NCT00077831

Brief Summary

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

Trial Health

87
On Track

Trial Health Score

Automated assessment based on enrollment pace, timeline, and geographic reach

Enrollment
535

participants targeted

Target at P75+ for all trials

Timeline
Completed

Started Sep 2001

Longer than P75 for all trials

Geographic Reach
1 country

4 active sites

Status
completed

Health score is calculated from publicly available data and should be used for screening purposes only.

Trial Relationships

Click on a node to explore related trials.

Study Timeline

Key milestones and dates

Study Start

First participant enrolled

September 1, 2001

Completed
2.4 years until next milestone

First Submitted

Initial submission to the registry

February 12, 2004

Completed
4 days until next milestone

First Posted

Study publicly available on registry

February 16, 2004

Completed
10.1 years until next milestone

Primary Completion

Last participant's last visit for primary outcome

April 1, 2014

Completed
1.2 years until next milestone

Study Completion

Last participant's last visit for all outcomes

June 1, 2015

Completed
Last Updated

July 21, 2015

Status Verified

July 1, 2015

Enrollment Period

12.6 years

First QC Date

February 12, 2004

Last Update Submit

July 19, 2015

Conditions

Craniosynostosis

Outcome Measures

Primary Outcomes (1)

  • 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.

Secondary Outcomes (1)

  • Neurodevelopment as assessed by the Wechsler Intelligence Scale for Children Version IV

    Target age 7 years

Interventions

neurobehavioral developmentBEHAVIORAL

observational study of infant and child development

Eligibility Criteria

Age2 Months - 7 Years
Sexall
Healthy VolunteersNo
Age GroupsChild (0-17)
Sampling MethodNon-Probability Sample
Study Population

Children with single-suture craniosynostosis between the ages of 2 months and 3 years were recruited and followed during phase 1. This same chort of children are now being followed at the age of 7 (phase 2). See eligibility criteria section for more information.

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.

Contact the study team to discuss eligibility requirements. They can help determine if this study is right for you.

Sponsors & Collaborators

Study Sites (4)

Children's Health Care of Atlanta

Atlanta, Georgia, 30342, United States

Location

Northwestern University, Cleft Lip and Palate Institute

Westchester, Illinois, 60154, United States

Location

St. Louis Children's Hospital / Washington University

St Louis, Missouri, 63110, United States

Location

Seattle Children's Hospital

Seattle, Washington, 98105, United States

Location

Related Publications (21)

  • 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. doi: 10.1093/jpepsy/jsh068.

    PMID: 15491988BACKGROUND

  • 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.

    PMID: 21848431BACKGROUND

  • 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;107(2 Suppl):103-10. doi: 10.3171/PED-07/08/103.

    PMID: 18459881RESULT

  • 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;2006:3450-5. doi: 10.1109/IEMBS.2006.260321.

    PMID: 17945777RESULT

  • 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. doi: 10.1597/06-190.1.

    PMID: 17760493RESULT

  • 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: 10.1080/09297040701594888. Epub 2007 Sep 26.

    PMID: 17899471RESULT

  • 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. doi: 10.1007/s00381-006-0251-z. Epub 2006 Dec 21.

    PMID: 17186250RESULT

  • 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. doi: 10.1597/04-044.1.

    PMID: 16001919RESULT

  • 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. doi: 10.1597/04-208.1.

    PMID: 16526927RESULT

  • 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.

    PMID: 21204214RESULT

  • 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):635-647. doi: 10.1097/PRS.0b013e31825dc18b.

    PMID: 22929249RESULT

  • 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 with metopic craniosynostosis. Am J Med Genet A. 2010 Sep;152A(9):2203-10. doi: 10.1002/ajmg.a.33557.

    PMID: 20683987RESULT

  • 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 Nov;22(11):1053-61. doi: 10.1111/j.1460-9592.2012.03843.x.

    PMID: 22502768RESULT

  • 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 Aug 19.

    PMID: 20815729RESULT

  • 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.0000316417.06500.DA.

    PMID: 18797362RESULT

  • 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. 2007 Apr 1;143A(7):678-86. doi: 10.1002/ajmg.a.31630.

    PMID: 17343269RESULT

  • 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):1874-1881. doi: 10.1097/01.prs.0000259184.88265.3f.

    PMID: 17440368RESULT

  • 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: 10.1001/archpediatrics.2011.1800.

    PMID: 22312170RESULT

  • 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. 2010 Aug;67(2):408-15; discussion 415-6. doi: 10.1227/01.NEU.0000371992.72539.8B.

    PMID: 20644427RESULT

  • 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.0b013e3182997885.

    PMID: 23851801RESULT

  • 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: 10.1597/13-239.

    PMID: 24841516RESULT

MeSH Terms

Conditions

Craniosynostoses

Condition Hierarchy (Ancestors)

SynostosisDysostosesBone Diseases, DevelopmentalBone DiseasesMusculoskeletal DiseasesCraniofacial AbnormalitiesMusculoskeletal AbnormalitiesCongenital AbnormalitiesCongenital, Hereditary, and Neonatal Diseases and Abnormalities

Study Officials

  • Matthew L. Speltz

    Seattle Children's Hospital

    PRINCIPAL INVESTIGATOR

Study Design

Study Type
observational
Observational Model
CASE CONTROL
Time Perspective
PROSPECTIVE
Sponsor Type
OTHER
Responsible Party
PRINCIPAL INVESTIGATOR
PI Title
Principal Investigator

Study Record Dates

First Submitted

February 12, 2004

First Posted

February 16, 2004

Study Start

September 1, 2001

Primary Completion

April 1, 2014

Study Completion

June 1, 2015

Last Updated

July 21, 2015

Record last verified: 2015-07

Locations

US(4)