A Single Lab Test to Aid Pierre Robin Sequence Severity Diagnosis

Fahradyan, Artur; Azadgoli, Beina; Tsuha, Michaela; Urata, Mark M.; Francis, Stacey H.

doi:10.1177/1055665618778400

Cited by 7 publications

(6 citation statements)

References 44 publications

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“…These characteristics were (1) M aximum CO 2 > 62 mm Hg; (2) AH I > 23; (3) O 2 S aturation nadir < 79.4%; and (4) T otal sleep time with < 90% O 2 saturation of > 5.7%. Interestingly, Fahradyan et al 19 found higher levels of carbon dioxide (pCO 2 ) through a heel stick capillary blood gas to better “predict” surgical intervention than PSG, using similar ROC analyses. Both studies suffer from similar methodological problems that inherently stem from identifying predictors for surgery ex post facto, as these “predictors” likely influenced the clinical decision to proceed with surgical intervention.…”

Section: Discussionmentioning

confidence: 99%

Current Trends in Surgical Airway Management of Neonates with Robin Sequence

Fan

Mandelbaum

Buro

et al. 2018

Plastic and Reconstructive Surgery - Global Open

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Background:Robin sequence is defined by the clinical triad of micrognathia, glossoptosis, and upper airway obstruction, and is frequently associated with cleft palate and failure to thrive. Though the efficacy of certain surgical interventions to relieve airway obstruction has been well established, algorithms dictating decision making and perioperative protocols are poorly defined.Methods:A 22-question survey was sent via e-mail to members of the American Cleft Palate-Craniofacial Association and International Society of Craniofacial Surgeons. Questions were related to surgeon experience in treating neonates with Robin sequence, and specific perioperative protocols.Results:One hundred fifty-one responses were collected. Most respondents were surgeons practicing in North America(82.8%), in a university hospital setting (81.5%) and had completed a fellowship in pediatric plastic surgery or craniofacial surgery (76.2%). Preoperative protocols varied widely by years in training and location of practice. Although 78.8% of respondents always performed direct laryngoscopy, only 49.7% of respondents routinely obtained preoperative polysomnography. Mandibular distraction osteogenesis was the most common primary surgical airway intervention reported by 74.2%, with only 12.6% primarily utilizing tongue-lip adhesion. Slightly less than half of respondents ever performed tongue-lip adhesion. Operative selection was influenced by surgeon experience, with 80% of those in practice 0–5 years primarily utilizing mandibular distraction, compared with 56% of respondents in practice >15 years.Conclusions:This study documents wide variations in preoperative, operative, and postoperative protocols for the surgical airway management of neonates with severe Robin sequence. These results underscore the need to acquire more objective data, to compare different protocols and outcome measures.

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Section: Discussionmentioning

confidence: 99%

Current Trends in Surgical Airway Management of Neonates with Robin Sequence

Fan

Mandelbaum

Buro

et al. 2018

Plastic and Reconstructive Surgery - Global Open

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“…Some authors believe capillary blood gas CO 2 may be predict the need for a surgical airway. Fahradyan et al (2019) reported the use of CBG CO 2 in 73 patients with RS and stated that ROC curves with cutoff value of pCO 2 level of 49.5 is the best among other variables in terms of specificity and sensitivity combination as an “indication” for surgical intervention. Kwan et al (2021) also noted that a PCO 2 cutoff of 55 mmHg (sensitivity = 55.7%; specificity = 91.3%; area under the curve = 0.78) and a HCO 3 cutoff of 30 mEq/L (sensitivity = 56.2%; specificity = 95.0%; area under the curve = 0.80) were most predictive of patients who “required” a surgical airway.…”

Section: Discussionmentioning

confidence: 99%

Defining Age-related OSA Features in Robin Sequence Using Polysomnographic-based Analyses of Respiratory Arousal Responses and Gas-exchange Parameters

Niño

Aziz

Weiss

et al. 2021

The Cleft Palate Craniofacial Journal

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Introduction Robin sequence (RS) is a leading cause of obstructive sleep apnea (OSA) in newborns. Most studies have focused on understanding anatomic factors leading to OSA and changes in apnea–hypopnea index (AHI) on polysomnography (PSG) beyond the neonatal period. This study aims to define age-related OSA features between patients with RS, without RS and healthy controls using PSG-based analyses of respiratory arousal responses and gas-exchange parameters. Design Retrospective comparison of PSG features in a total of 48 children encompassing three groups: (a) infants with RS (n = 24, <1-year old), (b) non-RS older children (1-2 years old) with severe OSA (obstructive AHI (OAHI) of ≥10 events; n = 12), and (c) control infants and children (0-2 years old) without sleep apnea (OAHI ≤1.5/h, n = 12). We examined OSA sleep-stage specific and position-specific indexes, and the relationship between OSA severity and respiratory arousal indexes (OAHI/respiratory arousal indexes). Results OSA sleep-stage specific indexes (rapid eye movement [REM] vs non-REM[NREM]) as well as position-specific indexes (supine vs nonsupine) were similar in individuals with and without RS. Relative to the non-RS groups, infants with RS have more sustained hypoxemia (time with SpO2 < 90%) and reduced arousal responses to OSA demonstrated by higher OAHI/respiratory arousal indexes. OAHI/respiratory arousal indexes significantly correlated with the severity of hypoxemia in infants with RS. Conclusion Infants with RS and OSA show reduced arousal responses to apneic events, which correlates with higher hypoxemia severity. OAHI/respiratory arousal indexes in RS may identify high-risk individuals with upper airway obstruction and reduced arousal protective responses.

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“…However, some of these techniques have been criticized as subjective or nonstandardized; moreover, not every institution has resources available for obtaining accurate PSG data on newborns. 5,6 Normative values for neonatal PSG are still debated, and PSG, which assesses for airway obstruction during sleep alone, may overlook dynamic airway obstruction which manifests during periods of activity. 7,8 One of the objective parameters measured by PSG is end-tidal carbon dioxide.…”

Section: Introductionmentioning

confidence: 99%

“…Current management algorithms for airway obstruction in infants with RS may incorporate physical examination findings, data related to oral intake (feeding curve), and polysomnography (PSG) findings to quantify the degree of obstruction present and identify surgical candidates. However, some of these techniques have been criticized as subjective or nonstandardized; moreover, not every institution has resources available for obtaining accurate PSG data on newborns 5,6 . Normative values for neonatal PSG are still debated, and PSG, which assesses for airway obstruction during sleep alone, may overlook dynamic airway obstruction which manifests during periods of activity 7,8 …”

Section: Introductionmentioning

confidence: 99%

“…Recently, the use of capillary blood gas (CBG) values was proposed as a quantifiable and objective measure of hypoventilation in infants with Robin sequence (RS) that may assist in therapeutic stratification 6 . However, the time frame by which infants with RS and chronic hypoventilation will demonstrate objective signs of CO 2 retention and compensatory metabolic alkalosis was not specified in that report; the ideal time point for detecting signs of hypoventilation during birth hospitalization among infants with RS has not been described.…”

Section: Introductionmentioning

confidence: 99%

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Detection of Chronic Hypoventilation Among Infants With Robin Sequence Using Capillary Blood Gas Sampling

et al. 2021

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Objectives/Hypothesis: To describe the use of capillary blood gas (CBG) sampling to detect and quantify hypoventilation in infants with Robin sequence (RS).Methods: Case series with chart review at two institutions. Infants with RS presenting over a 10-year period were identified using departmental databases. CBG values obtained during infancy or until airway intervention (AI) were reviewed.Results: From 2008 to 2018, 111 infants with RS were identified as having had been assessed and managed from birth or transfer until discharge home and having CBG data available. In most cases, CBG sampling was obtained every other day until intervention or discharge. A total of 81 (73%) infants required AI: 72 (89%) underwent mandibular distraction osteogenesis, five (6%) underwent tracheotomy, and four (5%) were discharged home with a nasopharyngeal airway. The mean PCO 2 at day of life (DOL) 7-30 for the AI group was 52.7 mmHg (95% confidence interval: 51.7-53.7) and for the no AI group was 45.9 mmHg (44.8-47.0; P < .0001). The mean HCO 3 at DOL 7-30 for the AI group was 29.8 mEq/L (29.4-30.1) and for the no AI group was 27.0 mEq/L (26.5-27.4; P < .0001). Receiver operating characteristic curves were created for maximum PCO 2 and HCO 3 values and cutoffs were established by optimizing a balance of sensitivity and specificity. Infants requiring AI surpassed the PCO 2 and HCO 3 cutoff at a median of DOL 9.Conclusions: Among infants with RS and hypoventilation, objective measures of respiratory acidosis may be apparent by DOL 9.

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A Single Lab Test to Aid Pierre Robin Sequence Severity Diagnosis

Abstract: A simple CBG heel stick may better predict the physiologic effects of obstructive apnea; therefore, it should be added to the algorithm of PRS workup.

Cited by 7 publications

References 44 publications

Current Trends in Surgical Airway Management of Neonates with Robin Sequence

Current Trends in Surgical Airway Management of Neonates with Robin Sequence

Defining Age-related OSA Features in Robin Sequence Using Polysomnographic-based Analyses of Respiratory Arousal Responses and Gas-exchange Parameters

Detection of Chronic Hypoventilation Among Infants With Robin Sequence Using Capillary Blood Gas Sampling

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