顱顏攝影、齒列咬合型態、測顱 X 光片與兒童睡眠呼吸障礙之關聯性研究

Association between craniofacial photogrammetry, dental arch morphology, and cephalometry in children with sleep-disordered breathing

薛婉儀(Wan-Yi Hsueh)

指導教授 ： 許巍鐘

國立臺灣大學/醫學院/臨床醫學研究所/碩士(2020年)

https://doi.org/10.6342/NTU202002210

摘要

背景
「腺樣體面容（adenoid facies）」是臨床上常見的特殊臉型，此種典型的臉部樣貌被認為和兒童睡眠呼吸中止症有關。這樣的關聯性讓我們聯想到顱顏形態可能是造成兒童睡眠呼吸中止症的重要因子。現有用以評估顱顏構造的工具包括測顱Ｘ光片(cephalometry)、電腦斷層(CT)、及核磁共振(MRI)，其缺點為價格高昂，具輻射線暴露，排檢時間過長。因此, 發展一項非侵入性,且可近性高的替代檢查實屬必要，而顱顏攝影(craniofacial photogrammetry)即為具備這些優點的檢查方法。顱顏攝影參數與睡眠呼吸中止症在成人已有文獻證實關聯性，唯在兒童睡眠呼吸中止症仍缺乏相關研究。本研究旨在探討顱顏攝影參數是否與兒童阻塞性睡眠呼吸中止症相關, 進而發展取代傳統檢查的方法，提高兒童睡眠呼吸中止症篩檢的普及率。
目的
本研究的第一個目標是找出顱顏攝影、測顱X光片、齒列咬合型態在兒童睡眠呼吸障礙不同嚴重度族群中具統計學上顯著差異的參數。第二個目標是檢驗顱顏攝影和測顱Ｘ光片所得參數的關聯性，期望顱顏攝影能取代測顱X光片作為兒童睡眠呼吸障礙的篩檢工具。
方法
本研究採前瞻性橫斷面研究，收集2019年4月到2020年2月間因打呼至台大醫院兒童耳鼻喉科就診，年齡3-18歲的兒童及青少年。若有張口呼吸或打呼症狀則納入收案。此外需接受睡眠多項生理檢查、顱顏攝影、測顱X光片及齒列咬合測量。受試者睡眠呼吸障礙的嚴重度依據睡眠多項生理檢查的結果分成單純打呼( primary snoring, AHI<1)、輕度睡眠呼吸中止症( mild OSA, 1≦AHI <5)、中重度睡眠呼吸中止症( moderate-severe OSA, AHI≧ 5)。顱顏攝影和測顱X光片的影像分別經過位點標定後,以軟體進行角度及長度的測量，所得到的顱顏及頸部下頦角參數與齒列咬合測量數據再經由統計方法找出具統計上顯著差異的參數。研究對象依據睡眠呼吸障礙的嚴重度分成三個模組進行分析，分析模組一: 單純打呼（AHI <1）和睡眠呼吸中止症組（AHI≥1）, 兩組患者的年齡，性別和BMI百分比經傾向分數配對後進行分析;分析模組二：單純打呼（AHI <1）、輕度睡眠呼吸中止症組（1≦AHI <5）、中重度睡眠呼吸中止症組( moderate-severe OSA, AHI≧ 5);分析模組三: 單純打呼（AHI <1）和中重度睡眠呼吸中止症組( moderate-severe OSA, AHI≧ 5)。邏輯回歸（Logistic regression）用來檢驗分析模組一中顱顏攝影、測顱X光片及齒列咬合型態各項參數數值增加時，發生睡眠呼吸中止症（AHI≥1）的勝算是否上升; 在分析模組三中用來檢驗各項參數數值增加時，發生中重度睡眠呼吸中止症（AHI≥5）的勝算是否上升。單因子變異數分析(One-way ANOVA)用於檢驗分析模組二中各項參數在睡眠呼吸障礙不同嚴重度間是否具有顯著差異。線性迴歸（linear regression）則是用來分析模組二中各項參數增加時與睡眠呼吸中止症嚴重度（log AHI）的線性關係。皮爾森積差相關分析(Pearson correlation)用以比較顱顏攝影與測顱X光片間的關聯性。分析結果以年齡、性別和BMI百分比經配對的模組一為主要判讀依據，模組二及三的結果提供判讀參考。
結果
本研究收案共納入103位兒童（平均年齡7.332.97歲；男孩75位），包括非睡眠呼吸中止症30位，輕度睡眠呼吸中止症47位和中度重度睡眠呼吸中止症26位。分析模組一是找出單純打呼（AHI <1）和睡眠呼吸中止症組（AHI≥1）之間各項檢查上具統計顯著差異的參數。兩組患者的年齡，性別和BMI百分比經統計配對後，每組包括29位受試者。根據邏輯回歸，下頷傾斜度（mego-tn，tn-gogn, gome-tsup）在顱顏攝影測量中具有顯著意義（分別為OR = 1.10、1.08、1.13, p <0.05）；測顱X光片中具統計顯著的參數為上咽氣道空間（SPAS）與中咽氣道空間（MAS）（分別為OR = 0.64、0.85, p <0.05）; 代表下頷傾斜度的參數(PP-MP、MP-SN、Sn-GoGn) 雖然在睡眠呼吸中止症組中大於單純打呼組，但未達統計顯著差異。在齒列咬合型態的部分雖然未發現具統計上顯著差異的參數, 但上顎寬度在犬齒、第一臼齒及第二臼齒間的距離皆是睡眠呼吸中止症組小於單純打呼組。顱顏攝影與測顱X光片之間各參數的相關性在下頷傾斜度（mandibular plane angle）及下顎後縮(retrusive mandible)兩項呈現中度相關; 在上顎長度(maxillary length)、下顎長度(mandibular length)、及前臉高度(anterior facial height)呈現強相關。
結論
顱顏攝影是評估兒童睡眠呼吸中止症具非侵入性、無輻射暴露、可近性高優點的新方法。本研究利用顱顏攝影發現下頜傾斜度增加與兒童睡眠呼吸中止症相關，是潛在可用於預測兒童睡眠呼吸中止症的顱顏特徵。但在齒列咬合型態測量的部分未發現具統計上顯著差異的參數。顱顏攝影與測顱X光片各參數間的相關性為中度至強相關。相對於測顱X光片，顱顏攝影也是一項適合評估顱顏構造的工具。為了證明顱顏攝影的有效性，未來還需要納入更多受試者進一部分析，且須配合適當的分組分層研究。

關鍵字

兒童睡眠呼吸中止症 ； 顱顏攝影 ； 測顱X光片 ； 齒列咬合

並列摘要

Introduction: “Adenoid facies” is a typical facial feature of pediatric sleep disordered breathing(SDB). The special characteristics give us a hint that craniofacial morphology might be an important predisposing factor of pediatric SDB. While cephalometry, computed tomography (CT) and magnetic resonance imaging (MRI) are recommended for evaluating craniofacial structures, these techniques were limited in clinical application due to radiation exposure, cost, or long waiting list.(Kaditis et al., 2016; R. W. Lee, Chan, Grunstein, Cistulli, 2009) Evolving an alternative technique with advantages of being noninvasive and readily accessible is warranted. Meanwhile, craniofacial photogrammetry seems a good candidate. Previous investigations have demonstrated craniofacial phenotypic differences in OSA adults by using photographic analysis technique. The information about children is lacking. Therefore, we conducted this study to assess the craniofacial morphology of children with and without obstructive sleep apnea (OSA) by the novel craniofacial phtogrammetry technique.

Objectives:The primary aim of this study is to see whether parameters of craniofacial photogrammetry, orthodontic measurements, and cephalometry differed between SDB groups. The additional goal is to compare craniofacial photogrammetry with cephalometry as being an alternative method for evaluating SDB.
Material and methods: Prospective, cross-sectional study. Children aged 3-18 years visit for snoring underwent routine ENT examinations. Craniofacial photogrammetry, cephalometry, orthodontic examinations, and overnight polysomnography(PSG) were arranged. Participants were stratified according to PSG diagnosis (primary snoring: AHI<1; OSA: AHI≥1) (mild OSA: 1≤AHI <5; moderate-severe OSA:AHI≥5). Craniofacial photogrammetry and cephalometry were analysed to derive craniofacial and cervicomental measurements. Orthodontic measurements were also performed. Analytic Model I, II, III were set up for statistical analysis according to SDB severity. Pearson correlation was performed to compare craniofacial photogrammetry and cephalometry in assessing craniofacial measurements of these eight categories: maxillary length, retrusive maxilla, mandibular length, mandibular angle, mandibular plane angle, retrusive mandible, anterior face height, and facial convexity. The result interpretation was mainly based on analytic Model I due to propensity score matched by age, sex, and BMI percentile between the two groups (AHI<1 vs. AHI≥1).
Results: A total of 103 children were enrolled (mean age, 7.332.97 years; 75 boys), including 30 primary snoring group, 47 mild OSA group, and 26 moderate-severe OSA group. Analytic Model I was to demonstrate parameters with significant difference between primary snoring group(AHI<1) and OSA group(AHI ≥ 1). The two groups were matched for age, sex, and BMI percentile, and 29 subjects were included in each group. Logistic regression was applied on Model I to see association between variables and OSA. Mandibular inclination(mego-tn, tn-gogn, gome-tsup) was noted with significant difference by craniofacial photogrammetry (odds ratio [OR] = 1.10、1.08、1.13, p <0.05). Significant decreased superior pharyngeal airway space(SPAS) and middle pharyngeal airway space (MAS) in OSA group was found by cephalometry (OR= 0.64 and 0.85 respectively, p <0.05) . No significant variable of orthodontic measurements was identified. The correlation between craniofacial photogrammetry and cephalometry was moderate in mandibular plane angle and retrusive mandible; strong correlation was demonstrated in maxillary length, mandibular length, and anterior face height.
Conclusion: Craniofacial photogrammetry is a novel technique with advantages of easily accessible and free from radiation. Increased mandibular inclination is a potential predictor for pediatric OSA by craniofacial photogrammetry. No significant variable of orthodontic measurements was identified in this study. Correlation between craniofacial photogrammetry and cephalometry was moderate to strong. Further investigation with larger sample size and adequate subgroup stratification is needed to clarify validity of craniofacial photogrammetry.

並列關鍵字

SDB ； OSA ； child ； sleep ； craniofacial photogrammetry ； cephalometry ； orthodontic ； dental

參考文獻

Adel, M., Yamaguchi, T., Nadim, M., Tomita, D., Hikita, Y. (2016). Evaluation of the craniofacial morphology of Egyptian adults undergoing orthodontic treatment. Dentistry, 6(379), 2161-1122.1000.

Amonoo-Kuofi, K., Phillips, S. P., Randhawa, P. S., Lane, R., Wyatt, M. E., Leighton, S. E. (2009). Adenotonsillectomy for sleep-disordered breathing in children with syndromic craniosynostosis. Journal of Craniofacial Surgery, 20(6), 1978-1980.

Bannink, N., Nout, E., Wolvius, E. B., Hoeve, H. L., Joosten, K. F., Mathijssen, I. M. (2010). Obstructive sleep apnea in children with syndromic craniosynostosis: long-term respiratory outcome of midface advancement. International journal of oral and maxillofacial surgery, 39(2), 115-121.

Berry, R. B., Budhiraja, R., Gottlieb, D. J., Gozal, D., Iber, C., Kapur, V. K., . . . Tangredi, M. M. (2012). Rules for scoring respiratory events in sleep: update of the 2007 AASM Manual for the Scoring of Sleep and Associated Events. Deliberations of the Sleep Apnea Definitions Task Force of the American Academy of Sleep Medicine. J Clin Sleep Med, 8(5), 597-619. doi:10.5664/jcsm.2172

Bonuck, K., Freeman, K., Henderson, J. (2009). Growth and growth biomarker changes after adenotonsillectomy: systematic review and meta-analysis. Archives of disease in childhood, 94(2), 83-91.