Section 4 Early Treatment

#### **Chapter 16**

## A Removable Class III Traction Appliance for Early Class III Treatment

*Kristin N. Moore, David R. Musich, Donald Taylor, Budi Kusnoto and Carla A. Evans*

#### **Abstract**

Maxillary, mandibular, and dental effects resulting from the use of a removable intraoral Class III traction appliance as well as the protraction facemask in treatment of Class III malocclusion were assessed. This is a retrospective study comparing measurements from pre-treatment and post-treatment lateral cephalometric radiographs of two groups. Group 1 consisted of 25 patients treated with rapid palatal expansion followed by a removable intraoral Class III traction appliance. Group 2 consisted of 25 patients treated with rapid palatal expansion followed by a protraction facemask. The subjects were Caucasian, both male and female, with an age range of 3 to 12 years. The only significant differences were in length of treatment time and the skeletal change of angle SNA. The mean treatment times were 6.96 months and 10.96 months in the removable Class III traction appliance and protraction facemask groups, respectively. The mean increase in SNA was 0.46 degrees in the removable Class III traction appliance group and 1.81 degrees in the protraction facemask group. A removable Class III traction appliance provides orthodontists with another useful Class III treatment modality.

**Keywords:** Class III malocclusion, Class III treatment, protraction facemask, traction, orthodontics

#### **1. Introduction**

Class III malocclusion can result from mandibular prognathism, maxillary skeletal retrusion or a combination of both [1]. Many treatment philosophies and appliances have been used to treat this problem, such as protraction facemask, chin cup, and Frankel's FR-III appliance and orthognathic surgery. Miniplates and temporary anchorage devices are also being used in order to minimize the negative side effects that can occur with treatment. In Class III malocclusion, an accurate diagnosis and timing of treatment are considerations in order to achieve optimal results.

The orthopedic facemask was developed in the 1960's by Delaire [2] and has been shown to be effective in treatment of Class III malocclusion in early mixed or late mixed dentition. It can assist in correction of maxillary skeletal retrusion, maxillary

dentoalveolar retrusion, mandibular prognathism, and decreased lower facial height. It can produce the following effects: correction of a centric occlusion to centric relation (CO-CR) discrepancy, forward movement of the maxilla, forward movement of the maxillary dentition, lingual tipping of the lower incisors, and the downward and backward movement of the mandible [3]. The protraction facemask applies an anterior force on the circummaxillary sutures and stimulates bone apposition in suture areas [4]. Generally the facemask is prescribed to be worn by the patient for 12 to 16 hours per day with forces ranging between 180 g and 500 g [4, 5]. It has been suggested that the facemask be worn until the patient achieves approximately 4-5 mm of positive overjet [3]. It is often used in combination with a rapid palatal expander.

Macdonald et al. [6] found that facemask treatment increased the convexity of the facial profile due to the forward displacement and downward and backward rotation of the maxilla as well an opening rotation of the mandible. The maxillary incisors moved forward as the mandibular incisors retruded. Ngan et al. [7] found that the maxilla moved forward an average of 2.1 mm and the molar relationship corrected to Class I or even Class II relationship. In addition, the lower face height increased and the overbite decreased by an average of 1.5 mm. Nartallo-Turley and Turley [8] found an increase in SNA, maxillary depth, and ANB as well as forward movement of A-point and ANS. The maxilla moved forward and rotated counter-clockwise and the mandible rotated clockwise as the SNB and facial depth decreased.

Intraoral devices for treatment of Class III malocclusions [9, 10] have been described. A removable Class III traction appliance using elastics to produce the desired vector of force (**Figures 1a** and **1b**) was developed in the 1980's to overcome issues of patient compliance with the protraction facemask. It can be used in conjunction with rapid palatal expansion or fixed appliances in Class III treatment. This removable appliance can be used at any age and aids in disclusion of the dentition as well as directional traction as it addresses maxillary skeletal retrusion, maxillary dentoalveolar retrusion, and functional shifts associated with mandibular prognathism [11]. Similar to protraction facemask, it is said to have the following effects: correction of a CO-CR discrepancy, forward and downward displacement of the maxilla, forward movement of the maxillary dentition, lingual tipping of the lower incisors, and the downward and backward movement of the mandible. The appliance is worn by the patient full time (20–22 hours per day) sometimes in conjunction with a rapid palatal expander and/or partial or full braces treatment until 3-4 mm of positive overjet is achieved [10].

The main advantages for the removable Class III traction device are the capacity to have light, continuous, full-time forces acting to disarticulate the occlusion and allow correction of the posterior and anterior crossbites with minimal occlusal interference. 4–8 ounce elastics are recommended for younger patients and heavier forces are recommended for older patients. 10–12 ounce elastics are sometimes recommended at night based on individual patient needs. If needed, a removable appliance could be used in conjunction with a facemask at night. Another advantage of the removable appliance is that it is easy to gain optimal compliance in patients and is tolerated well by the patient.

Some disadvantages have been reported with the removable orthodontic traction device. In the mixed dentition, strong retention from the composite ridges can accelerate exfoliation of the primary canines, compromising the anchor teeth and causing some discomfort to the patient. For this reason, it is recommended that the retentive ridges be used on teeth with the best root structure. The appliance can also experience significant wear if patients have a nocturnal bruxism habit. However, replacement of the appliance is simple and inexpensive.

*A Removable Class III Traction Appliance for Early Class III Treatment DOI: http://dx.doi.org/10.5772/intechopen.99885*

#### **Figure 1.**

*(a) Anterior crossbite correction using the removable traction appliance followed by a retention phase using the same appliance. Retention ridges can be seen in the bottom row. (b) Location of hooks on the lower removable traction appliance.*

This study was designed to determine whether treatment of Class III malocclusion with a removable Class III traction appliance has outcomes similar to a protraction facemask. Specifically, the objectives were to compare maxillary, mandibular, and dental effects resulting from use of both appliances.

#### **2. Comparison of removable traction appliance and protraction facemask**

#### **2.1 Methods**

The removable orthodontic traction device described in this chapter (**Figures 1a** and **1b**) is relatively inexpensive and easy to fabricate. The first step in making this appliance is the application of retentive ridges to several of the patient's mandibular teeth especially in the anterior region. This is done by etching the tooth and then applying a composite resin to the surface of the tooth in a ridge shape, about 75% of the mesial-distal tooth width and 2-3 mm in height. Next, an impression is taken of the arch and a stone model is fabricated. Separating medium is applied to the cast and allowed to dry. A vacuum formed type retainer using C+ plastic from DENTSPLY Raintree Essix (DENTSPLY Raintree Essix, Sarasota, Florida, USA) is fabricated on the model. Durasoft® or Biocryl® from Great Lakes Orthodontics

(Great Lakes Orthodontics, Ltd., Tonawanda, New York, USA) can also be used. If a hygienic fixed expander is in place, a similar removable appliance can be made for attachment of elastics to the maxillary arch. After trimming, Caplin hooks (DENTSPLY GAC International, Bohemia, NY, USA) are added to the retainers in the upper molar and lower canine regions by heating each hook with a torch and pressing it into the appropriate area on the appliance, ensuring it does not melt completely through the plastic (**Figure 1b**). After ensuring the hooks are secure, the appliance is inserted into the patient's mouth and traction is initiated using Class III elastics. The patient is instructed to wear the appliance full time. Monthly visits are recommended to monitor for progress [10].

In this retrospective study, Group 1 consisted of 25 Caucasian patients from a private orthodontic practice who had been treated with rapid palatal expansion (hygienic Hyrax™ expander) followed by the removable intraoral Class III traction appliance and 180 g force from Class III elastics. Group 2 consisted of 25 Caucasian patients treated with a rapid palatal expansion (hygienic Hyrax™ expander) followed by a protraction facemask (AD Protraction Facemask; Ormco, Orange, CA, USA) with 350-400 g traction, taken from a different private orthodontic practice. Patient data from both offices were collected, de-identified, and assigned case numbers by the private practice orthodontists. Patients from both groups were treated until positive overjet was achieved. The inclusion criteria for both groups were an initial diagnosis of a dental and skeletal Class III malocclusion based on an ANB angle less than 0 degrees, Wits appraisal less than 0, and at least 25% Class III molar relationship in permanent or primary molars. If any functional shifts were present, they were not recorded and thus not taken into consideration. Patients were excluded if any of the following were present: dentofacial deformities (i.e. cleft lip and palate), missing teeth, periodontal disease, or prior treatment elsewhere.

The patients' pre-treatment (T1) and post-treatment (T2) lateral cephalometric radiographs were collected, scanned and digitized. The radiographs were uploaded and traced using Dolphin software (Dolphin, Chatsworth, CA, USA). Skeletal and dental measurements were collected. The landmarks seen in **Figure 2** were used in the cephalometric analysis. The following cephalometric measurements were used: SNA, SNB, ANB, Wits appraisal, Y axis, angle of convexity, mandibular plane angle, facial angle, cant of occlusal plane, upper incisor to SN, lower incisor to mandibular plane, interincisal angle, upper incisor to NA, lower incisor to NB, overbite, overjet, millimeter measurement from sella perpendicular to palatal plane to maxillary molar, millimeter measurement from sella perpendicular to palatal plane to maxillary incisor, millimeter measurement from sella perpendicular to palatal plane to mandibular molar, millimeter measurement from sella perpendicular to palatal plane to mandibular incisor, millimeter measurement from sella to A point, millimeter measurement from PTM to ANS.

A statistical power analysis determined that a sample of 20–25 subjects would yield a power of 0.8 which would provide statistically significant results. Intrareliability and inter-reliability tests all had a correlation of 0.8 or above and those values were considered to be reliable. An independent *t*-test was used to compare sample descriptives, and to compare T1 values. An independent *t*-test for parametric data and a Mann–Whitney test for non-parametric data was utilized to evaluate mean differences between groups. Also, as another indicator of similarity of samples, cervical vertebral maturation (CVM) stage for T1 and T2 for both groups, means and standard deviations were calculated according to the method of Baccetti et al. [12].

*A Removable Class III Traction Appliance for Early Class III Treatment DOI: http://dx.doi.org/10.5772/intechopen.99885*

#### **Figure 2.**

*Landmarks: 1-Nasion (N); 2-Sella (S); 3-Porion (Po); 4-Basion (Ba); 5-Articulare (Ar); 6-Condylion (Co); 7-PT point; 8-Pterygomaxillare (PTM); 9-Orbitale (or); 10-anterior nasal spine (ANS); 11-Subspinale (a); 12-upper central incisor root tip; 13-posterior nasal spine (PNS); 14-upper first molar occlusal; 15-lower first molar occlusal; 16-lower central incisor crown; 17-upper central incisor crown; 18-Supramentale (B); 19-lower central incisor root; 20-Pogonion (Pog); 21-Gnathion (Gn); 22-Menton (me); 23-Gonion (go).*

#### **2.2 Results**

#### *2.2.1 Sample descriptives*

Means and standard deviations were calculated for both T1 and T2 chronological ages for both groups (**Table 1**). The mean age for Group 1 at T1 was 8 years, 8 months and at T2 was 10 years, 6 months. The mean age for Group 2 at T1 was 8 years, 9 months and at T2 was 11 years, 1 month. An independent *t*-test showed that no significant differences existed among the T1 and T2 chronological ages between groups (p>0.05). To examine cervical vertebral maturation (CVM) stages for T1 and T2 for both groups, means and standard deviations were calculated. The mean CVM


### **Table 1.**

for group 1 at T1 was 2.1 and at T2, 2.8. The mean CVM for group 2 at T1 was 2.4 and at T2, 3.1. An independent *t*-test showed that no significant differences occurred at T1 and T2 between groups. For length of treatment of both groups, an independent *t*-test was used and showed that significant differences existed between groups (p<0.05).

#### *2.2.2 Comparison of T1 values and T2 values between groups*

Independent *t*-tests were used to evaluate if any differences existed among the T1 values and the T2 values between groups. It was found that no significant differences existed among the T1 values between groups (p>0.05).

#### *2.2.3 Comparison of T2-T1 differences between groups*

Differences between T2 and T1 were calculated for each variable within each group (**Tables 2** and **3**). An independent *t*-test was used to evaluate if any significant differences existed among the changes from T1 to T2 between groups for parametric data. A Mann–Whitney test was used for non-parametric data (group 2 for sella to A point and millimeter measurement of Ptm to ANS). SNA showed that significant differences existed between groups (p<0.05). All other values showed no significant differences between group 1 and group 2 (p>0.05).

#### **2.3 Discussion**

Both groups started and ended treatment at similar chronologic ages. Since chronologic age is only a rough indicator of maturity, cervical vertebral maturation stage was examined for both groups. Peak mandibular growth or the pubertal growth spurt has been found to occur between stages 3 and 4 with active growth having been completed at stage 6 [12]. Baccetti et al. [12] suggested that Class III treatment with rapid maxillary expansion and protraction facemask therapy should be started during stages 1 and 2 in order to produce the most effective results on the maxilla. Both groups had a mean initial CVM of stage 2 which correlates to pre-pubertal growth peak. No significant differences in CVM stage existed at T1 and T2 between groups suggesting that both groups were similar with regards to skeletal maturation before and after treatment.

The significant difference in treatment times may have affected the outcomes between groups. The protraction facemask was used for a greater period of time on average than the removable Class III traction appliance and has a direct effect on the maxilla. Thus, with a greater treatment time one could expect more change at SNA, which may have contributed to the significantly increased SNA in the protraction facemask treated group when compared with the removable Class III traction appliance treated group. The outcomes of the protraction facemask treated group were consistent with studies conducted by Nartallo-Turley and Turley [8], Ngan et al. [7], and Macdonald et al. [6].

No significant differences were found between groups comparing Wits appraisal, ANB, FMA, Y-axis, cant of the occlusal plane, Sella to A point, PTM to ANS, and angle of convexity. This may suggest that both appliances produced similar results in the maxilla and rotation of the mandible. It was also found that both groups exhibited proclination of the upper incisors, mesial movement of the upper and lower dentition, uprighting of the lower incisors, increase in interincisal angle, increase in overjet and increase in overbite similar to the studies by Nartallo-Turley and Turley [8], Ngan et al. [7], and Macdonald et al. [6].


*A Removable Class III Traction Appliance for Early Class III Treatment DOI: http://dx.doi.org/10.5772/intechopen.99885*

#### **Table 2.**

*Comparison of T2-T1 angular differences between groups (N = 25).*

The strength of this study is that it evaluated the effects of using a removable Class III traction appliance. Since the results showed that no statistical differences existed for dental and all but one of the skeletal variables between groups, the removable Class III traction appliance could be used as another minimally invasive Class III treatment modality for patients. Further studies of removable Class III traction appliances should implement a randomized patient assignment prospectively as well as obtain long-term results in order to evaluate their overall effectiveness.

Conventional protraction facemask therapy has been found in multiple studies to be effective; however, compliance is a major limitation. Patients often view the protraction facemask as awkward at best and complain about it being difficult to wear and interfering with sleep. Cole [13] evaluated patient compliance using headgear to treat Class II malocclusion; patients were fitted with a commercially available timing


#### **Table 3.**

*Comparison of T2-T1 linear differences between groups (N = 25).*

headgear that measured the amount of headgear wear. Compliance levels varied from 5.6% to 107.7% with a mean of 74.4%. It was found that most patients reported more headgear wear than what actually took place. Poor patient compliance with headgear or facemask can contribute to poor outcomes in treatment.

Since the removable orthodontic traction device is an intraoral appliance, it is possible for patients to adapt to wearing the appliance full time. Patients may not view this removable intraoral appliance with the same annoyance as they do the protraction facemask. If any minimally invasive treatment modalities can be used with predictability, it has great benefit as significant risk and cost is reduced in the care of the patient.

Based on the outcomes of this study comparing a removable Class III traction appliance and protraction facemask for the treatment of Class III malocclusion, it seems that both appliances are effective treatment modalities. Each appliance has its advantages and disadvantages and each treatment modality should be selected on a patient-by-patient basis.

#### **3. Conclusion**

A removable intraoral Class III traction appliance provides orthodontists with a useful noninvasive treatment alternative to protraction facemask in young patients presenting with Class III malocclusions. Both treatments resolved the Class III dental *A Removable Class III Traction Appliance for Early Class III Treatment DOI: http://dx.doi.org/10.5772/intechopen.99885*

relationships; only slight differences in outcomes were found between the protraction facemask and removable Class III traction appliance, namely, time in treatment and change in angle SNA were both slightly larger in the protraction facemask patients. It is common for orthodontists to treat using a protraction facemask, but if similar results can be achieved by using a removable Class III removable traction appliance, then it may be advantageous to consider this appliance as an option for some Class III patients.

#### **Conflict of interest**

The authors declare no conflict of interest.

#### **Abbreviations**

CO centric occlusion CR centric relation CVM cervical vertebral maturation mm millimeter T1 pre-treatment T2 post-treatment

#### **Author details**

Kristin N. Moore1 , David R. Musich<sup>2</sup> , Donald Taylor3 , Budi Kusnoto4 and Carla A. Evans<sup>5</sup> \*

1 Lake Zurich, IL, USA

2 Schaumburg, IL, USA

3 Montreal, QC, Canada

4 University of Illinois at Chicago, Chicago, IL, USA

5 Boston University, Boston, MA, USA

\*Address all correspondence to: caevans@bu.edu

© 2021 The Author(s). Licensee IntechOpen. This chapter is distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

### **References**

[1] Guyer EC, Ellis EE, McNamara JA, Behrents RG. Components of class III malocclusion in juveniles and adolescents. Angle Orthod 1986;56:7-30.

[2] Delaire J. L'emploi physiologique des tractions extra-orales postero-anterieures sur masque orthopedique dans Ie traitement des classes III. Orthod Fr 1988;59:577-589.

[3] McNamara JA. Treatment of patients in the mixed dentition. In: Orthodontics: Current Principles and Techniques, ed. TM Graber, RL Vanarsdall, KWL Vig, Elsevier Mosby, St. Louis, 2005, pp. 543-577.

[4] Şar , Arman-Özçipici A, Uçkan S, Yazici AC. Comparative evaluation of maxillary protraction with or without skeletal anchorage. Am J Orthod Dentofacial Orthop 2011;139:636-649.

[5] Yepes E, Quintero P, Rueda ZV, Pedvoza A. Optimal force for maxillary protraction facemask therapy in the early treatment of class III malocclusion. Eur J Orthod 2014;36:586-594.

[6] Macdonald KE, Kapust AJ, Turley PK. Cephalometric changes after the correction of class III malocclusion with maxillary/facemask therapy. Am J Orthod Dentofacial Orthop 1999;116:13-24.

[7] Ngan, P., Yiu, C., Hu, A., Hägg, U., Wei, S.H., and Gunel, E. Cephalometric and occlusal changes following maxillary expansion and protraction. Eur. J. Orthod. 20:237-254, 1998.

[8] Nartallo-Turley PE, Turley PK. Cephalometric effects of combined palatal expansion and facemask therapy on class III malocclusion. Angle Orthod 1998;68:217-224.

[9] Vanlaecken R, Williams MO, Razmus T, Gunels E, Martin C, Ngan P. Class III correction using an inter-arch spring-loaded module. Prog Orthod 2014;15:32.

[10] Musich DR, Busch MJ. Clinical management of the class III growing patient. Int Orthod Kieferorthop 2008;40:13-26.

[11] White LW. Orthodontic Pearls: A Clinician's Guide. Self-Published, 2011, p. 115.

[12] Baccetti T, Franchi L, McNamara JA. The cervical vertebral maturation (CVM) method for the assessment of optimal treatment timing in dentofacial orthopedics. Semin Orthod 2005;11:119-129.

[13] Cole W. Accuracy of patient reporting as an indication of headgear compliance. Am J Orthod Dentofacial Orthop 2002;121:419-423.

#### **Chapter 17**

## Bridging the Gap: Nasoalveolar Moulding in Early Cleft Palate Rehabilitation

*Amanda Nadia Ferreira*

#### **Abstract**

Orofacial clefts (OFC) are among the commonest birth defect in developed and developing countries alike. In underdeveloped and developing countries, babies born with oral clefts are generally anaemic with low birth weight and may be unfit for surgery. The surgical reconstruction is also challenging and the aesthetic outcome cannot be guaranteed by the surgeon. Presurgical nasoalveolar moulding (PNAM) has been suggested to bridge the gap between the clefted segments before surgical repair. It is a simple yet effective technique that needs to be initiated at the right time and age to achieve ideal functional and aesthetic outcomes. This chapter highlights the effectiveness of the nasoalveolar moulding technique and details the manner in which the appliance is fabricated and activated.

**Keywords:** presurgical nasoalveolar moulding, feeder plate, clept lip and palate

#### **1. Introduction**

Each year, around 250,000 babies are born with some form of orofacial clefts [1]. Worldwide, the incidence of cleft is reported in one of every 600–800 newborns [2]. A vast majority of these babies are born in underdeveloped or developing countries. This already deplorable situation is aggravated by the fact that most of these cases are concentrated in rural areas where access to health care is severely inadequate or unavailable as compared to urban cities [3, 4].

In developed countries, cleft lip/palate (CL/P) is identified before birth by ultrasonography, which gives the parents much needed time for education and counselling regarding the additional care needed after birth. Consequently, due to the widespread access to medical care and scientific data, aetiology is scientifically understood to be due to a combination of genetic and environmental factors. In contrast, in developing countries prenatal care is less advanced or limited, a CL/P is usually unexpected and families rely less on medical explanations for the cleft and rely more on religion and folklore to explain the deformity [5].

Veau [6] classified clefts into (**Figure 1**).

Group I: Cleft involving the soft palate alone.

Group II: Cleft involving the hard and soft palate up to the incisive foramen.

Group III: Complete unilateral cleft involving the soft and hard palate, the lip and alveolar ridge on one side.

Group IV: Complete bilateral cleft involving the soft and hard palate, the lip and alveolar ridge on both sides.

Successful rehabilitation of all these cases requires a multidisciplinary approach. Patients with orofacial clefts need to be treated at the right time and age to achieve functional and aesthetic well-being. The management of the child born with a cleft lip and palate requires coordinated care provided by a cleft care team [7], comprising of different individuals belonging to several specialities in:


In many developing countries, there are several unrepaired cleft patients due to the mismatch between the volume of patients and resources. Furthermore, babies who are born underweight or anaemic are not suitable for surgery. There is also an acute shortage of qualified surgeons available to treat them [8]. This results in patients who cannot reach their full social and economic potential [9]. Surgical repair alone cannot address the multiple issues encountered in patients with cleft lip and palate. One specific task is the aesthetic recreation of the deficient columella. The earliest mention of

*Bridging the Gap: Nasoalveolar Moulding in Early Cleft Palate Rehabilitation DOI: http://dx.doi.org/10.5772/intechopen.101986*

presurgical infant orthopaedics was in the 1950s. This adjunctive therapy reduced the severity of the initial cleft deformity before surgery. This enabled the surgeon to enjoy the benefits associated with surgical repair in an infant with a minimal cleft deformity and reduced the need for a secondary surgery [10].

This chapter describes the technique of presurgical nasoalveolar moulding (PNAM), which was first described by Grayson et al. [11] in 1993 and had several modifications made over the years by Brecht et al. [12] in 1995, Grayson and Santiago [13] in 1997 and Cutting et al. [14] in 1998. This approach involves the active moulding and repositioning of the deformed nasal cartilages and alveolar process and lengthening the deficient columella, using the NAM appliance which consists of nasal stents attached to an intraoral moulding plate to aid in the moulding of the clefted alveolar ridge and nasal cartilage. The primary goal of PNAM is to achieve good arch form and eventually stabilisation.

The concept of NAM works on Matsuo's principle that a high degree of plasticity is seen in the cartilages of infants in the first few months after birth. A high amount of circulating maternal oestrogen causes an increase in the amount of hyaluronic acid in the fetal cartilage, rendering it plastic. Hence, active soft tissue and cartilage moulding are most successful if initiated within the first 6 weeks of life [15].

#### **2. Unilateral orofacial cleft lip and palate**

Clinical examinations of babies born with unilateral cleft lip and palate often show significant nasal deformities. The lower lateral alar cartilage is concave and

**Figure 2.** *Unilateral orofacial cleft lip and palate.*

depressed in the alar rim and separated from the contralateral cartilage. This results in a depressed nasal tip and possibly an overhang of the apex of the nostril. The columella and nasal septum are deviated towards the cleft, and the base towards the non-cleft side. Furthermore, the orbicularis oris muscle in the lateral lip segments contracts into a bulge with some fibres running superiorly along the margins of the cleft towards the nasal tip (**Figure 2**) [16, 17].

### **3. Bilateral orofacial cleft lip and palate**

Babies born with bilateral cleft lip and palate often present a challenge to the cleft care team. In these cases, the alar cartilages have failed to migrate up into the nasal tip and stretch the columella. So, the cartilages are positioned along the alar margins and are stretched over the cleft as flaring alae. The prolabium also lacks muscle tissue and is positioned directly on the end of the shortened columella. In the complete bilateral cleft, the premaxilla is suspended from the tip of the nasal septum, while the clefted alveolar segments stay behind (**Figure 3**) [18, 19]. The primary issue in these cases is that the premaxilla is unattached laterally and is positioned far too anteriorly by the time lip surgery is scheduled. Secondly, in some cases, the lateral width of the premaxilla exceeds the anterior space between the two lateral maxillary segments. A combination of these two challenges may also exist.

**Figure 3.** *Bilateral orofacial cleft lip and palate.*

#### **4. Procedure**

Before commencing any treatment procedures, the parents/caregivers are counselled about PNAM therapy. The procedure, goals, possible complications and their role is explained to them.

*Bridging the Gap: Nasoalveolar Moulding in Early Cleft Palate Rehabilitation DOI: http://dx.doi.org/10.5772/intechopen.101986*

#### **5. Impressions**

Several impression materials and techniques have been advocated for making the impression of the clefted alveolar segments. Grayson and Shetye [20] advised keeping the child nil orally for about 4 hours and making the impression while holding the baby upside down to prevent aspiration in the event of vomiting and asphyxia due to airway obstruction. A thick mix of tissue conditioning material was loaded onto the tray and inserted intraorally. The impression is allowed to set while the baby is making suckling actions in order to create the desired border seal and ensure the baby's ability to perform nasal breathing. The baby's oxygen level was monitored during the entire duration of impression making.

Retnakumari et al. [21] used heavy body silicone impression material with the baby in a supine position during the procedure. Dubey et al. [22] kept the baby in the mother's lap with the head facing downward and her hands supporting the baby's chest and lap region while making the impression. Yang et al. [23] advised alginate impressions using a beaded pretrimmed paediatric tray. Splengler et al. [24] made intraoral and extraoral alginate impressions with the baby under general anaesthesia. This method is generally not recommended as the patient is subjected to hospitalisation for an impression procedure.

Irrespective of the material and technique used, the sole objective of including all the available undercuts in the dental cast should be met. An ideal impression material must be rigid and set fairly quickly in the baby's mouth. The baby is positioned in an upright position, fully awake on the caregiver's lap. It is preferable if the baby is crying, as it allows better visuals of the extent of the cleft. The entire clefted palate should be recorded (**Figure 4**) and the size of the cleft should be determined on the resultant cast using a Vernier calliper.

**Figure 4.**

*Impression of the clefted segments in a unilateral cleft (A) and a bilateral cleft (B).*

#### **6. Appliance fabrication and design**

The moulding plate is fabricated on the dental stone cast obtained from the impression. All the undercuts and the cleft space are blocked with wax. The moulding plate is made up of clear acrylic. A 5 mm hole is incorporated to facilitate breathing in case of accidental dislodgement (**Figure 5**). The plate must be 2–3 mm in thickness to provide structural integrity and permit adjustments during the process of moulding.

#### **Figure 5.**

*On the obtained cast (A), cleft space is blocked out with wax (B) and the moulding plate is fabricated with a breathing hole (C).*

#### **Figure 6.** *Two retentive arms are incorporated in bilateral cases.*

A retentive acrylic arm is fabricated and positioned labially at an angle of 40 degrees to the plate. It should be placed at the junction of the upper and lower lip. The retentive arm adequately secures the moulding plate in the mouth with the help of orthodontic elastics and tapes. In bilateral cases, there is a need for two retentive arms (**Figure 6**) [13]. The appliance has to be finished and polished ensuring that no sharp borders are present.

#### **7. Appliance insertion and moulding**

The NAM appliance was tried on the baby. The intaglio surface of the plate was then modified to allow for selective pressure on the two segments of the arch using tissue conditioner. There is selective removal of acrylic in the region into which the movement of alveolar bone is desired; and tissue conditioner was added to regions from which, the alveolar bone needed to be reduced. Selective pressure was applied on the greater and lesser alveolar segments to permit moulding. 1 mm thickness of tissue conditioner was applied onto the outer surface in the region of the greater

#### *Bridging the Gap: Nasoalveolar Moulding in Early Cleft Palate Rehabilitation DOI: http://dx.doi.org/10.5772/intechopen.101986*

segment and the inner surface was relieved by 1 mm. Tissue conditioner was also applied on the inner surface in the region of the lesser segment and the outer region was relieved by 1 mm (**Figure 7**). This caused a force that was directed inward on the greater segment and outward on the lesser segment that would cause approximation of alveolar tissue [25].

The NAM appliance is secured extra orally to the cheeks and bilaterally by surgical tapes with orthodontic elastic bands at one end. A muslin head cap with Velcro strips at the side is tailor-made for the baby (**Figure 8**). The Velcro strips provided attachment of the elastic bands, as well as facilitated their placement and removal. The elastic band is looped on the retentive arm of the moulding plate and secured with tape to the cheeks. The elastics with an inner diameter of 0.25 inch, and heavy wall thickness, should be stretched to about twice their resting diameter in order to achieve an ideal activation force of about 100 g. The amount of force could vary depending on the clinical objective and the mucosal tolerance to ulceration. Additional tapes may be necessary to secure the horizontal tape to the cheeks.

The infant may require time to adjust to feeding with the NAM appliance in the first few days. The baby is seen weekly to make adjustments to the moulding plate. These adjustments are made by selectively removing the hard acrylic and adding the

**Figure 7.** *Selective pressure applied on the clefted alveolar segments.*

**Figure 8.** *A custom made muslin head cap used to secure the NAM appliance.*

soft tissue conditioner to the moulding plate. No more than 1 mm of modification of the moulding plate should be made per visit. The desired movement can usually be accomplished within 6 to 8 weeks.

The NAM appliance needs to be worn 24 hours a day and removed only for daily cleaning, and needs to be inserted back soon afterwards. Even after 3 weeks, most cases did not show any clinical evidence of tissue irritation or accumulation of debris.

The effectiveness of the selective moulding is enhanced by adequately supporting the appliance against the palatal tissues and taping the lip segments across the cheek. This tight apposition of the lip segments provides the same benefit of traditional lip adhesion, but without the consequent scarring. It also serves to improve the alignment of the nasal base by bringing the columella towards the midsagittal plane, thereby improving the symmetry of the nostrils. Lip adhesion in isolation produces an uncontrolled orthopaedic movement. However, if carried out along with the moulding plate, the movements can be more precise and controlled.

#### **8. Nasal stent**

The nasal stent is added to the NAM appliance when the width of the cleft is reduced to a size of ≥6 mm. The reasoning behind delaying the addition of the nasal stent is that when the cleft size reduces, the alignment of the base of the nose and the lip segment also improves. The alar rim, which was initially stretched over the clefted segments at birth, will show some laxity, now that the cleft size has reduced and thus can be elevated into a symmetrical and convex form with the nasal stent. Any attempt to correct this deformity before reducing the cleft size may result in an undesirable increase in the lateral alar wall [26].

Matsuo and Hirose [27] suggested a silicone nasal conformer, which can be used for presurgical nasal moulding. The height of the conformer is adjusted by gradually adding some soft resin or flat silicone sheets on the domes. It can be used for presurgical elongation of the columella in incomplete clefts or postoperative maintenance of the nostril configuration. Blanching occurs at the nasal tip as infant suckles and activates the appliance. It also exerts a reciprocal intraoral moulding force against the clefted alveolar segments.

Grayson and Shetye [20] adapted nasal stent to extend from the anterior flange of an intraoral moulding plate. The greatest advantage of NAM is that it enables the practitioner to apply force skilfully to shape the nasal cartilage. Figueroa's technique [28] involves the simultaneous moulding of the alveolar cleft and nasal cartilage using a rigid acrylic nasal extension attached to an acrylic plate. Elastics are attached to the acrylic plate to allow gentle retraction of the premaxilla. A soft resin ball may also be attached to the acrylic plate across the prolabium in order to maintain the nasolabial angle. In bilateral cases, there is a need for two retentive arms as well as two nasal stents which are similar in shape to the unilateral stent.

The nasal stent is made from 19 gauge (0.36 inch), round stainless-steel wire, in the shape of a 'Swan Neck' (**Figure 9**). The base of the stent should be located midway between the clefted lip segments. The superior loop is adjusted to fit passively in the nostril on the cleft side. The nasal portion of the wire is then covered with self-cure clear acrylic and then by a layer of the tissue conditioner until mild blanching is evident. This superior lobe gently lifts the nasal dome forward, while the lower lobe lifts the tip of the nose and defines the top of the columella.

*Bridging the Gap: Nasoalveolar Moulding in Early Cleft Palate Rehabilitation DOI: http://dx.doi.org/10.5772/intechopen.101986*

#### **Figure 9.** *Nasal stent.*

Through gradual increments of tissue conditioner, the nostril on the cleft side is lifted to achieve acceptable elevation, and symmetry moulding continued until the desired nasal cartilage and alveolar shape is achieved.

Shetty et al [29] used the following protocol for presurgical NAM therapy:

#### 1.**First visit:**


#### 3.**Periodic 3 weeks recall visits:**


#### 4.**Care and instructions**


#### 5.**Troubleshooting for parents**


#### 6.**Troubleshooting for cleft care team:**


*Bridging the Gap: Nasoalveolar Moulding in Early Cleft Palate Rehabilitation DOI: http://dx.doi.org/10.5772/intechopen.101986*

#### **9. Lip and nose surgery**

The success of PNAM depends upon the surgical procedure and the treating surgeon's skill. The surgical procedure, most commonly recommended is the modified gingivoperiosteoplasty (GPP), described by Millard and Lantham [30] carried out usually within 12–16 weeks of age. The surgery may be delayed in cases where additional weeks of PNAM therapy is needed. The surgical procedure involves a first stage primary lip nose repair to close the alveolar defect followed by one-stage palatal repair at 11–13 months of age when speech begins to develope (**Figure 10**) [31].

**Figure 10.** *Lip and nose surgery.*

#### **10. Extraoral nasal stent**

Postsurgery, an additional external nasal stent can be given for 1 year to improve the nasal morphology if it did not resemble the unaffected side and also maintain the nasal correction if needed. The postsurgical external nasal stent is fabricated by making an impression of the unaffected nostril using tissue conditioner, and using it to mould the nasal contour on the cleft side [32].

#### **11. Complications**

The most common complication with the NAM therapy is irritation of the oral mucosa, gingival tissue and nasal mucosa. These issues arise due to the forces applied by the appliance [20]. They can be avoided by careful examination and modification of the extent and fit of the appliance. Fungal infection is another complication that can occur due to poor oral hygiene and continuous wear of the appliance. This can be avoided by following a meticulous oral hygiene routine and following the wash care instructions for the NAM plate. In severe cases, local nystatin or systemic amphotericin can be used [33].

#### **12. Conclusion**

Presurgical infant orthopaedics by means of nasoalveolar moulding enables the surgeon to carry out gingivoperiosteoplasty, which decreases the need for a second surgery. Bilateral cases, especially benefit as columella lengthening is carried out nonsurgically. It also minimises scar tissue formation and provides for more consistent outcomes. PNAM is most successful when initiated early and through meticulous planning and collaboration between the various disciplines.

### **Author details**

Amanda Nadia Ferreira Department of Prosthodontics, Crown and Bridge, Goa Dental College and Hospital, Goa, India

\*Address all correspondence to: dr.amandaferreira@gmail.com

© 2022 The Author(s). Licensee IntechOpen. This chapter is distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

*Bridging the Gap: Nasoalveolar Moulding in Early Cleft Palate Rehabilitation DOI: http://dx.doi.org/10.5772/intechopen.101986*

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[3] Cubitt JJ, Hodges AM, Van Lierde KM, Swan MC. Global variation in cleft palate repairs: An analysis of 352,191 primary cleft repairs in low- to higher-middleincome countries. The Cleft Palate-Craniofacial Journal. 2014;**51**(5):553-556. DOI: 10.1597/12-270

[4] Mossey P, Little J. Addressing the challenges of cleft lip and palate research in India. Indian Journal of Plastic Surgery. 2009;**42**(Suppl):S9-S18

[5] Medwick L, Synder J, Schook C, Blood E, Brown S, White W. Casual attributes of cleft lip and palate across cultures. The Cleft Palate-Craniofacial Journal. 2013;**50**:655-667

[6] Veau V. Division Palatine. Paris: Masson; 1931. Cited in Whitaker et al. A proposed new classification of craniofacial anomalies. Cleft Palate Journal. 1981;**18**(3):161-76

[7] Dean JA, Mcdonald RE, Avery DR. Dentistry for the Child and Adolescent. 9th ed. Missouri, United States: Elsevier Mosby; 2012

[8] Sommerlad BC. A technique for palate repair. Plastic and Reconstructive Surgery. 2003;**112**:1542-1548

[9] Roberts-Thomson K. Epidemiology of cleft lip and palate. In: Peres MA, Antunes JLF, Watt RG, editors. Oral Epidemiology. Textbooks in

Contemporary Dentistry. Cham: Springer; 2021

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[11] Grayson BH, Cutting CB, Wood R. Preoperative columella lengthening in bilateral cleft lip and palate. Plastic and Reconstructive Surgery. 1993;**92**: 1422-1423

[12] Brecht LE, Grayson BH, Cutting CB. Columellar elongation in the bilateral cleft lip and nose patient. Journal of Dental Research. 1995;**74**:257

[13] Grayson BH, Santiago PE. Presurgical orthopedics for cleft lip and palate. In: Aston SJ, Beasley RW, CHM T, editors. Grabb and Smith's Plastic Surgery. 5th ed. Philadelphia: Lippincott-Raven; 1997. pp. 237-244

[14] Cutting CB, Grayson BH, Brecht LE, Santiago PE, Wood R, Kwon S. Presurgical columellar elongation and primary retrograde nasal reconstruction in one-stage bilateral cleft lip and nose repair. Plastic and Reconstructive Surgery. 1998;**101**:630-639

[15] Matsuo K, Hirose T, Tomono T, Iwasawa M, Katohda S, Takahashi N, et al. Nonsurgical correction of congenital auricular deformities in the early neonate: A preliminary report. Plastic and Reconstructive Surgery. 1984;**73**:38-51

[16] Huffman WC, Lierle DM. Studies on the pathologic anatomy of the unilateral harelip nose. Plastic and Reconstructive Surgery. 1949;**4**:225-234

[17] Hogan VM, Converse JM. Secondary deformity of unilateral cleft lip and nose. In: Grabb WC, Rosentein SE, Bzoch KR, editors. Cleft Lip and Palate-Surgical, Dental and Speech Aspects. Boston: Little Brown; 1971. pp. 245-264

[18] Broadbent TR, Woolf RM. Cleft lip and nasal deformity. Annals of Plastic Surgery. 1984;**12**:216-234

[19] Millard DR. Embryonic rationale for the primary correction of the classical congenital clefts of the lip and palate. Annals of the Royal College of Surgeons of England. 1994;**76**:150-160

[20] Grayson BH, Shetye PR. Presurgical nasoalveolar moulding treatment in cleft lip and palate patients. Indian Journal of Plastic Surgery. 2009;**42**:S56-S61

[21] Retnakumari et al. Nasolveolar molding in presurgical infant orthopedics. Archives of Medicine and Health Sciences. 2014;**2**(1)

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[23] Yang S, Stelnicki EJ, Lee MN. Use of nasoalveolar moulding appliance to direct growth in newborn patient with complete unilateral cleft lip and palate. Pediatric Dentistry. 2003;**25**:253-256

[24] Splengler LA, Chavarria C, Teichgraber FJ, Gatenes J, Xia JJ. Presurgical nasoalveolar moulding therapy for the treatment of bilateral cleft lip and palate: A preliminary study. The Cleft Palate-Craniofacial Journal. 2006;**43**:321-328

[25] Sarin SP, Parkhedkar RD, Deshpande SS, Patil PG, Kothe S. Journal of Indian Prosthodontic Society. 2010;**10**:67-70

[26] Grayson BH, Cutting CB, Santiago PE, Brecht LE. Presurgical nasoalveolar moulding in infants with cleft lip and palate. The Cleft Palate-Craniofacial Journal. 1999;**36**:486-498

[27] Matsuo K, Hirose T. Preoperative non surgical overcorrection of cleft lip nasal deformity. British Journal of Plastic Surgery. 1991;**44**:5-11

[28] Figueroa A. Orthodontics in cleft lip and palate management. In: Mathes SJ, Hentz UR, editors. Plastic Surgery. 2nd ed. Philadelphia, PA: Saunders; 2006. pp. 271-310

[29] Shetty V, Vyas HJ. A comparison of results using nasoalveolar moulding in cleft infants treated within 1 month of life versus those treated after this period: Development of a new protocol. International Journal of Oral and Maxillofacial Surgery. 2012;**41**:28-36

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[32] Bhutiani N, Tripathi T, Verma M, Bhandari PS, Rai P. Assessment of treatment outcome of presurgical nasoalveolar molding in patients with cleft lip and palate and its postsurgical stability. The Cleft Palate-Craniofacial Journal. 2020;**57**(6):700-706. DOI: 10.1177/1055665620906293

[33] Murthy PS, Deshmukh S, Bhagyalakshmi A, Srilantha KT. Presurgical nasoalveolar molding: Changing paradigms in early cleft lip and palate rehabilitation. Journal of International Oral Health. 2013; **5**(2):70-80

#### **Chapter 18**

## Deleterious Facial Effects Caused by Noninvasive Ventilation Mask Early Treatment, in Congenital Muscular Dystrophy

*David Andrade, Maria-João Palha, Ana Norton, Viviana Macho, Rui Andrade, Miguel Palha, Sandra Bussadori, Lurdes Morais and Manuela Santos*

#### **Abstract**

Neuromuscular disorders is a general term that encompasses a large number of diseases with different presentations. Progressive muscle weakness is the predominant condition of these disorders. Respiratory failure can occur in a significant number of diseases. The use of devices to assist ventilation is quite frequent in these types of patients. Noninvasive ventilation can be applied by various means, including nasal, oronasal, or facial masks. Masks, type bilevel positive airway pressure, continuous positive airway pressure, and similar are generally supported on the maxilla. Oral health in pediatric neuromuscular diseases has some peculiar aspects that we must consider in these patients' follow-up. Based on a clinical case, this chapter provides a better understanding of these patients. It will focus on the oral and maxillofacial morphological alterations and preventive measures and strategies for oral pathologies management in this population. Despite always aiming at esthetics, treating these patients should always prioritize the possibilities of improving the oral and general functions of the body.

**Keywords:** neuromuscular diseases, congenital muscular dystrophy, noninvasive ventilation, clinical case, adverse effects, palatal expansion technique, extraoral traction appliances, pediatric dentistry, maxillary retrusion, airway obstruction

#### **1. Introduction**

Neuromuscular disorders (NMD) is a general term that encompasses a large number of diseases with different presentations.

Progressive muscle weakness is the predominant condition of these disorders. Respiratory failure can occur in a significant number of diseases. The use of devices to assist ventilation is quite frequent in these types of patients. Noninvasive ventilation (NIV) can be applied by various means, including nasal, oronasal, or facial masks.

Masks, type bilevel positive airway pressure (BiPAP), continuous positive airway pressure (CPAP), and similar are generally supported on the maxilla.

Long-term noninvasive ventilation (LTNIV) has been increasingly used in children to manage chronic respiratory failure and airway obstruction. Interfaces are of paramount importance for NIV effectiveness and patient compliance. Factors such as the child's age, disease, craniofacial conformation, type of ventilator and ventilation mode, and children's and family's preferences should be considered when selecting the appropriate mask. Adverse events such as skin lesions, facial growth impairment, and leaks must be prevented and promptly corrected. Humidification is a controversial issue on NIV, but it may be helpful in certain circumstances. Regular cleaning and disinfection of interfaces and equipment must be addressed. During follow-up, educational programs, close supervision, and continuous support to children and families are crucial to the success of LTNIV therapy [1].

Oral health in pediatric neuromuscular diseases has some peculiar aspects that we must consider in these patients' follow-up. Based on a clinical case, this chapter provides a better understanding of these patients. It will focus on the oral and maxillofacial morphological alterations and preventive measures and strategies for oral pathologies management in this population. Despite always aiming at esthetics, treating these patients should always prioritize the possibilities of improving the oral and general functions of the body.

Also, future research on the oral health of patients will be discussed.

#### **2. Neuromuscular disorders**

Neuromuscular disorders (NMDs) include conditions affecting the anterior horn cell (e.g., Spinal muscular atrophy = SMA), the peripheral nerve (e.g., Charcot– Marie–Tooth disease = CMT), the neuromuscular junction (e.g., Congenital myasthenia), or the muscle itself (e.g., Duchenne muscular dystrophy = DMD). NMDs are progressive, impair motor function, and often reduce life expectancy and quality of life [2]. Progressive muscle weakness is the predominant condition of these disorders. All muscles may be involved, such as facial muscles (including orbicularis oris) and bulbar muscles. In some of the patients, congenital high arched palate and even temporomandibular arthrogryposis can be present. Feeding problems are common, with the need for a feeding tube or gastrostomy. Speech problems with anarthria or dysphonia with nasal speech can be present.

The terms "muscle disease," "myopathy," "neuromuscular conditions," and "neuromuscular disorders" all describe a group of conditions that affect either the muscles, those in the arms and legs or the heart and lungs, or the nerves which control the muscles [3].

#### **3. Noninvasive ventilation**

Positive airway pressure (PAP) may be invasive or not, depending on the techniques we use. Furthermore, it can be used in acute or chronic situations.

Noninvasive ventilation can be applied by various means, including nasal, oronasal, or facial mask, among others [4]. Masks, type BIPAP, CPAP, VPAP, and similar, are generally supported over the jaw. A nasal interface (or nasal cannula) is the preferred interface, and a nasobuccal interface can be used with caution in case of mouth breathing.

*Deleterious Facial Effects Caused by Noninvasive Ventilation Mask Early Treatment… DOI: http://dx.doi.org/10.5772/intechopen.100161*

Positive Long-term NIV is a highly efficacious type of noninvasive respiratory support that has transformed the scope of chronic respiratory failure and severe sleep-disordered breathing in children with NMD by avoiding tracheotomies and allowing the child to live at home with a good quality of life for a child and his family. The tremendous heterogeneity of the disorders, ages, prognosis, and outcomes of the patients underlines the necessity of management by experienced, multidisciplinary pediatric centers, having technical competencies in pediatric NIV, and expertise in sleep studies and therapeutic education [5].

#### **4. Major groups of risk**

If we look to the population that uses NIV, we identify the significant groups of risk, where respiratory muscles are weakened, or the airway is obstructed:


#### **5. Facial side effects during noninvasive positive pressure ventilation in children**

Retrognathia is a physical misalignment of the upper (maxilla) and lower (mandibular) jawbones in which either or both recede relative to the frontal plane of the forehead. In the maxilla, we use the term maxillary retrusion as synonymous [6].

Facial side effects during noninvasive positive pressure ventilation (NPPV) in children are not a new effect. Brigitte Faroux [7], in 2005, concluded about the use of nasal masks that global facial flattening was present in 68% of the patients, and a maxillary retrusion was present in 37% of patients. In that time, she suggested systematic maxillofacial follow-up so that these effects may be identified. Remedial measures could include the change of the interface or reducing the daily use of NPPV.

Continuous positive airway pressure (CPAP) is a type of positive airway pressure used to deliver a set pressure to the airways maintained throughout the respiratory cycle during inspiration and expiration [8].

Bariani, in 2020, reports that most of the studies demonstrated that long-term use of nasal positive airway pressure in childhood/adolescence is associated with midface hypoplasia [9].

Ma, in 2021, tries the fit and comfort evaluation of custom mask designs using a randomized fit test with a series of three-dimensional (3D) printed versus commercial standard masks. Results indicate that custom masks are more comfortable than conventional continuous positive airway pressure (CPAP) masks, particularly on fit, contact pressure, and comfort [10].

Martelly, in 2021, also makes 3D masks (BiPAP and CPAP) and reports that while the custom-fit mask did not reduce the average measured leakage for subjects, subjects reported experiencing less leakage. Overall, results suggest that the

custom-fit masks are more comfortable and tolerable than the provided off-the-shelf (OTS) mask option [11].

#### **6. Case report—deleterious facial effects caused by noninvasive ventilation mask early treatment in congenital muscular dystrophy**

With this clinical case, we want to familiarize professionals who work with these patients about the consequences that LTNIV may have in these children's face and oral functions and the importance of a pediatric dentist or an orthodontist in the team that treats these patients.

A 5-year-old child born with congenital muscular dystrophy type 1A (MDC1A) and total absence of merosin due to laminin α2 gene (*LAMA2*) mutation was referred by the pediatrician for the first time to the dentist. The child had a motor deficit, absence of gait, muscle hypotonia, feeding difficulty, breathing difficulties, sleep apnea, and normal intellectual development.

MDC1A is a rare autosomal recessive hereditary disease [12, 13] with severe consequences for its patients. It is characterized by motor deficits, muscle hypotonia, retractions, and progressive respiratory system impairment [14]. Affected individuals may show muscle hypertrophy [15]. The use of noninvasive ventilation devices is one of the strategies to manage the disease [16]. BiPAP and other similar units function as air compressor units and are used to treat respiratory insufficiencies [17], aiding ventilation with positive results in the pediatric population [18]. In this population, these devices are often used with a mask resting on the upper jaw.

Severe maxillary retrusion was diagnosed. Looking for the cause of such a severe facial deformity, it was hypothesized that force exerted by the noninvasive ventilation mask in this particular patient (with facial and bulbar weakness since birth) is one of the reasons for the deformity. The use of devices to assist ventilation is quite frequent in these patients. The use of these auxiliary ventilation devices, which are very important for the patient's well-being, must be carefully considered as they cause maxillary hypoplasia.

The retrusion of the middle floor of the face is maxillary hypoplasia or maxillary deficiency, which is an underdevelopment of the jawbones, with the greater concavity of the face and reduced nasolabial angle. This term represents underdevelopment of the maxilla (upper jaw) in length (decrease in the average height of the face) or depth (retrusion of the jaw) [19]. The maxillary retrusion resulting in non-development creates a pseudo-class III malocclusion resulting in esthetic disharmony, greater resistance of the nasal pathways [20, 21], narrowing of the pharyngeal airway, discomfort, and problems of eruption in mixed dentition [22, 23], with maxillary compression and crowding, in addition to pseudomacroglossia [24]. That is, the benefit obtained by the best oxygenation should always be equated with the relative anatomofunctional misfit that may result from prolonged use of the mask. Surveillance of the evolution of the maxilla development by pediatric dentists is necessary, and this professional should be part of the treatment and follow-up team.

The management of this disease and its comorbidity is predominant for the patient's quality of life, considering that there is no cure for it.

In this case, the use of the noninvasive ventilation device (BiPAP) occurred from the age of 15 months in the context of rest, nocturnal use (**Figure 1**). The device is used due to the child's ventilation difficulties, trying to compensate for the fragility of the respiratory muscles.

*Deleterious Facial Effects Caused by Noninvasive Ventilation Mask Early Treatment… DOI: http://dx.doi.org/10.5772/intechopen.100161*

#### **Figure 1.**

*At 15 months, the child started using noninvasive night ventilation with a nasal mask. So that there is a good adaptation of the nasal mask, without air escaping, a strap adapts the silicone to the face, anchored in the neck or nape, easily creating a retrusive force to the jaw.*

#### **Figure 2.**

*The panoramic X-ray is the best exam to test the child and parent collaboration. For a child who does not walk and needs constant help from the mother to position himself, the radiograph is of sufficient quality and should not be repeated.*

In our consultation, and despite the difficulties arising from age and the absence of gait that makes the child always dependent on the mother, good collaboration was achieved to perform a study [25], after obtaining additional diagnostic

#### **Figure 3.**

*Teleradiography - lateral radiography of the cranium from a 5-year-old female patient with Congenital Muscle Dystrophy. Severe maxillary retrusion, crossbite, open bite, and vertical growth of the mandible.*

#### **Figure 4.**

*Extra-oral photographs—as the child cannot hold herself standing, the photos were taken in the dental chair. The mother and child were very collaborative.*

*Deleterious Facial Effects Caused by Noninvasive Ventilation Mask Early Treatment… DOI: http://dx.doi.org/10.5772/intechopen.100161*

#### **Figure 5.**

*Intra-oral photographs—the lips were previously moistened with a lip care balm to avoid the child's sensitivity and facilitate the placement of the lips and cheeks retractors.*

#### **Figure 6.**

*On 22/01/2017—the educator's photo shows retrognathic profile and lingual protrusion (Pseudomacroglossia).*

elements: orthopantomography (**Figure 2**), teleradiography (**Figure 3**), extra-oral (**Figure 4**), and intra-oral photographs (**Figure 5**), and upper and lower molds of the child's mouth.

The child does not present dental problems, all teeth being healthy.

The study allowed to suggest starting the first phase of the correction of this problem, using an orthopedic maxillary therapy, with the placement of a rapid maxillary expansion appliance to widen the maxilla, followed by the simultaneous use of a facial mask for maxillary traction. At all times, the use of noninvasive ventilation was maintained.

The child must be regularly followed, and other therapeutical measures may have to be considered with the evolution of the treatment. At this moment, the focus is to recover as soon as possible the oral physiological functions.

Severe maxillary hypoplasia with progeny disharmony greater than 1 cm with a retracted appearance of the middle face is notorious. Retrusion, anterior and posterior crossbite, open bite, vertical growth of the mandible, and pseudomacroglossia (**Figures 2**–**6**).

Concavo profile and pseudomacroglossia (**Figure 6**) are visible in the resting position of the photo taken by the educators.

Further investigation of the facial features pre-use of NIV allowed us to conclude that the face was harmonic, without any hypoplasia or retrusion, as seen from the observation of several photographs and videos pre NIV (**Figures 7** and **8**). The profile was convex, with slight dystocclusion.

**Figure 7.**

*Photo of the child on 17/11/2011, taken before using the noninvasive ventilation appliance; note the convex facial profile, without maxillary retrognathia problems.*

*Deleterious Facial Effects Caused by Noninvasive Ventilation Mask Early Treatment… DOI: http://dx.doi.org/10.5772/intechopen.100161*

#### **Figure 8.**

*Child's photograph on 10/01/2012, also taken before using the noninvasive ventilation device; note the convex face profile, with a noticeable distal occlusion of the mandible and the middle third of the face well developed and proportional.*

#### **7. Discussion**

Noninvasive mechanical ventilation (NIV) is defined as a ventilatory support that does not require an orotracheal tube or tracheostomy, used through an interface, with the objective of promoting adequate ventilation, reducing respiratory work, preventing respiratory muscle fatigue, increasing alveolar ventilation, and improving gas exchange, thus avoiding intubation and promoting, in some cases, early extubation [26, 27]. The use of NIV may also reduce complications associated with invasive mechanical ventilation and, consequently, morbidity and mortality rates related to this ventilatory support [28].

Currently, NIV is considered an alternative ventilatory support in the Pediatric Neuromuscular Outpatient Clinic. It has good acceptability and high success rates. It is indicated in the presence of acute or chronic respiratory disorders, neuromuscular diseases, central nervous system disorders, obstructive sleep apnea, neuromuscular diseases, central nervous system disorders, obstructive sleep apnea, postoperative, post-extubation period, and early extubation [29, 30].

#### *Current Trends in Orthodontics*

A certain type of patient needs to use a ventilation aid for different reasons and at different ages, sometimes in the first years of life. The use of these noninvasive ventilation masks (BiPAP, CPAP, and the like) is sometimes maintained for years and a considerable period of daily use (more than eight or even 12 hours). The masks allow patients better oxygenation, but closure must be obtained around the place where the mask touches so that there is no air leakage. This peripheral closure of the mask on the face is obtained by the traction pressure of the mask against the tissues, and the mask can be nasal, nasal and oral, or facial.

The harmful effects caused using the mask will depend on the type of mask applied, the places where the forces are applied, the intensity of the forces, their direction, and the time of action of the forces in question. If this mask does not rely on solid structures of the face causes severe deformations in patients, especially if they are young and with the skeleton to form and easily moldable. Also, the substrate on which the forces act is important because a hard bone of an adult male does not deform in the same way as the soft bone of a baby or child.

Looking for the possible causes for maxillary retrusion and hypoplasia found, the feasible one that can be related is the use of noninvasive nasal mask ventilation in a child with facial and bulbar weakness. In this clinical case, everything indicates that the deformation found was iatrogenic, caused by the masks used in noninvasive ventilation. That is one of the main reasons for the deformity due to the mask.

Using this device from an early age (15 months) exerts an anchoring force in the skull (more difficult to deform) that causes an inhibition reaction of the growth of the maxilla and its retrusion since the predominant force was exerted in the cranial calotte. In this sense, the absence of well-distributed facial supports caused the applied forces to retort the entire anterior sector.

Given what was observed, it seemed that the alternative would be to counteract all this movement. So, we associate with the retrusive force of BiPAP a protrusive force, through a facial mask, complemented by the transverse expansion of the maxillary disjunction.

Maxillary reverse traction therapy is indicated for the solution of orthodontic cases as a non-surgical alternative for correcting malocclusion, allowing movement of the maxilla forward and down through the remodeling of the maxillary sutures. At the same time, the mandible shows a clockwise rotation, which corrects the concavity of the soft tissue profile. The technique may be associated with surgical procedures and rapid maxillary expansion orthopedical movements. Better results are obtained when therapy is used in young patients when compared to older patients [31].

The technique used allowed the child to continue to use BiPAP, necessary for his health and metabolism of development, but at the same time counteract the forces exerted by the NIV.

In this case, maxillary hypoplasia was reported in a child with congenital muscular dystrophy and a total absence of merosin. These children have severe breathing problems and ventilation difficulties.

The obstruction of the nasal airways is related to their volume [32, 33]. Some authors believe that rapid maxillary expansion may result in greater ventilatory capacity [34, 35] resulting from increased permeability and nasal volume [36, 37] as well as different tongue positioning [38, 39] and changes in voice [40].

The correct anatomy, good airways permeability, and positioning of the teeth and tongue permit better oral functions, such as chewing, drinking, speaking, breathing, and even earing [41].

#### *Deleterious Facial Effects Caused by Noninvasive Ventilation Mask Early Treatment… DOI: http://dx.doi.org/10.5772/intechopen.100161*

This procedure allowed an increase of the entire nasal pyramid, resulting from the rapid expansion of the maxillary, which in the future will allow better ventilation, tongue positioning, and drainage of secretions in addition to closer anatomy of normal. It allows better development and a smaller number of hours lost by parents in medical consultations.

Despite the importance of this intervention for the child's current development, the treatment must have a second phase, pluridisciplinary, that may involve pediatric dentistry, orthodontics, physiotherapy, speech therapy, and maxillofacial surgery.

The fact that we later found other cases in which the same etiological hypothesis is possible made us write this text as a starting point for investigation and as a warning to colleagues who put on auxiliary ventilation masks such as BiPAP device, CPAP, and similar, for the treatment of apnea, sleep apnea, or situations that require prolonged use of NIV. It is intended that this text is a warning so that the placement of these masks is supported whenever possible in an extensive area and with the involvement of multiple less deformable bones, besides making it necessary to control the forces used so that these masks do not act like an orthodontic device of maxillary retrusion. Whenever possible, use only nose tubes, or helmet-like devices, to avoid jaw pressure at early ages.

In NIV a mask supported by the maxilla is used. Suppose we hypothesized that its use since the age of 15 months might have retracted the jaw or prevented its growth, functioning as an orthodontic appliance and with an unequal distribution of forces. In that case, we are faced with a method of disease management that may end up being harmful in the long term, preventing the natural formation of the sinus and the consequent ventilatory improvement. The use of this method of disease management is a "two-beak stick" because it allows an improvement in ventilatory capacity when using the device but seems to decrease the typical anatomical characteristics necessary for adequate ventilation. In this sense, noninvasive ventilation devices with an average force distribution and forces supported in more extensive areas and independent bones should be preferred to prevent retrusion. The most extensive support surface can prevent retrusion by what would be ideal the realization of masks tailored to each face.

#### **8. Patient perspective**

The mother left a testimony, translated to English, after removing names:

Sharing: I, mother and primary caregiver, hereby share this testimony, with the greatest satisfaction of what this device gave to my child's mouth, as she has severe congenital muscular dystrophy, a total absence of merosin (strength protein).

As soon as the appliance was placed in her mouth, within days, there was massive notoriety in her teeth spacing, mouth opening, and "ogival" palate alteration (widening and less depth), to the point where the teeth fit (aligned) correctly, "teeth with teeth."

Something that had not happened before and that made chewing less effective, such as lack of muscle strength, which was another situation that made the situation worse.

After this situation was successful, we moved on to the next stage with the traction device. It rectifies the bite, which was misaligned, due to the BiPAP respiratory device for sleep apnea at a very early age. Bipap had a beneficial effect on oxygenation while sleeping, but at the jaw level, projected it backward.

The process was slower but managed to exceed expectations to the extent of the severity noted. Through this long but very advantageous process, the changes were notorious at all levels:


However, this could not happen if I had not chosen as an Orthodontist Prof. Doctor Casimiro de Andrade and his team in this very complex process. In this, there was a development of delivery, trust, and humanity par excellence, to which I fully thank.

He was gradually doing the treatment according to the child's condition, in the different phases, so that this process would not be invasive under any circumstances.

Moreover, the entire multidisciplinary team was delighted to see my daughter evolve and progress throughout her treatment (Dr. Lurdes Morais (pulmonologist), Dr. Julia Eça de Guimarães (pediatrician), and Physiotherapist Ana Moreira and her team.

Mother of the child. Grateful for everything.!!! Thank you all.

#### **9. Informed consent**

The patient responsible agreed to use and publish the disease-related article with personal information to be excluded.

#### **10. Conclusions**

Whenever VNI use is prolonged and at an early age, exclusively maxillary support should be avoided, if possible discarded, preferring the use of a mask with multi-site facial support, or in some instances, total and better-distributed loads, if necessary tailored to measure.

Pediatric dentistry and orthodontics play a vital role in promoting health and development in children with neuromuscular disorders.

A pediatric dentist is a vital element in the medical team that follows these patients and should always identify, prevent, and intercept these problems to achieve proper development.

#### **Acknowledgements**

We thank the fantastic collaboration of all the therapeutic teams that followed the child.

We thank the mother's child as an example of care and attention to her child.

*Deleterious Facial Effects Caused by Noninvasive Ventilation Mask Early Treatment… DOI: http://dx.doi.org/10.5772/intechopen.100161*

#### **Conflict of interest**

The authors supported this investigation.

The authors declare no conflict of interest.

David Casimiro de Andrade, Maria João Palha e José Rui Andrade: responsible for the conception and design.

David Casimiro de Andrade, Maria João Palha, Viviana Macho and Ana Norton were responsible for the data collection and manuscript redaction.

Ana Norton, Viviana Macho were responsible for the critical revision of its contents.

Sandra Kalil Bussadori, Miguel Palha, Lurdes Morais and Manuela Santos were responsible for the critical revision of its intellectual contents.

David Casimiro de Andrade was responsible for graphics and photos, the critical revision of its intellectual contents, and the final approval of the version to be published.

All authors declare that written informed consent was obtained from the patient (or other approved parties) to publish this research paper.

### **Author details**

David Andrade1 \*, Maria-João Palha<sup>2</sup> , Ana Norton1 , Viviana Macho3 , Rui Andrade5 , Miguel Palha4 , Sandra Bussadori6 , Lurdes Morais7 and Manuela Santos8

1 Pediatric Dentistry, Faculty of Dental Medicine, University of Porto, Porto, Portugal

2 Pediatrics, Santa Maria Hospital, Centro Hospitalar Universitário Lisboa Norte, Lisboa, Portugal

3 Pediatric Dentistry, Faculty of Health Sciences, University Fernando Pessoa, Porto, Portugal

4 Developmental Pediatrician, Child Development Centre Diferenças, Lisbon, Portugal

5 Nutritionism, Clínica Dentária de Espinho, Espinho, Portugal

6 Pediatric Dentistry, Uninove, Campus Vila Prudente, São Paulo, Brazil

7 Pediatric Pneumology, North Maternal and Child Centre from Centro Hospitalar do Porto, Porto, Portugal

8 Neuropediatrics, North Maternal and Child Centre from Centro Hospitalar do Porto, Porto, Portugal

\*Address all correspondence to: dandrade@fmd.up.pt

© 2021 The Author(s). Licensee IntechOpen. This chapter is distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

*Deleterious Facial Effects Caused by Noninvasive Ventilation Mask Early Treatment… DOI: http://dx.doi.org/10.5772/intechopen.100161*

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