Department of Orthodontics, Süleyman Demirel University, Isparta, Turkey
Received Date: October 24, 2014; Accepted Date: April 29, 2015; Published Date: May 05, 2015
Citation: Esenlik E. Presurgical Infant Orthopedics for Cleft Lip and Palate: A Review. Journal of Surgery [Jurnalul de chirurgie] 2015; 11(1): 9-14. doi:10.7438/1584-9341-11-1-2
Copyright: © 2015 Esenlik E. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
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Cleft lip and palate deformities are some of the most common facial and oral anomalies. Severe cleft forms are associated with severe nasolabial deformities, and present a significant surgical challenge in order to achieve a functional and aesthetic outcome. Presurgical infant orthopedics has been used in the treatment of cleft lip and palate for some centuries. Starting with the McNeil method, several methods and modifications had been developed by different clinicians over time. However, there is no consensus in the literature on infant orthopedic methods and their benefits. Therefore, the aim of this review is to discuss presurgical infant orthopedic methods and their advantages and disadvantages. Presurgical orthopedics allows not only the alignment of cleft segments, but also molding alar cartilages and nose tip. In addition, this procedure allows performing primary alveolar grafting or gingivoperiosteoplasty to establish a union bone at the cleft side as well. However, there have been some studies reporting that there was no positive effect of presurgical orthopedics on the maxilla and maxillary arch. There is still no consensus in the literature on the best protocol for orthopedic and surgery methods for the treatment of cleft lip and palate in infants.
Cleft lip and palate; Presurgical infant orthopedics;Nasoalveolar molding
Clefts of the lip and palate are some of the most common facialand oral anomalies. These anomalies can appear with considerablevariation in form and severity . The maxillary structure of the cleft lipand palate is divided in two or three segments by the cleft of the palateand alveolus. A unilateral cleft defect is characterized by a wide nostrilbase and separated lip segments on the cleft side. Severe cleft forms areassociated with severe nasolabial deformities, and present a significantsurgical challenge in order to achieve functional and aesthetic outcomes. The affected lower lateral nqsal cartilage is displaced laterally andinferiorly, resulting in a depressed dome, increased alar rim, obliquecolumella, and overhanging nostril apex . When associated withcleft palate, the nasal septum deviates to the non-cleft side, with anassociated shift of the nasal base . The bilateral cleft lip and palatemay be symmetrical or asymmetrical, depending on the equality ofinvolvement on both sides. In these patients, both nasal chambers arein direct communication with the oral cavity, and the turbinates areclearly visible within both nasal cavities. The premaxilla may be smallor large, and projects considerably forward from the facial aspect of themaxilla .
Cleft lip and/or palate patients have feeding, functioning, aesthetic,speech, and psychological problems; and therefore, are best managedthrough a team of experts . Management of cleft lip and/or cleftpalate is a process that starts in infancy and continues in adulthood.These patients undergo many surgical procedures throughout life.Numerous methods and treatment strategies have been developed overthe years to reduce the number of surgeries. Despite the fact that therehave been many advances in surgery, certain orthopedic correctionsprior to the primary surgery are still required in patients with cleftlip and palate. For this purpose, presurgical infant orthopedics issuggested for achieving better surgical outcomes. Since it is widelyaccepted the intervention of multidisciplinary teams for treating cleftpatients, various methods have been developed for presurgical infantorthopedics. The aim of this review is to summarize the current stateof knowledge of the effects of presurgical infant orthopedics (PSIO)on long-term outcomes of different treatment protocols. In thisreview, the advantages, disadvantages, effects on maxillary growth, anddentoalveolar arches, as well as in speech and complications of some presurgical infant orthopedics methods will be discussed. A PubMedsearch was performed using the terms PSIO, presurgical nasoalveolarmoulding and its long-term results and related articles were selectedfor the review. In addition, limitations of these studies will be discussed.
Some presurgical implementations in infants were developed afew centuries ago. Facial binding or adhesive tape strapping was usedcenturies ago to narrow clefts before surgery . The use of a bonnetand strapping to stabilize the premaxilla after surgical retraction hasalso been reported . Head is still used today to retract the premaxilla, and T traction using an external device has been reported as usefulfor surgical procedures in short-term . All procedures, which at thetime were mainly performed by orthodontists or by the surgeons, werebased on the ever proven assumption that a narrow and well aligned cleftwould be easier to repair, with less undermining and less mobilizationof soft tissues. A narrower cleft would also require less tension in therepaired lip, and thus, the aesthetic outcome, facilitation of feeding,and speech was improved [11-15]. Presurgical infant orthopedics aimsat securing a good maxillary arch form in an acceptable relation withthe mandible, and at restoring normal oral function . It also correctsangulation of the palatal shelves to a more horizontal position . It isgenerally accepted that modern presurgical infant orthopedics startedwith the McNeil technique . Since McNeil, many researchershave published their own methods for obtaining proper growth anddevelopment of the face, and for improving surgery results. The Hotzappliance, and then the Latham device were introduced for aligningthe cleft segments [18-23]. Some authors used a combination ofthese appliances like in the Brogan technique, which combines the McNeil technique and the Hotz plate . Later, Grayson and Cuttingdescribed the “presurgical nasoalveolar molding (PNAM)” concept formolding not only cleft segments but also nasal appearance . Certaininvestigations reporting short- and long-term results of these methodsmentioned above are summarized below.
McNeil Method: McNeil was the first in aligning presurgicallythe alveolar parts in cleft lip and palate patients . He suggestedthe use of serial appliances to approximate cleft alveolar segments. Bymolding the palatal segments into the correct position using a seriesof acrylic plates, McNeil believed that this would produce a normalmaxilla, while reducing the alveolar and palatal cleft at the same time.McNeil and Burston claimed that soft tissues overlying the hard palatewere stimulated to grow, and they also added that neonatal maxillaryorthopedics could control and modify the postnatal development ofthe maxilla . In addition, the use of a series of acrylic plates may befavorable for patients who have to travel long distances and are unableto visit the orthodontics clinic weekly. Another advantage may beforeseeing the final position of the alignment arches.
Latham device: The other appliance used for aligning cleft segmentsis the Latham device, which was introduced by Dr. Latham [25-27]. Inthis approach, forces are applied using a pinned palatal appliance inorder to manipulate mechanically the maxillary segments into closeapproximation, which is followed by alveoloperiosteoplasty and lipadhesion. According to Drs. Latham and Millard, these alignmentsallow the performance of gingivoperiosteoplasty (GPP), providingstabilization of the maxillary segments and reconstruction of the nasalfloor . Bercowitz et al. reported a longitudinal study in unilateraland bilateral cleft lip and palate treated with the Latham device. Theyalso performed periosteoplasty in all cases, and compared the results topatients treated with a non-orthopedics procedure without GPP, andtreated just with a lip adhesion method. They found a higher frequencyof anterior and posterior crossbite in the presurgical orthopedicsgroup . Some authors have commented that those findings mightbe the result of the periosteoplasty procedure . Dr. Latham appliedthe Latham device with less extensive surgery in cases of bilateral cleftlip and palate, and he assessed their dental occlusion and lateral headradiographs at 5 years of age. He found greater values for cephalometricmeasures in maxillary length, maxillary prominence, and ANB anglecompared to previous cases .
In another long-term study, more anterior open bites and posteriorcrossbites were found in unilateral and bilateral cases compared to nonorthopedics and periosteoplasty group . In the study of Chan et al.,in which they evaluated active appliances longitudinally, dental modelsof patients with unilateral cleft lip and palate (UCLP) were assessedusing the Goslon Yardstick. GPP and lip adhesion were performed bothin non-orthopedic and in orthopedic groups. No significant differenceswere found in Goslon scores between the two groups. The authorsconcluded that Latham procedures did not affect dental arch relationsin preadolescent children with UCLP . Similarly, Allareaddy et al.stated that, outcomes are predictable without any major adverse eventsor complications by using Latham device . Besides, it was statedthat Latham device could be useful in unusual cleft cases .
Hotz appliance: The Hotz appliance, also known as the Zurichapproach, in which arch alignment is achieved by grinding away theacrylic in specific areas, was introduced after the McNeil technique.Although there is no strict research about the outcomes of the Hotzplate, it was stated that this method had a tremendous impact oncleft patients . According to Hotz and Gnoinski, the primary aimof presurgical orthopedics is not to facilitate surgery or to stimulategrowth, as postulated by McNeil, but to take advantage of intrinsic developmental potentials. Therefore, the Zurich approach, aftera lip operation is performed at the age of 6 months, palate repair ispostponed until 5 years of age . These authors concluded thatorthopedic guidance combined with optimal timing of surgery hasbeneficial effects. In a study investigating the short-term effects of theHotz plate, a harmonization in the vertical and transverse positions ofthe segments was found in the plate group compared to the controlgroup . In another study with 4 years follow-up of the Hotz plategroup, the width of the palate was larger in the Hotz plate group than inthe control group, but no difference was observed in the anteroposteriordistance of the palate between the groups . In addition, Sasaguri etal., investigated the long-term effects of the use of the Hotz plate and lipadhesion. They found that arch width and length of the anterior part ofthe maxillary were larger in the Hotz (+) group than in the Hotz and lipadhesion group, and in the group without Hotz plate and palatoplasty,at 5 years of age. The anterior part of the maxillary arch was wider inthe Hotz group than in the other two groups . In another longterm study, Silvera et al. concluded that The two-stage palatoplasty incombination with application of the Hotz' plate had good effects on themaxillary growth than one stage palatoplasty without Hotz plate up tothe age of 12 years .
Nasoalveolar Molding Appliance: In 1993 Grayson et al.introduced the PNAM concept, which continues to play significantrole in neonatal cleft lip and/or palate treatment . This approachis preferred by certain orthodontists because it produces improvedresults, and allows repositioning of the maxillary alveolus andsurrounding soft tissues. Grayson and colleagues have reportedmany studies about PNAM treatment and they suggested the use ofthis appliance for improving nasal appearance, which results in lesssecondary nasal surgeries. This procedure also minimizes the need forlater alveolar bone grafting, allows GPP, as well as effective retractionof the protruded premaxilla, and lengthening of the deficient columella[37,38]. In addition, produces limited maxillary growth disturbance. PNAM has become very popular among orthodontists becauseof its nasal molding effect . It is also suggested to correct septaldeviation in early ages without surgery, since nasal cartilages are ableto mold easily in the first postnatal 2 months because maternal estrogenprovides the molding for the nasal cartilages.
In the PNAM approach the orthodontist adjusts the applianceevery 1–2 weeks in 1 mm increments by removing hard acrylic resin,and adding soft acrylic resin. Once the maxillary alveolar segment gap isless than 6 mm, a nasal stent can be added to the appliance using acrylicresin placed on 0.036 inch-thick wire. The stent is positioned 3–4 mminto the nostril just below the soft tissue triangle of the nose. The sizeand shape of the stent is adjusted by adding soft acrylic to help create a“tissue expander” effect on the length of the cleft-side columella, as wellas to reposition the malpositioned lower lateral cartilage. This processcan take several months and results in a delay of the definitive cleft liprepair until approximately 4–5 months of age. PNAM should ideallybegin before 6 weeks of age to take advantage of the early plasticity ofnasal cartilages .
The first goal of PNAM in bilateral cases is to move the premaxillarysegment posteriorly and medially, while preparing the lateral alveolarclefts to come in contact with the premaxilla . The posteriorlateral palatal shelves are molded to the appropriate width to acceptthe premaxilla. The premaxilla is retracted and derotated as necessaryusing the molding plate in conjunction with external tape and elastics.In addition, another important point is the elongation of the columella. Cutting et al. stated that a saddle should be placed at the lipand columella junction. This saddle produces a separation betweenlip and columella that is expanded along an anterior vector, whilethe prolabium is stretched downwards using tape. Several monthsof appliance adjustments are often required. They also reported thatmuch of the nasal tip shape produced through presurgical molding was lost within a few weeks because of the fibroadipose tissue depositedbetween the widely separated nasal domes. Therefore, they suggestedremoving the fibro-fat tissue from between the nasal domes of the lowerlateral cartilages, and suture them together in the midline without anexternal incision.
Grayson pointed out that multiple nasal surgical revisions areoften indicated to approximate the nasal symmetry, because surgicaltechniques for managing nasal deformity are lacking. He also pointedout that in bilateral cleft lip and palate (BCLP), the deficient columellaand ectopic premaxilla are the primary reconstructive challenges.Multiple nasal surgeries are required, which often result in excessivescarring at the columella-prolabial junction, and lack of nasal projection. Therefore, he emphasized the importance of nasal molding beforesurgery in the early neonatal period. In addition, some other appliancesfor nasal molding have been reported . However, the permanenceof the improvement in nasal symmetry and appearance using PNAMremains controversial; however, there is a trend towards a positive effect.Liou et al. reported that nasal asymmetry was significantly improvedafter nasoalveolar molding in infants with cleft lip and palate; but afterthe primary closure of the cleft lip and nose, there was a significantrelapse of the nasal asymmetry in the first year postsurgery, whichremained stable afterwards. This relapse was the result of a significantdifferential growth between cleft and noncleft sides in the first yearpostsurgery . Similarly, Pai et al., who used the nasoalveolarmolding (NAM) appliance in their study, concluded that there wassome relapse of nostril shape in width (10%), height (20%), and angleof columella (4.7%) at 1 year of age, compared to their presurgicalstatus . Therefore, the use of a nasal stent has been suggested afterprimary lip closure, at least for 6 months. Nonetheless, in a long-termstudy, it was found that the change in nasal shape is stable until earlychildhood, and it was emphasized that the symmetry in nose shape wasmaintained . In another longitudinal study with 8 year follow-up,the rate of residual fistula was assessed, and it was found that NAMin conjunction with nasal floor closure contributes to a low incidenceof oronasal fistulae . Another longitudinal study investigating theeffects of NAM approach on further surgery requirements reportedthat NAM-prepared patients were more likely to have less severe clefts,present the best surgical outcomes, and need less revision surgeriescompared to patients not prepared with NAM .
The average age at the time of appliance is another concern. Graysonand Maull focused on starting neonatal period (within first one month)for better nasal esthetics results, while in the Latham technique, startedwithin 8-11 weeks . However, Shetty et al., evaluate the effects ofnasoalveolar moulding (NAM) in complete unilateral cleft lip andpalate infants presenting for treatment at different ages. Study groupscomprised: group I treated with NAM within 1 month of age; groupII treated with NAM between 1 and 5 months of age. This studyconcluded that the effects of NAM were most significant in groupI. Group II patients also benefited from NAM, although to a lesserextent. This study validates the use of NAM in infants presenting latefor treatment .
Since cleft lip and palate treatment requires multidisplinaryapproaches, outcomes may be affected at any stage of treatment.Therefore, the pure effect of PSIO appliances is very difficult to assessbecause of the variety in timing and sequence of treatment protocolsfor both surgery and orthodontics. The major difficulty whencomparing different presurgical orthopedic methods is the type ofsurgical technique, and whether performing sequential palatal closure(one or two stage) or GPP, at what age, the experience of the surgeon[49-52]. The debate on the ideal time for hard palate closure is notover so far. Furthermore, because of the different timing for gatheringrepresentative sample sizes, and the inability to obtain untreated control groups, the comparison of the effects of different orthopedicappliances is an almost impossible task. Another limitation is thatprevious studies, except one, have not examined the severity of cleftsin infants. Peltomaki et al. found that patients with large clefts andsmall arch circumference, arch length, or both, showed less favorablemaxillary growth than those with small clefts. Therefore, furtherrandomized control studies comparing the effects of PSIO withindifferent orthopedic and surgical techniques are needed for assessinglong-term results .
Another surgical issue is completing the osseous union at the cleftregion at the time of primary lip closure, which may affect maxillarygrowth, and therefore, influence the outcome of presurgical infantorthopedics. Different methods have been used for this purpose, andsome of them, such as primary alveolar grafting, have been abandonedbecause of the detrimental effects on maxillary and facial growth [54-56]. As minimal invasive methods such as GPP provide bone union,there is a debate regarding the effect of the combination of this techniquewith presurgical infant orthopedics. Two cleft groups were comparedin a study; one group underwent presurgical alveolar molding followedby GPP at the time of lip repair, while the other group did not undergomolding and GPP. The authors investigated whether narrowing ofthe cleft parts and GPP diminished the need for bone-grafting later.The results of this study showed that all patients in the control grouprequired bone grafts, while 60% of patients treated with presurgicalorthopedics and GPP did not need a secondary alveolar bone graft inthe mixed dentition . However, although these benefits have beenstated by many authors, there is no consensus regarding the utility ofGPP or secondary alveolar bone grafting . Another issue is thatmost of the anterior growth of the maxilla takes place by the age ofsix years . Similarly, Wood et al. were unable to demonstrate anyclear impairment of maxillary growth in patients treated with GPPcompared to patients not treated with this technique .
Some studies have reported complications in soft and hard tissuesusing PNA6 therapy [59-61]. Grayson and Maull reported someproblems including soft tissue breakdown, intraoral ulcerations, andfailure to apply tapes and elastics, cooperation issues, and the eruptionof neonatal teeth during treatment. They reported that common areasof breakdown were the frenilum attachments, the anterior premaxilla,or the posterior fauces, as the molding plate is retracted. They alsoreported that the intranasal lining of the nasal tip can become inflamedif too much force was applied by the upper lobe of the nasal stent .The other most frequent problem was the development of cheek skinrashes . In the study of Lewy-Bercowsky et al., soft and hard tissuecomplications were mentioned. Contact dermatitis due to repeatedremoval of tapes, meganostril produced by improper positioning ofthe nasal stent, overactivation of the nasal stent resulting in bruises orpetechiae in the dome area were mentioned as soft tissue complications.Neonatal teeth eruption during treatment, or premature eruption ofthe incisors due to the pressure exerted by the acrylic plate, whichcreates a T-shape maxillary arch after the use of the molding plate, werereported as hard tissue complications .
In a unique study, the effects and complications of two PSIOtreatment methods were compared. The authors stated that bothGrayson and Figueroa nasoalveolar molding improved nasaldeformities, and reduced alveolar gaps in a similar manner; however,the Figueroa technique was associated with fewer oral mucosalcomplications and better efficiency .
Despite the fact that the usefulness of these methods has beenpointed out, presurgical orthopedic procedures have been stated asunnecessary in some studies of the Eurocleft project. One of these studies was performed by Kuijpers-Jagtman and Prahl Andersenanalyzing neonatal orthopedics of the Zurich approach, for over20 years. According to their longitudinal observations, neonatalorthopedics is not the best approach. They conducted a randomizedclinical trial named “Dutchcleft” in three centers, and comparedinfant orthopedics and non-orthopedics groups in relation to general,orthodontic, and cost effectiveness, as well as speech effects of theseapproaches. Regarding general effects, there was no difference betweenthe groups in weight for age, length for age, or weight for length. Whenthey assessed the maxillary arch form and dimensions, they found thatcleft gap was reduced significantly in the orthopedic group; however,no significant differences were found between the groups after lipclosure . Furthermore, Prahl et al. found that infant orthopedicsdid not prevent collapse of the maxillary arch . Therefore, in theDutchcleft study there were no observable effects on occlusion and jawrelationships at the ages of 4 and 6 years . Evaluation of speechand language development showed that at one year of age, childrenwho wore plates presented an enhanced production of alveolar sounds;however, at the age of 1.5 year, when the plate was no longer used,a limited effect on speech was observed . At 30 months of age,the phonologic development of the orthopedic group was normal ordelayed, while most children in the non-orthopedic group presentedan abnormal development . Taking into account the results of theDutchcleft trial, there is no need to perform infant orthopedics forunilateral cleft lip and palate.
Papadopoulos et al. also investigated the effectiveness of presurgicalinfant orthopedics using a systematic review . They showed thatthere were no significant differences in craniofacial and dentoalveolarchanges, indicating that PSIO treatment had no effect on cleft lip andpalate patients. The limited evidence derived from this study doesnot seem to support the short- or long-term effectiveness of PSIOin these patients. Furthermore, Van der Heijden et al. performed ameta-analysis, and inferred that the results of studies on nasoalveolarmolding were inconsistent in relation to changes in nasal symmetry,although there was a trend towards a positive effect . In a similarmanner, Uzel and Alparslan concluded in their systematic review thatpresurgical infant orthopedic appliances have no long-term positiveeffects in patients with cleft lip and palate and that more randomizedcontrolled trials are necessary. They also added that the encouragingresults on the effect of nasoalveolar molding appliances on nasalsymmetry need to be supported by future randomized controlled trials.
Presurgical infant orthopedics has been investigated in terms ofcost-effectiveness. The main principle of cost-effectiveness analysis isto estimate the cost and treatment outcome compared to an alternativetreatment. The total cost of presurgical orthopedics was higher in thetreatment group of the Dutchcleft study . In this study, the meanmedical cost for infant orthopedics treatment was US$852. The nonorthopedicstreatment group had a significantly lower mean medicalcost (US$304). Mean travel costs and indirect nonmedical costs wereUS$128 and US$231 for the orthopedics, and US$79 and US$130 forthe non-orthopedics groups, respectively. However, the additional costof neonatal maxillary orthopedics might be partly outweighed by thecosts in speech therapy in later years, as the group treated with neonatalorthopedics had a significantly better rating for speech . Based onthe results of the Dutchcleft study, the authors concluded that neonatalmaxillary orthopedics for unilateral cleft lip and palate is not necessary forfeeding, patient’s satisfaction or orthodontic reasons. Regarding speech, apositive but very limited effect was found until the age of 2.5 years.
Apparently, studies concerning presurgical orthopedics in cleft lipand palate have been heterogeneous and lacked adequate reporting. Inparticular, surgical time and sequence of surgery were stated as decisivefactors for the final success, rather than the presurgical orthopedic treatment type . It should be kept in mind that the best surgeryapproach for these patients was not described, and the outcomeswere affected not only by presurgical orthopedics, but also by surgerymethods [70-74]. There are many surgical alternatives as there aremany type of orthopedics. Future prospective longitudinal studies areneeded to achieve a consensus on the effect of presurgical orthopedics,as well as the best treatment approach.
It can be inferred from this review that presurgical orthopedicappliances are useful for aligning cleft segments, reducing softtissue tension and improving nasal aesthetics. Although in someinvestigations it was found that there were no differences between thegroups that underwent presurgical infant orthopedics and those whodid not; there is a trend towards a positive effect on nasal symmetrywith the use of the PNAM appliance. Assessments on the effects ofdifferent combinations of cleft surgery and orthopedics methods arestill needed. Therefore multidisciplinary treatment modalities are ofgreat importance for the rehabilitation of cleft patients.
Authors have no conflict of interests to disclose.