cited by 175 · see citing works
Sagittal craniostenosis: Fetal head constraint as one possible cause
Graham, J. M., deSaxe, M., & Smith, D. W. (1979) · The Journal of Pediatrics · doi:10.1016/s0022-3476(79)80728-3 · Cited by 128 · see citing works
Lapeer, R. J., & Prager, R. W. (2001) · Journal of Biomechanics · doi:10.1016/s0021-9290(01)00070-7 · Cited by 120 · see citing works
Dichorionic twin trajectories: the NICHD Fetal Growth Studies
Grantz, K. L., Grewal, J., Albert, P. S., Wapner, R. J., D’Alton, M. E., SCISCIONE, A. C., Grobman, W. A., Wing, D. A., Owen, J., Newman, R., et al. (2016) · American Journal of Obstetrics and Gynecology · doi:10.1016/j.ajog.2016.04.044 · Cited by 119 · see citing works
Changing Epidemiology of Nonsyndromic Craniosynostosis and Revisiting the Risk Factors
Lee, H. Q., Hutson, J. M., Wray, A. C., Lo, P. A., Chong, D. K., Holmes, A. D., & Greensmith, A. L. (2012) · Journal of Craniofacial Surgery · doi:10.1097/scs.0b013e318252d893 · Cited by 116 · see citing works
Fetal constraint as a potential risk factor for craniosynostosis
Sanchez‐Lara, P. A., Carmichael, S. L., Graham, J. M., Lammer, E. J., Shaw, G. M., Ma, C., & Rasmussen, S. A. (2010) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.33246 · Cited by 106 · see citing works
Differences in fetal growth patterns between twins and singletons*
Hiersch, L., Okby, R., Freeman, H., Rosen, H., Nevo, O., Barrett, J. F. R., & Melamed, N. (2018) · The Journal of Maternal-Fetal & Neonatal Medicine · doi:10.1080/14767058.2018.1555705 · Cited by 71 · see citing works
SMAD6 variants in craniosynostosis: genotype and phenotype evaluation
Calpena, E., Cuellar, A., Bala, K., Swagemakers, S. M. A., Koelling, N., McGowan, S. J., Phipps, J. M., Balasubramanian, M., Cunningham, M. L., Douzgou, S., et al. (2020) · Genetics in Medicine · doi:10.1038/s41436-020-0817-2 · Cited by 51 · see citing works
Self-citationTracing craniosynostosis to its developmental stage through bone center displacement.
Mathijssen, I. M. J., van Splunder, J., Vermeij‐Keers, C., Pieterman, H., de Jong, T. H. R., Mooney, M. P., & Vaandrager, J. M. (1999) · PubMed · Cited by 48 · see citing works
Self-citationTracing Craniosynostosis to Its Developmental Stage Through Bone Center Displacement
Mathijssen, I. M. J., van Splunder, J., Vermeij‐Keers, C., Pieterman, H., de Jong, T. H. R., Mooney, M. P., Vaandrager, J. M., & Eppley, B. L. (1999) · Journal of Craniofacial Surgery · doi:10.1097/00001665-199911000-00014 · Cited by 37 · see citing works
Self-citationGenetic and Epigenetic Influences of Twins on the Pathogenesis of Craniosynostosis
Lakin, G. E., Sinkin, J. C., Chen, R., Koltz, P. F., & Girotto, J. A. (2012) · Plastic & Reconstructive Surgery · doi:10.1097/prs.0b013e31824422a8 · Cited by 33 · see citing works
Identical Twins Discordant for Metopic Craniosynostosis
Magge, S. N., Snyder, K. A., Sajja, A. P., DeFreitas, T. A., Hofherr, S. E., Broth, R. E., Keating, R. F., & Rogers, G. F. (2017) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000003368 · Cited by 19 · see citing works
Clinical and genetic characteristics of craniosynostosis in Hungary
Bessenyei, B., Nagy, A., Szakszon, K., Mokánszki, A., Balogh, E., Ujfalusi, A., Tihanyi, M., Novák, L., Bognár, L., & Oláh, É. (2015) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.37298 · Cited by 16 · see citing works
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1 of the 24 references couldn’t be matched to a known paper.
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- Di Rocco F, Arnaud E, Renier D. Evolution in the frequency of nonsyndromic craniosynostosis. J Neurosurg Pediatr 2009;4:21-25
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Hermann, J., Raak, C. K., Ostermann, T., & Scharbrodt, W. (2025) · Orphanet Journal of Rare Diseases · doi:10.1186/s13023-025-03978-9 · Cited by 2 · see citing works
“More recently Cinca et al. proposed that mechanical constraints in twin pregnancies may contribute to sagittal suture fusion, highlighting the multifactorial nature of this condition [38].”
Segal, N. L. (2023) · Twin Research and Human Genetics · doi:10.1017/thg.2023.3
Segal, N. L. (2023) · Twin Research and Human Genetics · doi:10.1017/thg.2023.13
- Authors
- Cinca, K. P., de Planque, C. A., Peters, N. C. J., Versnel, S. L., & Mathijssen, I. M. J.
- Affiliations
- Erasmus MC (NL)
- Department of Plastic and Reconstructive Surgery and Hand Surgery, Erasmus MC
- Corresponding author
- Cinca, Kevin P.
- Versnel, Sarah L.
- Mathijssen, Irene M.J.
- Venue
- Journal of Craniofacial Surgery
- Published
- 2022-07-22
- Volume
- 33
- Issue
- 8
- Pages
- 2350-2353
- ISSN
- 1049-2275 (ISSN-L)
- OpenAlex
- W4286433110
- Cited by
- 3
- References
- 24
- Citations by year
- 2025: 1 · 2023: 2
- FWCI
- 0.45
- Type
- journal article
- Language
- en
- Open access
- green
- pure.eur.nl
- Licence
- other-oa
- Locations
- Journal of Craniofacial Surgery (publishedVersion)
- PubMed (publishedVersion)
- EUR Research Repository (Erasmus University Rotterdam) (submittedVersion, other-oa)
- EUR Research Repository (Erasmus University Rotterdam) (submittedVersion)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Craniofacial Disorders and Treatments
- Health Sciences › Medicine › Pediatrics, Perinatology and Child Health › Fetal and Pediatric Neurological Disorders
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Cleft Lip and Palate Research
- Keywords
- sagittal craniosynostosis, scaphocephaly, twins, prenatal ultrasound, cephalic index, biparietal diameter, head circumference, fetal growth, pathogenesis
- MeSH terms
- Child, Craniosynostoses, Female, Gestational Age, Humans, Jaw Abnormalities, Pregnancy, Retrospective Studies, Sutures, Twins, Ultrasonography, Prenatal
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