Abstract
Aim:
The main purpose was to evaluate the effect of complete denture rehabilitation on the jaw growth pattern in individuals with ectodermal dysplasia from an early age to maturity.
Settings and Design:
This was a prospective in vivo study performed in the Department of Prosthodontics, King George Medical University, Lucknow, India.
Materials and Methods:
Rehabilitation with three sets of conventional complete dentures was completed in an ectodermal dysplasia case at the age of 5, 10, and 17 years. Cephalometric and diagnostic cast analyses were the methods performed to evaluate jaw growth patterns. Linear and angular measurements obtained after denture rehabilitation were averaged and compared with mean standard values of nearly corresponding ages, as given by Sakamoto and Bolton. Conversely, alveolar ridge arch width and length were evaluated for their dimensional changes during the same age intervals.
Statistical Analysis Used:
Mann–Whitney U-test was used to check the difference between the groups. The significance of the level adopted was 5%.
Results:
Nasion-anterior nasal spine, anterior nasal spine-menton, anterior nasal spine-pterygomaxillary fissure, gonion-sella, and gonion-menton lengths were found to be not statistically significant than the mean standard values of nearly corresponding ages (P > 0.05). The decrease in facial plane angle, increase in Y-axis angle, and mandibular plane angle after complete denture rehabilitation were statistically significant when compared to their mean standard values (P < 0.05). Cast analysis showed more increase in the length compared to the width in both arches.
Conclusion:
Complete denture rehabilitation did not significantly affect the jaw growth pattern, although it improved facial esthetics and masticatory activity by establishing adequate vertical dimensions.
Keywords: Cast analysis, cephalometric analysis, complete denture, ectodermal dysplasia
INTRODUCTION
Ectodermal dysplasia syndrome (EDS) is an inherited type of disease impacting several structures of ectodermal origin, resulting in an immense and vast number of disturbances.[1] Approximately 150 different ectodermal dysplasia (ED) clinical types can be described, with chances of each case appearing per 10,000–100,000 birth.[2] A few mild type EDS variants exist, whereas vast of them can be detrimental. While female carriers exceed affected males, there are little or no signs seen in female carriers as the transmission usually occurs through a sex-linked recessive gene leading to predominant expression in males.[3] Based on the number and function of sweat glands, ED can be classified into two broad types: hidrotic ED (normal sweat glands) and hypohidrotic/anhidrotic ED (sparse/absent sweat glands).[4,5] Hypohidrotic ED is the most frequently occurring type and its characteristic features include hypotrichosis, hypodontia or anodontia, and hypohidrosis.[6,7]
ED children remain a challenge for a dentist toward their dental rehabilitation, including the patient’s self-confidence, attitude, and peer group interaction that, if intervened, can show significant signs of improvement.[8] Cephalometric studies have shown that dento-maxillary features such as hypoplasia and maxillary retrusion, counterclockwise rotation and mandibular protrusion, and reduced facial and ramus heights were common among EDs patients.[9] Therefore, clinical measures must be taken to intercept and correct these functional deformities at appropriate times. In complete anodontia cases, oral function and esthetics are restored using complete dentures. Very few follow-up studies on oral rehabilitation of ED cases have been discussed, and moreover, there seem to be scant data to interpret the results of dentures on jaw growth and development.[10,11,12,13,14] The current study aimed to evaluate the effect of complete denture rehabilitation on the jaw growth pattern in individuals with ED from an early age to maturity. The null hypothesis was that complete denture rehabilitation would affect the jaw growth pattern of completely edentulous ED cases.
MATERIALS AND METHODS
This study was performed in the Department of Prosthodontics, King George Medical University, Lucknow, to evaluate the consequences of complete denture treatment on craniofacial growth in hereditary ectodermal dysplasia (HED) cases. A completely edentulous hereditary ED case was followed from 5 to 17 years of age.[15,16] Classic characteristics of HED were displayed at the time of presentation, which included anodontia, prominent forehead, saddle nose, and hypotrichosis [Figure 1a]. A noncontributory family history was detected as both parents and siblings had no alike features. Radiographic examination disclosed complete deprivation of teeth and tooth buds [Figure 2]. Patient consent was obtained before the study, and ethical standards were followed accordingly.
Figure 1.
Preoperative (a) and postoperative (b) facial frontal view
Figure 2.
Orthopantomogram view
The three sets of complete dentures were constructed for the patient when he was at the age of 5 years, 10 years, and 17 years, respectively [Figure 3]. At all ages, the complete denture fabrication method was the same and mainly consisted of a selection of age-appropriate teeth sets, as discussed in detail in the previous publications.[15,16] Maxillary and mandibular impressions were taken following the selective pressure impression technique. Type IV dental stone (Kalrock; Kalabhai Karson Pvt. Ltd., Mumbai, India) was used to pour the impression to construct master casts. Face-bow transfer and mounting procedures were done on a semi-adjustable articulator (Hanau™ Wide-Vue; Whip Mix Corporation, Louisville, USA). Physiological rest position using phonetic and esthetic techniques was applied to establish the occlusal vertical dimension. The centric relation was established according to the dynamic records formed by unforced jaw movements in the terminal hinge position accomplished by the patient and manually guided. The denture was processed in centric occlusion with balanced articulation using anatomically structured age-appropriate acrylic teeth, finished, and polished [Figure 1b]. After denture insertion, postoperative instructions were given to the patient.
Figure 3.
Three sets of complete dentures at the age of 5 (a), 10 (b), and 17 (c) years
The future recall was scheduled at all years of follow-up to check for the requirement of necessary relining or construction of a new denture. The recurring occlusal changes noticed were loss of posterior tooth contact and anterior tooth contact occurrence, which was subsequently corrected by the addition of self-cure acrylic resin (Trevalon, Dentsply Ltd., Gurgaon, India) to the posterior denture teeth’s occlusal surface.
Analysis of jaw growth
Cephalometric and diagnostic cast analyses were the methods used to evaluate jaw growth. Assessments were made on diagnostic casts and lateral cephalograms taken at the ages of 5, 10, and 17 years to evaluate jaw growth [Figure 4].
Figure 4.
Lateral cephalograms at the age of 5 (a), 10 (b), and 17 (c) years
Measurements for size alterations in alveolar arch length and width were made on diagnostic casts. A segmental method was applied to calculate the arch length and width, where four reference points divided each side of the arch into three sections. The reference points taken were as follows: arch midline (a), the canine eminence (b), the hamular notch for the maxillary arch or posterior extent of the retromolar pad for the mandibular arch (d), and an equidistant point from point b and point d (c) [Figures 5 and 6].
Figure 5.
Diagnostic cast analysis of maxillary arch at age of 5 (a), 10 (b) and 17 (c and d) years
Figure 6.
Diagnostic cast analysis of mandibular arch at age of 5 (a), 10 (b) and 17 (c and d) years
Angular measurements such as facial angle, Y-axis, and mandibular plane angle on lateral cephalograms were analyzed to differentiate the positional association of craniofacial landmarks, while linear assessments such as nasion-anterior nasal spine (N-ANS), anterior nasal spine-menton (ANS-ME), anterior nasal spine-pterygomaxillary fissure (ANS-PTM), gonion-sella (GO-S), and gonion-menton (GO-ME) were measured for dimensions. Derived cephalometric values were collated, averaged, and compared with the mean standard values of nearly corresponding ages, as given by Sakamoto and Bolton standards[17] [Tables 1 and 2].
Table 1.
Cephalometric analysis of the patient at 5, 10, and 17 years of age and values compared with the average values reported by Sakamoto T 195917]
| Parameters | Patient values | Average values | |||||
|---|---|---|---|---|---|---|---|
|
|
|
||||||
| 5 years (with dentures) | 10 years (with dentures) | 17 years (without denture) | 17 years (with denture) | 5 years | 10 years | 17 years | |
| Angle (°) | |||||||
| Facial angle | 92.2 | 85.2 | 100 | 92 | 81.1 | 83.1 | 84.1 |
| Y-axis | 48.6 | 59.6 | 44 | 54 | 65.7 | 65.2 | 66 |
| Mandibular plane angle | 13 | 13.5 | 5 | 14 | 32.8 | 31.6 | 32 |
| Length (mm) | |||||||
| N-ANS | 46.1 | 50.2 | 52.1 | 52.1 | 43.5 | 50.6 | 55.7 |
| ANS-ME | 52.5 | 61.8 | 42.1 | 64.3 | 58.4 | 61.1 | 69.7 |
| ANS-PTM | 40.3 | 44.1 | 54.0 | 54.0 | 49.9 | 51.8 | 57.2 |
| GO-S | 60.2 | 70.3 | 74.3 | 76.2 | 62.3 | 71.8 | 80.6 |
| GO-ME | 58.7 | 67.7 | 65.0 | 65.0 | 55.5 | 65.5 | 72.7 |
| Ratio of lengths (%) | |||||||
| N-ANS/ANS-ME | 87.8 | 81.2 | 123.8 | 81.0 | 74.4 | 82.8 | 79.9 |
| ANS-ME/GO-S | 87.2 | 87.9 | 56.7 | 84.4 | 93.7 | 85.1 | 86.4 |
N-ANS: Nasion-anterior nasal spine, ANS-ME: Anterior nasal spine-menton, GO-S: Gonion-sella, GO-ME: Gonion-menton, ANS-PTM: Anterior nasal spine-pterygomaxillary fissure
Table 2.
Cephalometric analysis of the patient at 5, 10, and 17 years of age and values compared with the skeletal values as derived from the Bolton standards for age and sex
| Length (mm) | Patient values | Standard values | |||||
|---|---|---|---|---|---|---|---|
|
|
|
||||||
| 5 years (with dentures) | 10 years (with dentures) | 17 years (without dentures) | 17 years (with dentures) | 5 years | 10 years | 17 years | |
| Co-point A (maxillary length) | 73.4 | 77.1 | 90.0 | 90.0 | 81.7 | 87.7 | 98.9 |
| Co-Gn (mandibular length) | 95.2 | 100.2 | 110.1 | 110.1 | 99.3 | 107.7 | 126.8 |
Co-Point A: Condylion-Point A, Co-Gn: Condylion-gnathion
All collected data were tabulated and analyzed with software (IBM SPSS Statistics for Windows, Version 25.0. Armonk, NY: IBM Corp.). Linear and angular measurements of cephalometric analysis were represented as mean ± standard deviation and compared with mean standard values using Mann–Whitney U-test. A two-tailed (α = 2) P < 0.05 was considered statistically significant.
RESULTS
Lateral cephalograms showed a continuous increase in length of N-ANS, ANS-ME, ANS-PTM, and Go-S when measured after complete denture rehabilitation at 5, 10, and 17 years of age, revealing continuous growth but remaining posterior to standard values. In comparison, the mean N-ANS, ANS-ME, ANS-PTM, Go-S, and Go-ME were not statistically significant than the mean standard values of nearly corresponding ages (P > 0.05). There were a decrease in facial plane angle and an increase in Y-axis angle and mandibular plane angle after complete denture rehabilitation at 17 years of age. The mean values of facial plane angle (89.80 ± 3.98 vs. 82.76 ± 1.52, P = 0.04), Y-axis angle (54.06 ± 5.50 vs. 65.63 ± 0.40, P = 0.02), and mandibular plane angle (13.50 ± 0.50 vs. 32.13 ± 0.61, P < 0.01) after complete denture rehabilitation were found to be statistically significant when compared to their mean standard values (P < 0.05) [Table 3].
Table 3.
Cephalometric values of the patient at all ages compared with the standard mean values using Mann–Whitney U-test
| Parameters | Means | Normal standard | Mann–Whitney U-test | P |
|---|---|---|---|---|
| Facial angle | 89.80±3.98 | 82.76±1.52 | 0.00 | 0.04 |
| Y-axis | 54.06±5.50 | 65.63±0.40 | 0.00 | 0.02 |
| Mandibular plane angle | 13.50±0.50 | 32.13±0.61 | 0.00 | <0.01 |
| N-ANS | 49.46±3.06 | 49.93±6.13 | 4.00 | 0.91 |
| ANS-ME | 59.53±6.21 | 63.06±5.90 | 4.00 | 0.51 |
| ANS-PTM | 46.13±7.07 | 52.96±3.78 | 2.00 | 0.21 |
| GO-S | 68.90±8.09 | 71.56±9.15 | 3.00 | 0.72 |
| GO-ME | 63.80±4.61 | 64.56±8.63 | 4.00 | 0.89 |
| N-ANS/ANS-ME | 86.50±10.65 | 88.40±4.63 | 4.00 | 0.54 |
| ANS-ME/GO-S | 83.33±3.86 | 79.03±4.26 | 2.00 | 0.26 |
| Co-point A (maxillary length) | 80.16±8.71 | 89.43±8.73 | 2.00 | 0.26 |
| Co-Gn (mandibular length) | 101.83±7.58 | 111.26±14.09 | 3.00 | 0.36 |
N-ANS: Nasion-anterior nasal spine, ANS-ME: Anterior nasal spine-menton, GO-S: Gonion-sella, GO-ME: Gonion-menton, ANS-PTM: Anterior nasal spine-pterygomaxillary fissure, Co-point A: Condylion-point A, Co-Gn: Condylion-gnathion
The ratio of maxillary facial height to mandibular facial height (N-ANS/ANS-ME ratio) after complete denture rehabilitation at 5, 10, and 17 years of age was more than the standard values of nearly corresponding ages, denoting a decreased mandibular facial height, hence showing reduced Y-axis and mandibular plane angle in comparison to standard values. The mean value of the N-ANS/ANS-ME ratio (86.50 ± 10.65 vs. 88.40 ± 4.63, P = 0.54) was statistically insignificant to the mean standard values of nearly corresponding ages (P > 0.05) [Table 3]. The measured maxillary length (Co-Point A) and mandibular length (Co-Gn) showed a maxillary length increase of 16.6 mm and a mandibular length increase of 14.9 mm from 5 years to 17 years of age, but their mean values did not differ statistically when compared to their mean standard values (P > 0.05).
Maxillary cast analysis showed an overall increase in arch length of 26 mm and arch width of 11 mm in the anterior section, 7 mm in the middle section, and 13 mm in the posterior section from 5 to 17 years of age. Similarly, the mandibular cast analysis revealed an overall increase in arch length of 35 mm and arch width of 10 mm in the anterior section, 8 mm in the middle section, and 9 mm in the posterior section from age 5–17 years.
DISCUSSION
The present study showed comparable linear measurements of cephalometric analysis after complete denture rehabilitation in the case of ED with mean standard values of nearly corresponding ages. Although improvement was observed in mandibular plane angle after denture rehabilitation and raised vertical dimension, however, lower values were noticed when compared to standard values of nearly corresponding ages indicating a reduced lower facial height. Hence, complete denture rehabilitation did not significantly affect the jaw growth pattern, although it improved the facial esthetics by establishing adequate vertical dimensions, therefore rejecting the null hypothesis of the study. The outcome of our study favored the interpretations of Sarnat et al.[10] and Ochiai et al.,[18] stating that the absence of teeth did not affect jaw growth, and additionally, the denture flange perhaps did not hinder jaw growth but instead enhanced the masticatory activity by imparting better denture stability and retention.
A tissue-supported removable denture is a reliable treatment option for an ED patient with respect to age and growth of the alveolar bone. The insertion of endosseous implants at an early age, before 18 years of age, can cause implant submergence, as the growth of jaws continues, which consequently increases the risk of implant failure.[19] Therefore, a removable complete denture was chosen as a treatment option in this study. Clinically, vertical dimension was determined during denture construction using physiologic rest position and verified with the aid of facial support, esthetics, and phonetics. The established vertical dimension, besides preventing the development of class 3 malocclusion, also improved the child’s skeletal relationship during his growth period.
The periodic recall examination was scheduled at 6 months to make necessary adjustments implying its importance for addressing the growth changes arising in a child. As reported by Shaw,[20] denture underextension and posterior open bite were constant setbacks associated with jaw growth. The posterior contact absence was controlled by the addition of self-cure acrylic resin to the posterior denture teeth’s occlusal surface, and subsequently, posterior contact was evaluated using standard means of articulating paper. We thus agree that the application of autopolymerizing acrylic resin and acrylic teeth are satisfactory choices for managing growth alterations in such patients.
During the duration of 5–17 years of age, forward growth was markedly limited at the ANS point as observed on cephalogram, which was also reported by Sarnat et al.[10] A minor deflection from average was noticed in the anteroposterior mandibular body length and ramus height. A 16.6 mm maxillary length increase and a 14.9 mm mandibular length increase were detected. Therefore, more growth was perceived in the maxilla than the mandible, although both were almost at par. These growth changes might occur as a consequence of the presence of complete dentures during the period of growth; however, confirmation is inadequate with the present study design. Hamano and Nakata[21] studied anhidrotic ED cases with anodontia and agreed upon the fact that interaction between the lack of teeth and the atrophic rhinitis was the cause of deficient forward growth of the maxilla. Tocchini et al.[22] in their anodontia study found a reduced maxillary forward growth, however, they were unable to distinguish whether it was due to teeth absence or prosthetic replacement.
In this study, the ratio of maxillary facial height to mandibular facial height (N-ANS/ANS-ME) values after complete denture rehabilitation was more than the standard values of nearly corresponding ages, denoting a decreased mandibular facial height, hence showing reduced Y-axis and mandibular plane angle in comparison to standard values. N-ANS/ANS-ME value was noticed to be lesser with complete dentures than without dentures because of the establishment of an adequate vertical dimension. Similar results were reported by Sarnat et al.[10] who studied the craniofacial growth in various anodontia cases using cephalometric measurements, portraying the facial structure and jaw growth as placed close to the lower limit of normal. Furthermore, the presence of dentures in our study allowed a backward, downward rotation of the mandible, as also proved by Franchi et al.[23] in their early prosthetic rehabilitation for complete anodontia case of ED.
The observations of our study detected an increased arch length compared to the arch width. Studies undertaken individually by Shirakawa et al.[24] and Tocchini et al.[22] also supported our results. Shirakawa et al.[24] evaluated the study casts and measured the alveolar ridge arch for 2 years. They reported a maxillary arch length increase of 5 mm and arch width increase of 6 mm whereas a mandibular arch length increase of 12 mm and arch width increase of 6 mm. The results of Shirakawa et al.[24] therefore proved an increased mandibular arch length compared to arch width while there was an overall increase in the dimensions of the maxillary arch. Cast analysis evidently supported the increase in the size of maxillary and mandibular dental arches, as also shown by de Castro et al.[12]
This was a long-term, preliminary study but was limited to comparatively analyzing the growth pattern of an ED case from early life to maturity. However, a multicentric, long-term study with a higher sample size is required to elevate the authenticity of data.
CONCLUSION
Within the limitations of the study, it was concluded that the lack of teeth in ED cases did not influence the jaw growth pattern following complete denture rehabilitation, although it improved the masticatory function, speech, and facial esthetics by establishing the adequate vertical dimension.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
Acknowledgment
We thank the Journal of Indian Prosthodontic Society and the Journal of Prosthodontics for contributing to our previous publications in their esteemed journals.
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