Objective Measurements for Upper Airway Obstruction in Infants with Robin Sequence: What Are We Measuring? A Systematic Review

Overview

Journal J Clin Sleep Med

Specialties Neurology
Psychiatry

Date 2021 May 7

PMID 33960296

Citations 8

Authors

Robrecht J H Logjes

Joanna E MacLean

Noor W de Cort

Christian F Poets

Veronique Abadie

Koen F M Joosten

Cory M Resnick

Ivy K Trindade-Suedam

Carlton J Zdanski

Christopher R Forrest

Frea H Kruisinga

Roberto L Flores

Kelly N Evans

Corstiaan C Breugem

Affiliations

Soon will be listed here.

Abstract

Study Objectives: Identifying optimal treatment for infants with Robin sequence (RS) is challenging due to substantial variability in the presentation of upper airway obstruction (UAO) in this population. Objective assessments of UAO and treatments are not standardized. A systematic review of objective measures of UAO was conducted as a step toward evidence-based clinical decision-making for RS.

Methods: A literature search was performed in the PubMed and Embase databases (1990-2020) following PRISMA guidelines. Articles reporting on RS and UAO treatment were included if the following objective measures were studied: oximetry, polysomnography, and blood gas. Quality was appraised by the methodological index for nonrandomized studies (range: 0-24).

Results: A total of 91 articles met the inclusion criteria. The mean methodological index for nonrandomized studies score was 7.1 (range: 3-14). Polysomnography was most frequently used (76%) followed by oximetry (20%) and blood gas (11%). Sleep position of the infant was reported in 35% of studies, with supine position most frequently, and monitoring time in 42%, including overnight recordings, in more than half. Of 71 studies that evaluated UAO interventions, the majority used polysomnography (90%), of which 61% did not specify the polysomnography technique. Reported polysomnography metrics included oxygen saturation (61%), apnea-hypopnea index (52%), carbon dioxide levels (31%), obstructive apnea-hypopnea index (27%), and oxygen desaturation index (16%). Only 42 studies reported indications for UAO intervention, with oximetry and polysomnography thresholds used equally (both 40%). In total, 34 distinct indications for treatment were identified.

Conclusions: This systematic review demonstrates a lack of standardization, interpretation, and reporting of assessment and treatment indications for UAO in RS. An international, multidisciplinary consensus protocol is needed to guide clinicians on optimal UAO assessment in RS.

Citation: Logjes RJH, MacLean JE, de Cort NW, et al. Objective measurements for upper airway obstruction in infants with Robin sequence: what are we measuring? A systematic review. . 2021;17(8):1717-1729.

Citing Articles

Diagnosis and Early Management of Robin Sequence.

Rickart A, Sikdar O, Jenkinson A, Greenough A Children (Basel). 2024; 11(9).

PMID: 39334626 PMC: 11430236. DOI: 10.3390/children11091094.

Nonsurgical improvement of severe upper airway obstruction in infants with Robin sequence and cleft palate using Stanford orthodontic airway plate treatment.

Choo H, Sidell D, Kim J, Ahn H, Day H, Sullivan S J Clin Sleep Med. 2024; 20(11):1807-1817.

PMID: 38963072 PMC: 11530975. DOI: 10.5664/jcsm.11282.

Respiratory outcomes after cleft palate closure in Robin sequence: a retrospective study.

Sullivan N, Lachkar N, Don Griot J, Kruisinga F, Leeuwenburgh-Pronk W, Broers C Clin Oral Investig. 2024; 28(5):247.

PMID: 38602599 PMC: 11008067. DOI: 10.1007/s00784-024-05647-w.

Spectrum of Disease in Hospitalized Newborns with Congenital Micrognathia: A Cohort of 3,236 Infants at North American Tertiary-Care Intensive Care Units.

Padula M, Naing K, Wenger T, Ahmad I, Coghill 3rd C, Wild K J Pediatr. 2023; 265:113799.

PMID: 37879601 PMC: 10872910. DOI: 10.1016/j.jpeds.2023.113799.

Non-Surgical Respiratory Management in Relation to Feeding and Growth in Patients with Robin Sequence; a Prospective Longitudinal Study.

van der Plas P, van Heesch G, Koudstaal M, Pullens B, Mathijssen I, Bernard S Cleft Palate Craniofac J. 2023; :10556656231199840.

PMID: 37728101 PMC: 11580327. DOI: 10.1177/10556656231199840.

References

Paes E, van Nunen D, Basart H, Don Griot J, van Hagen J, van der Horst C . Birth prevalence of Robin sequence in the Netherlands from 2000-2010: a retrospective population-based study in a large Dutch cohort and review of the literature. Am J Med Genet A. 2015; 167A(9):1972-82. DOI: 10.1002/ajmg.a.37150. View

Slim K, Nini E, Forestier D, Kwiatkowski F, Panis Y, Chipponi J . Methodological index for non-randomized studies (minors): development and validation of a new instrument. ANZ J Surg. 2003; 73(9):712-6. DOI: 10.1046/j.1445-2197.2003.02748.x. View

Wilson A, Moore D, Moore M, Martin A, Staugas R, Kennedy J . Late presentation of upper airway obstruction in Pierre Robin sequence. Arch Dis Child. 2000; 83(5):435-8. PMC: 1718546. DOI: 10.1136/adc.83.5.435. View

van Lieshout M, Joosten K, Mathijssen I, Koudstaal M, Hoeve H, van der Schroeff M . Robin sequence: A European survey on current practice patterns. J Craniomaxillofac Surg. 2015; 43(8):1626-31. DOI: 10.1016/j.jcms.2015.07.008. View

Katz S, Witmans M, Barrowman N, Hoey L, Su S, Reddy D . Paediatric sleep resources in Canada: The scope of the problem. Paediatr Child Health. 2014; 19(7):367-72. PMC: 4173906. DOI: 10.1093/pch/19.7.367. View

Hong H, Wee C, Haynes K, Urata M, Hammoudeh J, Davidson Ward S . Evaluation of Obstructive Sleep Apnea in Prone Versus Nonprone Body Positioning With Polysomnography in Infants With Robin Sequence. Cleft Palate Craniofac J. 2019; 57(2):141-147. DOI: 10.1177/1055665619867228. View

Kaditis A, Alonso Alvarez M, Boudewyns A, Abel F, Alexopoulos E, Ersu R . ERS statement on obstructive sleep disordered breathing in 1- to 23-month-old children. Eur Respir J. 2017; 50(6). DOI: 10.1183/13993003.00985-2017. View

Coutier L, Guyon A, Reix P, Franco P . Impact of prone positioning in infants with Pierre Robin sequence: a polysomnography study. Sleep Med. 2019; 54:257-261. DOI: 10.1016/j.sleep.2018.10.037. View

Kimple A, Baldassari C, Cohen A, Landry A, Ishman S . Polysomnographic results of prone versus supine positioning in micrognathia. Int J Pediatr Otorhinolaryngol. 2014; 78(12):2056-9. DOI: 10.1016/j.ijporl.2014.08.042. View

10.

Breugem C, Evans K, Poets C, Suri S, Picard A, Filip C . Best Practices for the Diagnosis and Evaluation of Infants With Robin Sequence: A Clinical Consensus Report. JAMA Pediatr. 2016; 170(9):894-902. DOI: 10.1001/jamapediatrics.2016.0796. View

11.

DeHaan K, Seton C, Fitzgerald D, Waters K, MacLean J . Polysomnography for the diagnosis of sleep disordered breathing in children under 2 years of age. Pediatr Pulmonol. 2015; 50(12):1346-53. PMC: 6680200. DOI: 10.1002/ppul.23169. View

12.

Carpenter R, Irgens L, Blair P, England P, Fleming P, Huber J . Sudden unexplained infant death in 20 regions in Europe: case control study. Lancet. 2004; 363(9404):185-91. DOI: 10.1016/s0140-6736(03)15323-8. View

13.

Duenas-Meza E, Bazurto-Zapata M, Gozal D, Gonzalez-Garcia M, Duran-Cantolla J, Torres-Duque C . Overnight Polysomnographic Characteristics and Oxygen Saturation of Healthy Infants, 1 to 18 Months of Age, Born and Residing At High Altitude (2,640 Meters). Chest. 2015; 148(1):120-127. PMC: 4493868. DOI: 10.1378/chest.14-3207. View

14.

Daftary A, Jalou H, Shively L, Slaven J, Davis S . Polysomnography Reference Values in Healthy Newborns. J Clin Sleep Med. 2019; 15(3):437-443. PMC: 6411184. DOI: 10.5664/jcsm.7670. View

15.

van Lieshout M, Joosten K, Mathijssen I, Koudstaal M, Wolvius E, van der Schroeff M . Non-surgical and surgical interventions for airway obstruction in children with Robin Sequence. J Craniomaxillofac Surg. 2016; 44(12):1871-1879. DOI: 10.1016/j.jcms.2016.06.021. View

16.

Resnick C, Levine J, Calabrese C, Padwa B, Hansen A, Katwa U . Early Management of Infants With Robin Sequence: An International Survey and Algorithm. J Oral Maxillofac Surg. 2019; 77(1):136-156. DOI: 10.1016/j.joms.2018.05.031. View

17.

Drescher F, Jotzo M, Goelz R, Meyer T, Bacher M, Poets C . Cognitive and psychosocial development of children with Pierre Robin sequence. Acta Paediatr. 2008; 97(5):653-6. DOI: 10.1111/j.1651-2227.2008.00742.x. View

18.

Evans K, Sie K, Hopper R, Glass R, Hing A, Cunningham M . Robin sequence: from diagnosis to development of an effective management plan. Pediatrics. 2011; 127(5):936-48. PMC: 3387866. DOI: 10.1542/peds.2010-2615. View

19.

Caouette-Laberge L, Bayet B, Larocque Y . The Pierre Robin sequence: review of 125 cases and evolution of treatment modalities. Plast Reconstr Surg. 1994; 93(5):934-42. View

20.

Costa M, Tu M, Murage K, Tholpady S, Engle W, Flores R . Robin sequence: mortality, causes of death, and clinical outcomes. Plast Reconstr Surg. 2014; 134(4):738-745. DOI: 10.1097/PRS.0000000000000510. View