CRANIOFACIAL MORPHOLOGY AND SLEEP DISORDERED BREATHING

IN CHILDREN

by

Reza Aran

DMD, The University of British Columbia, 2007

A THESIS SUBMITTED IN PARTIAL FULFILLMENT OF

THE REQUIREMENTS FOR THE DEGREE OF

MASTER OF SCIENCE

in

THE FACULTY OF GRADUATE STUDIES

(Craniofacial Science)

THE UNIVERSITY OF BRITISH COLUMBIA

(Vancouver)

April 2013

© Reza Aran, 2013

Abstract

Objective: The aim of this study is to understand how craniofacial morphology and the

severity of a malocclusion can contribute to sleep disordered breathing (SDB) symptoms

in children when controlled for age, gender and body mass index (BMI).

Methods: A total of 301 subjects with complete records were included in this study.

Two hundred and thirty-seven were preadolescents, of which 97 were male and 140 were

female (mean age 9.9±1.6); 64 were adolescents, of which 24 were male and 40 were

female (mean age 13.8±0.9). All the subjects’ parents were asked to complete a SDB

questionnaire. Lateral cephalometric images were analyzed to assess the position of the

hyoid bone, length of the soft palate, and the maxillary and mandibular dental and

skeletal relationship. A clinical examination was performed to determine the Angle

classification, Mallampati score, tonsil size (Brodesky), and BMI.

Results: Data from 301 children that completed the questionnaires and underwent a

cephalometric analysis were evaluated. Subjects were divided into two groups based on

their age and each group was further divided based on gender. By comparing

preadolescents with adolescents we found that preadolescents presented a significantly

higher incidence of hyperactivity, morning headaches, more frequent snoring, and

bedwetting. Adolescents exhibited significantly higher daytime sleepiness, difficulty

getting up, and impaired daytime function. When comparing female and male subjects,

we found that frequent snoring, and morning headaches were more prevalent among

females, while daytime sleepiness, and hyperactivity were more common among males.

  ii  
Craniofacial features that have a significant relationship with SDB symptoms are, a lower

position of the hyoid bone, retruded mandible, steeper mandibular plane angle, and

retroclined lower incisors. There was no statistically significant relationship between

Angle classifications, tonsil size, Mallampati score, and BMI with SDB symptoms in this

sample.

Conclusion: This study suggests that craniofacial morphology, but not severity of

malocclusions, could be a potential contributing factor to SDB symptom severity.

  iii  
Preface

The research topic of this project was suggested by Dr. Alan Lowe and the research

question was identified and the project designed by Reza Aran under the guidance of Drs.

Alan Lowe and Fernanda Almeida. The data was collected and analyzed by Reza Aran.

Reza Aran prepared the Manuscript with content editing by Drs. Alan Lowe and

Fernanda Almeida, and copy editing by Clare Davies.

The study was approved by the University of British Columbia office of Research

Services, Humans Research Ethics Board (Certificate Number: H12-00024).

  iv  
Table of Contents

Abstract.............................................................................................................................. ii  
Preface ............................................................................................................................... iv  
Table of Contents .............................................................................................................. v  
List of Tables ................................................................................................................... vii  
List of Figures................................................................................................................... ix  
List of Abbreviations ........................................................................................................ x  
Acknowledgements .......................................................................................................... xi  
Dedication ....................................................................................................................... xiii  
Chapter 1: Introduction .................................................................................................. 1  
1.1      Sleep  Disordered  Breathing  (SDB)  .....................................................................................  1  
1.2      SDB  Prevalence  .........................................................................................................................  1  
1.3      Pathophysiology  .......................................................................................................................  2  
1.3.1      Three  Major  Contributing  Factors  to  SDB  ...............................................................................  2  
1.4      Cephalometric  Characteristics  of  Patients  with  SDB  ...................................................  4  
1.5      Diagnosis  .....................................................................................................................................  8  
1.6      Treatment  of  SDB  .....................................................................................................................  9  
1.6.1      Continuous  Positive  Airway  Pressure  (CPAP)  and  Positive  Airway  Pressure  
(PAP)  .....................................................................................................................................................................  9  
1.6.2      Adenotonsilectomy  .........................................................................................................................  10  
1.6.3      Orthodontic  Treatment  ................................................................................................................  11  
1.7      Age  and  Gender  Differences  in  SDB  ................................................................................  15  
1.8      Objectives  .................................................................................................................................  16  
1.9      Hypothesis  ...............................................................................................................................  16  
Chapter 2: Materials and Methods ............................................................................... 18  
2.1      Subjects  .....................................................................................................................................  18  
2.2      Clinical  Findings  ....................................................................................................................  19  
2.2.1      Mallampati  Scores  and  Tonsil  Size  ..........................................................................................  19  
2.2.2      Angle  Classification  ........................................................................................................................  22  
2.3      Questionnaires  .......................................................................................................................  24  
2.4      Cephalometric  Analysis  .......................................................................................................  26  
2.5      Method  Error  ..........................................................................................................................  28  
2.6      Statistical  Analysis  ................................................................................................................  28  
Chapter 3: Results........................................................................................................... 30  
3.1      Subjects  .....................................................................................................................................  30  
3.2      Clinical  Findings  ....................................................................................................................  31  
3.2.1      Mallampati  Scores  and  Tonsils  Size  ........................................................................................  31  
3.2.2      Body  Mass  Index  ..............................................................................................................................  33  
3.2.3      Angle  Classification  ........................................................................................................................  34  
3.3      Questionnaires  .......................................................................................................................  34  
3.3.1      OSA-­‐18  Questionnaire  ...................................................................................................................  38  
3.3.2      PSQ-­‐22  Questionnaire  ...................................................................................................................  40  
3.4      Cephalometric  Analysis  .......................................................................................................  43  
3.5      Correlation  Between  Cephalometric  Variables  and  SDB  Symptoms  ...................  45  
Chapter 4: Discussion ..................................................................................................... 49  
Chapter 5: Conclusion .................................................................................................... 60  

  v  
References ........................................................................................................................ 61  
Appendix .......................................................................................................................... 68  
Appendix  A  .......................................................................................................................................  68  
A.1      OSA-­‐18  Questionnaire  ......................................................................................................................  69  
A.  2    PSQ-­‐22  Questionnaire  ......................................................................................................................  70  
 

   

  vi  
List of Tables

 
Table 1 Demographic data and questionnaire scores (mean)  ..................................................  30  

Table 2 Significant differences between age and gender in Mallampati scores  ................  32  

Table 3 Body mass index distribution among the four groups based on age and gender

 ...............................................................................................................................................................  33  

Table 4 Angle classification  ................................................................................................................  34  

Table 5 OSA-18 Percentages of individual item responses  .....................................................  35  

Table 6 PSQ-22 Percentages of individual item responses  ......................................................  37  

Table 7 Most common SDB symptoms among the children in our sample according to

responses given to the OSA-18 and PSQ-22 questionnaires  ............................................  38  

Table 8 Difference between PreAd and Ad in percentages  ......................................................  39  

Table 9 Differences between PreAd males and Ad males in percentages  ...........................  39  

Table 10 Differences between PreAd females and Ad females in percentages  .................  39  

Table 11 Percentage of subjects who chose answered "YES" to the SDB Symptoms and

the differences between preadolescents and adolescents  ...................................................  40  

Table 12 The percentage of male, female and total sample who answered "YES" to the

following SDB symptoms in the PS22 questionnaire and significant P-Value  .........  41  

Table 13 A summary of differences between two age groups according to the OSA-18

and PSQ-22 questionnaire  ............................................................................................................  42  

Table 14 Cephalometric analyses with mean and standard deviation for the whole

sample and both PreAd and Ad  ..................................................................................................  44  

Table 15 Corrections between domains and cephalometric variables  ..................................  45  

  vii  
Table 16 A summary of important craniofacial features that have significant correlation

with SDB symptoms  ......................................................................................................................  46  

Table 17 A summary of SDB symptoms which are correlated with each individual

cephalometric variable  ...................................................................................................................  47  

Table 18 Comparison between males and females of aged 13-16  .........................................  48  

  viii  
List of Figures

 
Figure 1 A summary of important craniofacial features and the supporting literature  ......  7  

Figure 2 Mallapati Airway Classification (I-V Scores).  ............................................................  20  

Figure 3 Brodsky Classification (0-4 Grades)  ..............................................................................  21  

Figure 4 Orhtodontic models and intraoral photos were used to determine Angle

classification  .....................................................................................................................................  23  

Figure 5 Cephalometric landmarks, and linear and angular cephalometric variables.  ....  27  

Figure 6 Percentage of children with different Mallampati scores and the differences

between PreAd and Ad groups  ...................................................................................................  31  

Figure 7 Percentage of female patients with different Mallampati scores and the

differences between PreAd females and Ad females  ..........................................................  32  

Figure 8 Classification of patients according to their age and gender who had SDB

symptoms according to responses given to the PSQ-22  ....................................................  42  

  ix  
List of Abbreviations

Ad: adolescent

AHI: apnea hypopnea index

BMI: body mass index

CPAP: continuous positive air pressure

MPAP: mean pulmonary artery pressure

OA: oral appliances

OSA: obstructive sleep apnea

OSAS: obstructive sleep apnea

PAP: positive air pressure

PreAd: preadolescent

PSQ: pediatric sleep questionnaire

RME: rapid maxillary expansion

SDB: sleep disordered breathing  

  x  
Acknowledgements

I would like to acknowledge Dr. Edwin Yen, Dr. Alan Lowe, Dr. David Kennedy and the

other members of the Graduate Admission Committee for accepting me into the UBC

Graduate Orthodontic Program. I am so grateful to be a part of such an incredible diverse

program with such great mentors. Specializing in orthodontics has been a dream and

long-term goal for me and I am extremely thankful for the opportunity that I was given.

Dr. Yen, your guidance, continued support and wisdom have been a source of inspiration.

Research was something undiscovered for me until I started my Master’s project. This

was the time that I received research training and I have learned new things and

experienced more enthusiasm for research over time. These could not happen without

supervision of wonderful, erudite mentors such as Drs. Alan Lowe and Fernanda

Almeida. I would like to express my deepest gratitude to Dr. Alan Lowe for supervising

my project and giving me such a great insight into the research world of Sleep Disordered

Breathing. I am so thankful to Dr. Fernanda Almeida in her co-supervisor role for my

project and for her kindness, patience and encouragement, and for teaching me how to be

a good researcher. Every time that I meet with you for your feedback, you have been so

welcoming and gave me a wonderful energy and a desire to learn and achieve a great

improvement. Also, I want to thank Dr. Benjamin Pliska for his participation in my

committee, as well as his outstanding clinical knowledge and skills that he generously

shared with me in my everyday learning and orthodontic clinics. I want to thank Dr. Hui

Chen for her participation in my committee, and for being so positive and encouraging

every time I saw her in clinics or seminars.

  xi  
I want to thank Dr. Kazutomo Yagi for his great help through out this project. I have

learned a lot from you. You are an exceptional and incredible scholar. And, of course,

two wonderful people that I would never forget for their great help and kindness are

Mary Wong for the data base and statistical analyses and Sandy Harrison for patient data

collection. Thank you to Gail Furlong and Mitchel Wong for helping me to collect the

data for my project. I would like to thank Clare Davies, for helping me with copy editing

and without whose help I would not have been able to complete the task in a timely way.

Lastly, my fellow residents or so called “Dream Team”, I will miss every moment that

we have spent together, from literature reviews to the clinic sessions and to the social

events. We have had a wonderful and unforgettable three years together and I wish you

the most beautiful, successful and peaceful years ahead.

  xii  
Dedication

There is only one person that I can truly dedicate this to and that is my incredible wife,

Katayoun, for her unconditional love and support not only within these past three years,

but also throughout all the years of our being together. There have been many others who

have helped and guided me along the journey of my life and career, and I am forever

grateful for to all of those who have been there to support me. However, I would not be

the person who I am today, nor would I be at this place today, without the persistent

support, incredible caring, friendship, kindness, constancy, and deep true love that you,

Katayoun have given me.

I dedicate this to you as a symbol of my deep gratitude and love and look forward to

spending the rest of my life making every moment with you and our lovely children as

happy and exciting as possible, as that is what you really deserve.

  xiii  
Chapter 1: Introduction

1.1 Sleep Disordered Breathing (SDB)

Sleep disordered breathing (SDB) is a general term for several chronic conditions such as

habitual snoring, hypopnea, and the more severe condition known as obstructive sleep

apnea (OSA).1, 2 Over 20% of children have some signs of SDB such as loud snoring,

mouth breathing, difficulty wakening, excessive daytime sleepiness and behavioral

changes such as hyperactivity.3, 4 SDB is also known to be an important contributing

factor to the problematic behaviors observed in children who suffer from sleep

deprivation.5, 6 Other clinical symptoms include failure to thrive,7, 8 hypertension,9

cardiovascular failure,10 and frequent upper airway infections.3, 11, 12 Additionally, Ross

et al found that children with SDB have about a 3.6 times higher chance of having severe

asthma.13 In summary, SDB children are at a higher risk of metabolic, cardiovascular,

and neurobehavioral morbidity.5, 14-19

1.2 SDB Prevalence

Schechter found the prevalence of snoring and OSA among children to be 3.2%-12.1%

and 0.7%-10.3% respectively.14 Bixler et al, 2009 investigated the prevalence and risk

factors of SDB in a general population. They found that the prevalence of moderate SDB

was 1.2% and the two main risk factors were BMI and nasal abnormalities.11 In a

technical report by Marcus et al, the prevalence of habitual snoring was reported to be

1.5% to 27.6 % and the prevalence of OSA was reported to be 1.2% to 5.7%.20 An adult

  1  
with an apnea hypopnea index (AHI) of more than five events per hour is considered to

have OSA, however a child with an AHI of 1 to 2 is considered to have OSA.20, 21

Metabolic and inflammatory factors may play an important role in whether a child

develops SDB in adulthood.11 Li et al studied 20,152 children with habitual snoring and

found a prevalence of 12% with 14.5% for males and 9.5% for females. Children with

SDB had three times more behavioral problems and neurocognitive abnormalities than

those without.14 The prevalence of hyperactivity behaviors is higher in younger children

with SDB.22

1.3 Pathophysiology

For a better understanding, proper diagnosis and accurate treatment, one needs to know

the pathophysiology and contributing factors of SDB. The etiology of OSA syndrome is

different in adults compared with children. In adults, obesity is the main etiological

factor, while in children, adenotonsilar hypertrophy appears to be the main factor.20 In

addition to adenotonsilar hypertrophy, two other main contributing factors to OSA

syndrome in children are obesity and alterations of the craniofacial morphology.3, 12, 23-25

1.3.1 Three Major Contributing Factors to SDB

Approximately 80% of SDB in young children is associated with adenotonsilar

hypertrophy26, which is more prevalent among young children (2-8 years).27

Adenotonsilar hypertrophy increases upper airway obstruction27 and this can negatively

influence craniofacial development.28 Children with large tonsils may have retrognathic,

posteriorly and inferiorly rotated mandibles.23, 29, 30 Children with snoring symptoms at

  2  
age four showed a more constricted maxilla with a greater amount of anterior open bite

and posterior crossbite.31 Richards et al reported that in patients who had dysphagia,

choking and vomiting experiences, a complete cessation of these symptoms occurred

after removal of the adenoid and tonsilar tissues.32 In patients with habitual mouth

breathing and asthma, removal of the adenoid and tonsils also brought about significant

improvement. It has been suggested that the morbidity of OSA may be increased if

diagnosis and treatment of adenotonsilar hypertrophy is delayed.32

While large tonsil size is associated with SDB in young children, in children over six

years of age, obesity may contribute to disease severity.33 Larger pharyngeal tonsils in

obese SDB children have a greater effect on airway obstruction than they do in normal

weight SDB children.34 The severity of SDB is significantly higher in obese children

compared to normal weight children35-37 and the risk of SDB in children has been shown

to increases by 12% for every 1 kg/m2 BMI increment above the mean BMI.27 In adult

obese SDB patients, soft tissue in conjunction with adipose tissue increases the risk of

airway obstruction while in non-obese SDB adults, the bony structures of the craniofacial

complex may be the main contributing factors to SDB.38 To what extent this could be true

in SDB children is unknown. In both obese and non-obese SDB children, skeletal

changes are evident but obese children tend to have a larger anterior facial height.36

In addition to adenotonsilar hypertrophy and obesity, craniofacial morphology is another

major contributing factor to SDB in young children.3, 12 As previously described, many

changes in craniofacial features are closely related to adenotonsilar hypertrophy and

  3  
obesity.28, 36 A steeper mandibular plane angle, larger anterior facial height and tendency

to a vertical growth pattern are all characteristics of children who breathe through their

mouth.39 A retruded mandible, steep mandibular plane, and longer anterior lower facial

height are the main craniofacial characteristics of individuals with SDB symptoms.40-42

Some other craniofacial features related to SDB include a short nasal floor and

retroclined lower incisors,23 an increased interincisal angle,42 and a long soft palate.3, 23

Several studies have concluded that a lower position of the hyoid bone is an important

craniofacial feature contributing to SDB symptoms.23, 40, 43-45 A higher palatal vault,3, 28

narrow maxilla and posterior crossbite29, 46 have also been reported as important

craniofacial characteristics of OSA children.

1.4 Cephalometric Characteristics of Patients with SDB

Several studies based on cephalometric variables have suggested that some craniofacial

morphometric features are associated with SDB symptoms.28, 29, 41, 46, 47 There is a

significant correlation between cephalometric data and AHI scores in that patients with

higher AHI scores showed an increased mandibular plane angle, and a decreased length

of the mandibular plane (Go-Gn).48 Kawashima et al found that preschool children with

enlarged tonsils had retrusive mandibles with a downward and backward rotation.23 SDB

children had more hyperdivergent facial types compared with normal subjects.29, 48

Kulnis et al looked at two groups of children, snoring and non-snoring, between the ages

of 7-14 years. The snoring children had a different craniofacial morphology when

compared with non-snoring children; they had a significantly narrower anteroposterior

  4  
dimension of the pharynx and also a lower hyoid bone compared to the mandibular

plane.44

Parkkinen et al compared normal children with subjects who showed SDB symptoms.

Children with OSA had an increased anteroposterior jaw relationship, a mandibular plane

inclination, total lower anterior facial height, a longer and thicker soft palate, and a lower

hyoid bone position relative to the inferior border of the mandible.45, 49

Children who breathe through their mouth tend to have certain cephalometric craniofacial

features such as a steeper mandibular plane angle, larger anterior facial height, and a

tendency to a vertical growth pattern.39 Juliano et al analyzed cephalometric radiographs

of 52 mouth breathing and 90 nose breathing children. They found that mouth breathing

children have cephalometric patterns similar to those of adult OSA patients. The common

craniofacial features of a mouth breather were a retruded mandible, steep mandibular and

occlusal plane angles, and more proclined upper incisors. They found no significant

difference between the groups in the position of the hyoid bone relative to the mandibular

plane but the hyoid bone was located more anteriorly to the vertebrae in mouth

breathers.30

Ozdemir et al looked at eleven measurements of bony structures in lateral cephalometric

images. They also looked at other confounding factors such as size of the airway and the

adenotonsilar hypertrophy. Patients with higher AHI scores showed increased cranial

base angles, a decreased length of the body of the mandible and decreased posterior

airway space. They did not find any correlation between protrusion of the maxilla or

  5  
mandible and SDB, but they did find a significant correlation between cephalometric data

and adenotonsilar hypertrophy in addition to higher AHI scores.48

Kawashima et al completed a study of preschool children and found that cephalometric

craniofacial features of SDB children included a short nasal floor, retrusive mandible,

increased interincisal angle, increased posterior facial height, retroclined lower incisors,

narrow pharyngeal airway space, anterior tongue base position and long soft palate. They

did not find any significant relationship between the hyoid bone position and SDB

symptoms.23

Figure 1 provides a summary of the craniofacial features that are correlated with SDB in

children and the publications that support these findings.

  6  
Craniofacial features Literature

Narrow maxilla and posterior crossbite Agren, 1998; Zucconi, 1999; Lofstrand-

Tidstrom, 1999

Steep mandibular plane angle Zucconi, 1999; Ozdmir, 2004; Zettergren-

Wijik, 2006; Juliano, 2009; Kawashima,

2000;

Increased lower anterior face height Zucconi, 1999; Zettergren-Wijik, 2006;

Juliano, 2009; Marino, 2009; Kawashima,

2000; Lofstrand-Tidstrom, 1999

Retruded/smaller mandible Jamieson, 1986; Zucconi, 1999; Ozdmir,

2004; Zettergren-Wijik, 2006; Juliano,

2009; Marino, 2009; Kawashima, 2000;

Lofstrand-Tidstrom, 1999; Kim, 2011

Lower hyoid bone Jamieson, 1986; Kawashima 2002; Cuccia,

2007; Ozdmir, 2004;
Retroclined lower incisors Kawashima, 2002; Zettergren-Wijk et al,

2006

Proclined upper incisors Juliano et al, 2009; Tsuda, 2010

Long soft palate Jamieson, 1086; Kawashima, 2002; Tsuda,

2010

High palatal height Lofstrand-Tidstrom, 1999; Tsuda, 2010;

Kim, 2011

Figure 1 A summary of important craniofacial features and the supporting literature

  7  
1.5 Diagnosis

 
SDB has an effect on the growth of children and therefore it is important that it be

diagnosed as early as preschool.3, 16, 23 Clinical signs and symptoms that are good

predictors of SDB include enlarged pharyngeal tonsils, apnea during sleep, nocturnal

snoring, daytime sleepiness, and mouth breathing.50 In addition to these clinical signs and

symptoms, there are important radiographic features associated with pediatric SDB such

as a retruded or small mandible, increased mandibular plane inclination, increased total

and lower anterior facial height, a longer and thicker soft palate, retroclined lower

incisors, and a lower hyoid bone position.3, 25, 41, 45

Huynh et al investigated the relationship between responses to a sleep-disordered

breathing questionnaire and a clinical orthodontic examination of a subject’s facial and

dentoalveolar morphology. A sample of 604 children less than 18 years old underwent a

clinical assessment to determine their dental, skeletal, facial and esthetic conditions.

Children who suffered from SDB were characterized by hypertrophic adenoids and

tonsils, a long and narrow face (dolichocephalic), a high mandibular plane angle, narrow

palate, severe crowding in both the maxilla and mandible, allergies, frequent colds, and

habitual mouth breathing. They concluded that children with the signs and symptoms of

SDB should be referred to a sleep medicine specialist for diagnosis and to an orthodontist

for treatment of any dentoskeletal abnormalities.12 In a recent publication from the

American Academy of Sleep Medicine (AASM) regarding the indications of

polysomnography in children with SDB, it was suggested that clinical evaluation alone is

not sufficient to establish a diagnosis of OSA in children. Integration of

  8  
polysomnographic findings with a clinical evaluation is the best way to achieve an

accurate diagnosis of SBD in pediatric populations.51

1.6 Treatment of SDB

 
Marcus et al published a recent technical report on the diagnosis and management of

childhood OSA. According to this report, adenotonsilectomy is considered the first line

of treatment to improve SDB in both obese and non-obese children. However, there is

still a 13%-29% chance for persistent OSA symptoms after adenotonsilectomy. The

majority of obese children need continuous positive airway pressure (CPAP) even after

removal of the tonsil and adenoid tissues. CPAP is not considered as first-line therapy for

children with OSA when adenotonsilectomy is an option. However, for obese individuals

who have had an adenotonsilectomy and for those whom surgery is contraindicated,

CPAP should still be used. In addition, exercise and weight loss can reduce the severity

of OSA in obese children. Orthodontic treatments such as rapid maxillary expansion

(RME) or the use of mandibular advancement appliances have been reported as effective

for obese and non-obese SDB children.20

1.6.1 Continuous Positive Airway Pressure (CPAP) and Positive Airway Pressure

(PAP)

 
Literature supports the use of nasal CPAP as an effective way of treating the symptoms

of OSAS in young children.52-54 However, compliance can sometimes be a barrier for

effective use of CPAP. Marcus et al compared CPAP with PAP devices in OSA children

  9  
aged 2-16 years. After three months of PAP use, a significant improvement in

neurobehavioral function was observed. The investigators recommended PAP for

children with OSAS especially for those for whom compliance might be a barrier to

effective use of CPAP.55 In those children where surgery is a contraindication or those

who do not respond adequately to surgical removal of the adenoid and tonsils, CPAP or

PAP could be a useful aids to reduce OAS symptoms.20

1.6.2 Adenotonsilectomy

 
Surgical removal of the hypertrophic adenoid and tonsils in children has been found to

significantly improve SDB symptoms and consequently quality of life.19, 56, 57 Lofstrand-

Tidestrom et al reported improvement in SDB symptoms in children who had an

adenotonsilectomy at an early age. However, they have observed significant relapse in

terms of both OSA symptoms and dentofacial development during the teen-age years.31

Lipton et al analyzed the published literature on the treatment and morbidity of OSA

children and found that adenotonsilectomy remains the first line of treatment.15 However,

Kim et al found that over 40% of children with SDB symptoms had persistent pre-

surgical symptoms even after an adenotonsilectomy.4 Nevertheless, in a systemic review

and meta-analysis by Bonuck et al, they found that several studies reported a significant

improvement in children’s SDB symptoms and the growth after surgical removal of the

adenoid and tonsils.58 Ozdmir et al also observed significant improvement in craniofacial

features and SDB symptoms after removal of the tonsils and adenoid tissues.48 While

surgical removal of hypertrophic adenoid and tonsils is generally recommended, a recent

study by Tatlipinar et al suggested that adenotonsilar hypertrophy increases the risk of

  10  
cardiopulmonary complications and may affect a child’s quality of life.19 Lofstrand-

Tidestrom et al found that surgical removal of the adenoid and tonsils in snoring children

did not change the development of dentofacial features. They recommended collaboration

between professionals from all relevant disciplines including orthodontists,

otolaryngologists, and speech and language specialists.31

1.6.3 Orthodontic Treatment

 
Sleep related disorders could induce unfavorable effects on craniofacial and dental

development in young children. Early diagnosis and treatment of SDB has a significant

impact on the normalization of dentofacial morphology as a child grows.42 O’Brein et al

emphasized the need for early diagnosis and treatment of SDB in young children to

achieve a better prognosis.16

Orthodontic treatment may be necessary for those children with SDB who have a

constricted maxilla, an anterior open bite, and a long lower facial height especially when

they do not experience significant improvement in their craniofacial features and SDB

symptoms even after removal of the adenoid and tonsils at an early age.31 Normal weight

children who do not respond well to the surgical removal of the adenoid and tonsilar

tissues often show specific craniofacial features that result in the overall reduction of the

upper airway space.4 In cases where craniofacial features and malocclusions are

contributing factors to the persistence of symptoms even after removal of the adenoid and

tonsilar tissues, a systematic approach is recommended such as an adenotonsilectomy

followed by orthodontic treatment.4

  11  
Orthodontic therapy at an early age should be encouraged in OSA children because this

treatment may permanently modify these children’s nasal breathing and respiration and

consequently prevent obstruction of the upper airway.59 In a recent study by Villa et al it

is recommended that orthodontic appliances such as a rapid maxillary expansion (RME),

a lower jaw positioner, or a modified monobloc are effective for treating OSA in children

with malocclusions.60

1.6.3.1 Palatal Expansion

 
Orthodontic treatment such as maxillary expansion (RME) in conjunction with

adenotonsilectomy may improve symptoms in children with SDB.61 In orthodontic

treatment with RME there may be an increase in nasal cavity width and a subsequent

decrease in nasal airway resistance. This can improve the patient’s natural function and

finally reduce the chance of developing breathing-related issues.61, 62Pirelli et al studied a

group of 60 OSA children between the ages of 6-13 years with constricted maxillae. They

all received RME and CT scans were used to analyze the children’s craniofacial features

before and after expansion. There was great improvement in the size of the upper

airway/nasomaxillary complex and consequently an improvement in the symptoms of

OSA.63 When Villa et al looked at OSA improvement in children 12 months after RME

treatment, they found that there was a significant reduction in SDB symptoms and the

effect of treatment remained 24 months after the end of the orthodontic treatment.64

  12  
1.6.3.2 Functional and Mandibular Advancement Appliances

 
The use of mandibular advancement appliances for sleep disorders is not new. The first

report of a device used for sleep disordered breathing problems was by Pierre Robin in

1939.65 Mandibular advancement devices have been an effective way of treating OSA

symptoms in adults,66 and are considered a moderately effective treatment for snoring

and mild to moderate OSA in adult patients.67

Functional orthopedic appliances and oral appliances may be helpful in the treatment of

those children with craniofacial anomalies who are at a higher risk of SDB. With the

increasing prevalence of OSA in children, oral appliance therapy is also becoming a

promising option for children.68

In their review of oral appliances (OAs) for snoring and obstructive sleep apnea in adults,

Ferguson et al believe that the mechanisms of action of oral OAs during sleep are

improvement of upper airway muscle tone that in turn improves the upper airway patency

and consequently brings about a reduction in upper airway collapsibility.69 To what

extent this mechanism is similar in children warrants further investigation.

Both the Herbst and the Twin Block are functional appliances that could be used in

patients with retruded mandibles based on patient-specific dentoalveolar conditions and

the need for treatment.70 Lawton et al compared the Herbst with the Twin block in a

randomized crossover study. They found no significant difference between the efficacy of

the two appliances while both appliances in OSA patients.70

  13  
The majority of the literature on OAs is based on studies of adult subjects; most studies

done of children are non-randomized and non-controlled. Furthermore, many of the

studies done of children are undertaken with only a small sample size and short follow up

times, such as the investigations by Villa et al 2002, Rose et al, 2006 and Schutz et al,

2011.

In a randomized controlled study of a small group of children by Villa et al, jaw-

positioning appliances were used and a significant improvement in OSA symptoms was

observed compared with the control group. They concluded that treatment of OSAS with

an OA in children with malocclusions is an effective and well-tolerated method.

Although their sample was small and the time for follow up was short, the result was

nevertheless promising.59

In a case report study, Rose et al used RME followed by Frannkel-II and functional

Regulator Type-II appliances in OSA patients with more severe craniofacial anomalies;

significant improvement in SDB symptoms was observed in both groups.71 Schutz et al

found a significant improvement in SDB symptoms after one year of treatment in OSA

patients using a Herbst appliance which included a palatal expansion screw to advance

the lower jaw and expand the upper jaw at the same time.72 Further randomized

controlled trials on OAs and functional orthodontic/orthopedic appliances are needed.

  14  
1.7 Age and Gender Differences in SDB

 
Kawashima et al looked at the differences between males and females with SDB. They

found that preschool males with SDB have a longer anterior facial height and anteriorly

positioned hyoid bone when compared with females of the same age. On the other hand,

females had a narrower pharyngeal airway.73 Redline et al studied risk factors for sleep-

disordered breathing, including obesity, in young children and found that SDB symptoms

were associated with obesity but not age or gender.27 In a recent study by Kim et al, SDB

children in different age groups revealed different clinical symptoms. Problems with

breathing, sleep terrors, bruxism and delayed growth were more common among

preadolescents while impaired daytime function, daytime sleepiness, insomnia, morning

headaches, difficulties going to sleep and unrefreshing sleep were more common among

adolescents. A smaller mandible and a high and narrow palatal vault were evident among

the children with SDB without any significant differences between genders or age

groups.4 Kawashima et al looked at gender-dependent differences in craniofacial features.

They compared craniofacial morphology and hyoid bone position of preschool children

with SDB. The results showed that males had a longer anterior facial height, and a hyoid

bone positioned more anteriorly compared with SDB females. On the other hand, SDB

females had a narrower pharyngeal airway space. There was no significant difference

between the two genders with grade 3+ tonsils in terms of SDB symptoms (when

controlled for tonsil size). SDB males compared with normal males had a larger

interincisal angle, a longer lower facial height, retroclined lower incisors, a short nasal

floor, a narrower pharyngeal space, and a longer soft palate, while SDB females

compared with normal females had a narrower pharyngeal space, and longer soft palate.

  15  
It appears that in preschool children, males with SDB have skeletal risk factors and

females with SDB have airway risk factors.73

1.8 Objectives

 
Children with SDB have characteristic craniofacial morphological features that can affect

their growth from an early age. Prevention of SDB by altering the morphology and

function of the craniofacial complex may be one of the options for young children.

However, to what extent the craniofacial morphology of children impacts the upper

airway and increases the incidence of sleep apnea is still poorly understood. The goal of

this project is to understand how the craniofacial morphology and severity of

malocclusion may contribute to SDB symptoms in children according to their age and

gender. Such knowledge will facilitate early treatment and reduce the risk of continuing

development of the SDB profile.

1.9 Hypothesis

 
Adenotonsilar hypertrophy, obesity and craniofacial morphology and the degree of their

contribution to SDB varies among different age groups and genders. It has been well

established that certain craniofacial features are a risk factor for SDB in adults. However,

to what extent these craniofacial features could be related to SDB in children remains

unclear. We hypothesize that in young children, craniofacial complex dimensions and

severity of malocclusion are associated with SDB symptoms. The aim of this study is to

  16  
determine if craniofacial morphology and severity of malocclusion are related to SDB

symptoms in children aged 5-16 years when controlled for age, gender, and BMI.

  17  
Chapter 2: Materials and Methods

2.1 Subjects

 
In this retrospective study, we reviewed 450 patient records from the undergraduate and

graduate orthodontic clinics at the University of British Columbia. The study was

comprised of subjects from 5-16 years old, of both genders and of different ethnicities

including Caucasian, Asian, Spanish, Indian, and some other smaller ethnicities.

Exclusion criteria were individuals less than 5 and more than 16 years old, those with

significant craniofacial abnormalities, incomplete records, or if their images had been

taken on a different cephalostat.

Patients were divided into two groups: those from the undergraduate clinic who were put

in the group PreAd (preadolescents, 5-12 years) and those from the graduate clinic who

were put in the group Ad (adolescents, 13-16 years old). Patients from the undergraduate

clinic generally had less severe malocclusions and were in need of more basic

orthodontic treatment, while the patients from the graduate clinic had more severe

malocclusions and were in of more extensive orthodontic treatments. We further divided

each group into two subgroups for comparison between genders of the same age range:

PreAd males, PreAd females, Ad males, and Ad females.

  18  
2.2 Clinical Findings

2.2.1 Mallampati Scores and Tonsil Size

 
Validated diagnostic diagrams (Mallampati and Brodsky) were used to categorize the

visibility of the entrance of the upper airway (Mallampati) and the size of the palatal

tonsils in transverse (Brodsky). Mallampati airway classification uses four categories

(grades I-IV). Class I: the soft palate and entire uvula are visible, Class II: the soft palate

and a portion of the uvula are visible, Class III: the soft palate is visible and the base of

the uvula may be visible, Class IV: neither the soft palate nor the uvula are visible

(Figure 2). During assessment, each patient was instructed to open his or her mouth wide

and protrude his or her tongue forward as far as possible while trying not to emit any

sound. The Brodsky classification shows the transverse occupancy of both the right and

left pharyngeal tonsils. This classification has five grades (grades 0-4). 0: if the tonsils do

not extending beyond the tonsilar pillar, 1: if the tonsils occupy less than 25% of the

airway, 2: if the tonsils occupy 25%-50% of the airway, 3: if the tonsils occupy 50%-75%

of the airway, 4: if the tonsils occupy more than 75% of the airway (Figure 3).

  19  
Figure 2 Mallapati Airway Classification (I-V Scores). During assessment, the patient is
instructed to open his or her mouth as wide as possible, while protruding the tongue as far
as possible. Patients are instructed to not emit sounds during the assessment. Class I: soft
palate and entire uvula is visible, Class II: soft palate and portion of uvula visible, Class
III: soft palate visible (may include base of uvula), Class IV: Soft palpate not visible74

  20  
 !

!
Figure 3 Brodsky Classification (0-4 Grades) 75 !
0: Tonsils are in fossa
1: If the tonsils occupied less than 25% of the airway
2: If the tonsils occupied 25% to 50% of the airway
3: If the tonsils occupied 50% to 75% of the airway
4: If the tonsils occupied greater than 75% of the airway

  21  
2.2.2 Angle Classification

 
To determine the Angle classification, orthodontic models and intraoral photos of each

individual patient were analyzed by the author. We used both photos and digital

orthodontic models for greater accuracy and reliability (Figure 4). Angle Class I: the

mesiobuccal cusp of the upper first molar occludes on the buccal grove of the lower first

molar, Class II division 1: the mesiobuccal cusp of the upper first molar occludes anterior

to the buccal grove of the lower first molar with the proclined upper incisors, Class II

division 2: the mesiobuccal cusp of the upper first molar occludes mesial to the buccal

grove with retroclined upper incisors, Class III: the mesiobuccal cusp of the upper first

molar occludes distal to the buccal grove of the lower first molar.

  22  
 !!
!
!
!
!
!
!
!
!
!
!
!
!
!
!
Figure 4 Orthodontic models and intraoral photos were used to determine Angle
classification

  23  
2.2.3      Body  Mass  Index  (BMI)  

Each individual’s height and weight were measured in inches and pounds respectively.

These measurements were then converted to meters and kilograms and the body mass

index (BMI) was calculated. Using growth charts showing BMI for age from the Center

of Disease Control, we divided our sample into four groups: 0: underweight, 1: normal, 2:

overweight, 3: obese. 76

2.3 Questionnaires

 
Franco et al developed the OSA-18 questionnaire based on responses from the caregivers

of 61 OSA children. This study was comprised of subjects from 6 months to12 years old,

of both genders and of different ethnicities including Caucasian (8%), African American

(85), and Spanish (7%). The test-retest reliability was done according to Litwin et al,

1995 and the R-value was more than 0.74. The Respiratory Disturbance Index (RDI) was

classified as normal to mild OSA if RDI ≤ 5, moderate OSA for RDI = 6-9 and severe

OSA for RDI ≥ 10. The impact of OSA on quality of life was large for 36%, moderate for

31%, and small for 33%. The investigators concluded that the OSA-18 is a useful way to

determine the quality of life in OSA children.77

Chervin et al developed the PSQ-22 questionnaire based on responses from the parents of

54 SDB children after polysomography and from the parents of 108 children before

polysomography as a control group. They applied a simple questionnaire with a

“yes/no/don’t know” response format. The SDB subjects and the control group included

  24  
children of both genders aged 2-18 with means of 9.3±4.1 and 7.0±3.8 years respectively.

Sensitivity and specificity of the test and correct classification were found to be similar

for both groups. The scales showed good internal consistency and in a separate sample

(n=21), a good test-retest stability. They recommended the PSQ-22 as a useful way to

identify SDB in clinical research where polysomnography is not feasible.6

We used the two validated standardized questionnaires previously described (part one

OSA-18 and part two, PSQ-22) to evaluate SDB symptoms. Parents or guardians who

were able to monitor their children both during the day and at night while the children

were sleeping completed the questionnaires. The OSA questionnaire (OSA-18)

comprised 18 questions related to SDB symptoms in children. Each question was scored

based on a graded scale of 1 to 7. The higher the grade, the higher the possibility of SDB

and so reduced quality of life. Part one was divided into five domains including daytime

function, sleep disturbances, physical symptoms, emotional symptoms and

parents’/caregivers’ concerns (Appendix A.1). Based on previous reports, those patients

who scored over 60 were considered to have a high likelihood of SDB. Part two, the

pediatric sleep questionnaire (PSQ-22), consisted of 22 questions to evaluate sleep related

breathing disorder symptoms (Chervin et al 2000) (Appendix A.2). Each question could

be answered with “yes”, “no”, or “don’t know”. Only “yes” answers were considered as

indicative of the presence of SDB symptoms.

  25  
2.4 Cephalometric Analysis

 
All cephalometric images were obtained from the same cephalostat with subjects holding

their heads in a natural position with the Frankfort horizontal plane parallel to the floor.

Dolphin imaging software, version 10.2 premium, was used to digitize and analyze the

data with the UBC standardized lateral cephalometric analysis for OSA. The reference

points and lines were used in the cephalometric analysis are shown in Figure 5 and

include 14 linear and 9 angular measurements. The linear measurements were palatal

height (PALHT), upper facial height (UFH), lower facial height (LFH), total facial height

(TFH), length of soft palate (PNS-P), mandibular body length (Go-Gn), hyoid bone

position (MP-H), vertical airway length (PNS-Eb [V]), position of the maxillary central

to Nasion-A point line (U1-NA), position of the lower incisor relative to Nasion-B point

line (L1-NB), hard tissue chin button prominence (Pog-NB), facial convexity (A-NPog),

overjet and overbite. To measure the palatal height, we used a McKee analysis to

measured, in millimeters, from the mesiobucal cup tip of the upper first molar to the

palatal plane. The angular measurements included the antroposterior position of the

maxilla and the mandible relative to the anterior cranial base and to each other (SNA,

SNB and ANB respectively), the chin position relative to the anterior cranial base (SN-

Pog), the upper incisor to the anterior cranial base (U1-SN), the lower incisor to the

mandibular plane (L1-MP), the interincisal angle (U1-L1), and the mandibular plane

angle (MP-SN).

  26  
 !
"#

$!"# /0# %!"#

%#
$#
).# &$#
10# 2#
,# $(#
)*#
'+# '$#
-#

Figure 5 Cephalometric landmarks, and linear and angular cephalometric variables. Point S sella, N
nasion, A point A, B point B, ANS anterior nasal spine, PNS posterior nasal spine, Pg pogonion, Gn
gnathion, Me menton, Go gonion, H hyoid bone, Eb base of epiglottis, P tip of soft palate, SN anterior
cranial base, MP (Me-Go) mandibular plane, OP occlusal plane (midpoint between maxillary and
mandibular incisor edge to midpoint between maxillary and mandibular molar mesial cusps). Linear
variables; PALHT (palatal height) mesiobucal cups tip of upper first molar to palatal plane, OB overbite
(vertical overlap of upper and lower incisors), OJ (horizontal overlap of upper and lower incisors), UFH
(N-ANS) upper anterior face height, LFH (ANS-Me) lower anterior face height, TFH (N-Me) total anterior
face height, U1-NA distance from incisal, edge of upper incisor to NA plane, L1-NB distance from incisal
edge of lower incisor to NB plane, Pg-NB distance between Pogonion and vertical continues of NB plane,
CONV (facial convexity) distance from NPg to A point, Go-Gn mandibular body length, MP-H position of
hyoid bone to mandibular plane, PNS-P length of soft palate, VAL (PNS-Eb) vertical airway length.
Angular measurements; SNA antroposterior position of maxilla relative to anterior cranial base, SNB
antroposterior position of mandible relative to anterior cranial base, ANB anteroposterior relationship of
maxilla to mandible relative to anterior cranial base, U1-SN inclination of upper incisor relative to anterior
cranial base, L1-MP inclination of lower incisor relative to mandibular plane, U1-L1 interincisal angle, SN-
Pg chin position relative to anterior cranial base, SN-MP mandibular plane angle relative to cranial base, H
ANGLE soft tissue facial convexity (based on Tsuda et al, 2010).3

  27  
2.5 Method Error

 
The author digitized ten randomly selected lateral cephalometric radiographs on three

separate occasions to calculate the error of method according to Bland J. & Altman D.

Methodology of error measurement.78 The standard deviation of repeated measurements

on the same subject enabled us to measure the size of the measurement errors. A method

error within 0.94mm was found for the linear measurement variables and within 0.71

degrees was found for the angular measurements.78

2.6 Statistical Analysis

 
Statistical software (SPSS software, Chicago, II USA) was used to analyze the data. Data

were presented as mean and standard deviations. A P value of less than 0.05 was

considered significant. We used nonparametric tests to analyse the questionnaires, which

were not normally distributed. Spearman’s rho test was used to identify correlations

between the cephalometric variables and the questionnaires (questions and domains). The

Mann-Whitney Test was used to compare the responses to the questionnaires between the

two different age groups and also between genders. A 2-tailed t-test was then used to find

the significant differences between all the groups in terms of age and gender. We used the

Kruskal-Willias Test to determine the correlation between the Angle classification and

the SDB symptoms among all the groups according to genders and age. In addition to

these nonparametric tests, we also used parametric tests for the data, which related to the

cephalometric variables, age, and BMI and were considered normally distributed. The

  28  
Leven’s Test was used to determine the equality of variances across the samples and to

predetermine for the t-test. A paired t-test was also used to identify the mean value of the

cephalometric variables between the genders of different age groups. A one-way

ANOVA test was used to find the significance of the mean value between and within the

four groups. After finding the significant differences between the groups by ANOVA, we

used the Tucky HSD to find the mean differences of P value of less than 0.05, between

each group with other groups. A Chi-Square test was used to find the significance of the

questionnaires in terms of the Mallampati and Tonsil scores.

  29  
Chapter 3: Results

3.1 Subjects

 
A total of 405 patient records were reviewed. Of these, 79 patient records were excluded

because the children were older than 16 years, 67 records were excluded because the

child’s height and weight had not been recorded at the time SDB questionnaires were

completed, and three cases were excluded because images had been taken on a different

cephalostat. We analyzed 301 cases of both genders and different ethnicities including

Caucasian, Asian, Spanish, Indian and some other smaller ethnicities. Our sample of 301

was divided into two groups according to age. The group PreAd (preadolescents=5-12

years) included 237 children, 97 male and 140 female with a mean age of 9.9±1.6, and

the group Ad (adolescents=13-16) included 64 children, 24 male and 40 female with a

mean age of 13.8±0.9. We also divided each group into two subgroups for comparison

between genders of the same age range. A mean score for the questionnaire OSA-18 was

29.6±12.5. A summary of these findings is provided in Table 1.

Table 1 Demographic data and questionnaire scores (mean)

Total Group PreAd/5-12 years Group Ad/13-16 years
N 301 237 64
Age 5-16 9.9±1.6 13.8±0.9
Male/Female 121/180 97/140 24/40
Questionnaire score 29.6±12.5 29.1±11.7 31.6±15.2

  30  
3.2 Clinical Findings

3.2.1 Mallampati Scores and Tonsils Size

 
We assessed the Mallampati scores of all subjects by clinical examination and determined

the percentage of each group (PreAd and Ad) with a score of I to IV (Figure 6).

45  
40  
35  
30  
25  
Preadolescents  
20  
Adolescents  
15  
10  
5  
0  
Mallam  I   Mallam  II   Mallam  III   Mallam  IV  

Figure 6 Percentage of children with different Mallampati scores and the differences
between PreAd and Ad groups

There was no significant correlation between the Mallampati scores and SDB symptoms

for all 301 children, but when comparing the two different age groups (PreAd and Ad) we

found that group Ad had higher Mallampati scores (Median=3, P=008) than PreAd

(Median=2, P=0.295) (Table 2 and Figure 6). There was no significant difference

between PreAd and Ad males. However Ad females had higher Mallampati scores (grade

  31  
III and IV) when compared with PreAd females (Median=3, P=0.009) (Table 2. and

Figure 7).

Table 2 Significant differences between age and gender in Mallampati scores

Mallampati Median 2-tailed

PreAd vs Ad 2/3* 0.009**

PreAd Females vs Ad Females 2/3* 0.008*

PreAd Males vs Ad Males 2/2 0.295

*
Median for Mallampati scores
**
P<0.05

40  

35  

30  

25  

20   Female  5-­‐12  
Female  13-­‐16  
15  

10  

5  

0  
Mallam  I   Mallam  II   Mallam  III   Mallam  IV  

Figure 7 Percentage of female patients with different Mallampati scores and the
differences between PreAd females and Ad females

  32  
We did not find any significant correlation between tonsil size, SDB symptoms, and

cephalometric variables in our sample when we looked at individual groups and when we

pooled the groups together as one sample.

3.2.2 Body Mass Index

 
According to the Body Mass Index (BMI) charts from the Center of Disease Control

(CDC), 4.7% of our sample were considered underweight, 71.1% normal/healthy weight,

16.6% overweight and finally 7.6% obese. We also divided the sample into four groups

based on age and gender to determine the distribution of BMI (PreAd-M= males 5-12,

Ad-M= males 13-16, PreAd-F= females 5-12, and Ad-F= females 13-16). We found that

the majority of overweight and obese children were preadolescents of both genders

(Table 3).

Table 3 Body mass index distribution among the four groups based on age and gender
Gender and Age Groups Total (n/%)

BMI groups PreAd-M Ad-M PreAd-F Ad-F

0: Underweight 6 0 7 1 14/4.7

1: Normal 62 18 104 30 214/71.1

2: Overweight 19 4 19 8 50/16.6

3: Obese 10 2 10 1 23/7.6

Total 97 24 140 40 301/100

There was no significant correlation between BMI and SDB symptoms in our sample

when we looked at individual groups and when we pooled the groups together as one

  33  
sample. The only sleep-related symptom listed in the PSQ-22 questionnaire that showed a

moderate correlation with BMI was sleepiness during the daytime (P=0.032).

3.2.3 Angle Classification

 
The Angle classification distribution among the sample was 28.9% Class I, 52.8% Class

II division 1, 9.0% Class II division 2, and 8.3% Class III malocclusion (Table 4). There

was no statistically significant difference between the Angle classifications with respect

to SDB symptoms when controlled for tonsil size, age, gender, and Mallampati scores.

Table 4 Angle classification

Cl. I Cl. II Div 1 Cl. II Div 2 Cl. III Total

N/% 87/28.9% 162/52.8% 27/9% 25/8.3% 301/100%

3.3 Questionnaires

 
The data from the OSA-18 and PSQ-22 questionnaires show that some SDB symptoms

were reported in more than 15% of individuals in our sample. We categorized the range

of answers to questions on the OSA-18 from 1 - 7 into three main categories as follow:

Category 1: None of the time (1) and Hardly any of the time (2), Category 2: A little of

the time (3) and Some of the time (4), and Category 3: Good bit of the time (5), Most of

the time (6), and All of the time (7).

  34  
According to responses given to the OSA-18 questionnaire by all 301 children, the

combined symptoms most commonly seen were loud snoring (15%), mouth breathing

(16%) and difficulty waking (21.2%)(Table 5).

Table 5 OSA-18 Percentages of individual item responses

Domains Questions None/hardly Little/some Good
bit/most/all
D1: Sleep Q1 Loud snoring* 85.0 12.0 3.0
disturbance
Q2 Breath holding/pauses 97.0 2.7 0.3

Q3 Chocking or gasping 97.3 2.3 0.3

Q4 Fragmented sleep 90.0 7.3 2.3
D2: Physical Q5 Mouth breathing* 84.0 10.0 6.0
symptom
Q6 Frequent colds or URIs 89.7 9.0 1.3
Q7 Rhinorrhea 86.7 12.0 1.3
Q8 Dysphagia 97.3 2.3 0.3
D3: Emotional Q9 Mood swings or 86.7 11.0 2.3
symptoms tantrums
Q10 92.7 6.0 1.3
Aggression/hyperactivity
Q11 Discipline problems 92.4 6.6 1.0
D4: Daytime Q12 Daytime drowsiness 92.7 6.6 0.7
function
Q13 Poor attention span 88.7 9.3 2.0
Q14 Difficulty waking* 78.7 14.6 6.6
D4: Caregiver Q15 Worried over 93.0 5.3 1.7
concern children’s health
Q16 Concerned not enough 97.0 2.0 1.0
air
Q17 Missed activities 96.0 3.3 0.7
Q18 Frustration 88.7 8.6 2.7
* over 15%

We analyzed patient responses to the PSQ-22 questionnaire and summarized the findings

as provided below in Table 6. This table shows the percentage of children that responded

with Yes, No, or Don’t Know to each single SDB symptom. According to the PSQ-22

questionnaire as completed by the entire sample of 301 children, the symptoms most

  35  
commonly seen were mouth breathing during the day (18.9%), dry mouth on waking

(28.2%), waking unrefreshed in the morning (20.6), hard to wake up in the morning

(2,4.6), not listening when spoken to directly (19.6), and interrupts or intrudes on others

(15.9).

  36  
Table 6 PSQ-22 Percentages of individual item responses
Questions Yes No Don’t Know

PSQ 1: snores more often 10.3 82.7 7

PSQ 2: always snores 7 89.7 3.3

PSQ 3: snores loudly 10.6 88 1.3

PSQ 4: heavy or loud breathing 12.3 83.7 4

PSQ 5: have trouble breathing 4 92 4

PSQ 6: stop breathing during night 2.7 91.4 6

PSQ 7: mouth breathing during day* 18.9 75.1 6

PSQ 8: dry mouth on waking up 28.2 63.5 8.3

PSQ 9: waking unrefreshed in the morning 20.6 72.4 7

PSQ 10: sleepiness during the day 11.6 84.7 3.7

PSQ 11: sleepiness at school 5.3 93 1.7

PSQ 12: hard to wake up in morning 24.6 75.4 0

PSQ 13: not listening when spoken to directly 19.6 79.1 1.3

PSQ 14: difficulty organizing tasks 14.3 84.1 1.7

PSQ15: easily distracted 21.3 74.4 4.3

PSQ 16: fidgets with hands and feet 18.6 78.1 3.3

PSQ 17: “On the go”/hyperactive 14 80.4 5.6

PSQ 18: interrupts or intrudes on others 15.9 81.7 2.3

PSQ 19: occasionally wetting the bed 4.3 95.3 0.3

PSQ 20: wakes up with headache 7 90.4 2.7

PSQ 21: stops growing 4 94 2

PSQ 22: overweight 6 90.7 3.3

*
Over 15%

  37  
Table 7 shows a summary of the most common SDB symptoms among the children in

our sample according to responses given to the OSA-18 and PSQ-22 questionnaires.

Table 7 Most common SDB symptoms among the children in our sample according to
responses given to the OSA-18 and PSQ-22 questionnaires

OSA-18 PSQ-22

Daytime Sleepiness Breathing Pauses

Difficulty Getting Up Bed Wetting

Frequent Colds Snores More Often

Daytime Function Hyperactivity

Headache in Morning

3.3.1 OSA-18 Questionnaire

 
The next three tables (Tables 8,9, and10) document the severity of SDB symptoms for

each age and gender group according to responses given to the OSA-18.

By comparing PreAd with Ad, we found that the percentage of Ad who had “Some” or

“Good Bit” of daytime sleepiness and difficulty getting up was significantly higher than

PreAd (Table 8).

  38  
Table 8 Difference between PreAd and Ad in percentages

Daytime Sleepiness Difficulty Getting Up

Preadolescents Adolescents Preadolescents Adolescents
Hardly 94.9 84.4 81.9 67.2
Some 4.6 14.1* 13.1 20.3*
Good bit 0.4 1.6* 5.1 6.6*
*
Significant percentage

By comparing PreAd males with Ad males, we found that frequent colds were more

common among PreAd males, while daytime sleepiness was more prevalent among Ad

males (Table 9).

Table 9 Differences between PreAd males and Ad males in percentages

Frequent Colds Daytime Sleepiness

Male 5-12 Male 13-16 Male 5-12 Male 13-16
Hardly 79.2 94.8 92.8 79.2
*
Some 20.8 5.2 7.2 16.7*
Good bit 0.0 0.0 0.0 4.2*
*
Significant percentage

By comparing PreAd females with Ad females, we found that daytime sleepiness and

difficulty getting up were more prevalent among Ad females then PreAd females (Table

10).

Table 10 Differences between PreAd females and Ad females in percentages

Daytime sleepiness Difficulty Getting up

Female Female Female Female
5-12 13-16 5-12 13-16
Hardly 96.4 87.5 83.6 62.5
Some 2.9 12.5* 11.4 25.0*
Good bit 0.7 0.0 5.0 12.5*
*
Significant percentage

  39  
Overall, based on the OSA-18 questionnaire, several significant differences between age

groups and genders were found. There was a higher incidence of daytime sleepiness

among adolescents compared to preadolescents of both genders. Adolescents, especially

females, showed a greater degree of difficulty getting up in the morning. Frequent colds

appeared to be more prevalent among younger males.

3.3.2 PSQ-22 Questionnaire

The next two tables (Tables 11 and12) identify the severity of PSQ-22 SDB symptoms

based on age and gender. When comparing age groups, we found that the prevalence of

breathing pauses and bed wetting among preadolescents was 3.2% and 7.2% respectively,

while the prevalence was zero in adolescents for both symptoms (Table 11).

Table 11 Percentage of subjects who chose answered "YES" to the SDB Symptoms and
the differences between preadolescents and adolescents

Preadolescents Adolescents

SDB Symptoms YES NO YES NO

Breathing Pause 3.2 96.8 0.0 100

Bed wetting 7.2 92.8 0.0 100

When comparing different genders, several PSQ-22 SDB symptoms were significant.

Snoring more often and morning headaches were more frequent among females than

males (13.3% vs 5.8% and 10.6% vs 1.7% respectively), while sleepiness during the day

  40  
and hyperactivity were more prevalent among males (9.1% vs 2.8% and 19% vs 10.6%

respectively) (Table 12).

Table 12 The percentage of male, female and total sample who answered "YES" to the
following SDB symptoms in the PS22 questionnaire and significant P-Value

Male=121 Female=180 Total=301 P*

Snore more often 5.8 13.3 10.3 0.045

Sleepy during day 9.1 2.8 5.3 0.038

“On the go”/Hyperactivity 19 10.6 13.6 0.031

Headache in morning 1.7 10.6 7 0.005

*
P<0.05

Figure 8 shows the number of patients from each group based on age and gender who

presented with all four SDB symptoms. There were significant differences between

PreAd and Ad for both genders. Snoring and morning headaches were more evident in

PreAd females compared with Ad females. Sleepiness during the day and hyperactivity

were more common among PreAd males than Ad males. We also found that hyperactivity

was more common among preadolescents of both genders when controlled for age

(Figure 8).

  41  
 20  
18  
16  
14  
12   Male  5-­‐12  
10   Male  13-­‐16  
8  
Female  5-­‐12  
6  
Female  13-­‐16  
4  
2  
0  
Snore  more   Sleepy   On  the  go   Headache  in  
often   during  day   morning  

Figure 8 Classification of patients according to their age and gender who had SDB
symptoms according to responses given to the PSQ-22

In summary, when comparing preadolescents with adolescents based on the OSA-18 and

PSQ-22 questionnaires, we found that snoring frequency, hyperactivity, morning

headache and bedwetting were more prevalent among preadolescents, while difficulty

getting up, daytime sleepiness and impaired daytime function were more common among

adolescents. (Table13).

Table 13 A summary of differences between two age groups according to the OSA-18
and PSQ-22 questionnaire

More Common Among Preadolescents More Common Among Adolescents

Snoring often Difficulty getting up in the morning

“On the go”/hyperactivity Impaired daytime function

Morning headaches Daytime sleepiness

Bed wetting

  42  
3.4 Cephalometric Analysis

Table 14 shows cephalometric variables with mean and standard deviations for normal

(according to UBC Sleep Study analysis), the whole sample and the two main groups,

PreAd and Ad. We have divided the cephalometric analysis into four categories including

the maxilla, mandible, intermaxillary relationship and upper airway. Looking at the

maxilla and mandible individually, all variables are within normal limit of mean and

standard deviation of norm. But looking at the intermaxillary relationship, four variables

(ANB, H-Angel, Convexity and Overjet) showed significant variation from the normal

standard. They all showed an increased anteroposterior measurement between the maxilla

and mandible. This could be normal in growing children to some extent (Table 14).

  43  
Table 14 Cephalometric analyses with mean and standard deviation for the whole
sample and both PreAd and Ad

Mean and Standard Deviation

Cephalometric Variables
Maxilla PreAd (n=180) Ad (n=121) Total (n=301) Normal

U1-SN 105.7±8.0 105.9±8.5 105.8±8.1 102.6±2.5

SNA 82.4±3.9 83.0±3.6 82.5±3.8 82.0±3.5
U1-NA (mm) 3.2±2.4 3.4±2.9 3.2±2.5 4.3±2.7
PALHT (mm) 17.95±2.3 21.0±2.7 18.6±2.7 N/A

Mandible
SN-Pg 77.8±3.4 79.1±3.8 78.1±3.5 80.0±3.5
SNB 77.4±3.5 78.5±3.7 77.6±3.5 80.9±3.4
L1-MP 93.6±6.1 94.1±7.6 93.7±6.4 95.0±7.0
LI-NB (mm) 4.7±2.2 5.1±2.4 4.8±2.2 4.0±1.8
Pg-NB 0.7±1.6 1.2±1.4 0.8±1.6 2.1±1.7
GoGn (mm) 69.6±5.8 74.9±5.1 70.7±6.0 72.6±4.4
Mp-H (m) 11.4±4.6 13.1±4.1 11.6±4.5 15.0±2.0

InterMaxillary
relationship
ANB 5.0±2.3 4.5±2.0 4.9±2.3 1.6±1.5
U1-L1 126.5±9.9 126.3±11.2 126.4±10.1 130.0±6.0
MP-SN 34.1±5.2 33.6±5.7 34.0±5.3 33.0±6.0
UFH (mm) 46.3±3.6 49.6±2.9 47.0±3.5 50.0±2.5
LFH (mm) 60.7±4.9 65.2±7.4 61.7±5.8 65.0±4.5
TFH (mm) 105.6±6.8 113.5±8.9 107.3±8.0 115.0±5.5
H-Angle 17.2±4.8 15.6±4.8 16.8±4.8 10.0±4.0
Convexity (mm) 4.2±2.4 3.8±2.2 4.1±2.3 1.0±2.0
Overjet (mm) 4.8±2.6 4.5±2.2 4.8±2.6 2.5±2.5
Overbite (mm) 3.0±2.1 2.7±2.1 2.9±2.1 2.5±2.0

Upper airway
PNS-P (mm) 29.6±3.1 31.6±3.3 30.1±3.2 N/A
PNS-V (mm) 53.0±5.8 59.2±6.2 54.3±6.4 N/A

  44  
3.5 Correlation Between Cephalometric Variables and SDB Symptoms

 
Three main domains from the OSA-18 questionnaire that appeared to have a significant

correlation with cephalometric variables in SDB children were sleep disturbances,

daytime function and caregiver concern. Cephalometric variables that showed significant

correlation with SDB symptoms were a lower position of the hyoid bone in relation to the

inferior border of the mandible (MP-P), retroclination of the lower incisor to the

mandibular plane (L1-MP), retroposition of the mandible in relation to the cranial base

(SN-Pog), and a steeper inclination of the mandibular plane to the cranial base (MP-SN).

Children with a lower position of the hyoid bone and retroclined lower incisors had more

sleep disturbances during the night. Those with a retruded mandible and a higher

mandibular plane angle relative to the anterior cranial base had more impaired daytime

function. Individuals with a lower position of the hyoid bone raised caregiver concerns

more often (Table 15).

Table 15 Corrections between domains and cephalometric variables

Domains
Cephalometric Sleep Disturbance Daytime Function Caregiver Concern

Variables R P R P R P

MP-H 0.188 0.001 0.096 0.097 0.177 0.002

L1-MP -0.152 0.008 0.014 0.804 -0.056 0.306

SN-Pog 0.031 0.583 0.168 0.003 0.060 0.299

MP-SN 0.094 0.105 -0.162 0.005* 0.018 0.753

R- Pearson correlation r
*
P<0.01

  45  
By looking at the relationship between individual questions and cephalometric variables,

we found a significant correlation between seven SDB symptoms and four cephalometric

variables (Table 16). A lower position of the hyoid bone was correlated with all of the

SDB symptoms in Table 17 except for difficulty organizing tasks. Retroclined lower

incisors had a significant correlation with three main symptoms including loud snoring,

breath-holding, and the feeling of choking, which could all affect sleep quality. A

retruded mandibular position was related to excessive daytime sleepiness. A larger

mandibular plane angle relative to the cranial base was correlated with loud snoring and

difficulty organizing tasks (Tables 16 and 17).

Table 16 A summary of important craniofacial features that have significant correlation

with SDB symptoms (R-value)

SDB Symptoms
Loud Holding Choking Restless Excessive Parent Difficulty
Cephalometric snoring breath sleep sleep daytime worries/ organizing
Variables sleepiness frustration task

MP-H 0.151 0.221 0.178 0.155 0.155 0.167

L1-MP - 0.192 - 0.211 - 0.197

SN-Pog 0.165

MP-SN 0.175 0.182

R – Pearson correlation r
P<0.01

  46  
Table 17 A summary of SDB symptoms which are correlated with each individual
cephalometric variable

By comparing cephalometric variables between PreAd males and PreAd females found

no significant difference between the two groups. When evaluating the differences

between male and female adolescents (ages 13-16), we found that males presented higher

values in palatal height, upper facial height (UFH), lower facial height (LFH), total facial

height (TFH), proclination of the lower incisors (L1-NB), and lower facial convexity (H-

Angle), while females presented higher values of the angle between the upper and lower

incisors (U1-L1) and the distance between the posterior nasal spine and the base of the

epiglottis (PNS-V) (Table 18). Despite the significant differences between the two

groups, there was no significant deviation from the normal mean and standard deviation

based on the UBC Sleep Study analysis.

  47  
Table 18 Comparison between males and females of aged 13-16

Males 13-16 Females 13-16 Sig. (2-tailed)

Palatal Height mm 22.2 ± 2.7 20.2 ± 2.4 P=0.003
UFH mm 51.2 ± 2.9 48.7 ± 2.4 P=0.000
LFH mm 68.9 ± 7.3 62.9 ± 6.6 P=0.001
TFH mm 118.7 ± 8.3 110.4 ± 7.7 P=0.000
U1-L1° 122.2 ± 9.1 128.8 ± 11.7 P=0.020
L1-NB° 6.3 ± 2.1 4.4 ± 2.2 P=0.001
H-ANGLE° 17.2 ± 3.9 14.7 ± 5.0 P=0.037
PNS-Vmm 61.91 ± 6.3 67.62 ± 5.7 P=0.007

In summary, when comparing preadolescents and adolescents, we found that

preadolescents presented a significantly higher incidence of hyperactivity, headaches in

the morning, snoring and bedwetting. Adolescents showed significantly higher daytime

sleepiness, difficulty getting up, and impaired daytime function. By comparing females

with males we found that frequent snoring and morning headaches were more prevalent

among females, while daytime sleepiness, and hyperactivity were more common among

males. Craniofacial features that have a significant relationship with SDB symptoms are a

lower position of the hyoid bone, a retruded mandible, a steeper mandibular plane angle

and retroclined lower incisors. There was no statistically significant relationship between

SDB symptoms and the Angle classification, tonsil size, Mallampati scores or BMI.

  48  
Chapter 4: Discussion

The present study supports the hypothesis that craniofacial morphology is correlated to

SDB symptoms in children. The majority of studies on the relationship between SDB

symptoms and craniofacial morphology have been of children with SDB symptoms

compared to children with no SDB symptoms.12, 23, 39, 43, 45 In the present study, subjects

were recruited from both undergraduate and graduate orthodontic clinics at the University

of British Columbia. These children were attending the clinics solely to receive

orthodontic treatment. In terms of the severity of malocclusion, the children recruited

from the undergraduate clinic were considered to have less severe malocclusions and in

need of more basic orthodontic treatment; however the children recruited from the

graduate clinic were considered to have more severe malocclusions and in need of more

extensive orthodontic treatment, such as rapid maxillary expansion, extractions with full

edgewise therapy and/or orthognathic surgery.

In a previous study in our laboratory, (Tsuda et al, 2010), we looked at the incidence of

SDB symptoms and their correlation with craniofacial features in otherwise healthy

preadolescent children. In the present study, in addition to the preadolescents from the

undergraduate orthodontic clinic, we included adolescent orthodontic patients from the

graduate orthodontic clinic. Supplemental to the incidence of SDB symptoms and their

correlation with the craniofacial morphology in both groups, we divided our subjects into

different groups based on their age and gender to determine any possible correlation

between SDB symptoms and age, gender, BMI, and severity of malocclusion. The most

  49  
common SDB symptoms were snoring, difficulty getting up, daytime sleepiness, morning

headaches, hyperactivity and frequent colds. All these symptoms were significantly

correlated with several craniofacial features. These craniofacial features included a

retruded mandible (greater SN-Pog), a steeper mandibular plane (increased MP-SN

angle), a lower position of the hyoid bone (longer MP-H), and retroclined lower incisors

(smaller L1-MP) as shown in tables 17 and 18.

Snoring, difficulty waking, and daytime sleepiness were three important symptoms

significantly correlated with SDB in this study, which is in accordance with previous

reports.3, 4 Corbo et al found that habitual snoring was more common among males than

females, and Li et al reported a higher prevalence of habitual snoring among males

(14.6%) than females, (9.5%).79, 80 Corbo et al also found that obese males older than 15

were at significantly higher risk of being habitual snorers.79 However, our results showed

a higher prevalence of snoring in females (13.3%) compared with males (5.8%). We also

found a higher prevalence among younger children and we did not find any correlation

between obesity and snoring. In accordance to the findings of Chang et al and Kim et al

who found a higher prevalence of daytime sleepiness in older children.4, 81 In our study,

difficulty waking was more common among adolescents regardless of their gender, but

Laberge et al found that difficulty waking was more common in early adolescent females

with possible causes being hormonal changes and higher pubertal status.82 Individual

preferences in sleep-wake timing and age-related changes in sleep-wake timing can vary

among subjects.83 Changes in reproductive hormones during puberty can affect circadian

rhythms and circadian physiology during early adulthood.84 Therefore, in adolescents,

  50  
difficulty waking, and daytime sleepiness could be due in part to circadian rhythms and

hormonal changes in addition to the presence of SDB.

Another SDB symptom that related to the quality of sleep in children was the presence of

morning headaches.4, 85 In our study, morning headaches were more prevalent among

preadolescent females. In contrast, in a study reported by Kim et al, morning headaches

and unrefreshing sleep appeared to be more prevalent among adolescents regardless of

gender.4 Zwart et al found that the overall frequency of recurrent headaches did not vary

significantly with the age of the children, but the frequency of recurrent headaches was

significantly higher among females.85 There is hardly any published data about the

pathophysiology of morning headaches and OSA in young children.

Poor quality of sleep in SDB children can have an adverse effect on their daytime

behavior and school performance.2, 7, 8 Results from the present study as well as from

several other studies have shown that hyperactivity was more common among younger

SDB children.16, 22, 81, 86 Chervin et al suggested that there was a link between sleepiness

and hyperactive behavior in both males and females during childhood and adolescence.22

Our data suggests a higher prevalence of hyperactivity among preadolescent males

compared with preadolescent females. In a recent meta-analysis review by Willcutt, the

prevalence of attention-deficit/hyperactivity disorder was reported to be higher among

young males.87 Further research is needed to determine the etiology of this disorder and

investigate its prevalence among children with SDB symptoms.

  51  
Frequent colds or upper airway infections have been reported as a symptom in subjects

with SDB.2, 3 Bixler et al reported nasal abnormalities (e.g. chronic sinusitis/rhinitis) as

an important risk factor for SDB in children between 5-12 years.11 During the completion

of the SDB questionnaires pertaining to younger children, we also found that parents

repeatedly reported their children as having frequent colds. The obstruction of the nasal

airway forces children to breathe through their mouth during the day and at night during

sleep. This might affect their growth pattern leading to a more vertical face pattern.12 A

steeper mandibular plane angle, a larger anterior facial height and a tendency to a vertical

growth pattern are characteristics of children who breathe through their mouth.39 It seems

that obstruction of the nasopharyngeal complex and craniofacial anomalies in growing

children are correlated and further study is needed to identify the cause and effect

relationship between these two variables.

Gozal et al and Bixler et al reported obesity as an important risk factor for SDB in

children.7, 11 However, this was not the case for the group of children in the present study

when controlled for age and gender. In our sample, more than 75% of the population was

either normal weight or underweight (71.1% and 4.7% respectively) and the percentage

of overweight and obese children was significantly less (16.6% and 7.6% respectively).

In their review, Cuccia et al, looked at common craniofacial changes in patients suffering

from OSA with regards to degree of obesity. They found that in both obese and non-

obese OSA children, skeletal changes were evident, but the obese subjects presented with

greater intermaxillary divergence and an increased angle between the palatal plane (ANS-

PNS) and the mandibular plane (Go-Me).36

  52  
Neither Mallampati scores nor tonsilar grades were revealed to be significantly related to

SDB symptoms in our sample when we controlled for age, gender and BMI. However,

Kim et al found a significant correlation between higher Mallampati scores and the

severity of SDB in children.4 The majority of the studies reporting a significant

correlation between adenotonsilar hypertrophy and SDB symptoms in children included

younger subjects.26, 27, 31, 34, 80 In the present study we did not find any significant

correlation between tonsilar size and SDB symptoms. This could be explained by the

majority of our sample being older than 6-8 years and having tonsils significantly

reduced in size because of growth and developmental changes. In the study by

Kawashima et al, no difference was found between either a tonsil size of grade II (25%-

75% tonsil visible) or a tonsil size of grade III (>75% visible) as contributing to SDB

symptoms in preschool children.41 Parkkenin et al reported no association between size of

tonsils and increased AHI.45 We measured tonsil grades with a visual scale using a

Brodesky chart, which is a 2D method that measures the tonsils in a transverse direction.

This may not be as accurate as a 3D method that can measures not only the transverse but

also the anteroposterior and vertical dimensions.

According to our data, Angle classification cannot be used to determine SDB patients as

confirmed by previous studies.3, 29, 46 However, Parkkinen et al found a class II molar

relationship in children with SDB.49 When we controlled for age, gender and BMI, there

was no correlation between the Angle classification and cephalometric variables with

regards to SDB symptoms. The Angle classification shows the dental relationship

  53  
between the upper and lower first molars but it does not represent the skeletal facial type

of children. A child can have a Class I skeletal facial type with a Class II or a Class III

dental relationship often because of early loss of primary teeth. We feel that the Angle

classification alone without consideration of the mandibular and maxillary skeletal

relationship is not a good indicator of a patient’s facial and/or skeletal type.

A retruded or smaller mandible was one of the craniofacial features that had a significant

correlation with SDB symptoms in the children in our study and also in several other

studies.23, 29, 30, 40, 41, 45, 47, 48, 88 Lofstrand-Tidestrom et al found that a shorter lower dental

arch was significantly correlated with SDB in four year-old children.46 Children with

smaller mandibles and longer anterior facial heights showed more symptoms of SDB.89

Schiffman et al used magnetic resonance imaging (MRI) to determine the mandible

dimensions in both OSA and normal children. The 3D analysis of the mandible size and

shape showed no significant differences between the two groups. However, the position

of the mandible relative to the cranial base and facial skeleton was not evaluated in this

study.90 Kawashima et al reported an anterior displacement of the tongue base in patients

having large tonsils and a retruded mandible.23 However, one must always remember that

short mandibles in children may simply reflect normal growth and associations with SDB

should be interpreted with caution.

In an orthodontic clinic, cephalometric radiographs are routinely taken when patients are

awake and have a natural head posture. In this position, patients may position their

tongue more anteriorly for better respiration. While a patient is asleep, the muscles that

  54  
keep the tongue in a forward and upward position, such as the genioglossus and

palatoglossus, may become more relaxed and the subsequent retropositioning of the

tongue may contribute to the obstruction of the upper pharyngeal airway.

In addition to a retruded mandible, a steep mandibular plane angle was another important

craniofacial feature shown to have a significant correlation with SDB symptoms in our

study sample and in several other studies. 23, 29, 30, 40, 45, 47, 48, 88 We found that a steeper

mandibular plane angle (greater MP-SN angle) had a significant correlation with some of

the SDB symptoms observed in children such as impaired daytime function, loud snoring

and difficulty organizing tasks.

A lower position of the hyoid bone also had a significant correlation with SDB symptoms

in the present study as well as in many other studies.3, 40, 43, 45, 48, 73, 89 Kawashima et al

studied the position of the hyoid bone in males and females with SDB. They found that

the hyoid bone was located more anteriorly in relation to the third cervical vertebra in

male subjects compared with females.73 Finkelstein et al found a larger distance between

the hyoid bone and the mandibular plane of children with increasing severity of SDB.89

On the other hand, Bates et al found that the hyoid bone in OSA adult patients was

rotated counterclockwise and the distance from the most anterior superior part to the

mandibular plane was consequently decreased.91 Kawashima et al looked at patients with

normal faces, high angle faces, and low angle faces, and they found no significant

differences in the position of the hyoid bone in relation to the mandibular plane between

the three groups.88 Characteristics of a high angle face are a longer anterior facial height,

  55  
steeper mandibular plane, and tendency to an anterior open bite. Several studies have

shown that the above features are common among OSA subjects,29, 30, 42, 46 but in the

study by Kawashima et al, 2002 on normal subjects with three different facial types, the

position of the hyoid bone did not differ in high angle faces compared with the other two

groups. Our data showed that a lower position of the hyoid bone in children is

significantly correlated with several SDB symptoms, including loud snoring, breath-

holding during sleep, choking or gasping during sleep, restless sleep, and excessive

daytime sleepiness.

Van De Graaff et al studied the suprahyoid muscles of dogs and the musculature

relationship with upper airway resistance and concluded that the hyoid bone and muscles

could strongly affect upper airway flow resistance.92 The hyoid bone has no direct

attachment to the craniofacial bones. It attached to the mandible, cranial base, tongue and

pharyngeal cartilages by supra and infra hyoid muscles and tendons. Any change or

displacement on the supra or infra hyoid structures can change the position of the hyoid

bone. We assume that a retroposition of the mandible as seen in severe Class II skeletal

patients, a downward and backward rotation of the mandible as seen in high angle cases,

and a lower position of the tongue base may affect the position of the hyoid bone. Further

studies with 3D imaging in SDB children may be able to determine the contribution of a

lower position of hyoid bone to obstruction of the pharyngeal area.

We support the idea that a steep mandibular plane, and a lower position of the hyoid bone

are correlated and are characteristics of SDB children. Further study is needed to

  56  
understand the craniofacial development, airway dimensions and respiratory functions

and their correlations in growing OSA children with more varying degrees of SDB.

In accordance with our study, retroclined lower incisors in SDB patients have been

reported by a few other investigators to be an important cephalometric finding.41, 42 In the

present study, we found that retroclination of the lower incisors was correlated to SDB

symptoms to a higher degree in adolescent females compared with adolescent males.

A longer soft palate is another craniofacial feature that was found to be correlated with

SDB in children.3, 23, 40 Parkkinen et al compared normal children with children who

presented more severe SDB symptoms. They found that a longer and thicker soft plate

was correlated with OSA in children.45 However, in our study there was not a significant

correlation identified between the length of the soft palate and SDB symptoms.

Increased palatal height in patients with SDB is a craniofacial feature that has been

reported by several studies.3, 4, 46 Tsuda et al studied the height of the palatal vault by

measuring from orthodontic models and found a significant correlation between a higher

palatal vault and SDB symptoms in children.3 Another study by Parkkinen et al, analyzed

orthodontic models of three groups of children including OSA children and snoring

children compared with normal subjects as a control group. They found that there was no

statistically significant difference between all three groups in terms of the height of the

palate.49 We used a McKee analysis to measure the palatal height from lateral

cephalometrics by measuring the distance from the mesiobuccal cusp tip of the upper first

  57  
molar to the palatal plane (ANS-PNS line) in millimeters. We did not find a significant

correlation between palatal height and SDB symptoms in the whole sample or when we

controlled for age and gender. The technique we used to measure the palatal height as

well as the lower severity of SDB in our sample could be why we did not find a positive

relationship.

There are other potential limitations with our study. The sample consisted of a group of

children who were enrolled in orthodontic treatment clinics and who had a lower degree

of SDB disease severity. Measurement of tonsil size by a visual scale (Brodesky) may

have less accuracy than a 3D method even though this technique has been widely used in

previous studies. We did not quantify adenoid size because the majority of the children in

our sample were older than 6-7 years. Our sample consisted of some ethnicities that may

have different cephalometric variables. Even though validity of the OSA-18 and PSQ-22

questionnaire were assessed by Franco et al, 2000 and Chervin et al, 2000 respectively,

there potentially could be some bias in the inclusion criteria.

Several studies have suggested early orthodontic intervention for children with

craniofacial abnormalities and SDB symptoms.3, 23, 42, 59 The results of this study suggest

that SDB in children is a multifactorial disorder and these factors may vary based on a

child’s age and gender. We may need a multidisciplinary approach toward the diagnosis

and treatment of these cases to achieve better outcomes. According to the review of the

literature done by Marcus et al, the first line of treatment for OSAS in young children is

still adenotonsilectomy even though there might be some residual symptoms after

  58  
surgery.20 One important difference between SDB children and adults is that children are

still growing. Any factor that may interfere with a child’s development may have an

impact on their craniofacial features over the course of their lifetime. We recommend an

orthodontic examination of SDB children to obtain a proper diagnosis of the craniofacial

features and, if necessary, timely orthodontic treatment such as palatal expansion or

mandibular advancement appliances. Further studies are needed to find out if combining

adenotonsilectomy and orthodontic treatment in young children with craniofacial

abnormalities could have a significant impact on reducing SDB symptoms and achieving

more permanent results.

To improve our understanding of the causal relationship between craniofacial

morphology and SDB, a population-based study is needed with a large sample size

including different races, genders and age groups, with diagnosed SDB children and a

control group.

  59  
Chapter 5: Conclusion

 
Snoring, difficulty getting up, daytime sleepiness, morning headaches, hyperactivity and

frequent colds were the most common SDB symptoms observed in the SDB children in

our sample. The craniofacial features that had a significant relationship with SDB

symptoms were a lower position of the hyoid bone, a retruded mandible, steep

mandibular plane angle, and retroclined lower incisors. There was no statistically

significant relationship between SDB symptoms and the Angle classification, tonsil size,

Mallampati scores or BMI. Craniofacial morphology, but not severity of malocclusion,

could be an important contributing factor to the severity of SDB symptoms. SDB is a

multifactorial disease and warrants a multidisciplinary approach for a better diagnosis

and proper treatment. A child may present with some sleep related symptoms and specific

craniofacial characteristics that could be suggestive for SDB. The important role of the

dental practitioner is to recognize the need for further investigation and possible

treatment in the future to improve the quality of life of a child with SDB.

  60  
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Appendix

Appendix A

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A.1 OSA-18 Questionnaire
Please circle the number that best describes how often each symptom or problem has occurred during the
past four weeks.
Sleep Disturbances None of Hardly A little Some of A good Most of All of the
During the past 4 weeks how often the time any of the of the the time bit of the the time time
has your child had…. time time time

1….loud snoring? 1 2 3 4 5 6 7
2. …breath-holding spells or pauses 1 2 3 4 5 6 7
in breathing at night?
3…. choking or made gasping sounds 1 2 3 4 5 6 7
while asleep?
4…. restless sleep or frequent waking 1 2 3 4 5 6 7
from sleep?
Physical symptoms
During the past four weeks, how
often has your child had….
5… mouth breathing because of nasal 1 2 3 4 5 6 7
obstruction?
6…frequent colds or upper 1 2 3 4 5 6 7
respiratory infections?
7…. nasal discharge or runny nose 1 2 3 4 5 6 7

8…. difficulty in swallowing foods? 1 2 3 4 5 6 7

Emotional symptoms
During the last four weeks, how often
your child had…..
9…. mood swings or temper 1 2 3 4 5 6 7
tantrums?
10… aggressive or hyperactive 1 2 3 4 5 6 7
behavior?
11… discipline problems? 1 2 3 4 5 6 7
Daytime functions 1 2 3 4 5 6 7
During the past four weeks, how
often has your child had….
12… excessive daytime sleepiness? 1 2 3 4 5 6 7
13… a poor attention span or 1 2 3 4 5 6 7
concentration?
14…. difficulties getting up in the 1 2 3 4 5 6 7
morning?
Caregiver concerns 1 2 3 4 5 6 7
During the past four weeks, how
often have the problems described
above…
15…caused you to worry about your 1 2 3 4 5 6 7
child’s general health?
16… created concern that your child 1 2 3 4 5 6 7
is not getting enough air?
17… interfered with your ability to 1 2 3 4 5 6 7
perform daily activities?
18… made you frustrated? 1 2 3 4 5 6 7

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A. 2 PSQ-22 Questionnaire
For each of the questions below, please circle the answer that best describes your child’s condition.
While sleeping, does your child….
1. …snore more often than half the time? Yes No Don’t Know
2. …always snore? Yes No Don’t know
3. …snores loudly? Yes No Don’t know
4. … have “heavy” or loud breathing? Yes No Don’t know
5. …have trouble breathing, or struggle to breathe? Yes No Don’t know
6. …have you ever seen your child stop breathing at night? Yes No Don’t know

Does your child…

7. …tend to breathe through the mouth during the day? Yes No Don’t know
8. …have a dry mouth on waking up in the morning? Yes No Don’t know
9. …wake up feeling unrefreshed in the morning? Yes No Don’t know
10. …have a problem with sleepiness during the day? Yes No Don’t know
11. …has a teacher or other supervisor commented that your Yes No Don’t know
child appears sleepy during the day?
12. …is it hard to wake your child up in the morning? Yes No Don’t know

This child often…

13. …does not seem to listen when spoken to directly Yes No Don’t know
14. …has difficulty organizing task and activities Yes No Don’t know
15. …is easily distracted by extraneous stimuli Yes No Don’t know
16. …fidgets with hands or feet or squirms in seat Yes No Don’t know
17. …is ‘on the go’ or often acts as if ‘driven by a motor’ Yes No Don’t know
18. …interrupts or intrudes on others Yes No Don’t know
19. …does your child occasionally wet the bed Yes No Don’t know
20. …did your child wake up with morning headaches Yes No Don’t know
21. …did your child stop growing at the normal rate at any Yes No Don’t know
time since birth?
22. …is your child overweight? Yes No Don’t know

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