chapter 1 [contents]

In children born with a cleft lip and palate infant orthopedics (IO) is performed during infancy mainly in order to facilitate feeding, to reduce the cleft width and to normalize and maintain the shape of the upper dental arch, pre and post surgery. This treatment modality has been part of the unilateral cleft lip and palate (UCLP) treatment protocol in almost half of the European cleft lip and palate centers.1,2 However, since the introduction by Mc Neil2-5 half a century ago, proponents and opponents have been discussing its’ effectiveness. The main reason for this long lasting controversy was the lack of scientific evidence to support the indication and use of IO. This ongoing discussion led to the great debate on the effectiveness of IO at the 47th meeting of the American cleft palate association in St Louis.6 The debate as well as the impossibility to perform a systematic review or a meta-analysis into the effectiveness of IO, due to lack of evidence based information, marked the initiation of a comprehensive randomized controlled trial into the effects of infant orthopedics in the Netherlands, called “Dutchcleft”.

chapter 2 [contents]

Since the introduction of infant orthopedics (IO) about half a century ago, IO has been part of the comprehensive care of cleft lip and palate patients in many cleft palate centers. In Europe in the year 2000 about 54% of the 201 operational centers use infant orthopaedics.1 Infant orthopedics was developed and introduced on theoretical grounds, and it became part of the treatment protocol in many centers, although the actual effectiveness of IO had never been tested, nor were the possible adverse effects properly looked into.1-4 McNeil5-6 speculated on the possibility of molding the alveolar segments during the growth spurt in early infancy instead of puberty. His argument for IO was to actively align the segments to facilitate surgery.5 He also promoted the use of molding appliances after lip closure to control arch form and prevent collapse. Today’s main objective for IO7 has not really changed since McNeil, but more and more unsupported benefits were attributed to the treatment.

Proponents of the use of infant orthopedics in UCLP, performed with passive acrylic plates from birth up to 18 months of age, have had a major influence, especially in Europe. Gnoinski8 stated in a review that this approach allows a more normalized pattern of deglutition, prevents twisting and dorsal position of the tongue in the cleft, improves arch form and position of the alar base, facilitates surgery, and improves outcome in general. Other advantages claimed in the literature are flattening of the palatal shelves, prevention of cross bites, straightening of the nasal septum, less danger of aspiration, better speech development, better nose breathing, better middle ear conditions, and additional psychological support for parents. Because of these positive effects, less speech therapy and orthodontic and surgical treatment is assumed to be needed in the long term (see among other studies8-18).

Opponents of IO claim that infant orthopedics is a complex and expensive therapy and is not evidence based. Parents are obliged to travel frequently for treatment during the first year of the infant’s life and put up with the burden of compliance. Furthermore, maxillary growth is restricted artificially3,19 and speech is negatively influenced due to delayed surgery of the palate, inherent to IO.20,21

Most studies concerning IO are dealing with the effect on maxillary arch dimensions measured on dental casts.8,15-17,19,22-24 The results of these studies remain inconclusive due to shortcomings in the design of the studies. The most common shortcomings are retrospective study design, small sample size, lack of well described group characteristics like gender and type of cleft, lack of a (randomized) control group of UCLP children without IO, and lack of a proper description of the treatment itself. Also frequently no clear outcome measures are given, confounding variables are not taken into account, and competence of the professionals, who perform the treatment, is unclear.25-27

To investigate the effect of IO in children with a complete UCLP, a prospective randomized trial started in 1993 in three academic Cleft Palate Centers in the Netherlands, i.e. the Cleft Palate Centers of Nijmegen, Amsterdam and Rotterdam. The primary objective of the study was to assess general effects (feeding, general body growth and parent’s satisfaction), orthodontic and surgical effects, and effects on speech. A comprehensive cost-effectiveness analysis at 4 yr of age will be part of the study. The part of the study reported here deals with the effects of IO (= IO+) and no IO (= IO-) on maxillary arch dimensions until 18 months of age.

chapter 3 [contents]

Maxillary arch form in patients with complete unilateral cleft lip and palate (UCLP) changes substantially during in-fancy as a result of treatment and growth.1-9 The anterior alveolar arch width narrows with time, accelerated by surgical closure of the lip and palate. Unfortunately, the arch form narrows in many cases to the extent that the alveolar segments overlap each other in the transversal direction (collapse) to a greater degree than would be considered ideal ridge relationship.10 The major concern with the collapsed arch forms is the development of crossbite in the deciduous and permanent dentition.

Even though infants with complete UCLP show a certain variety of arch forms and dimensions, this group rarely shows collapse of the maxillary segments at birth. Bacher et al.11 showed that at birth the proximal end of the major segment at the cleft side was always anterior to the minor segment, no overlap of the segments occurred, and the midpoint of the premaxilla was shifted to the cleft side. Kramer et al.2 showed that patients with UCLP initially demonstrated larger anterior and posterior arch width and arch depth dimensions than the noncleft population. These differences were reversed when compared with noncleft individuals at 18 months. In general, treated patients with UCLP have a smaller maxillary arch width and higher prevalence of lateral and anterior crossbites, compared with the noncleft population. For some cleft palate centers, this is one of the objectives leading to the inclusion of infant orthopedics (IO) into their treatment protocols, to prevent the occurrence and severity of collapsed arch forms.12,13

Only a few studies have evaluated the effects of IO on maxillary arch form using passive plates on maxillary arch form.5-7,14 None, however, have compared the effects of IO on maxillary arch form in a randomized controlled trial. Mazaheri et al.10 qualitatively studied the change of arch form in patients with UCLP in an observational study. It was their view that good arch form could be achieved without additional therapeutic interventions like IO. They found that at the age of 1 month, 50% of the children presented overlap (collapse) of the alveolar segments. This percentage increased with time to reach 80% at the age of 18 months and decreased to 50% at the age of 4 years. Supporters of IO,  5-8,12,13,15,16 using passive acrylic plates, claimed that the treatment molds the alveolar segments into a better arch form and prevents collapse, improving dentomaxillary development. However, little research has been conducted to ascertain whether this holds true or to elucidate what the desired arch form should be at different stages or ages to achieve optimal results in later life. It is generally assumed that a noncollapsed arch form during infancy is a reasonable starting point for normalized dental and skeletal development.

To investigate the effect of IO in children with a complete UCLP, a prospective randomized trial was initiated in 1993 in three academic cleft palate centers in the Netherlands (i.e., the cleft palate centers of Nijmegen, Amsterdam, and Rotterdam). The primary objective of the study was to assess general effects (feeding, general body growth, and parents’ satisfaction), orthodontic and surgical effects, and effects on speech. A comprehensive cost-effectiveness analysis at 4 years of age was part of the study. The portion of the larger study reported here deals with the effects of IO (IO+) versus no IO treatment (IO-) on maxillary arch form and the position of the maxillary segments in children with UCLP until 18 months of age.

The hypotheses to be tested were whether the first contact between the maxillary alveolar segments is reached earlier and the frequency of contact is higher, when IO is used; whether IO prevents collapse of the maxillary alveolar segments; and, in case of collapse, whether severity of the collapse is reduced by IO.

chapter 4 [contents]

Infants with unilateral cleft lip and palate (UCLP) experience feeding difficulties due to the defect. Stabilization of the nipple is difficult, and infants with a UCLP are unable to generate or maintain negative pressure because air flows through the cleft, decompressing the negative pressure in the oral cavity.1,2 The only way they can extract milk from the nipple is by mechanical movements of the lips, tongue, and mandible that push the nipple against the cleft palate. This often results in prolonged feeding times or inadequate food intake, as well as increased risk for backflow through the nose (nasal regurgitation), choking, aspiration, and vomiting. Moreover, when feeding is unsuccessful, both parent and child become frustrated, so it is crucial that good feeding is acquired as soon as possible.3

A general feeding solution in UCLP is to deliver milk directly into the mouth1 and to educate parents about feeding techniques, bottles, nipples, formula volume goals, and use of energy-dense additives.3 The most common instructions are to use a bigger cross-carved hole in the nipple, in combination with a squeeze bottle.4 It is also advised to hold the child in a more upright position.3

Infant orthopedics (IO) is thought to have a favorable effect on feeding and to reduce feeding problems.3,5-7 Little sound evidence is available about the effects of IO on the feeding process. During IO, the cleft is covered artificially and the oral environment is normalized. This is thought to result in more effective feeding movements. However, Choi et al.2 found that, even in the presence of an orthopedic plate, negative pressure generation was not possible with UCLP. Turner et al.8 found an increase in flow-rate with bottle feeding combined with a feeding plate and lactation education. Although not to an adequate level, breastfeeding has been managed in some cases using a specially designed appliance.9 Bokhout10 found that the presence of cariogenic lactobacilli were associated with the use of IO. This early colonization may imply a high risk for dental caries in the primary dentition and should also be taken into account when IO is used.

Growth of infants with cleft lip and palate is generally found to lag behind during infancy.11-17 However, authors who have reported on long(er) term results found catch-up gains in weight and/or height.11,13,17 The factors most frequently mentioned regarding growth inhibition include feeding problems, recurrent upper airway and ear infections, and surgical interventions. Felix-Schollaart18 and Felix-Schollaart et al.19 were the first to record the occurrence of feeding difficulties, as well as their influence on length and weight, among other variables. They reported a very low frequency of feeding difficulties, but that the explained variance (14%) of having feeding difficulties at a relatively late age (12 to 18 months) influenced weight and length negatively. Head circumference was not significantly influenced by feeding difficulties.

Available information in the literature on the effects of IO on feeding and, subsequently, on weight and length seemed to be inadequate at the time of the preparation of this trial. Therefore, feeding variables and the anthropometric variables recommended by the World Health Organization,20 weight, and length were measured in Dutchcleft. In this prospective randomized clinical trial, the effects of IO in infants with a complete UCLP were evaluated. The trial started in 1993, in cooperation with the academic cleft palate centers of Nijmegen, Amsterdam, and Rotterdam, all in the Netherlands.21 The primary objective of the study was to assess general effects (feeding, infant growth, and parent’s satisfaction), orthodontic and surgical effects, speech, and cost-effectiveness. The present paper reports on the effects of IO on bottle feeding during the first 24 weeks and on weight and length during the first year. The aims were to test whether IO increases the feeding velocity (mL/min), the amount of food intake per feeding, and the indices of weight-for-length (g/cm) and length-for-age (cm).

Table 1       Mean ages in weeks ± standard deviation (SD) at which questionnaires were filled in. N IO- and N IO+ are the number of returned questionnaires at a certain stage.

* IO- = no passive maxillary plate; IO+ = passive maxillary plate.

Table 2       Background variables for both groups IO- and IO+; Some skewed variables are presented as percentiles.

* IO- = no passive maxillary plate; IO+ = passive maxillary plate.

chapter 5 [contents]

The facial appearance of an infant born with a complete unilateral cleft lip and palate (UCLP) changes with time and is at each moment in time the result of intrinsic, functional, and extrinsic factors.1,2 At birth, the cleft lip usually is widely flared open and the maxillary segments are separated with a mean of approximately 12 mm at the level of the alveolar cleft.3 For the infant, the main problem is feeding. However, the initial facial impairment also will have its subsequent effects on parents, family, and later, social network. Fortunately the facial appearance of infants with cleft lip and palate (CLP) does not appear to affect the early mother-infant relationship adversely.4,5 Later, a normalized facial appearance in combination with good intelligibility6 generally is preferred by peers and is of great importance when socializing.7,8,9

As the child grows older, several interventions (i.e., surgery, orthodontics, speech therapy) will have occurred to normalize function and facial appearance. In almost half of the European cleft palate teams, infant orthopedics (IO) is part of the treatment protocol.10 IO was introduced by McNeil.11,12 One of the arguments for the use of IO was to improve the presurgical alignment of the maxillary segments to facilitate lip/nose surgery. It was assumed by proponents of IO with a passive maxillary plate, as used in this study, that the cleft width would be reduced significantly and relevantly. The approximation of the alveolar segments and the cleft lip were assumed to result in reduced tension on the repaired lip and in a better treatment outcome.13,14 However, this claim has never been established.14

The purpose of this study was to evaluate the effect of IO, using passive maxillary plates, on facial appearance at 18 months of age. The included infants were born with a complete UCLP. Two treatment groups were evaluated; one group received IO treatment during the first year of life, the other group did not. The material was collected as part of a clinical trial into the effects of IO, executed in cooperation with the academic cleft palate centers of Nijmegen, Amsterdam, and Rotterdam, in the Netherlands.3,15,16 The main objectives of the trial were to assess general effects (i.e., feeding, nutritional status, maxillary growth, facial appearance, and parent’s satisfaction), orthodontic and surgical effects, speech, and cost-effectiveness.

chapter 6 [contents]

In children born with cleft lip and palate (CLP) the quality of life (QoL) is already influenced at birth, by the impact of the conditions on the infant, mother and family. Not only does the birth of a child with CLP raise controversial emotions in the parents especially in the mother.1 But the parents also have to adjust to the loss of the anticipated perfect child.2 The contradictory feelings, clashing interests between the parents and others and the conflicting ideas about what is best for the child illustrate the complexity of the relationship between parents and child.1 On top of this the infant is bound to have reconstructive surgery of the cleft lip/nose and soft palate in the first year of life, and in half of the European CLP teams, infants also receive infant orthopedics (IO).3 This latter type of early treatment with palatal appliances was introduced about 50 years ago in order to narrow the cleft presurgically. Since its introduction different type of appliances with different objectives have been introduced of which many are still in use.4,5 Opponents claim that that there is insufficient scientific data to support the use of IO, that IO is expensive and not cost effective. Claimed advantages of IO in the literature are amongst others: facilitation of lip surgery due to the narrowed cleft, a more normalized tongue tip function; less feeding problems; restoring the symmetry of the maxilla and nose; straightening of the nasal septum; better speech development; minimization of the severity of skeletal and dental deformities, thus less orthodontics and surgery later on and thus cost-effective; psychological support for parents and psychological advantages for the child due to better child-parent interaction.5 A positive treatment experience with IO during the early phases of care could affect the mother-child relationship positively. Psychological advantages and support for the mother and child due to a more frequent child-parent-team interaction as well as being able to participate actively in treatment as a parent due to IO have been claimed and discussed.6-9 The claimed psychological advantage of IO would certainly be welcome since parents of infants with CLP are reported to have higher levels of parental stress in infancy and toddler hood.10 In addition Speltz et al.11 found that mothers of CLP reported less favorable social support than mothers of children with less visible malformations. Perceived family support was according to Bradbury2 the only significant variable related to parental adjustment. On the other hand most children with CLP emerge from the first 2 years of life with secure maternal attachment.12

So far all evidence on “psychological effects of IO” has emerged from expert opinion but was never evaluated scientifically. Therefore, in this study, the satisfaction in motherhood in relation to the care of their child with or without IO was studied in a randomized controlled trial design.

chapter 7 [contents]

This thesis is part of an ongoing randomized clinical trial (RCT) on the effects of infant orthopedics (IO) in UCLP of which the results regarding maxillary growth and development, feeding and nutritional status, esthetics, and satisfaction in motherhood, from birth until 1.5 yr of age are presented and discussed. The continuous discussion between proponents en opponents of IO, the lack of scientific evidence for the use of IO as well as the growing notion by the orthodontists of the participating CLP-teams that the contribution of IO might be irrelevant and therefore not costeffective marked the onset of this RCT. The outcome was measured comprehensively and the use of reproducible and valid outcome measures was strived for.