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Skeletal expansion via craniofacial distraction osteogenesis technique in syndromic craniosynostosis: impact on ophthalmic parameters

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Abstract

Background

This study aims to compare the changes in ophthalmic parameters among syndromic craniosynostosis patients who underwent craniofacial skeletal expansion procedures via distraction osteogenesis (DO).

Method

A retrospective study was conducted involving syndromic craniosynostosis patients who underwent surgical expansion via the DO technique from the year 2012 to March 2022. Changes in six parameters which consist of visual acuity, refractive error, optic disc health, intraocular pressure, degree of proptosis and orbital volume were measured objectively pre and post-surgery. For categorical parameters, the Chi-square cross-tab test was done. Paired sample T-test was used for normally distributed variables. Wilcoxon signed-rank test was used for non-normally distributed data.

Results

Visual impairment was present in 21.4% of eyes before surgery and increased to 28.5% post-surgery. Three patients had changes of refractive error post-surgery with one developed hypermetropia, another developed anisometropia and the last had improvement to no refractive error. Two patients had optic disc swelling which was resolved post-surgery. Intraocular pressure changes were inconsistent post-surgery. All patients achieved a significant reduction in the degree of proptosis post-surgery. Orbital volume calculation using computed tomography (CT) scans shows a significant increase in volume post-surgery for all patients.

Conclusion

Our study shows a significant increase in orbital volume post-surgery with a reduction in the degree of proptosis. Optic disc and nerve health improved after the surgery. Changes in terms of visual acuity, refractive error and IOP were inconsistent after the surgical intervention.

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Availability of data and materials

All data generated or analyzed during this study are included in this published article.

References

  1. Sawh-Martinez R, Steinbacher DM (2019) Syndromic craniosynostosis. Clin Plast Surg 46(2):141–155

    Article  PubMed  Google Scholar 

  2. Wilkie AO, Byren JC, Hurst JA, Jayamohan J, Johnson D, Knight SJ et al (2010) Prevalence and complications of single-gene and chromosomal disorders in craniosynostosis. Pediatrics 126(2):e391–e400

    Article  PubMed  Google Scholar 

  3. Azoury SC, Reddy S, Shukla V, Deng CX (2017) Fibroblast growth factor receptor 2 (FGFR2) mutation related syndromic craniosynostosis. Int J Biol Sci 13(12):1479–1488

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  4. Ganesh A, Edmond J, Forbes B, Katowitz WR, Nischal KK, Miller M et al (2019) An update of ophthalmic management in craniosynostosis. J AAPOS 23(2):66–76

    Article  PubMed  Google Scholar 

  5. Buchanan EP, Xue Y, Xue AS, Olshinka A, Lam S (2017) Multidisciplinary care of craniosynostosis. J Multidiscip Healthc 10:263–270

    Article  PubMed  PubMed Central  Google Scholar 

  6. Hariri F, Farhana NA, Abdullah NA, Ibrahim N, Ramli NM, Mohd Abdullah AA et al (2021) Optic canal characteristics in pediatric syndromic craniosynostosis. J Cranio-Maxillofac Surg 49(12):1175–1181

    Article  Google Scholar 

  7. Tuite GF, Chong WK, Evanson J, Narita A, Taylor D, Harkness WF et al (1996) The effectiveness of papilledema as an indicator of raised intracranial pressure in children with craniosynostosis. Neurosurgery 38(2):272–278

    Article  CAS  PubMed  Google Scholar 

  8. Derderian C, Seaward J (2012) Syndromic craniosynostosis. Semin Plast Surg 26(2):64–75

    Article  PubMed  PubMed Central  Google Scholar 

  9. Mathijssen IM (2015) Guideline for care of patients with the diagnoses of craniosynostosis: working group on craniosynostosis. J Craniofac Surg 26(6):1735–1807

    Article  PubMed  PubMed Central  Google Scholar 

  10. Tahiri Y, Taylor J (2014) An update on midface advancement using Le Fort II and III distraction osteogenesis. Semin Plast Surg 28(4):184–192

    Article  PubMed  PubMed Central  Google Scholar 

  11. Rafique Ali AA, Ismail F, May May C, Mohd Abdullah AA, Khaliddin N, Hariri F et al (2022) Ophthalmic features of craniosynostosis: a Malaysian experience. Eur J Ophthalmol 32(3):1417–1423

    Article  PubMed  Google Scholar 

  12. Park NR, Moon JH, Lee JK (2019) Hertel exophthalmometer versus computed tomography scan in proptosis estimation in thyroid-associated orbitopathy. Clin Ophthalmol 13:1461–1467

    Article  PubMed  PubMed Central  Google Scholar 

  13. Shyu VBH, Hsu C-E, Chen C-H, Chen C-T (2015) The 3D-assisted quantitative assessment of orbital volume using an open-source software platform in a Taiwanese population. PLoS One 10(3):e0119589

    Article  PubMed  PubMed Central  Google Scholar 

  14. Goh PP, Abqariyah Y, Pokharel GP, Ellwein LB (2005) Refractive error and visual impairment in school-age children in Gombak District, Malaysia. Ophthalmology 112(4):678–685

    Article  PubMed  Google Scholar 

  15. Chew FLM, Thavaratnam LK, Shukor INC, Ramasamy S, Rahmat J, Reidpath DD et al (2018) Visual impairment and amblyopia in Malaysian pre-school children – the SEGPAEDS study. Med J Malaysia 73(1):25–30

    CAS  PubMed  Google Scholar 

  16. Khan SH, Nischal KK, Dean F, Hayward RD, Walker J (2003) Visual outcomes and amblyogenic risk factors in craniosynostotic syndromes: a review of 141 cases. Br J Ophthalmol 87(8):999–1003

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  17. Tay T, Martin F, Rowe N, Johnson K, Poole M, Tan K et al (2006) Prevalence and causes of visual impairment in craniosynostotic syndromes. Clin Experiment Ophthalmol 34(5):434–440

    Article  PubMed  Google Scholar 

  18. Liasis A, Nischal KK, Walters B, Thompson D, Hardy S, Towell A et al (2006) Monitoring visual function in children with syndromic craniosynostosis: a comparison of 3 methods. Arch Ophthalmol 124(8):1119–1126

    Article  PubMed  Google Scholar 

  19. Duan M, Skoch J, Pan BS, Shah V (2021) Neuro-ophthalmological manifestations of craniosynostosis: current perspectives. Eye Brain 13:29–40

    Article  PubMed  PubMed Central  Google Scholar 

  20. Bentley RP, Sgouros S, Natarajan K, Dover MS, Hockley AD (2002) Normal changes in orbital volume during childhood. J Neurosurg 96(4):742–746

    Article  PubMed  Google Scholar 

  21. Touze R, Bremond-Gignac D, Robert MP (2019) Ophthalmological management in craniosynostosis. Neurochirurgie 65(5):310–317

    Article  CAS  PubMed  Google Scholar 

  22. Sharma N, Greenwell T, Hammerton M, David DJ, Selva D, Anderson PJ (2016) The ophthalmic sequelae of Pfeiffer syndrome and the long-term visual outcomes after craniofacial surgery. J AAPOS 20(4):315–319

    Article  PubMed  Google Scholar 

  23. Lenroot RK, Giedd JN (2006) Brain development in children and adolescents: Insights from anatomical magnetic resonance imaging. Neurosci Biobehav Rev 30(6):718–729

    Article  PubMed  Google Scholar 

  24. Schultz KP, Wiggins CJ, Streff H, Shah VS, Buchanan EP (2019) Syndromic multisuture craniosynostosis with associated anterior segment dysgenesis, optic nerve hypoplasia, and congenital glaucoma. Cleft Palate Craniofac J 56(6):823–826

    Article  PubMed  Google Scholar 

  25. Alshamrani AA, Al-Shahwan S (2018) Glaucoma with Crouzon syndrome. J Glaucoma 27(6):e110–e112

    Article  PubMed  Google Scholar 

  26. Forte AJ, Steinbacher DM, Persing JA, Brooks ED, Andrew TW, Alonso N (2015) Orbital dysmorphology in untreated children with Crouzon and Apert syndromes. Plast Reconstr Surg 136(5):1054–1062

    Article  CAS  PubMed  Google Scholar 

  27. Way BLM, Khonsari RH, Karunakaran T, Nysjö J, Nyström I, Dunaway DJ et al (2019) Correcting exorbitism by monobloc frontofacial advancement in Crouzon-Pfeiffer syndrome: an age-specific, time-related controlled study. Plast Reconstr Surg 143(1):121e–132e

    Article  CAS  PubMed  Google Scholar 

  28. Lu X, Forte AJ, Junn A, Dinis J, Alperovich M, Alonso N et al (2022) Orbitofacial morphology changes with different suture synostoses in Crouzon syndrome. Cranio-Maxillofac Surg 50(5):406–418

    Article  CAS  Google Scholar 

  29. Britto JA, Greig A, Abela C, Hearst D, Dunaway DJ, Evans RD (2014) Frontofacial surgery in children and adolescents: techniques, indications, outcomes. Semin Plast Surg 28(3):121–129

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  30. Bender CA, Veneman W, Veenland JF, Mathijssen IMJ, Hop WCJ, Koudstaal MJ et al (2013) Orbital aspects following monobloc advancement in syndromic craniosynostosis. J Cranio-Maxillofac Surg 41(7):e146–e153

    Article  Google Scholar 

  31. Ramli N, Kala S, Samsudin A, Rahmat K, Abidin ZZ (2015) Proptosis-correlation and agreement between Hertel exophthalmometry and computed tomography. Orbit 34(5):257–262

    Article  PubMed  Google Scholar 

  32. Nout E, van Bezooijen JS, Koudstaal MJ, Veenland JF, Hop WC, Wolvius EB et al (2012) Orbital change following Le Fort III advancement in syndromic craniosynostosis: quantitative evaluation of orbital volume, infra-orbital rim and globe position. J Craniomaxillofac Surg 40(3):223–228

    Article  PubMed  Google Scholar 

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Acknowledgements

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Funding

The authors received no financial support for the research, authorship, and/or publication of this article.

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Authors and Affiliations

  1. Department of Oral and Maxillofacial Clinical Sciences, Faculty of Dentistry, Universiti Malaya, 50603, Kuala Lumpur, Malaysia

    Lim Chin Kai, Muhammad Kamil Hassan & Firdaus Hariri

  2. Department of Ophthalmology, Faculty of Medicine, Universiti Malaya, Kuala Lumpur, Malaysia

    Nurliza Khaliddin

Authors
  1. Lim Chin Kai
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  2. Nurliza Khaliddin
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  3. Muhammad Kamil Hassan
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  4. Firdaus Hariri
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Contributions

LCK involved in the conceptualization of the study, analyzed and interpreted the patient data and also writing of the manuscript draft. FH and NI were involved in conceptualization of the study along with major review and editing of the manuscript. MKH was involved with review and editing of the manuscript. All authors read and approved the final manuscript.

Corresponding author

Correspondence to Firdaus Hariri.

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Conflict of interest

The authors declare no competing interests.

Ethical approval and consent to participate

Ethical approval was obtained from the Medical Research Ethics Committee of the Universiti Malaya Medical Centre, Kuala Lumpur, Malaysia. (Approval Number- DF OS2022/0083 (P)).

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Kai, L.C., Khaliddin, N., Hassan, M.K. et al. Skeletal expansion via craniofacial distraction osteogenesis technique in syndromic craniosynostosis: impact on ophthalmic parameters. Int Ophthalmol 44, 147 (2024). https://doi.org/10.1007/s10792-024-03084-y

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  • DOI: https://doi.org/10.1007/s10792-024-03084-y

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