Obstructive Sleep Apnoea in Children with Down Syndrome: A Multidisciplinary Approach
Abstract
:1. Introduction
2. Materials and Methods
2.1. Parent Report of Sleep by the CSHQ-IT
2.2. ENT Examination
- Palatine tonsillar hypertrophy evaluation. Tonsil size was graded from 1 to 4: size 1 if tonsils were hidden within the pillars, size 2 if tonsils extended to the pillars, size 3 if tonsils extended beyond the pillars but not to the midline (Figure 1a), and size 4 if tonsils extended to the midline [14].
- Nasal flexible fibreoptic endoscopy (Storz, Tuttlingen, Germany) without decongestant or local anaesthesia to evaluate the following:
- (a)
- Adenoid tonsil hypertrophy. The adenoid tonsil size was graded into four degrees, i.e. Grade I when fibreoptic endoscopy imaging revealed adenoid tissue occupying only the upper segment of the nasopharyngeal cavity (≤25%) with almost completely free choanal openings; Grade II if the adenoid tissue was confined to the upper half part (≤50%) of the nasopharyngeal cavity with sufficiently pervious choana and perfect visualization of the tube ostium; Grade III if adenoid tissue occupied around 75% of the nasopharynx with partial involvement of the tube ostium and considerable obstruction of choanal openings; Grade IV if the adenoid tissue reached the lower choanal border without allowing visualization of the tube ostium (>75%) (Figure 1b) [15].
- (b)
- Presence or absence of nasal turbinate hypertrophy or septum deviation.
- (c)
- Nasal mucosa oedema, secretions, and polyps according to the Lund and Kennedy quantifying system [16]:
- (c.1.)
- Nasal mucosal oedema: 0, absent; 1, mild/moderate; 2, polypoid degeneration.
- (c.2.)
- Secretion: 0, absent; 1, hyaline; 2, thickened and/or mucopurulent.
- (c.3.)
- Polyps: 0, absent; 1, restricted to the middle meatus; 2, extending to the nasal cavity.
2.3. Orthodontic Evaluation
- Facial profile, classified as convex, concave, or orthognathic [17].
- Molar relationship, classified on both the left and right sides, according to Angle’s classification as Class I, II, or III [18].
- Overbite (OVB), i.e. the extent of vertical overlap of the maxillary central incisors over the mandibular central incisors, which was measured by an intra-oral ruler. A final value of ≥4 mm and ≤0 mm indicated deep bite and open bite, respectively, while 0 < OVB < 4 was considered normal [18].
- Transversal molar discrepancy with skeletal contraction, recorded on both sides in centric relation, is classified as unilateral or bilateral posterior cross-bite [19].
2.4. Sleep Study
- The overnight PG was performed using a cardiorespiratory device (Embletta MPR, Medcare Flaga, Reykjavík, Iceland) for a period of at least 5 h. Patients went to bed at a time of their preference, and studies were terminated when they awoke spontaneously in accordance with their home wake times. The following parameters were recorded: airflow through nasal pressure transducer, oxygen saturation by pulse oximetry (SpO2), pulse signals, thoracic and abdominal movements by inductance plethysmography, and body position. We manually scored PG according to the American Academy of Sleep Medicine Paediatric criteria [21]. Obstructive apnoea was defined as the presence of continued inspiratory effort associated with a >90% decrease in airflow for a duration of ≥2 breaths. Hypopnoea was defined as a ≥30% decrease in airflow for a duration of ≥2 breaths associated with a decrease in SpO2 by ≥3%. The PG results included the following:Apnoea–hypopnoea index (AHI), defined as the sum of all obstructive, central, and mixed apnoeas and hypopnoeas divided by hours of total sleep time.
- Oxygen desaturation index (ODI), defined as the number of times per sleep hour with SpO2 decrease ≥3%.
- Mean SpO2.
- Percentage of the total recording time spent with SpO2 below 90% (T90).
2.5. Statistical Analysis
3. Results
4. Discussion
Supplementary Materials
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Conflicts of Interest
References
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Total Score | 63 ± 5.1 |
---|---|
Bedtime resistance (items 1-3-4-5-6-8) | 11.3 ± 3.3 |
Sleep onset delay (item 2) | 1.4 ± 0.7 |
Sleep anxiety (items 5-7-8-21) | 7.3 ± 2.2 |
Sleep duration (items 9-10-11) | 4.6 ± 1.6 |
Night wakings (items 16-24-25) | 4.8 ± 1.6 |
Parasomnia (items 12-13-14-15-17-22-23) | 9.8 ± 1.8 |
Sleep-disordered breathing (items 18-19-20) | 4.4 ± 1.7 |
Daytime sleepiness (items 26-27-28-29-30-31-32-33) | 14.5 ± 3.5 |
n | % | ||
---|---|---|---|
Palatine tonsil hypertrophy size | Size 1 | 3 | 9 |
Size 2 | 10 | 29 | |
Size 3 | 14 | 41 | |
Size 4 | 7 | 21 | |
Adenoid tonsil hypertrophy size | grade I | 5 | 15 |
grade II | 14 | 41 | |
grade III | 15 | 44 | |
grade IV | 0 | 0 | |
Chronic rhinosinusitis score | 0 | 1 | 3 |
2 | 4 | 12 | |
4 | 11 | 32 | |
6 | 9 | 26 | |
8 | 9 | 26 | |
Nasal turbinate hypertrophy | Present | 23 | 68 |
Absent | 11 | 32 | |
Nasal septum deviation | Present | 17 | 50 |
Absent | 17 | 50 |
n | % | ||
---|---|---|---|
Facial profile | Orthognathic | 32 | 68 |
Concave | 10 | 21 | |
Convex | 5 | 11 | |
Molar class relationship | I | 24 | 63 |
II | 6 | 16 | |
III | 8 | 21 | |
NA * | 9 | - | |
Overbite | Normal | 38 | 81 |
Open bite | 8 | 17 | |
Deep bite | 1 | 2 | |
Cross-bite | 24 | 51 | |
Mother-reported sleep bruxism | 22 | 47 | |
Oral parafunctional behaviours | 14 | 30 |
Total sleep time (min) | 480 |
(478; 480) | |
Obstructive apnea index | 0 |
(0; 1.5) | |
Central apnea index | 0 |
(0; 0) | |
Hypopnea index | 1 |
(0; 2.75) | |
Apnea-hypopnea index | 2.2 |
(0.9; 5.1) | |
Oxygen desaturation index | 4 |
(1.3; 7.6) | |
Mean SpO2 saturation (%) | 96.3 |
(95.6; 97) | |
T90 | 0.2 |
(0; 1.5) |
Total Score | Bedtime Resistance | Sleep Onset Delay | Sleep Duration | Sleep Anxiety | Night Wakings | Parasomnias | Sleep-Disordered Breathing | Daytime Sleepiness | |
---|---|---|---|---|---|---|---|---|---|
AHI | 0.07 | 0.04 | −0.05 | 0.09 | 0.01 | −0.04 | −0.13 | 0.16 | 0.12 |
ODI | −0.01 | 0.13 | −0.18 | 0.09 | −0.01 | −0.14 | −0.28 | 0.04 | 0.13 |
Mean SpO2 | −0.17 | 0.05 | 0.02 | −0.22 | 0.14 | −0.18 | 0.03 | −0.35 * | −0.14 |
T90 | 0.11 | −0.03 | −0.04 | 0.33 * | 0.01 | 0.23 | −0.06 | 0.19 | −0.07 |
Adenoid Tonsils Hypertrophy Size | Palatine Tonsils Hypertrophy Size | Nasal Turbinate Hypertrophy | Nasal Septum Deviation | |||||||||
---|---|---|---|---|---|---|---|---|---|---|---|---|
I/II Grade | III Grade | p * | I/II Grade | III/IV Grade | p * | Present | Absent | p * | Present | Absent | p * | |
AHI | 1 (0.6; 4.5) | 5 (0.9; 8.1) | 0.04 | 1.5 (0.6; 5.5) | 2.9 (0.9; 7.1) | 0.36 | 2.1 (0.9; 5.5) | 2.2 (0.8; 6) | 1 | 2.4 (1; 6.2) | 1.5 (0.8; 5.5) | 0.62 |
ODI | 4 (1.8; 5.7) | 8 (1.7; 9.6) | 0.08 | 4.1 (2.3; 6.7) | 4.6 (1.4; 8.9) | 0.92 | 4.7 (1.7; 8.6) | 2.6 (1.5; 8.3) | 0.60 | 4.9 (2; 8.8) | 2.6 (1.6; 8.3) | 0.59 |
Mean SpO2 | 96.3 (95.5; 97) | 96.3 (94.8; 97.1) | 0.58 | 96.1 (95; 96.8) | 96.7 (95.7; 97.1) | 0.27 | 96.3 (95.6; 97.1) | 96.7 (94.3; 97) | 0.71 | 96.3 (95.7; 96.8) | 96.7 (94.5; 97.1) | 0.62 |
T90 | 0.1 (0; 0.2) | 1.2 (0.1; 3.4) | 0.01 | 0.2 (0; 1.8) | 0.1 (0; 1.8) | 0.75 | 0.1 (0; 1.5) | 0.3 (0; 7.7) | 0.46 | 0.1 (0; 1.4) | 0.4 (0; 4.9) | 0.12 |
Group A * (n = 6; 15.8%) | Group B # (n = 32; 74.2%) | p ** | |
---|---|---|---|
Apnea-hypopnea index | 7.1 (4.3; 9.4) | 1.4 (0.8; 4.6) | 0.01 |
Oxygen desaturation index | 8.9 (4.6; 10.2) | 3.3 (1.2; 7.1) | 0.04 |
Mean SpO2 | 96.3 (91.9; 97.3) | 96.3 (95.6; 96.9) | 0.98 |
T90 | 0.1 (0; 26.1) | 0.2 (0; 2) | 0.76 |
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Borrelli, M.; Corcione, A.; Rongo, R.; Cantone, E.; Scala, I.; Bruzzese, D.; Martina, S.; Strisciuglio, P.; Michelotti, A.; Santamaria, F. Obstructive Sleep Apnoea in Children with Down Syndrome: A Multidisciplinary Approach. J. Pers. Med. 2023, 13, 71. https://doi.org/10.3390/jpm13010071
Borrelli M, Corcione A, Rongo R, Cantone E, Scala I, Bruzzese D, Martina S, Strisciuglio P, Michelotti A, Santamaria F. Obstructive Sleep Apnoea in Children with Down Syndrome: A Multidisciplinary Approach. Journal of Personalized Medicine. 2023; 13(1):71. https://doi.org/10.3390/jpm13010071
Chicago/Turabian StyleBorrelli, Melissa, Adele Corcione, Roberto Rongo, Elena Cantone, Iris Scala, Dario Bruzzese, Stefano Martina, Pietro Strisciuglio, Ambrosina Michelotti, and Francesca Santamaria. 2023. "Obstructive Sleep Apnoea in Children with Down Syndrome: A Multidisciplinary Approach" Journal of Personalized Medicine 13, no. 1: 71. https://doi.org/10.3390/jpm13010071
APA StyleBorrelli, M., Corcione, A., Rongo, R., Cantone, E., Scala, I., Bruzzese, D., Martina, S., Strisciuglio, P., Michelotti, A., & Santamaria, F. (2023). Obstructive Sleep Apnoea in Children with Down Syndrome: A Multidisciplinary Approach. Journal of Personalized Medicine, 13(1), 71. https://doi.org/10.3390/jpm13010071