Awake Flexible Fiberoptic Laryngoscopy to diagnose glossoptosis in Robin Sequence patients

Basart, Hanneke; König, Astrid; Bretschneider, Jochen H.; Hoekstra, Carlijn; Oomen, Karin P. Q.; Pullens, Bas; Rinkel, Rico N. P. M.; Gogh, Christine D. L. van; Horst, Chantal M.A.M. van der; Hennekam, Raoul C. M.

doi:10.1111/coa.12552

Cited by 5 publications

(7 citation statements)

References 21 publications

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“…Facial 3D-landmarks. The Landmarks 4 (labium inferius), 2 (gnathion) and 1/3 (otobasion inferior) define the tetrahedron (1,2,3,4). The volume of the tetrahedron is calculated as (1/6)* det (ax ay az 1; bx by bz 1; cx cy cz 1; dx dy dz 1).…”

Section: Discussionmentioning

confidence: 99%

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Objectifying Micrognathia Using Three-Dimensional Photogrammetric Analysis

Basart

Suttie

Ibrahim

et al. 2018

Journal of Craniofacial Surgery

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Background: Micrognathia occurs isolated and as part of entities like Robin sequence (RS). An objective measurement of mandible size and growth is needed to determine the degree of micrognathia and enable a comparison of treatment outcomes. We undertook a pilot study to investigate the usability of 3D facial photogrammetry, a fast, non-invasive method, to estimate mandible size and growth in a small cohort of newborns and infants. Methods: We estimated exterior mandibular volume using a tetrahedron defined by four facial landmarks. We selected 12 patients with RS with different etiologies and obtained photogrammetric images prospectively in three patients with RS in whom mandibular growth in the first year of life was determined. We used three tetrahedra defined by six landmarks on mandibular CT scans to estimate an interior mandibular volume, which we compared to the exterior mandibular volume in 10 patients. Results: The exterior mandibular volume using 3D photography could be determined in all patients. Signature heat maps allowed visualization of facial dysmorphism in three dimensions; signature graphs demonstrated similarities of facial dysmorphism in patients with the same etiology and differences from those with other diagnoses and from controls. The correlation between interior (3D photogrammetry) and exterior mandibular volumes (CT imaging) was 0.8789. Conclusion: 3D facial photogrammetry delineates the general facial characteristics in patients with different syndromes involving micrognathia, and can objectively estimate mandibular volume and growth, with excellent correlation with bony measurement. We conclude that 3D facial photogrammetry could be a clinically effective instrument for delineating and quantifying micrognathia.

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Section: Discussionmentioning

confidence: 99%

“…The co-existence of these signs typically causes upper airway obstructions, which result in respiratory and feeding problems of varying severity. A cleft palate is present in the majority of RS patients (1)(2). In 66% of the patients, RS is part of a more extended syndrome or accompanied by other abnormalities (3)(4).…”

Section: Introductionmentioning

confidence: 99%

Objectifying Micrognathia Using Three-Dimensional Photogrammetric Analysis

Basart

Suttie

Ibrahim

et al. 2018

Journal of Craniofacial Surgery

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“…Ultimately, flexible fiberoptic laryngoscopy (FFL) of the upper airway should determine the extent of the upper airway obstruction, but validated scoring systems that objectify the obstruction of the airway are lacking. A recent attempt to score glossoptosis in RS patients using awake FFL was not successful, due to disappointing inter- and intra-agreement in the analysis of awake FFL videos of RS patients compared to non-RS patients [ 27 ]. Another complicating factor is that the degree of micrognathia does not seem to correlate well with the degree of airway compromise and the higher Cormack–Lehane grades diagnosed with laryngoscopy [ 28 ].…”

Section: Discussionmentioning

confidence: 99%

Management and outcomes of obstructive sleep apnea in children with Robin sequence, a cross-sectional study

et al. 2016

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ObjectiveThe objective of this cross-sectional study is to assess the prevalence, course, and management of obstructive sleep apnea (OSA) in children with Robin sequence (RS) aged 1–18 years.Materials and methodsA cross-sectional study was conducted in 63 children aged 1 to18 years with RS. Patient data were collected on baseline characteristics and management. OSA was evaluated by polysomnography.ResultsSixty-three children with RS were included (median age 8.0 years) and divided into two groups based on the initial treatment: prone positioning or respiratory support. Respiratory support was more often indicated in children with a non-isolated RS (p < 0.05). At cross section, in the prone positioning group (n = 32), one child was diagnosed with OSA. In the respiratory support group (n = 31), 13 children (42 %) had respiratory problems of whom 10 needed respiratory support.ConclusionsBetween the age of 1 and 18 years, almost one out of four children with RS still has respiratory problems. Children with RS, who can be treated with prone positioning only as an infant, are not likely to develop obstructive airway problems at a later age. In contrast, children who need respiratory support early after birth are at risk of continuing or re-developing OSA after the age of 1 year.Clinical relevanceThis study shows that those who need respiratory support at an early age need careful monitoring until adulthood.

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“…For example, the interrater reliability of laryngoscopy for assessment of UAO has been shown to be modest at best, with poorer reliability demonstrated in patients with RS as well as when performed in awake patients (versus drug-induced sleep endoscopy). [7][8][9] Computed tomography is a familiar diagnostic tool for most providers not trained in FFL and may provide useful information and objective data with regard to RS-associated UAO. 10 However, standard 3-dimensional computed tomography (3D-CT) is unable to evaluate the dynamic processes that characterize UAO.…”

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confidence: 99%

“…Information obtained from FFL is qualitative and subjective. For example, the interrater reliability of laryngoscopy for assessment of UAO has been shown to be modest at best, with poorer reliability demonstrated in patients with RS as well as when performed in awake patients (versus drug‐induced sleep endoscopy) 7–9 …”

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confidence: 99%

Accuracy and Reliability of 4D‐CT and Flexible Laryngoscopy in Upper Airway Evaluation in Robin Sequence

Lam

Bindschadler

Evans

et al. 2021

Otolaryngol.--head neck surg.

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Objectives To evaluate the performance of 4-dimensional computed tomography (4D-CT) in assessing upper airway obstruction (UAO) in patients with Robin sequence (RS) and compare the accuracy and reliability of 4D-CT and flexible fiber-optic laryngoscopy (FFL). Study Design Prospective survey of retrospective clinical data. Setting Single, tertiary care pediatric hospital. Methods At initial and 30-day time points, a multidisciplinary group of 11 clinicians who treat RS rated UAO severity in 32 sets of 4D-CT visualizations and FFL videos (dynamic modalities) and static CT images. Raters assessed UAO at the velopharynx and oropharynx (1 = none to 5 = complete) and noted confidence levels of each rating. Intraclass correlation and Krippendorff alpha were used to assess intra- and interrater reliability, respectively. Accuracy was assessed by comparing clinician ratings with quantitative percentage constriction (QPC) ratings, calculated based on 4D-CT airway cross-sectional area. Results were compared using Wilcoxon rank-sum and signed-rank tests. Results There was similar intrarater agreement (moderate to substantial) with 4D-CT and FFL, and both demonstrated fair interrater agreement. Both modalities underestimated UAO severity, although 4D-CT ratings were significantly more accurate, as determined by QPC similarity, than FFL (−1.06 and −1.46 vs QPC ratings, P = .004). Overall confidence levels were similar for 4D-CT and FFL, but other specialists were significantly less confident in FFL ratings than were otolaryngologists (2.25 and 3.92, P < .0001). Conclusion Although 4D-CT may be more accurate in assessing the degree of UAO in patients with RS, 4D-CT and FFL assessments demonstrate similar reliability. Additionally, 4D-CT may be interpreted with greater confidence by nonotolaryngologists who care for these patients.

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Awake Flexible Fiberoptic Laryngoscopy to diagnose glossoptosis in Robin Sequence patients

Cited by 5 publications

References 21 publications

Objectifying Micrognathia Using Three-Dimensional Photogrammetric Analysis

Objectifying Micrognathia Using Three-Dimensional Photogrammetric Analysis

Management and outcomes of obstructive sleep apnea in children with Robin sequence, a cross-sectional study

Accuracy and Reliability of 4D‐CT and Flexible Laryngoscopy in Upper Airway Evaluation in Robin Sequence

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