Child and Infant Learning Project

Craniosynostosis Clinical Trial

— CILP  

Official title:

Neurobehavioral Correlates of Craniosynostosis

Clinical Trial Details — Status: Completed

Administrative data

• NCT number: NCT00077831
• Other study ID #: NIDCR-13813
• Secondary ID: R01DE013813
• Status: Completed
• Phase: N/A
• First received: February 12, 2004
• Last updated: July 19, 2015
• Start date: September 2001
• Est. completion date: June 2015

Study information

• Verified date: July 2015
• Source: Seattle Children's Hospital
• Contact: n/a
• Is FDA regulated: No
• Health authority: United States: Federal Government
• Study type: Observational

Clinical Trial Summary

To learn more about the cognitive and motor development of infants and young children born with a craniofacial defect called craniosynostosis.

Description:

In the first phase of this multi-site, 10-year longitudinal study, infants with one of four types of single-suture craniosynostosis were recruited: sagittal, metopic, right unilateral coronal, and left unilateral coronal. A case-matched "control" group of healthy, normal infants was also followed. This study, which is now in its second phase, is following this same cohort of children at the age of 7 years.

Recruitment information / eligibility

• Status: Completed
• Enrollment: 535
• Est. completion date: June 2015
• Est. primary completion date: April 2014
• Accepts healthy volunteers: No
• Gender: Both
• Age group: 2 Months to 7 Years

Eligibility

Enrollment criteria at Phase 1:

• Confirmed diagnosis of single-suture, nonsyndromic craniosynostosis (sagittal, metopic, unilateral coronal, or lambdoid)

• Corrective (cranioplastic) surgery not yet performed

• Child born at 34 weeks gestation or later

• Absence of neurological conditions/significant health problems

• 33 months or younger at time of enrollment (male or female).

Enrollment criteria at Phase 2:

All participants enrolled in phase 1 are invited to participate in phase 2.

Study Design

Observational Model: Case Control, Time Perspective: Prospective

Related Conditions & MeSH terms

• Craniosynostoses
• Craniosynostosis

Intervention

Behavioral:
• neurobehavioral development
observational study of infant and child development

Locations

Country	Name	City	State
United States	Children's Health Care of Atlanta	Atlanta	Georgia
United States	Seattle Children's Hospital	Seattle	Washington
United States	St. Louis Children's Hospital / Washington University	St. Louis	Missouri
United States	Northwestern University, Cleft Lip and Palate Institute	Westchester	Illinois

Sponsors (2)

Lead Sponsor	Collaborator
Seattle Children's Hospital	National Institute of Dental and Craniofacial Research (NIDCR)

Country where clinical trial is conducted

United States, 

References & Publications (21)

Collett BR, Gray KE, Kapp-Simon KA, Birgfeld C, Cunningham M, Rudo-Stern J, Ung D, Buono L, Speltz ML. Laypersons' ratings of appearance in children with and without single-suture craniosynostosis. J Craniofac Surg. 2013 Jul;24(4):1331-5. doi: 10.1097/SCS — View Citation

Cunningham ML, Horst JA, Rieder MJ, Hing AV, Stanaway IB, Park SS, Samudrala R, Speltz ML. IGF1R variants associated with isolated single suture craniosynostosis. Am J Med Genet A. 2011 Jan;155A(1):91-7. doi: 10.1002/ajmg.a.33781. — View Citation

Gray KE, Cradock MM, Kapp-Simon KA, Collett BR, Pullmann LD, Speltz ML. Longitudinal analysis of parenting stress in mothers and fathers of infants with and without single-suture craniosynostosis. Cleft Palate Craniofac J. 2015 Jan;52(1):3-11. doi: 10.159 — View Citation

Kapp-Simon KA, Collett BR, Barr-Schinzel MA, Cradock MM, Buono LA, Pietila KE, Speltz ML. Behavioral adjustment of toddler and preschool-aged children with single-suture craniosynostosis. Plast Reconstr Surg. 2012 Sep;130(3):635-47. — View Citation

Kapp-Simon KA, Leroux B, Cunningham M, Speltz ML. Multisite study of infants with single-suture craniosynostosis: preliminary report of presurgery development. Cleft Palate Craniofac J. 2005 Jul;42(4):377-84. — View Citation

Kapp-Simon KA, Speltz ML, Cunningham ML, Patel PK, Tomita T. Neurodevelopment of children with single suture craniosynostosis: a review. Childs Nerv Syst. 2007 Mar;23(3):269-81. Epub 2006 Dec 21. Review. — View Citation

Lin HJ, Ruiz-Correa S, Shapiro LG, Speltz ML, Cunningham ML, Sze RW. Predicting neuropsychological development from skull imaging. Conf Proc IEEE Eng Med Biol Soc. 2006;1:3450-5. — View Citation

McCarthy JG, Warren SM, Bernstein J, Burnett W, Cunningham ML, Edmond JC, Figueroa AA, Kapp-Simon KA, Labow BI, Peterson-Falzone SJ, Proctor MR, Rubin MS, Sze RW, Yemen TA; Craniosynostosis Working Group. Parameters of care for craniosynostosis. Cleft Palate Craniofac J. 2012 Jan;49 Suppl:1S-24S. doi: 10.1597/11-138. Epub 2011 Aug 17. — View Citation

Mefford HC, Shafer N, Antonacci F, Tsai JM, Park SS, Hing AV, Rieder MJ, Smyth MD, Speltz ML, Eichler EE, Cunningham ML. Copy number variation analysis in single-suture craniosynostosis: multiple rare variants including RUNX2 duplication in two cousins wi — View Citation

Naumann HL, Haberkern CM, Pietila KE, Birgfeld CB, Starr JR, Kapp-Simon KA, Hopper RA, Speltz ML. Duration of exposure to cranial vault surgery: associations with neurodevelopment among children with single-suture craniosynostosis. Paediatr Anaesth. 2012 — View Citation

Rosenberg JM, Kapp-Simon KA, Starr JR, Cradock MM, Speltz ML. Mothers' and fathers' reports of stress in families of infants with and without single-suture craniosynostosis. Cleft Palate Craniofac J. 2011 Sep;48(5):509-18. doi: 10.1597/09-210. Epub 2010 A — View Citation

Ruiz-Correa S, Starr JR, Lin HJ, Kapp-Simon KA, Cunningham ML, Speltz ML. Severity of skull malformation is unrelated to presurgery neurobehavioral status of infants with sagittal synostosis. Cleft Palate Craniofac J. 2007 Sep;44(5):548-54. — View Citation

Ruiz-Correa S, Starr JR, Lin HJ, Kapp-Simon KA, Sze RW, Ellenbogen RG, Speltz ML, Cunningham ML. New severity indices for quantifying single-suture metopic craniosynostosis. Neurosurgery. 2008 Aug;63(2):318-24; discussion 324-5. doi: 10.1227/01.NEU.000031 — View Citation

Ruiz-Correa S, Sze RW, Starr JR, Lin HT, Speltz ML, Cunningham ML, Hing AV. New scaphocephaly severity indices of sagittal craniosynostosis: a comparative study with cranial index quantifications. Cleft Palate Craniofac J. 2006 Mar;43(2):211-21. — View Citation

Seto ML, Hing AV, Chang J, Hu M, Kapp-Simon KA, Patel PK, Burton BK, Kane AA, Smyth MD, Hopper R, Ellenbogen RG, Stevenson K, Speltz ML, Cunningham ML. Isolated sagittal and coronal craniosynostosis associated with TWIST box mutations. Am J Med Genet A. 2 — View Citation

Speltz ML, Kapp-Simon K, Collett B, Keich Y, Gaither R, Cradock MM, Buono L, Cunningham ML. Neurodevelopment of infants with single-suture craniosynostosis: presurgery comparisons with case-matched controls. Plast Reconstr Surg. 2007 May;119(6):1874-81. — View Citation

Speltz ML, Kapp-Simon KA, Cunningham M, Marsh J, Dawson G. Single-suture craniosynostosis: a review of neurobehavioral research and theory. J Pediatr Psychol. 2004 Dec;29(8):651-68. Review. — View Citation

Starr JR, Collett BR, Gaither R, Kapp-Simon KA, Cradock MM, Cunningham ML, Speltz ML. Multicenter study of neurodevelopment in 3-year-old children with and without single-suture craniosynostosis. Arch Pediatr Adolesc Med. 2012 Jun 1;166(6):536-42. doi: 10 — View Citation

Starr JR, Kapp-Simon KA, Cloonan YK, Collett BR, Cradock MM, Buono L, Cunningham ML, Speltz ML. Presurgical and postsurgical assessment of the neurodevelopment of infants with single-suture craniosynostosis: comparison with controls. J Neurosurg. 2007 Aug — View Citation

Starr JR, Lin HJ, Ruiz-Correa S, Cunningham ML, Ellenbogen RG, Collett BR, Kapp-Simon KA, Speltz ML. Little evidence of association between severity of trigonocephaly and cognitive development in infants with single-suture metopic synostosis. Neurosurgery — View Citation

Toth K, Collett B, Kapp-Simon KA, Cloonan YK, Gaither R, Cradock MM, Buono L, Cunningham ML, Dawson G, Starr J, Speltz ML. Memory and response inhibition in young children with single-suture craniosynostosis. Child Neuropsychol. 2008 Jul;14(4):339-52. doi — View Citation

* Note: There are 21 references in all — Click here to view all references

Outcome

Type	Measure	Description	Time frame	Safety issue
Primary	Neurodevelopment as assessed by the Bayley Scales of Infant Development II		1st assessment - post diagnosis, pre surgery. 2nd assessment - Target age 18 months. 3rd assessment - Target age 36 months.	No
Secondary	Neurodevelopment as assessed by the Wechsler Intelligence Scale for Children Version IV		Target age 7 years	No