This systematic review aims to evaluate nasoalveolar molding (NAM) in the context of burden of care defined as physical, psychosocial, or financial burden on caregivers.
Search Methods:
Following Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines, 5 databases were searched from inception through December 24, 2019, for keywords and subject headings pertaining to cleft lip and/or palate and NAM.
Eligibility Criteria:
Clinical studies on NAM with reference to physical (access to care, number of visits, distance traveled), psychosocial (caregiver perceptions, family interactions, breast milk feeding), and financial (direct and indirect costs) burden were included.
Data Collection and Analysis:
Study selection was performed by 2 independent reviewers.
Results:
The search identified 1107 articles and 114 articles remained for qualitative synthesis. Burden of care domains were discussed but not measured in 43% of articles and only 25% assessed burden of care through a primary outcome. Of these, 20 articles reported on physical, 8 articles on psychosocial, and 12 articles on financial burden. Quality of evidence is limited by study design and risk of bias.
Conclusion:
Nasoalveolar molding has been indiscriminately associated with burden of care in the literature. Although NAM may not be the ideal treatment option for all patients and families, the physical considerations are limited when accounting for the observed psychosocial advantages. Financial burden appears to be offset, but further research is required. Teams should directly assess the impact of this early intervention on the well-being of caregivers and advance strategies that improve access to care.
Akarsu-GuvenBArisanAOzgurFAksuM. Influence of nasoalveolar molding on skeletal development in patients with unilateral cleft lip and palate at 5 years of age. Am J Orthod Dentofacial Orthop. 2018;153(4):489–495.
3.
AlberconiTFSiqueiraGLCSathlerRKellyKAGaribDG. Assessment of orthodontic burden of care in patients with unilateral complete cleft lip and palate. Cleft Palate Craniofac J. 2018;55(1):74–78.
4.
AlperovichMFreyJDShetyePRGraysonBHVyasRM. Breast milk feeding rates in patients with cleft lip and palate at a north American craniofacial center. Cleft Palate Craniofac J. 2017;54(3):334–337.
BauerFXSchonbergerMGattingerJEblenkampMWintermantelERauAGüllFDWolffKLoeffelbeinDJ. RapidNAM: generative manufacturing approach of nasoalveolar molding devices for presurgical cleft lip and palate treatment. Biomed Tech (Berl). 2017;62(4):407–414.
8.
BlumeCPHensonTB. An access to care study for the pre-surgical nasoalveolar molding and other treatments for cleft lip and palate. Tex Dent J. 2011;128(7):639–645.
9.
BroderHLFloresRLCloustonSKirschnerREGarfinkleJSSischoLPhilipsC. Surgeon’s and caregivers’ appraisal of primary cleft lip treatment with and without nasoalveolar molding: a prospective multicenter pilot study. Plast Reconstr Surg. 2016;137(3):938–945.
10.
ChangSYLonicDPaiBCLoLJ. Primary repair in patients with unilateral complete cleft of lip and primary palate: assessment of outcomes. Ann Plast Surg. 2018;80(2S suppl 1):S2–S6.
11.
ChangCSWallaceCGPaiBCChiuYTHsiehYJChenIJLiaoYFLiouEJChenPK. Comparison of two nasoalveolar molding techniques in unilateral complete cleft lip patients: a randomized, prospective, single-blind trial to compare nasal outcomes. Plast Reconstr Surg. 2014;134(2):275–282.
12.
ChenYFLiaoYF. A modified nasoalveolar molding technique for correction of unilateral cleft nose deformity. J Craniomaxillofac Surg. 2015;43(10):2100–2105.
13.
DeanRAWainwrightDJDoringoILTeichgraeberJFGreivesMR. assessing burden of care in the patient with cleft lip and palate: factors influencing completion and noncompletion of nasoalveolar molding. Cleft Palate Craniofac J. 2019;56(6):759–765.
14.
DecWShetyePRDavidsonEHGraysonBHBrechtLECuttingCBWarrenSM. Presurgical nasoalveolar molding and primary gingivoperiosteoplasty reduce the need for bone grafting in patients with bilateral clefts. J Craniofac Surg. 2013;24(1):186–190.
15.
DesparsJPeterCBorghiniAPierrehumbertBHabersaatSMuller-NixCAnsermetFHohlfeldJ. Impact of a cleft lip and/or palate on maternal stress and attachment representations. Cleft Palate Craniofac J. 2011:48(4):419–424.
16.
EsmondeNOGarfinkleJSChenYLambertWEKuangAA. Factors associated with adherence to nasoalveolar molding (NAM) by caregivers of infants born with cleft lip and palate. Cleft Palate Craniofac J. 2018:55(2):252–258.
17.
FloresRLShetyePR. The nasoalveolar molding (NAM) treatment protocol. Cleft Palate Craniofac J. 2019;56(8):1124–1125.
18.
GarfinkleJSKingTWGraysonBHBrechtLECuttingCB. A 12-year anthropometric evaluation of the nose in bilateral cleft lip-cleft palate patients following nasoalveolar molding and cutting bilateral cleft lip and nose reconstruction. Plast Reconstr Surg. 2011;127(4):1659–1667.
19.
GBD 2016 Disease and Injury Incidence and Prevalence Collaborators. Global, regional, and national incidence, prevalence, and years lived with disability for 328 diseases and injuries for 195 countries, 1990-2016: a systematic analysis for the global burden of disease study 2016. Lancet. 2017;390(10100):1211–1259.
20.
GeorgeLKGwytherLP. Caregiver well-being: a multidimensional examination of family caregivers of demented adults. Gerontologist. 1986:26(3):253–259.
21.
GraysonBHGarfinkleJS. Early cleft management: the case for nasoalveolar molding. Am J Orthod Dentofacial Orthop. 2014;145(2):134–142.
22.
GrillFDRitschlLMDikelHRauARothMEblenkampMWolffKLoeffelbeinDJBauerFX. Facilitating CAD/CAM nasoalveolar molding therapy with a novel click-in system for nasal stents ensuring a quick and user-friendly chairside nasal stent exchange. Sci Rep. 2018;8(1):12084.
23.
HabersaatSMonnierMPeterCBolomeyLBorghiniADesparsJPierrehumbertBMüller-NixCAnsermetFHohlfeldJ. Early mother-child interaction and later quality of attachment in infants with an orofacial cleft compared to infants without cleft. Cleft Palate Craniofac J. 2013;50(6):704–712.
24.
HigginsJPTSterneJACSavovićJPageMPHróbjartssonABoutronIReevesBEldridgeS. A revised tool for assessing risk of bias in randomized trials. In: ChandlerJMcKenzieJBoutronIWelchV (eds). Cochrane Methods. Cochrane Database of Systematic Reviews. 2016; 10(suppl 1):29–31.
25.
HopkinsEEGazzaEMarazitaML. Parental experience caring for cleft lip and palate infants with nasoalveolar moulding. J Adv Nurs. 2016;72(10):2413–2422.
26.
KhavaninNJennyHJodehDSScottMARottgersSASteinbergJP. Cleft and craniofacial team orthodontic care in the United States: a survey of the ACPA. Cleft Palate Craniofac J. 2019;56(7):860–866.
27.
Levy-BercowskiDAbreuADeLeonELooneySStockstillJWeilerMSantiagoPE. Complications and solutions in presurgical nasoalveolar molding therapy. Cleft Palate Craniofac J. 2009;46(5):521–528.
28.
LiangZYaoJChenPKTZhengCYangJ. Effect of presurgical nasoalveolar molding on nasal symmetry in unilateral complete cleft lip/palate patients after primary cheiloplasty without concomitant nasal cartilage dissection: early childhood evaluation. Cleft Palate Craniofac J. 2018;55(7):935–940.
29.
LiaoYF. A modified nasoalveolar molding technique for correction of unilateral cleft nose deformity. Cleft Palate-Craniofac J. 2015;53(4):e152–e153.
30.
LiaoYFChenIJPaiCJKoWCWangYC. Comparative outcomes of two nasoalveolar molding techniques for bilateral cleft nose deformity. Plast Reconstr Surg. 2014;133(1):103–110.
31.
LiaoYFHsiehYJChenIJKoWCChenPK. Comparative outcomes of two nasoalveolar molding techniques for unilateral cleft nose deformity. Plast Reconstr Surg. 2012;130(6):1289–1295.
32.
MaillardSRetrouveyJMAhmedMKTaubPJ. correlation between nasoalveolar molding and surgical, aesthetic, functional and socioeconomic outcomes following primary repair surgery: a systematic review. J Oral Maxillofac Res. 2017;8(3):e2.
33.
MatsuoKHiroseTTomonoTIwasawaMKatohdaSTakahashiNKohB. Nonsurgical correction of congenital auricular deformities in the early neonate: a preliminary report. Plast Reconstr Surg. 1984;73(1):38–51.
34.
MoherDLiberatiATetzlaffJAltmanDG, The PRISMA Group. Preferred Reporting Items for Systematic Reviews and Meta-Analyses: the PRISMA statement. Int J Surg. 2010;8(5):336–341.
35.
Nazarian MobinSSKaratsonyiAVidarENGamerSGroperJHammoudehJAUrataMM. Is presurgical nasoalveolar molding therapy more effective in unilateral or bilateral cleft lip-cleft palate patients?Plast Reconstr Surg. 2011;127(3):1263–1269.
36.
Nur YilmazRBGermec CakanDTurkyilmaz UyarE. Maternal and paternal well-being during nasoalveolar molding and primary surgery periods. J Craniofac Surg. 2019;30(7):2227–2232.
37.
O’NeillGRossMM. Burden of care: an important concept for nurses. Health Care Women Int. 1991;12(1):111–121.
38.
PatelPARubinMSCloustonSLalezaradehFBrechtLECuttingCBShetyePRWarrenSMGraysonBH. Comparative study of early secondary nasal revisions and costs in patients with clefts treated with and without nasoalveolar molding. J Craniofac Surg. 2015;26(4):1229–1233.
39.
PetersonPE, WHO Oral Health Programme. The world oral health report 2003: continuous improvement of oral health in the 21st century—the approach of the WHO global oral health Programme/Poul Erik Petersen. 2003. Accessed August 4, 2019. https://apps.who.int/iris/handle/10665/68506.
40.
PfeiferTMGraysonBHCuttingCB. Nasoalveolar molding and gingivoperiosteoplasty versus alveolar bone graft: an outcome analysis of costs in the treatment of unilateral cleft alveolus. The Cleft palate-craniofacial journal. 2002;39(1):26–29.
41.
PremkumarSKunthavaiCRTovani-PaloneMR. Clinical application of customized presurgical nasoalveolar molding for the treatment of unilateral complete cleft lip and alveolus: case report. Electron J Gen Med. 2018;15(5):em76.
42.
QuastAWaschkauJSaptschakJDaratsianosNJordanKFrombergerPMüllerJLMeyer-MarcottyP. Facial perception of infants with cleft lip and palate with/without the NAM appliance. J Orofac Orthop. 2018;79(6):380–388.
43.
RitschlLMRauAGüllFDdi BoraBWolffKDSchönbergerMWintermantelEBauerFXLoeffelbeinDJ. Pitfalls and solutions in virtual design of nasoalveolar molding plates by using CAD/CAM technology—a preliminary clinical study. Journal of Cranio-Maxillofacial Surgery. 2016;44(4):453–459.
44.
RobertsJMJacobsAMorrowBHauckRSamsonTD. Current trends in unilateral cleft lip care: a 10-year update on practice patterns [published online ahead of print]. Ann Plast Surg. 2019.
45.
RubinMSCloustonSAhmedMMLoweKShetyePRBroderHLWarrenSMGraysonBH. Surgeons’ assessment of presurgical clefts and predicted surgical outcome in patients treated with and without nasoalveolar molding. J Craniofac Surg. 2015;26(1):71–75.
46.
SaadMSFataMFaroukAHabibAMAGadMTayelMBMareiMK. Early progressive maxillary changes with nasoalveolar molding: randomized controlled clinical trial. JDR Clin Trans Res. 2019:20(10):1–13.
47.
ShayPLGoldsteinJAPaligaJTWinkJJacksonOALowDBartlettSPTaylorJA. A comparative cost analysis of cleft lip adhesion and nasoalveolar molding before formal cleft lip repair. Plast Reconstr Surg. 2015;136(6):1264–1271.
48.
ShenCYaoCAMageeWIIIChaiGZhangY. Presurgical nasoalveolar molding for cleft lip and palate: the application of digitally designed molds. Plastic and reconstructive surgery. 2015;135(6):1007e–1015e.
49.
ShettyVAgrawalRKSailerHF. Long-term effect of presurgical nasoalveolar molding on growth of maxillary arch in unilateral cleft lip and palate: randomized controlled trial. Int J Oral Maxillofac Surg. 2017;46:977–987.
50.
ShettyVThakralASreekumarC. Comparison of early onset nasoalveolar molding with patients who presented for molding up to 1 year of age. J Oral Maxillofac Surg. 2016;74(4):811–827.
51.
ShettyVVyasHJSharmaSMSailerHF. A comparison of results using nasoalveolar moulding in cleft infants treated within 1 month of life versus those treated after this period: development of a new protocol. Int J Oral Maxillofac Surg. 2012;41(1):28–36.
52.
ShetyePKGraysonBH. Nasoalveolar molding treatment protocol in patients with cleft lip and palate. Semin Orthodont. 2017;23(3):261–267.
53.
SingerEDaskalogiannakisJRussellKAMercadoAMHathawayRRStoutlandALongREFesslerJSembGShawWC. Burden of care of various infant orthopedic protocols for improvement of nasolabial esthetics in patients with CUCLP. Cleft Palate Craniofac J. 2018;55(9):1236–1243.
54.
SinghAThakurSSinghalPDiwanaVKRaniA. A comparative evaluation of efficacy and efficiency of Grayson’s presurgical nasoalveolar molding technique in patients with complete unilateral cleft lip and palate with those treated with Figueroa’s modified technique. Contemp Clin Dent. 2018;9(suppl 1):S28–S33.
55.
SischoLBroderHLPhillipsC. Coping with cleft: a conceptual framework of caregiver responses to nasoalveolar molding. Cleft Palate Craniofac J. 2015;52(6):640–650.
56.
SischoLChanJWSteinMSmithCvan AalstJBroderHL. Nasoalveolar molding: prevalence of cleft centers offering NAM and who seeks it. Cleft Palate Craniofac J. 2012;49(3):70–275.
57.
SischoLCloustonSAPhillipsCBroderHL. Caregiver responses to early cleft palate care: a mixed method approach. Health Psychol. 2016;35(5):474–482.
58.
SitzmanTJCoyneSMBrittoMT. The burden of care for children with unilateral cleft lip: a systematic review of revision surgery. Cleft Palate Craniofac J. 2016;53(4):84–94.
59.
SitzmanTJGirottoJAMarcusJR. Current surgical practices in cleft care: unilateral cleft lip repair. Plast Reconstr Surg. 2008;121(5):261e–270e.
60.
St JohnDPaiLBelferMLMullikenJB. Effects of a child with a craniofacial anomaly on stability of the parental relationship. J Craniofac Surg. 2003;14(5):704–708.
61.
SteenlandKArmstrongB. An overview of methods for calculating the burden of disease due to specific risk factors. Epidemiology. 2006;17(5):512–519.
62.
SterneJACHernánMAReevesBCSavovićJBerkmanNDViswanathanMHenryDAltmanDGAnsariMTBoutronI, et al.ROBINS-I: a tool for assessing risk of bias in non-randomised studies of interventions. BMJ. 2016;355:i4919.
63.
SubramanianCSPrasadNKChitharanjanABLiouEJ. A modified presurgical orthopedic (nasoalveolar molding) device in the treatment of unilateral cleft lip and palate. Eur J Dent. 2016;10(3):435–438.
64.
ViswanathanMPatnodeCDBerkmanNDBassEBChangSHartlingLMuradHTreadwellJRKaneRL. Assessing the risk of bias in systematic reviews of health care interventions. In: Methods Guide for Effectiveness and Comparative Effectiveness Reviews [Internet]. Agency for Healthcare Research and Quality (US); 2017. Accessed December 26, 2019. https://www.ncbi.nlm.nih.gov/books/NBK519366/
65.
WangQZhouLZhaoJZKoEW. An extraoral nasoalveolar molding technique in complete unilateral cleft lip and palate. Plast Reconstr Surg Glob Open. 2013;1(4):e26.
66.
WeinfeldABHollierLHSpiraMStalS. International trends in the treatment of cleft lip and palate. Clin Plast Surg. 2005;32(1):19–23.
67.
WoodsSMGarfinkleJSCovellDAWangMBuschLSDoyleLM. Early weight gain in infants with cleft lip and palate treated with and without nasoalveolar molding: a retrospective study. Cleft Palate Craniofac J. 2018;56(7):902–907.
68.
World Health Organization. Global strategies to reduce the health-care burden of craniofacial anomalies: report of WHO meetings on international collaborative meetings on craniofacial anomalies. 2001. Accessed August 4, 2019. https://apps.who.int/iris/handle/10665/42594
69.
YuQGongXShenG. CAD presurgical nasoalveolar molding effects on the maxillary morphology in infants with UCLP. Oral Surg Oral Med Oral Pathol Oral Radiol. 2013;116(4):418–426.
70.
ZuhaibMBonanthayaKParmarRShettyPNSharmaP. Presurgical nasoalveolar moulding in unilateral cleft lip and palate. Indian J Plast Surg. 2016;49(1):42–52.
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