Anatomical Basis of Sleep-Related Breathing Abnormalities in Children with Nasal Obstruction
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ORIGINAL ARTICLE Anatomical Basis of Sleep-Related Breathing Abnormalities in Children With Nasal Obstruction Yehuda Finkelstein, MD; David Wexler, MD; Gilead Berger, MD; Ariela Nachmany, MA; Myra Shapiro-Feinberg, MD; Dov Ophir, MD Objective: To define, in a group of children with nasal tified in patients prone to obstructive breathing pat- obstruction, the anatomical differences that differenti- terns: increased flexure of the cranial base and bony na- ate those with quiet, unobstructed nocturnal respira- sopharynx, opening of the gonial angle, shortened tion from those with obstructive sleep-related breathing mandibular length, dorsocaudal location of the hyoid, re- abnormalities (snoring and obstructive sleep apnea). duced posterior airway space, and increased velar thick- ness. Design: Case series. Conclusions: A number of anatomical abnormalities may Patients: Fifty-nine children aged 3 to 13 years (35 boys contribute to sleep-related abnormal breathing in oth- and 24 girls) with nasal obstruction and without tonsil- erwise normal children with nasal obstruction. Our re- lar hypertrophy, known craniofacial syndromes, or neu- sults suggest that symptomatic children show some of romuscular diseases were included in the study. the same skeletal and soft-tissue configurations that are found in adults with obstructive sleep apnea. While ad- Main Outcome Measures: Each patient was catego- enoidectomy is generally an effective treatment in chil- rized as to severity of nocturnal obstructive breathing dren with obstructive sleep-related breathing abnormali- symptoms. Angular and linear cephalometric measure- ties, the underlying craniofacial variances that remain after ments were used for assessment of craniofacial features. adenoidectomy may predispose these patients to rede- Clinical symptom scores were correlated with the cepha- velopment of obstructive breathing abnormalities in adult- lometric measurements. hood. Results: Significant craniofacial abnormalities were iden- Arch Otolaryngol Head Neck Surg. 2000;126:593-600 ANIFESTATIONS of up- adenotonsillar hypertrophy. Children with per airway obstruc- adenoidal hypertrophy have nasal obstruc- tion are common in tion with consequent chronic mouth children. An esti- breathing. However, some of these chil- mated 12% of chil- dren present with mouth breathing as an dren exhibit habitual snoring,1 and about isolated manifestation, while others de- M 2 1% suffer from obstructive sleep apnea. velop SRBAs of varying severity, from snor- Snoring and other sleep-related breath- ing to obstructive sleep-disordered breath- ing abnormalities (SRBAs) may be asso- ing. In most of these children, snoring and ciated with deleterious effects, including SRBAs are cured if the obstruction of the From the Palate Surgery Unit impaired daytime psychomotor perfor- nose is removed, usually by adenoidec- (Dr Finkelstein and mance, enuresis, hyperactivity, and poor tomy. In our experience and according to Ms Nachmany), Department of sleep quality.1,3,4 Several predisposing ana- reports in the literature, parents report that, Otolaryngology–Head and tomical and physiologic factors have been after surgical treatment, “they now have Neck Surgery (Drs Wexler, identified in children with SRBAs. These a different child” who is more alert and ac- Berger, and Ophir), and include localized sites of narrowing in the tive in all respects.5 Department of Radiology upper airways, neuromuscular disorders, It is not yet known why some chil- (Dr Shapiro-Feinberg), Meir and major craniofacial abnormalities, such dren with obstructive adenoid hypertro- Hospital, Sapir Medical Center, Kfar Saba; and the Sackler as Stickler syndrome, Crouzon syn- phy develop SRBAs while others with iden- School of Medicine, Tel-Aviv drome, Treacher Collins syndrome, and tical nasal obstruction remain otherwise University, Tel Aviv Pierre Robin syndrome. asymptomatic chronic mouth breathers. A (Drs Finkelstein, Berger, and The broadest subset of children with lack of correlation between adenoid size Ophir); Israel. SRBAs comprises those with obstructive and severity of apnea has also been docu- ARCH OTOLARYNGOL HEAD NECK SURG/ VOL 126, MAY 2000 WWW.ARCHOTO.COM 593 ©2000 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 10/02/2021 PATIENTS AND METHODS In addition, special attention was paid to malocclusion, mainly anterior open bites and overjet of the maxillary in- PATIENTS cisors. Lateral cephalometric radiographs were obtained for Fifty-nine healthy children ranging in age from 3 to 13 years all subjects. For 38 patients, the radiographs were taken (mean ± SD, 8.6±3.1 years) were included in this study. in an authorized laboratory. Standard technique was em- There were 35 boys and 24 girls. Forty-six patients se- ployed: The patients were instructed to fix their distant gaze lected had nasal obstruction caused by adenoid hypertro- on an imaginary horizon to reproduce their habitual oc- phy and 3 patients had obstructions caused by severe chronic clusion, with the lips together, and to allow the tongue to rhinitis with symptoms persisting at least 3 months. Ex- relax in the floor of the mouth. Exposures were taken with clusion criteria were tonsillar enlargement, personal or fam- the patient slowly exhaling through the nose. Exposures ily history of neuromuscular disorder or craniofacial syn- were optimized to demonstrate both the bony landmarks drome, and obesity. and the superimposed soft tissues. For the remaining 21 patients, the cephalometric measurements were made from METHODS lateral head radiographs. To allow correction for projec- tion enlargement of the linear measurements, these radio- The determination of obesity was made according to pe- graphs were made with a 10-mm round steel median cali- diatric growth charts. A thorough history was recorded for bration marker in place. The head was oriented in the each patient following a structured interview format ap- Frankfurt plane. plied in a consistent fashion. Each patient was accord- The cephalometric landmarks, angles, and linear mea- ingly classified as to level of obstructive symptom severity: surements are defined in Table 1 and shown in Figure 2. Grade Symptoms 0 Oral breathing with no snoring STATISTICAL ANALYSIS 1 Mild snoring or snoring only while sleeping on back Mean±SEM was calculated for each variable at each level 2 Habitual snoring in all positions of symptom severity. Correlation coefficients were deter- 3 Habitual snoring associated with a history of mild mined by the Pearson method for the associations be- to moderate apnea and/or restless sleep tween the cephalometric variables and symptom severity. 4 Habitual snoring associated with a The Pearson correlation method was also used to test the marked clinical presentation of apnea and restless sleep relationships of the craniofacial variables with age and the relationships of each craniofacial variable with the others. Ears, nose, and throat examination was performed with Analysis of variance was used to determine statistically sig- special attention to mouth breathing, daytime loud oral nificant differences of individual cephalometric variables breathing during wakefulness, adenoidal face (ie, long face), at each level of symptom severity. Between-sex differ- steep mandibular planes, and receding chin (Figure 1). ences were tested by independent t test. mented.6 These discrepancies point to a lack of under- Cephalometric analysis has been used to character- standing of the substrates of SRBAs in children. ize skeletal and soft tissue relationships in adults with Two hypothetical mechanisms have been sug- SRBAs.9-14 Cephalometry can provide extensive data on gested to explain how nasal obstruction may induce the landmarks pertinent to the upper airways. However, SRBAs.7 According to the functional theory, upper air- correlative data between cephalometric parameters and way obstruction could lead to apneas via disturbed re- children with SRBA symptoms are sparse. Therefore, we flex mechanisms, possibly trigeminally or vagally medi- studied a series of children with chronic nasal obstruc- ated, that normally act to preserve airway patency in the tion without tonsillar hypertrophy, without any known presence of negative pressure in the upper airway.8 Con- craniofacial anomaly, and without hypotonia or neuro- tributing factors might be poor tone of the pharynx and muscular disease. tongue during sleep, leading to collapse of the orophar- Our hypothesis is that specific anatomical patterns ynx and obstruction. According to the mechanical theory, correlate with obstructive symptom severity in children with a direct mechanical effect—ie, with obstruction or with nasal obstructions. The present study was de- narrowing of the upper airway—the pressure gradient for signed to define the anatomical markers by craniofacial airflow increases and the pressures within the upper air- cephalometric evaluation of nonsyndromic young pa- way become more negative relative to the atmospheric tients who develop SRBAs in the presence of nasal ob- pressure. This could lead to a greater tendency toward struction. This is one component of our research pro- airway closure. gram on development of a general structural analytic We believe that airway stability during sleep is re- model of sleep-related upper airway obstruction. lated to structural parameters of the upper airways and that even the airflow-related soft tissue movements dur- RESULTS ing sleep are influenced by the underlying