Infant Orthopedics in Cleft Lip and Palate have No Effect on Maxillary Arch Dimensions in Children Younger than Age 6

Infant Orthopedics in Cleft Lip and Palate have No Effect on Maxillary Arch Dimensions in Children Younger than Age 6

DIAGNOSIS/TREATMENT/PROGNOSIS ARTICLE ANALYSIS & EVALUATION ARTICLE TITLE AND BIBLIOGRAPHIC INFORMATION Infant orthopedics has no effect on maxillary...

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DIAGNOSIS/TREATMENT/PROGNOSIS

ARTICLE ANALYSIS & EVALUATION ARTICLE TITLE AND BIBLIOGRAPHIC INFORMATION Infant orthopedics has no effect on maxillary arch dimensions in the deciduous dentition of children with complete unilateral cleft lip and palate (Dutchcleft). Bongaarts CAM, van ’t Hof MA, PrahlAndersen B, Dirks IV, Kuijpers-Jagtman AM. Cleft Palate Craniofac J 2006;43:665-72.

REVIEWER Deborah A. Redford-Badwal, DDS, PhD

PURPOSE/QUESTION Are there any differences at ages 4 and 6 years on maxillary arch dimensions in the deciduous dentition in children with unilateral cleft lip, alveolus, and palate who receive infant orthopedics before surgical closure of the soft palate versus those who do not receive orthopedics?

SOURCE OF FUNDING The source of funding was not directly stated, rather it was reported that it was part of a Dutch intercenter study carried out in a collaboration among the Cleft Palate Centers of the University of Nijmegen, Academic Center of Dentistry in Amsterdam, and University Medical Center Rotterdam in Rotterdam

TYPE OF STUDY/DESIGN Randomized controlled trial

LEVEL OF EVIDENCE Level 1: Good-quality, patientoriented evidence

STRENGTH OF RECOMMENDATION GRADE Not applicable

J Evid Base Dent Pract 2011;11:38-40 1532-3382/$36.00 Ó 2011 Elsevier Inc. All rights reserved. doi:10.1016/j.jebdp.2010.11.002

Infant Orthopedics in Cleft Lip and Palate have No Effect on Maxillary Arch Dimensions in Children Younger than Age 6 SUMMARY Subjects The children were all recruited from 1 of 3 academic cleft palate centers in the Netherlands from January 1993 to June 1996. The parents were all Caucasian and fluent in the Dutch language and their children were required to be enrolled in the trial within 2 weeks of birth. The patients all had complete unilateral cleft of the lip, alveolus, and soft palate but no other congenital anomalies (except for syndactyly). Once the parents agreed to participate in the study, a computerized balanced allocation method was used to assign patients to the groups. Patients were allocated based on birth weight (less than 3300 grams, or 3300 grams or greater) and alveolar cleft width (less than 8 mm, between 8 and 12 mm, or greater than 12 mm). Initial subject recruitment was 54 infants (41 boys and 13 girls, 27 in each treatment group), but a few were eliminated for various reasons throughout the study, including misdiagnosis and lack of required study material. The subjects were examined for 6 years, with impressions taken at 4 and 6 years of age and subsequent fabrication of plaster casts, which were used for the analysis. Overall, 45 children participated in the study and among those who received the infant orthopedic therapy (IOþ), 23 were evaluated at age 4 years and 22 were evaluated at age 6 years. Among those not receiving infant orthopedic therapy (IO–), 22 were evaluated at age 4 years and 23 were evaluated at age 6 years.

Key Exposure/Study Factor The primary difference examined in this study was the use of passive molding plates (infant orthopedic device) before surgical closure of the soft palate at 52 weeks on one group of randomized infants with a complete unilateral cleft lip, alveolus, and palate (IOþ) compared with a group not receiving the therapy (IO–) to determine the effect the intervention on future maxillary arch dimensions in the primary dentition (up to age 6).

Main Outcome Measures Digitized measurements of dental casts made from maxillary impressions taken at ages 4 and 6 from subjects of both groups (IOþ and IO–) and maxillary arch dimensions were assessed by using arch width, arch depth, arch length, arch form, and vertical position of the lesser segment. Segment collapse and contact were also determined.

Main Results Maxillary arch dimensions were not different whether the infants received infant orthopedic therapy or not (IOþ or IO–) in almost all of the parameters measured (95% confidence interval). The only 2 parameters that were found to have any significant difference were the mean arch depth measurement (I-TT’ OI–x = 30.14 6 2.62 versus OIþx = 32.81 6 1.98, P > .001) and the mean angle M-T-C(5) (OI–x = 40.55 6 3.05 versus OIþx = 43.37 6 3.85; P > .50) being slightly but statistically significant

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larger in the IOþ group at 4 years. These differences were not present at 6 years of age.

Conclusion Infant orthopedic therapy had no measurable effects on maxillary arch dimensions or on scores measuring the collapse or contact of the lesser segments in the primary dentition (ages 4 and 6).

COMMENTARY AND ANALYSIS This article is part of a larger collaborative study on a long-term project known as the Dutchcleft that examined the effects of infants treated with passive molding plates (IOþ) compared with those not receiving the therapy (IO–). Other publications from this study have commented on infant maxillary arch dimensions;1 maternal satisfaction;2 feeding;3 nasolabial appearance;4 occlusion;5 and facial growth.6 Although the total number of subjects in each group (IOþ versus IO–) was not that large (22 or 23), they were able to follow these children for multiple years, and it remains one of the largest randomized clinical trials investigating cleft lip and palate.7 The authors have concluded that the within the context of their study, IO therapy has no positive effects except for a small (but reportedly significant) improvement in speech development.8 The authors have expressed their outcomes in terms of financial and possibly emotional costs of the treatment when compared with the relatively little gain that they found with the IO. The research question that the authors were hoping to conclusively answer was whether IO therapy has value. One of the major problems underlying this question is the existence of different infant orthopedic therapies. It is difficult to extrapolate the results of this particular appliance to all other therapies, some of which include active treatment. Prospective examination of all the varying methods is difficult because different cleft treatment teams are often unable or unwilling to examine various outcome measures of the IO. Generally, presurgical IO therapies have claimed to help improve arch form and facilitate surgical closure with subsequent esthetic outcome improvement, ease of feeding, and improvement of speech.8-10 A more recent appliance used is called the nasoalveolar molding appliance (NAM). Advocates of the NAM claim that there is improvement in nasal symmetry and lip esthetics while elongating the columella and correcting the nasal cartilage.11-14 In this article, Bongaarts et al have carefully constructed and reported a study that has attempted to reduce previous errors in the collection of data by using a prospective randomized clinical design where infants with complete unilateral clefts of the lip, alveolus, and palate were placed into the treatment or control group by computerized allocation based on birth weight and cleft size. They closely followed these children for at least Volume 11, Number 1

6 years and examined many parameters in their attempt to be able to definitively state that IO therapy had no advantages over no treatment. The authors used duplicate casts and duplicate examiners blinded to IO group status to ensure accuracy. When interexaminer reliability was not considered acceptable, they excluded most of those data point measurements (vertical direction), except where they wanted to make direct comparisons to measurements previously reported in their 2001 article on maxillary arch growth in infants.1 There is some clinical data to support this study’s findings, specifically that there is little gained by the use of the infant orthopedics, but clear clinical comparisons have not been completely replicated. This article offers the major long-term results of this type of appliance therapy, corroborated with other clinical trials. Uzul and Alparslan7 have recently reported on a systematic review of long-term effects of presurgical infant orthopedics in patients with cleft lip and palate, with the finding of only 12 clinical outcomes of IO fulfilling their criteria of 4 to 6 years. One controlled study used the NAM;15 active plates were evaluated in 2 articles,16,17 and passive plates were reported in 8 studies, one of which was this article.2-6,18-20 In addition, long-term randomized controlled trial projects that also evaluate all outcomes of treatment using active plates, NAM, and Latham appliances (another presurgical appliance) should be evaluated on all types of clefts (unilateral and bilateral complete lips, alveolus, palates, as well as cleft types such as incomplete lips or complete lip and alveolus without cleft palates). These studies will also need to be followed throughout the growth of the face, requiring long follow-up, so difficulties in subject participation continuity will need to be overcome. Before different cleft treatment teams can be convinced to change their method of approaching the multiplicity of issues arising in children born with clefts, clear evidence has to be available that would support the use or lack of use of these infant orthopedic appliances. These data are not available to date, but more studies with the thoroughness that the Dutchcleft study used will help promote further agreement among experts. If IO therapies are indeed unnecessary and are an expense that adds little benefit to the child, the standard of care for the treatment of children born with clefts could be adjusted to include this information.

REFERENCES 1. Prahl C, Kuijpers-Jagtman AM, van’t Hof MA, Prahl-Andersen B. A randomized prospective clinical trial into the effect of infant orthopedics on maxillary arch dimensions in unilateral cleft lip and palate (Dutchcleft). Eur J Oral Sci 2001;109:297-305. 2. Prahl C, Prahl-Andersen B, Van’t Hof MA, Kuijpers-Jagtman AM. Presurgical orthopedics and satisfaction in motherhood: a randomized clinical trial (Dutchcleft). Cleft Palate Craniofac J 2008;45:284-8. 3. Prahl C, Kuijpers-Jagtman AM, Van ’t Hof MA, Prahl-Andersen B. Infant orthopedics in UCLP: effect on feeding, weight, and length:

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a randomized clinical trial (Dutchcleft). Cleft Palate Craniofac J 2005;42:171-7. Bongaarts CA, Prahl-Andersen B, Bronkhorst EM, Spauwen PH, Mulder JW, Vaandrager JM, et al. Effect of infant orthopedics on facial appearance of toddlers with complete unilateral cleft lip and palate (Dutchcleft). Cleft Palate Craniofac J 2008;45:407-12. Bongaarts CA, Kuijpers-Jagtman AM, van ’t Hof MA, PrahlAndersen B. The effect of infant orthopedics on the occlusion of the deciduous dentition in children with complete unilateral cleft lip and palate (Dutchcleft). Cleft Palate Craniofac J 2004;41:632-41. Bongaarts CA, Prahl-Andersen B, Bronkhorst EM, Prahl C, Ongkosuwito EM, Borstlap WA, et al. Infant orthopedics and facial growth in complete unilateral cleft lip and palate until 6 years of age (Dutchcleft). Cleft Palate Craniofac J 2009;46:654-63. Uzel A, Alparslan ZN. Long-term effects of presurgical infant orthopedics in patients with cleft lip and palate: a systematic review. Cleft Palate Craniofac J 2010, Sept 17. Epub ahead of print. Konst EM, Prahl C, Weersink-Braks H, De Boo T, Prahl-Andersen B, Kuijpers-Jagtman AM, et al. Cost-effectiveness of infant orthopedic treatment regarding speech in patients with complete unilateral cleft lip and palate: a randomized three-center trial in the Netherlands (Dutchcleft). Cleft Palate Craniofac J 2004;41:71-7. Berkowitz S, editor. Cleft lip and palate. 2nd ed. Berlin: Springer; 2006. Kuijpers-Jagtman AM, Ross EL Jr. The influence of surgery and orthopedic treatment on maxillofacial growth and maxillary arch development in patients treated for orofacial clefts. Cleft Palate Craniofac J 2000;37:527-39. Grayson BH, Santiago PE, Brecht LE, Cutting CB. Presurgical nasoalveolar molding in infants with cleft lip and palate. Cleft Palate Craniofac J 1999;36:486-98. Grayson BH, Cutting CB. Presurgical nasoalveolar orthopedic molding in primary correction of the nose, lip, and alveolus of infants born with unilateral and bilateral clefts. Cleft Palate Craniofac J 2001;38:193-8. Liou EJ, Subramanian M, Chen PK, Huang CS. The progressive changes of nasal symmetry and growth after nasoalveolar molding: a three-year follow-up study. Plast Reconstr Surg 2004;114:858-64.

14. Lee CT, Garfinkle JS, Warren SM, Brecht LE, Cutting CB, Grayson BH. Nasoalveolar molding improves appearance of children with bilateral cleft lip-palate. Plast Reconstr Surg 2008;122:1131-7. 15. Barillas I, Dec W, Warren SM, Cutting CB, Grayson BH. Nasoalveolar molding improves long-term nasal symmetry in complete unilateral cleft lip-cleft palate patients. Plast Reconstr Surg 2009;123:1002-6. 16. Chan TC, Hayes C, Shusterman S, Mulliken JB, Will LA. The effects of active infant orthopedics on occlusal relationships in unilateral complete cleft lip and palate. Cleft Palate Craniofac J 2003;40:511-7. 17. Masarei AG, Wade A, Mars M, Sell D. A randomized control trial investigating the effect of presurgical orthopedics on feeding in infants with cleft lip and/or palate. Cleft Palate Craniofac J 2007;44:182-93. 18. Ross RB, MacNamera MC. Effect of presurgical infant orthopedics on facial esthetics in complete bilateral cleft lip and palate. Cleft Palate Craniofac J 1994;31:68-73. 19. Konst EM, Rietveld T, Peters HFM, Kuijpers-Jagtman AM. Language skills of young children with unilateral cleft lip and palate following infant orthopedics: a randomized clinical trial. Cleft Palate Craniofac J 2003;40:356-62. 20. Bongaarts CA, van ’t Hof MA, Prahl-Andersen B, Dirks IV, KuijpersJagtman AM. Infant orthopedics has no effect on maxillary arch dimensions in the deciduous dentition of children with complete unilateral cleft lip and palate (Dutchcleft). Cleft Palate Craniofac J 2006;43:665-72.

REVIEWER Deborah A. Redford-Badwal, DDS, PhD Associate Professor Division of Pediatric Dentistry MC1610 University of Connecticut Health Center 263 Farmington Avenue Farmington CT 06030-1610 [email protected]

March 2011