The authors performed a systematic review to evaluate the potential beneficial effects of the nasoalveolar molding appliance on nonsyndromic unilateral clefts of the lip and/or palate prior to primary lip repair.
Material and Methods
A literature search was performed using three electronic databases (PubMed, Embase, Web of Science) and three journals (“Cleft Palate-Craniofacial Journal”, “Plastic and Reconstructive Surgery Journal” and “American Journal of Orthodontics and Dentofacial Orthopaedic”) from January 1980 to April 2017. Data extraction was performed with tables treating different subjects: surgical, aesthetical, functional, socio-economical effects of nasoalveolar molding (NAM) appliances and the evolution of NAM appliances, especially three-dimensional technology.
Results
Of the 145 articles retrieved in the literature surveys, 28 were qualified for the final analysis and 20 studies were excluded because of their small sample size (less than 10 patients) and/or too long follow-up (exceeded 18 months). Four randomized controlled trials were available. Although literature allowed discussing the short-term benefits of NAM appliance and the three-dimensional technology, scientific evidence is lacking.
Conclusions
Based on the results, nasoalveolar molding appliances have positive surgical, aesthetical, functional and socio-economical effects on unilateral clefts of the lip and/or palate treatment before the primary repair surgeries. Three-dimensional technology results in a more efficient and predictable nasoalveolar molding appliance treatment. However, nasoalveolar molding appliance effect in a short term remains unclear with the available literature. Further studies that integrate three-dimensional technology in a large scale are still needed.
Clefts of the lip and palate result from a fusion failure of the left and right maxillary prominences during the 6th and 12th weeks of gestation. The resultant congenital deformity leads to malformation of the upper lip, nose and alveolar and results in functional disabilities (i.e. mastication, feeding, speaking). This affects approximately 1/700 live birth in North America and the majority of clefts are unilateral and non-syndromic [1].
Two general strategies are used to close the lip and correct the nasal asymmetry. One approach involves lip and nose repair around three months of age irrespective of the size of the alveolar gap and may be followed by secondary correction of any residual deformity sometime during childhood. The second approach utilizes presurgical orthopaedic molding early after birth for approximately three months and must be performed prior to primary repair surgeries. With the latter, orthodontists and surgeons aim to provide symmetry and elongation to the deformed nasal cartilage and reduce the severity of the cleft palate prior to the initial surgical intervention [2].
Since the 1950s, McNeil [3,4] described the first intraoral presurgical orthopaedic appliance able to stimulate the tissue growth and reduce the width of the alveolar and palatal cleft. Then, clinicians have proposed several presurgical appliances to mold the alveolar arch and reduce lip and palate cleft. First, passive appliances, such as the Hotz plate, composed of a simple plate, aim to create alveolar alignment by spontaneous development of the segments without external force. Then, in the 1980s, active orthopaedics, such as the Latham appliance, are retained by surgically installed pins and delivers controlled forces to reduce the cleft gap and align the alveolar arch. Finally, in the 1990, Grayson used Matsuo et al. [5] concept and described a third category of presurgical infant appliances, semi-active, called the nasoalveolar molding (NAM) appliance. The appliance is composed of a passive alveolar plate with one (for unilateral cleft lip and/or palate [UCLP]) or two (for bilateral cleft lip and/or palate [BCLP]) nasal stents. NAM is the first appliance able to mold presurgically the alveolar arch into a predictable preferred alignment and correct the nasal cartilage at the same time [6,7]. To achieve its goal, the appliance is introduced within the first two weeks of life in order to benefit from the maximum growth potential. It must be worn full-time until the alveolar cleft is sufficiently narrowed and the lip segments are brought together. A nasal stent extending from the intraoral plate may be added to improve nasal symmetry. Primary lip repair is then performed once the effect of NAM is deemed adequate, usually after the 18 months. This surgery is more or less invasive depending on the efficiency of the NAM.
Recent reviews have been conducted to evaluate the efficacy of the presurgical orthopaedic treatment on patients with nonsyndromic unilateral clefting of the lip and/or palate. However the heterogeneity of these studies [8-12] prevents the construction of substantial evidence of the NAM effect (Table 1). Moreover, many studies evaluate the potential beneficial effects of the NAM appliance after the primary lip repair, whereas the efficiency of the appliance itself should be assessed prior to the primary lip repair (during the first 18 months).
Assessment of the molding of the nasal cartilage [27,44], molding of the alveolar possesses [40,42].
Mixed criteria for evaluating results
Landmarks on casts [26], cephalometry, facial photographs [43,44], photographs on cast [45,46],
three-dimensional optical scanner on the cast [64], three-dimensional optical facial scanner [62,63].
Inadequate sample sizes
Small sample size [17,19], without control group [27].
Inconsistent follow-up period
Before the primary surgery (during the first 18 months), after the primary surgeries [22,23,26].
Heterogeneity of appliances
Hotz plate [41,43], Grayson NAM [40].
Variable age of the patient
When the appliance was used, when primary surgeries were performed.
Mixed level of surgeon experience [12]
NAM = nasoalveolar molding.
The aim of this study was to undertaken a systematic review to compare the surgical, aesthetic, functional and socio-economical effects before the primary lip repair between patients with nonsyndromic unilateral clefting of the lip and/or palate treated either with the NAM appliance or with other presurgical appliances or without any presurgical treatment. The objective of the study was also to highlight complications and inefficiency of the NAM appliance. The final aim of the systematic review was to introduce the evolution of the NAM appliance technique, especially ones involving three-dimensional technology that have the potential to overcome some of the inefficiencies of the current NAM appliance approach will be introduced.
MATERIAL AND METHODS
The materials and methods of the literature employed by the authors in the systematic reviews are based on comprehensive search strategies that have been discussed and standardized.
Protocol
PRISMA-P (2015) recommendations were used to methodically build this review [13].
Focus questions
The four PICO elements (population, intervention, comparison and outcome) were employed to construct the systematic reviews. The focus questions:
What are the outcomes of NAM prior to primary repair surgeries on the UCLP compare to the outcomes without NAM or with others appliances?
What are the applications of three-dimensional technology in NAM approach nowadays?
Search strategy
An electronic search of three databases (PubMed, Medline, Web of Science) was performed and included articles from January 1980 to April 2017. The research terms used to identify articles discussing NAM appliance were: MeSH terms “cleft palate” OR “cleft lip” AND non-MeSH terms: “nasoalveolar molding, “presurgical nasoalveolar molding”, “presurgical orthopaedics”, “presurgical orthopaedic appliance”. Beside manual search in 3 journals was conducted: “Cleft Palate-Craniofacial Journal”, “Plastic and Reconstructive Surgery Journal” and “American Journal of Orthodontics and Dentofacial Orthopaedic”.
Type of study
The review includes all human prospective and retrospective follow-up, cohort studies, case series and randomized control studies related to NAM appliance outcomes on the UCLP.
Domain being studies
Surgical, aesthetic, functional, socio-economical effects of NAM appliances and evolution of NAM appliances, especially CAD/CAM technologies were the domain being studied in this systematic review. The outcomes were compared to results on patients treated with other presurgical appliances or patients who did not receive any presurgical treatment before the primary repair surgeries.
Inclusion criteria
Search criteria include a minimum of ten patients and a follow-up not exceeding 18 months, before the first primary repair, corresponding on the period when the facial growth is the most important. Within these studies, only the ones written in English and with clear description of treatment protocol, objective outcome measurements and proof that significant results could be attributed to use of NAM were included in this review. Studies relating to surgical, aesthetic, functional, socio-economical effects of NAM appliances and evolution of NAM appliances, especially CAD/CAM technologies were included.
Exclusion criteria
Articles that were excluded were related to the following reasons: presurgical infant orthopaedics (PSIO) different from NAM, small sample size (< 10 patients), long-term NAM efficiency reports (more than 18 months, usually after primary surgeries), bilateral clefts of the lip and/or palate and articles that lacked a control group.
Sequential search strategy
First, article titles were screened in order to exclude the impertinent studies. Then, abstracts were screened in order to exclude studies without inclusion criteria. Next, the selection of articles was performed after reviewing the “Material and Methods” and “Results” sections based on inclusion and exclusion criteria. The final stage of inclusion articles was based on evidence ratings according to the Oxford Center for Evidence-based Medicine-Level of Evidence (March 2009) [14].
Data extraction
Data extraction was performed with tables addressing different subjects: surgical, aesthetical, functional, socio-economical effects of NAM appliances and the evolution of NAM appliances, especially three-dimensional technology. Each table includes: author, year of publication, type of study, type of cleft lip and/or palate, aim of the study, sample size, presence or not of a control group, method assessment, effect of nasal molding, effect of alveolar molding, “other” outcomes, follow-up and study limit.
Assessment of methodological quality
Two independent researchers performed the literature search. Any disagreement regarding inclusion and exclusion criteria were discussed and resolved. Recommendations proposed in the general methods for Cochrane reviews in order to reduce the risk of bias assessment in studies were followed in the selection of articles [15].
Statistical analysis
Meta-analyses were to be conducted only if there were studies of similar comparison, reporting the same outcome measures. However, the studies included revealed considerable variations in design (i.e. large diversity of no-NAM presurgical appliances, methodology and landmark used to assess the outcomes of NAM compare to other appliances).
RESULTS
Study selection and characteristics
Out of 356 articles, 145 were further evaluated (Figure 1), 48 were considered relevant according to their abstract but 20 were excluded (Table 2). Twenty-eight articles were included and classified by category in tables (Table 3 and 4): “NAM impact on primary repair surgery”, “Aesthetical and functional NAM outcomes”, “Efficiency of NAM compared to other appliance without nasal stents”, “NAM technique improvement”, “Three-dimensional technology in NAM approach”, “Socio-economical impact with NAM approach”, “Complications with NAM treatment”. In each category author, year of publication, type of study, type of cleft lip and/or palate, aim of the study, sample size, presence or not of a control group, method assessment, effect of nasal molding, effect of alveolar molding, “other” outcomes, follow-up and study limit could be extracted.
Flow diagram of studies selection according PRISMA guidelines.
Excluded articles, classified by topic used for data extraction: surgical, aesthetical, functional, socio-economical effects of nasoalveolar molding (NAM) appliances, the improvements of NAM appliances and the three-dimensional technology (CAD/CAM)
Topic
Study
Year of publication
Study design
Cleft
Aim of the study
Sample size
Control group
Method assessment
Effect of nasal molding
Effect of alveolar molding
Other outcomes
Follow-up
Study limit
Reason for exclusion
Impacts on surgeries
Hsieh et al. [22]
2010
Retrospective study
UCLP
Effect of gingivoperiosteoplasty on facial growth
62 (NAM + GPP: 26; NAM: 40)
-
-
-
-
-
5 years
-
Follow-up exceed 18 months
Dec et al. [23]
2013
Retrospective study
UCLP;
BCLP
Assess if NAM can decrease fistula formation complication after primary repair
178
No control group
-
NAM may reduce nasolabial fistula formation
-
-
Mean: 11 years;
median: 9 years
-
Follow-up exceed 18 months
Patel et al.
[24]
2015
Retrospective study
UCLP;
BCLP
Assessment necessity of secondary nasal revision surgery with and without NAM
NAM: 172 UCLP, 71 BCLP;
non NAM: 28 UCLP, 5BCLP
-
-
NAM:
UCLP: 3%;
non NAM: 21%
-
NAM treatment saves between $491 and $4893 depending on the type of cleft
5 to 14 years
-
Follow-up exceed 18 months
Aesthetic and functional impacts
Maull et al. [25]
1999
Retrospective randomized study
UCLP
Impact of NAM on long-term nasal shape
20 (presurgical nasal stent: 10; NAM: 10)
-
-
-
-
-
-
-
Follow-up exceed 18 months
Chang et al. [26]
2010
Retrospective study
UCLP
Long-term outcome of four different techniques of nasal reconstruction
76 (NAM only: 16; NAM + rhinosplasty: 14; NAM + rhinoplasty + overcorrection: 46)
-
Two-dimensional photographs
NAM + rhino + overcorrection (20%) have best results
-
-
5 years
-
Follow-up exceed 18 months
Liou et al. [27]
2004
Case series
UCLP
Evaluate nasal symmetry with NAM
25
-
Two-dimensional photographs
Improve nasal symmetry
-
-
3 years
No control group, small study
Follow-up exceed 18 months
Barillas et al. [28]
2009
Retrospective study
UCLP
Long-term outcome of NAM techniques of nasal reconstruction
Included articles, classified by topic used for data extraction: impact on primary repair surgeries, on nasal and alveolar molding, nasal molding improvement with nasoalveolar molding (NAM), nasal and alveolar molding improvement with NAM and evolution of the NAM procedure
Topic
Study
Year of publication
Study design
Evidence level
Cleft
Aim of the study
Sample size
Control group
Method assessment
Effect of nasal molding
Effect of alveolar molding
Other outcomes
Follow-up
Impact on primary repair surgeries
Santiago et al. [36]
1998
Retrospective blind study
III
UCLP
Compare the need of bone graft between patients who undergo NAM + GPP and no-NAM
32 (NAM + GPP: 18)
No-NAM: 14
Clinical assessment
-
NAM group: 8 required bone graft;
no-NAM group: all required bone graft
-
Before surgery
Rubin et al. [37]
2015
Quasi-experimental study
III
UCLP
Assessment by the surgeon: necessity of secondary nasal revision surgery with and without NAM
176 of the 731 surgeons accepted to answer the survey. NAM: 10;
no-NAM: 10
No-NAM: 10
Two-dimensional photographs
Necessity secondary nasal revision surgery:
NAM group: 3%;
no-NAM group: 21%.
Not statistically significant
-
Patient with NAM cost $500 less
Before the surgery
Broder et al. [38]
2016
Prospective non-randomized study
II
UCLP BCLP
Examines clinician and caregiver appraisals of primary cleft lip and nasal reconstruction
NAM: 62;
No-NAM: 48
-
Two-dimensional photographs
Better postsurgery outcomes in the NAM group
-
-
13 months
Nasal stent impact on nasal and alveolar molding
Punga and Sharma [39]
2013
Prospective study
II
UCLP BCLP
Comparison between treatment with a presurgical appliance with and without nasal stent
20 (with nasal stents: 10; without nasal stents: 10)
-
Two-dimensional photographs
Increase the columella length with a nasal stents
-
-
Every 2 - 3 weeks until lip repair
Monasterio et al. [40]
2013
Prospective study
II
UCLP
Compare two techniques: nasal elevator and NAM-Grayson
40 (NAM Grayson: 20;
nasal elevator: 20)
-
Two-dimensional photographs casts
Two methods improved significantly the nasal asymmetry
Two methods reduced significantly the cleft width
-
3 months, (before the surgery)
Isogawa et al. [41]
2010
Prospective study
II
UCLP
Compare effect between Hotz plate modified by adding a nasal stent and modified NAM
10 (NAM: 5; Hotz: 5)
-
Casts
Favourable effect obtains with PNAM
Favourable effect with both techniques
-
Around 130 days
(4 months)
Sasaki et al. [42]
2012
Prospective study
II
UCLP
Compare the effect between appliance with NAM and passive method with only action of alveolar plate
28 (NAM: 13;
Hotz plate: 15)
Control with the symmetric nostril
Two-dimensional photographs casts
Better naris morphology in the NAM group
Cleft gap smaller in the NAM group
-
Just before the surgery and after
Nakamura et al. [43]
2009
Prospective study
II
UCLP
Assess outcome nasal correction after NAM, compare with Hotz plate
30 (NAM: 15; Hotz: 15)
-
Two-dimensional photographs photographs
Better nasal shape in the NAM group
-
-
1 and 5 years post-operative
Kozel [44]
2007
Retrospective study
III
UCLP
Compare presurical orthopaedic without nasal stents and with stents
With nasal stents: 16;
without nasal stents: 16
-
Two-dimensional photographs photographs
Nose was more symmetric with nasal stents
-
-
1 year after lip repair
Nasal molding improvment with NAM
López-Palacio et al. [45]
2012
Prospective study
II
UCLP
Nasal improvement with NAM
17
Non-cleft nostril
Two-dimensional photographs of casts
Improved of nasal tip projection, alar cartilage depression and
-
-
Before the primary rhinocheiloplasty (103 days),
Gomez et al. [46]
2012
Prospective study
II
UCLP
Nasal improvement with NAM
30
-
Two-dimensional photographs of casts
Reduction of cleft columella deviation, improved columella length
-
-
Before lip surgery (146 days)
Nasal and alveolar molding improvment with NAM
Keçik and Enacar [47]
2009
Prospective study
III
UCLP
NAM effect on nasal and alveolar tissues
22
-
Two-dimensional photographs scan on cast
Reduction alar base width and the deviation of the columella
Reduction of the cleft width, arch length
-
6 months (before surgeries)
Jaeger. et al. [48]
2007
Prospective study
III
UCLP
Evaluate nasal symmetry, gap reduction
11
-
Two-dimensional photographs
Improvement nasal symmetry and nostril shape
Cleft gap reduction
-
After NAM treatment (max 23 weeks)
Pai et al. [49]
2005
Case-series
III
UCLP
Evaluate nasal symmetry and width
57
Non affected side
Two-dimensional photographs
Effect on nasal symmetry, height, and columella angle
-
Relapse of nostril shape in width
1 year
Ezzat et al.
[50]
2007
Prospective, blinded measurement study
II
UCLP
Evaluate improvement alveolar cleft and nose symmetry
12
-
Intra-oral and extra-oral casts
Nasal symmetry improvement
Cleft gap reduction
-
Mean 110 days
Shetty et al. [51]
2012
Prospective study
II
UCLP
Evaluation of the NAM treatment depending on the moment when the treatment is started
Four types of design study were found: randomized control trials, prospective, retrospective longitudinal studies and case series. 20 studies were excluded because of their small sample size (< 10 cleft patients) [16-21] or/and their follow-up exceeding 18 months [22-31]. Two studies relating to nasal and alveolar outcomes after PSIO treatment was excluded because the used appliance was not NAM [32,33]. Two studies were excluded because of lack of evidence [34,35].
Risk of bias within studies
Each article has been ranked according to the Oxford Center for Evidence-based Medicine-Level [14] and at two external reviewers reviewed every manuscript.
Statistical analysis
No meta-analyses could not be performed due to the heterogeneity between the studies.
NAM impact on primary repair surgeries
The purpose of the PSIO is to facilitate primary repair of the lip and reduce the secondary revision. In the present review, three articles, using control groups, were selected to illustrate the benefit of NAM. In 1998, a retrospective review on 32 patients (18 cleft patients treated with NAM and 14 treated without NAM) by Santiago et al. [36] showed that NAM associated with gingivoperiosteoplasty (GGP) decreases the number of patients who require a secondary alveolar bone graft procedure. In a recent quasi-experimental study, surgeons from the American Cleft Palate-Craniofacial Association assessed 20 photographs of cleft lip and/or palate patients [37]. They were asked to evaluate the outcome after NAM treatment, and the likelihood of these patients requiring revision surgeries. They reported that NAM seems to reduce the necessity for over corrective surgeries. The advantage of the NAM treatment is confirmed with another recent study by Broder et al. [38]. It highlighted that caregivers reported better postsurgery outcomes in the NAM group compared with no-NAM group (P < 0.05), particularly in relation to the appearance of the nose.
Aesthetic and functional NAM outcomes
NAM is composed of an alveolar plate and an attached nasal stent. The two parts of this device take advantage of the high plasticity of the skeletal maxillary alveolar bone and the neonatal nasal cartilage to induce an alveolar and a nasal positive remodelling. The main goal of NAM is to reduce the width of the gap between the alveolar segments. Nasal stents may be added to the palatal plate to improve the deformity of the nasal cartilages and reduce the nostril asymmetry.
Five prospective studies [39-43] and one retrospective study [44] compared PSIO with and without a nasal stent. All demonstrated that presurgical devices with a nasal component have the ability elongate the columella, and improve nasal asymmetry. Beside its nasal effect, three prospective studies showed that the nasal component has also a positive effect on the cleft defect [40-42]. Among the group of studies that assesses only the nasal molding, 2 studies were included. These reported an improvement of nasal symmetry and alar cartilage depression with NAM before primary surgeries [45,46].
In the present review, four publications assessed nasal and alveolar effects. All showed that the alveolar gap, nasal width and nasal asymmetry were significantly reduced at the end of the PSIO therapy, prior to the primary lip repair [47-50]. Results presented by Shetty et al. [51], highlighted that a treatment started prior to one month of age has a greater impact on the nasal symmetry than treatments started after this period. In another study by Shetty et al. [52], are comparing the patients that initiate the treatment before 1 month, between 1 and 6 months and patients that started NAM after 6 months. It showed that beneficial NAM outcomes are increased if the treatment is started as soon as possible.
Reduction of the cost of presurgical treatment
Among the included studies, the one introducing three-dimensional technology report that NAM using three-dimensional technology has the potential to further decrease the cost of treatment by reducing the clinical chair time and the cost of NAM appliance [53,54].
Soft tissue, hard tissue and compliance complications
Despite the non-invasive effect of NAM therapy, some complications during and after NAM treatment can occur. One publication relating to NAM complications was included in the present review: a retrospective research [55]. According to Levy-Bercowski et al. [55], 74% of patients developed complications involving the soft tissue (mucosal ulceration, bleeding, tissue fungal infections and irritation); 7% involving the hard tissue (asymmetrical configuration of the arch) and 39% reported a lack of compliance. Among the soft and hard tissue complications, only ulcerations may be taken in account as a significant complication since the sample size is statistically small (27 patients). This suggests that improvements can be made to the traditional NAM appliance in order to reduce soft tissue problems [56].
Previous evolution of Grayson technique
Since the description of the NAM appliance by Grayson et al. [6], several modifications have been proposed. Mitsuyoshi et al. [57] reported the first modification in 2004. They suggested the use of a nasal stent constructed in cobalt-chrome wire, which is believed to enhance manual control of the force and direction of the stent by the operator [57]. Bennun and Figueroa [35] provided another modification known as dynamic presurgical nasal remodelling. Two of the selected studies assessed this last modified technique and both concluded that the Figueroa appliance (with a large nasal stent) decreases the risk of ulceration compared to the traditional (Grayson) appliance [58,59].
However, all these techniques have the potential to irritate the soft tissues, are time consuming and can discourage patients and family. After taking the impression, NAM requires manual fabrication of the appliance. The region of the cleft is filled with wax, the cast is duplicated, the plate is fabricated with heat-cured acrylic resin, and finally the surface is polished [60]. Moreover, frequent visits are needed to adjust regularly the appliance by adding sequential selective resin. Finally, all these techniques do not follow a strict and repeatable protocol and the fabrication method is totally operator dependent. They may be considered as outdated compared to innovative three-dimensional technology used in other fields of dentistry, which have embraced digital technology.
Emergence and benefit of three-dimensional analysis and printing
Rapid prototyping technology was introduced in the 1990s to medicine and dentistry. Computer-aided design (CAD) made it possible to capture digital stereo-photogrammetry with laser scanning and provided high-accurate measurements from defined landmarks. Computer-aided manufacturing (CAM) allowed printing of a sequential set of appliances obtained from the treatment design software. Three-dimensional technology for presurgical cleft treatment is widely adopted in the medical industry. Several studies have been conducted to assess this technology. Two types of studies were selected. One type assessed diagnostic efficiency with the help of CAD system, the other type evaluated the treatment effectiveness of the CAM system. Three prospective studies have been selected and all of them confirm that CAD systems are very reliable techniques to assess and quantify the nasal and alveolar improvement after presurgical treatment [61-63].
Among the studies that evaluated CAM system, one prospective study and one randomized control trial were selected [53,64]. Both showed that this method is a more efficient, more precise and more predictable technology. First, laser scanning and digital model construction simplify the NAM procedure by manufacturing a series of appliances at once, saving clinic time by reducing chair-side adjustment time. Secondly, the patient’s parents are able to change the NAM appliance weekly and come to the treatment center less frequently. Third, the ability to visualize the procedure on a desktop or laptop computer can simplify communication between orthodontist and patient.
DISCUSSION
In the present review, the authors sought to answer two questions regarding the efficacy of NAM on nonsyndromic unilateral lip and/or cleft patients before primary surgeries. Does NAM appliance help presurgical orthopeadics users to better achieve cleft treatment goals, especially reduce primary surgical needs? And, how does the three-dimensional technology improve NAM therapy?
The first question addressed the benefits of NAM on the subsequent lip repair at approximately 18 months. A study published by Santiago et al. [36] demonstrated that patients undergoing NAM followed by GGP had a reduced need of further bone grafting procedure. This trend is confirmed by surgeons and caregivers’ assessments when they found that NAM treatment improve UCLP outcomes. Excluded studies (follow-up exceeding 18 months) [22,23] reinforce these findings, particularly the retrospective study conducted by Patel et al. [24]. This study showed indeed that patients with complete UCLP and BCLP treated with NAM had a lower risk of early secondary nasal revisions compare to patient without NAM. Subsequently this phenomenon explained the reduction of the cost of care with NAM treatment compare to no-NAM treatment.
This question was also related to the aesthetic and functional success of the NAM treatment. Prior studies noted that NAM significantly improves the shape of the nose and reduces the alveolar clef just before the primary repair surgeries [38-51]. Despite these positive reports, alveolar molding is still not universally utilized, and this situation is not always due to inexperience or unavailability but to scepticism regarding its efficacy. Several studies [65-70] have showed no significant effect of NAM in long-term follow-up (between 2 to 10 years old). Critical review of these has determined them to be high quality (prospective in design, large sample size, and long term follow-up). They have assessed different appliances: Latham-Millard appliance [71,72], other active appliance [65] and passive presurgical orthopaedics [66-68]. The randomized Dutchcleft series conducted in 2004 [73], 2006 [67], 2008 [74], 2009 [68] and 2015 [69,70] are some of the most cited. However, none of these latter studies assessed NAM effect in particular. Only one study, conducted by Clark et al. [75] challenged the effect of NAM by demonstrating that long-term alveolar improvement after NAM treatment was not confirmed with the three-dimensional measurements.
Another issue related to the first question is the socio-economical impact of NAM therapy because although successful treatment of the aesthetic and functional aspects of orofacial cleft anomalies is possible, it is still technically challenging, lengthy and costly. Therefore, it is important to take into consideration the economic impact of the NAM on the overall cost of the therapy and the satisfaction of parents during the presurgical orthopaedic treatment. Among included studies, Shen et al. [53] and Chen et al. [54] evaluated the cost of cleft treatment and demonstrated that the NAM appliance has the potential to decrease the overall cost of cleft treatment. Two excluded retrospective studies confirmed this trend [30,31], NAM appeared indeed as a great mean to reduce the complexity, the number of the repair surgeries and subsequently the cost of care. One of them published by Shay et al. in 2015 [31], gives interesting results and showed that the cost of a group of patients treated with NAM (mean costs $ 3550.24 ± $ 667.27) is significantly lower compare to a group of patients who received a surgical clef lip adhesion without NAM (mean costs $ 9370.55 ± $ 1691.79). No study related to parent’s cooperation during NAM therapy was included. If parental compliance during NAM therapy may be compared to other presurgical orthopaedics, it is important to mention a relevant Dutchcleft prospective randomized controlled trial conducted by Prahl et al. [33]. This study compares the acceptance of presurgical orthopaedics by the mother. An analysis of questionnaires filled out by the mothers of cleft patients at 6 weeks, 24 weeks and 58 weeks showed that families were motivated to follow the orthopaedic treatment despite the increased care time required [33]. A small descriptive study by Hopkins et al. [34] that captures on parent’s lived experiences, highlights that education and providing support can substantially improve NAM treatment.
A final issue that needed to be addressed was the incidence of complications that occur during NAM treatment. Two publications highlighted different sets of complications related to use of the traditional NAM appliance (vomiting, ulceration, and non-compliance). Levy-Bercowski et al. [55] offer some preventive measures to facilitate NAM fabrication and reduce complications. These include specific techniques in taking the impression, limiting the posterior limit of the device, and minimizing the thickness of the occlusal plate (2 to 3 mm) [55,76].
The second question the authors sought to answer was: does the three-dimensional technology improve NAM therapy? Three-dimensional technology is widely used in medicine [77], especially in maxillofacial surgery. CAD/CAM is able to fabricate a reproducible, accurate and individual appliance in a short period of time, which suits the goal of individualized medicine where each patient requires a specific therapeutic approach using predictive simulation systems [78]. It is now possible to produce sequential NAM devices with a three-dimensional printer from stereolithographic files obtained from predictive simulation of cleft segment manipulation. According to Shen et al. [53], CAD [62,63] and CAM [19,53], results in a more efficient and predictable NAM treatment. Other advantages are the potential decrease in cost and time for fabrication. This technology is also more reliable and it allows for visualisation of the treatment objective, assessment of the improvement and adaptation of the appliance). Three-dimensional technology is already widely used to treat cleft deformities. A preliminary prospective study by Ritschl et al. [20] is comparing the efficiency and presence of complications between NAM made manually (n = 6) and NAM produced with three-dimensional technology (n = 6) highlighted no difference between these two methods. However; the small sample size prevents any conclusion. Prospective studies with a large sample size should be conducted to assess the real potential of three-dimensional-printed NAM devices. This innovative solution could address the shortcomings of NAM therapy and insure that NAM therapy becomes an integral part of the standard of care for unilateral cleft palate treatment.
CONCLUSIONS
According to the literature review, nasoalveolar molding appliance therapy offer positive surgical, aesthetical, functional and socio-economical effects on unilateral clefts of the lip and/or palate treatment when performed prior to primary repair surgeries. Three-dimensional technology seems to result in a more efficient and predictable nasoalveolar molding treatment. However, scientific evidence is lacking regarding the short-term effect of nasoalveolar molding appliances and the three-dimensional technology.
ACKNOWLEDGMENTS AND DISCLOSURE STATEMENTS
The authors do not have any financial interests, either directly or indirectly, in the products or information listed in the paper.
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