Increased utilization of healthcare services in children with craniosynostosis

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  • 1 Department of Neurosurgery, Stanford University School of Medicine; and
  • | 2 Division of Pediatric Neurosurgery, Lucile Packard Children’s Hospital, Stanford, California
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OBJECTIVE

Craniosynostosis is characterized by the premature fusion of at least one cranial suture. Although evidence suggests that patients with both syndromic and nonsyndromic craniosynostosis may benefit from developmental, behavioral, and mental health support, data on utilization of healthcare services are lacking. In this study the authors compared utilization of mental health care, rehabilitation therapies, and other specialty medical services among children with craniosynostosis, children with plagiocephaly, and healthy controls.

METHODS

The Optum Clinformatics Data Mart database was queried to identify 1340 patients with craniosynostosis, of whom 200 had syndromic craniosynostosis. Long-term utilization of mental health care, rehabilitation therapies, and other medical services up to the age of 6 years was calculated. Rates of utilization were compared to healthy controls (n = 1577) and children with plagiocephaly (n = 1249).

RESULTS

Patients with syndromic and nonsyndromic craniosynostosis used mental health care, occupational therapy, speech–language pathology, and other medical services at similar rates (p = 0.1198, p > 0.9999, p = 0.1097, and p = 0.8119, respectively). Mental health services were used more frequently by patients with craniosynostosis (11.0% in patients with syndromic craniosynostosis and 7.5% in those with nonsyndromic craniosynostosis) compared to patients in the plagiocephaly (5.0%, p = 0.0020) and healthy control (2.9%, p < 0.0001) cohorts. Rehabilitation therapies were more frequently used by patients with syndromic craniosynostosis and plagiocephaly (16.0% and 14.1%, respectively), which was significantly higher than use by healthy controls (p < 0.0001). Other medical subspecialty services (developmental pediatrics, ophthalmology, optometry, and audiology) were used by 37.0% of patients with craniosynostosis, compared with 20.9% (p < 0.0001) and 15.1% (p < 0.0001) of patients with plagiocephaly and healthy controls, respectively. Among patients with craniosynostosis, utilization did not differ by race or household income, but it was not uniform by age. Whereas ophthalmology utilization did not differ by age (p = 0.1003), mental health care was most commonly used among older children (p = 0.0107).

CONCLUSIONS

In this study, the authors demonstrate that rates of utilization of mental health care, rehabilitation therapies, and other medical subspecialty services are similar between patients with syndromic and those with nonsyndromic craniosynostosis, but higher than in healthy controls. Although surgical correction may be considered an isolated event, providers and parents need to monitor all children with craniosynostosis—syndromic and nonsyndromic—for developmental and mental health support longitudinally. Future work should explore risk factors driving utilization, including suture involvement, repair type, and comorbidities.

ABBREVIATIONS

CPT = Current Procedural Terminology; ICD-9 = International Classification of Diseases, Ninth Revision; ICD-10 = International Classification of Diseases, Tenth Revision; OT = occupational therapy; PT = physical therapy; SLP = speech–language pathology.

Supplementary Materials

    • Supplementary Tables 1 and 2 (PDF 202 KB)
Illustration from Cinalli et al. (pp 119–127). Printed with permission from © CC Medical Arts.

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  • 1

    Persing JA. MOC-PS(SM) CME article: management considerations in the treatment of craniosynostosis. Plast Reconstr Surg. 2008;121(4)(suppl):111.

  • 2

    Wilkie AOM, Johnson D, Wall SA. Clinical genetics of craniosynostosis. Curr Opin Pediatr. 2017;29(6):622628.

  • 3

    Governale LS. Craniosynostosis. Pediatr Neurol. 2015;53(5):394401.

  • 4

    Ko JM. Genetic syndromes associated with craniosynostosis. J Korean Neurosurg Soc. 2016;59(3):187191.

  • 5

    Boyadjiev SA. Genetic analysis of non-syndromic craniosynostosis. Orthod Craniofac Res. 2007;10(3):129137.

  • 6

    Wenger TL, Hing AV, Evans KN. Apert syndrome. In: GeneReviews. University of Washington,Seattle;1993.Accessed February 28, 2022. https://europepmc.org/article/NBK/nbk541728

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Lu X, Jorge Forte A, Sawh-Martinez R, et al. Association of regional cranial base deformity and ultimate structure in Crouzon syndrome. Plast Reconstr Surg. 2019;143(6):1233e1243e.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Moore MH, Cantrell SB, Trott JA, David DJ. Pfeiffer syndrome: a clinical review. Cleft Palate Craniofac J. 1995;32(1):6270.

  • 9

    Maliepaard M, Mathijssen IMJ, Oosterlaan J, Okkerse JME. Intellectual, behavioral, and emotional functioning in children with syndromic craniosynostosis. Pediatrics. 2014;133(6):e1608e1615.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10

    Bellew M, Liddington M, Chumas P, Russell J. Preoperative and postoperative developmental attainment in patients with sagittal synostosis: 5-year follow-up. J Neurosurg Pediatr. 2011;7(2):121126.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 11

    Kapp-Simon KA, Wallace E, Collett BR, Cradock MM, Crerand CE, Speltz ML. Language, learning, and memory in children with and without single-suture craniosynostosis. J Neurosurg Pediatr. 2016;17(5):578588.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 12

    Becker DB, Petersen JD, Kane AA, Cradock MM, Pilgram TK, Marsh JL. Speech, cognitive, and behavioral outcomes in nonsyndromic craniosynostosis. Plast Reconstr Surg. 2005;116(2):400407.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 13

    Di Chiara A, La Rosa E, Ramieri V, Vellone V, Cascone P. Treatment of deformational plagiocephaly with physiotherapy. J Craniofac Surg. 2019;30(7):20082013.

    • Crossref
    • Search Google Scholar
    • Export Citation
  • 14

    Stata Statistical Software. Version 16.StataCorp LLC;2019.

  • 15

    GraphPad Prism. Version 9. Accessed May 31, 2021. https://www.graphpad.com/

  • 16

    Tillman KK, Höijer J, Ramklint M, Ekselius L, Nowinski D, Papadopoulos FC. Nonsyndromic craniosynostosis is associated with increased risk for psychiatric disorders. Plast Reconstr Surg. 2020;146(2):355365.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 17

    Bannink N, Maliepaard M, Raat H, Joosten KFM, Mathijssen IMJ. Health-related quality of life in children and adolescents with syndromic craniosynostosis. J Plast Reconstr Aesthet Surg. 2010;63(12):19721981.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 18

    Shipster C, Hearst D, Somerville A, Stackhouse J, Hayward R, Wade A. Speech, language, and cognitive development in children with isolated sagittal synostosis. Dev Med Child Neurol. 2003;45(1):3443.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 19

    Kelleher MO, Murray DJ, McGillivary A, Kamel MH, Allcutt D, Earley MJ. Behavioral, developmental, and educational problems in children with nonsyndromic trigonocephaly. J Neurosurg. 2006;105(5)(suppl):382384.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 20

    van Vlimmeren LA, van der Graaf Y, Boere-Boonekamp MM, L’Hoir MP, Helders PJM, Engelbert RHH. Effect of pediatric physical therapy on deformational plagiocephaly in children with positional preference: a randomized controlled trial. Arch Pediatr Adolesc Med. 2008;162(8):712718.

    • Crossref
    • Search Google Scholar
    • Export Citation
  • 21

    Robinson S, Proctor M. Diagnosis and management of deformational plagiocephaly. J Neurosurg Pediatr. 2009;3(4):284295.

  • 22

    Baird LC, Klimo P Jr, Flannery AM, et al. Congress of Neurological Surgeons Systematic review and evidence-based guideline for the management of patients with positional plagiocephaly: the role of physical therapy. Neurosurgery. 2016;79(5):E630E631.

    • Crossref
    • Search Google Scholar
    • Export Citation
  • 23

    Ridgway EB, Berry-Candelario J, Grondin RT, Rogers GF, Proctor MR. The management of sagittal synostosis using endoscopic suturectomy and postoperative helmet therapy. J Neurosurg Pediatr. 2011;7(6):620626.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 24

    Abbott MM, Rogers GF, Proctor MR, Busa K, Meara JG. Cost of treating sagittal synostosis in the first year of life. J Craniofac Surg. 2012;23(1):8893.

  • 25

    Korpilahti P, Saarinen P, Hukki J. Deficient language acquisition in children with single suture craniosynostosis and deformational posterior plagiocephaly. Childs Nerv Syst. 2012;28(3):419425.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 26

    Florisson JMG, van Veelen MLC, Bannink N, et al. Papilledema in isolated single-suture craniosynostosis: prevalence and predictive factors. J Craniofac Surg. 2010;21(1):2024.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 27

    Duan M, Skoch J, Pan BS, Shah V. Neuro-ophthalmological manifestations of craniosynostosis: current perspectives. Eye Brain. 2021;13:2940.

  • 28

    Lehman S. Strabismus in craniosynostosis. Curr Opin Ophthalmol. 2006;17(5):432434.

  • 29

    Chung SA, Yun IS, Moon JW, Lee JB. Ophthalmic findings in children with nonsyndromic craniosynostosis treated by expansion cranioplasty. J Craniofac Surg. 2015;26(1):7983.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 30

    Ricci D, Vasco G, Baranello G, et al. Visual function in infants with non-syndromic craniosynostosis. Dev Med Child Neurol. 2007;49(8):574576.

  • 31

    Brown ZD, Bey AK, Bonfield CM, et al. Racial disparities in health care access among pediatric patients with craniosynostosis. J Neurosurg Pediatr. 2016;18(3):269274.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 32

    Lin Y, Pan IW, Harris DA, Luerssen TG, Lam S. The impact of insurance, race, and ethnicity on age at surgical intervention among children with nonsyndromic craniosynostosis. J Pediatr. 2015;166(5):12891296.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 33

    Shweikeh F, Foulad D, Nuño M, Drazin D, Adamo MA. Differences in surgical outcomes for patients with craniosynostosis in the US: impact of socioeconomic variables and race. J Neurosurg Pediatr. 2016;17(1):2733.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 34

    Hoffman C, Valenti AB, Odigie E, Warren K, Premaratne ID, Imahiyerobo TA. Impact of health disparities on treatment for single-suture craniosynostosis in an era of multimodal care. Neurosurg Focus. 2021;50(4):E13.

    • Crossref
    • Search Google Scholar
    • Export Citation
  • 35

    Gandolfi BM, Sobol DL, Farjat AE, Allori AC, Muh CR, Marcus JR. Risk factors for delayed referral to a craniofacial specialist for treatment of craniosynostosis. J Pediatr. 2017;186:165171.e2.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 36

    Sergesketter AR, Elsamadicy AA, Lubkin DT, et al. Socioeconomic factors, perioperative complications, and 30-day readmission rates associated with delayed cranial vault reconstruction for craniosynostosis. J Craniofac Surg. 2018;29(7):17671771.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 37

    Rubinger L, Chan C, Andrade D, et al. Socioeconomic status influences time to surgery and surgical outcome in pediatric epilepsy surgery. Epilepsy Behav. 2016;55:133138.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 38

    Attenello FJ, Ng A, Wen T, et al. Racial and socioeconomic disparities in outcomes following pediatric cerebrospinal fluid shunt procedures. J Neurosurg Pediatr. 2015;15(6):560566.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 39

    Bellew M, Chumas P. Long-term developmental follow-up in children with nonsyndromic craniosynostosis. J Neurosurg Pediatr. 2015;16(4):445451.

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