Orofacial clefts associated with cardiac anomalies
27 years of experience of a multidisciplinary group in a tertiary hospital in Portugal
DOI:
https://doi.org/10.15448/1980-6108.2021.1.37355Keywords:
Cleft lip, cleft palate, congenital abnormalities, congenital heart defectAbstract
AIMS: Orofacial clefts (OFC) are a heterogeneous group of birth defects arising in about 1.7/1000 newborns. They can occur with other congenital anomalies, including heart defects. We aim to describe a population with orofacial clefts and associated cardiac anomalies.
METHODS: Retrospective study of patients attended in the Cleft Lip and Palate Multidisciplinary Group outpatient clinic at Hospital Universitario São João, Porto-Portugal. Medical records from January 1992 through December 2018 were reviewed. Patients were divided into four groups according to the Spina classification: cleft lip (CL), cleft lip and palate (CLP), isolated cleft palate (CP) and atypical cleft (AC). Further categorization included gender, affected relatives, associated congenital anomalies and syndromes.
RESULTS: From the 588 patients included, 77 (13%) presented cardiac anomalies. Of those with orofacial cleft and cardiac anomalies, 53% were males and 17% had known affected relatives. CP was the most common cleft among patients with cardiac anomaly (~56%). Additional congenital anomalies were found in 89.7% of patients, namely facial defects, central nervous system, renal and skeletal malformations. A recognizable syndrome was identified in 61.5%, being Pierre-Robin the most common (n=22), followed by 22q11.2 microdeletion (n=9). Both additional congenital anomalies and recognizable syndromes were significantly more prevalent in patients with heart disease (p<0.05). The main groups of cardiac anomalies were left-to-right shunt (n=47) and right ventricular outflow tract obstruction (n=14). From these, 26 had a ventricular septal defect, 15 atrial septal defect and seven patients had tetralogy of Fallot. Five patients had dysrhythmias.
CONCLUSIONS: Due to the high prevalence of cardiac anomalies in the cleft population, a routine cardiac evaluation should be performed in all these patients.
Downloads
References
Kasatwar A, Borle R, Bhola N, K R, Prasad GSV, Jadhav A. Prevalence of congenital cardiac anomalies in patients with cleft lip and palate – Its implications in surgical management. Journal of Oral Biology and Craniofacial Research. Setembro de 2018;8(3):241–4. https://doi.org/10.1016/j.jobcr.2017.09.009.
Hadadi AI, Al Wohaibi D, Almtrok N, Aljahdali N, AlMeshal O, Badri M. Congenital anomalies associated with syndromic and non-syndromic cleft lip and palate. JPRAS Open. Dezembro de 2017;14:5–15. https://doi.org/10.1016/j.jpra.2017.06.001.
Allori AC, Mulliken JB, Meara JG, Shusterman S, Marcus JR. Classification of cleft lip/palate: then and now. The Cleft Palate-Craniofacial Journal. Março de 2017;54(2):175–88. https://doi.org/10.1597/14-080.
Rodrigues R, Fernandes MH, Monteiro AB, Furfuro R, Sequeira T, Silva CC, et al. SPINA classification of cleft lip and palate: A suggestion for a complement. Archives de Pédiatrie. Outubro de 2018;25(7):439–41. https://doi.org/10.1016/j.arcped.2018.08.001.
Chan K.W., Lee K.H., Pang K.K., Mou J.W., Tam Y.H. Clinical characteristics of children with orofacial cleft in a tertiary centre in Hong Kong. Hong Kong J Paediatr. 2013;18:147–151.
Santana TM de, Silva MD de P, Brandão SR, Gomes A de OC, Pereira RMR, Rodrigues M, et al. Nascidos vivos com fissura de lábio e/ou palato: as contribuições da fonoaudiologia para o Sinasc. Revista CEFAC. Abril de 2015;17(2):485–91. https://doi.org/10.1590/1982-021620158014.
Rizk F Salameh P, Hamadé A., Congenital Anomalies: Prevalence and Risk Factors, Universal Journal of Public Health, Vol. 2, No. 2, pp. 58 - 63, 2014. DOI: 10.13189/ujph.2014.020204.
Harry BL, Tebockhorst S, Deleyiannis FW-B. The impact of congenital cardiovascular malformations on the assessment and surgical management of infants with cleft lip and/or palate. The Cleft Palate-Craniofacial Journal. Maio de 2013;50(3):323–9. https://doi.org/10.1597/12-131.
Asani M, Aliyu I. Pattern of congenital heart defects among children with orofacial clefts in Northern Nigeria. J Cleft Lip Palate Craniofac Anomal. 2014;1(2):85. https://doi.org/10.4103/2348-2125.137895.
Wyse RKH, Mars M, Al-Mahdawi S, Russell-Eggitt IM, Blake KD. Congenital heart anomalies in patients with clefts of the lip and/or palate. Cleft Palate Journal. Julho de 1990;27(3):258–65. https://doi.org/10.1597/1545-1569_1990_027_0258_chaipw_2.3.co_2.
Barbosa MM, Rocha CMG, Katina T, Caldas M, Codorniz A, Medeiros C. Prevalence of congenital heart diseases in oral cleft patients. Pediatric Cardiology. 1 de Julho de 2003;24(4):369–74. https://doi.org/10.1007/s00246-002-0335-9.
Leite G, Ururahy M, Bezerra J, Lima V, Costa M, Freire S, et al. Cardiovascular abnormalities in patients with oral cleft: a clinical-electrocardiographic-echocardiographic study. Clinics [Internet]. 29 de Abril de 2018 [citado 5 de Setembro de 2020];73. Disponível em: https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5890169/?report=classic. https://doi.org/10.6061/clinics/2018/e108.
Venkatesh R. Syndromes and anomalies associated with cleft. Indian J Plast Surg. 2009;42(3):51. https://doi.org/10.4103/0970-0358.57187.
Sekhon P.S., Ethunandan M., Markus A.F., Gopal Krishnan, Bhasker Rao C. Congenital anomalies associated with cleft lip and palate—an analysis of 1623 consecutive patients. Cleft Palate Craniofac J. 2011;48:371–378. https://doi.org/10.1597/09-264.
Geis N, Seto B, Bartoshesky L, Lewis MB, Pashayan HM. The prevalence of congenital heart disease among the population of a metropolitan cleft lip and palate clinic. Cleft Palate J. 1981;18(1):19-23.
Otaigbe B, Akadiri O, Eigbobo J. Clinical and echocardiographic findings in an African pediatric population of cleft lip/palate patients: A preliminary report. Nig J Cardiol. 2013;10(1):6. https://doi.org/10.4103/0189-7969.118574.
Beriaghi S, Myers SL, Jensen SA, Kaimal S, Chan CM, Schaefer GB. Cleft lip and palate: Association with other congenital malformations. J Clin Pediatr Dent 2009;33:207-10. https://doi.org/10.17796/jcpd.33.3.c244761467507721.
Aljohar A, Ravichandran K, Subhani S. Pattern of cleft lip and palate in hospital-based population in Saudi Arabia: retrospective study. Cleft Palate Craniofac J. 2008;45(6):592-596. http//doi.org/10.1597/06-246.1.
Keyte A, Hutson MR. The neural crest in cardiac congenital anomalies. Differentiation. Julho de 2012;84(1):25–40. https://doi.org/10.1016/j.diff.2012.04.005.
Obeid R, Holzgreve W, Pietrzik K. Folate supplementation for prevention of congenital heart defects and low birth weight: an update. Cardiovasc Diagn Ther. 2019;9(Suppl 2):S424-S433. http//doi:10.21037/cdt.2019.02.03
Impellizzeri, A., Giannantoni, I., Polimeni, A. et al. Epidemiological characteristic of Orofacial clefts and its associated congenital anomalies: retrospective study. BMC Oral Health 19, 290 (2019). https://doi.org/10.1186/s12903-019-0980-5
Downloads
Published
How to Cite
Issue
Section
License
Copyright (c) 2021 Scientia Medica
This work is licensed under a Creative Commons Attribution 4.0 International License.
Copyright
The submission of originals to Scientia Medica implies the transfer by the authors of the right for publication. Authors retain copyright and grant the journal right of first publication. If the authors wish to include the same data into another publication, they must cite Scientia Medica as the site of original publication.
Creative Commons License
Except where otherwise specified, material published in this journal is licensed under a Creative Commons Attribution 4.0 International license, which allows unrestricted use, distribution and reproduction in any medium, provided the original publication is correctly cited.
