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Background Intranasal corticosteroids (INCS) are used in the management of sinonasal conditions. Use of exogenous steroids can be associated with hypothalamic‐pituitary‐adrenal axis dysfunction and adrenal insufficiency (AI). We aimed to estimate the rate of AI after INCS use in a meta‐analysis, stratified by steroid type and treatment duration. Methods Ovid Medline, Embase Classic, PubMed, Web of Science, and CINAHL databases were searched following Preferred Reporting Items for Systematic Reviews and Meta‐Analyses guidelines to identify studies investigating INCS use and AI. AI was defined as morning serum cortisol <550 nmol/L and <80 nmol/L with and without adrenocorticotropic hormone stimulation. INCS were classified as first (beclomethasone dipropionate, triamcinolone acetonide, beclomethasone, budesonide, dexamethasone) and second (ciclesonide, mometasone furoate, and fluticasone propionate) generation. Duration of treatment was classified as short (<1 month), medium (1‐12 months), and long‐term (>12 months) time periods. Results This search identified 3668 articles. A total of 39 studies (1678 patients) were included in the final analysis. The pooled percentage of AI for routinely utilized first‐ and second‐generation INCS was 0.70% (95% confidence interval [CI], 0.29‐1.12%). Stratified by type, AI was observed in 0.78% (95% CI, 0.25‐1.30%) of first‐generation and 0.58% (95% CI, −0.1% to 1.26%) of second‐generation steroids. AI was seen in 0.48% (95% CI, −0.01% to 0.96%) of short‐term, 1.13% (95% CI, 0.2‐2.1%) of medium‐term, and 1.67% (95% CI, 0.37‐2.9%) of long‐term use of INCS. Conclusion Overall, the use of INCS carries a low risk for AI. Although modest, this risk may differ depending on the length of duration and type of INCS used. Informing patients of these risks is of importance for the treatment of chronic sinonasal conditions.

Objective Several cases of facial nerve paralysis (FNP) post‐COVID‐19 infection have been reported with varying presentations and management. This study aims to identify FNP clinical characteristics and recovery outcomes among patients acutely infected with COVID‐19. We hypothesize that FNP is a potentially unique sequalae associated with COVID‐19 infections. Methods A systematic review of PubMed‐Medline, OVID Embase, and Web of Science databases from inception to November 2021 was conducted following the Preferred Reporting Items for Systematic Reviews and Meta‐Analyses guidelines. Results This search identified 630 studies with 53 meeting inclusion criteria. This resulted in 72 patients, of which 30 (42%) were diagnosed with Guillain‐Barré Syndrome (GBS). Non‐GBS patients were on average younger (36 vs. 53 years) and more likely to present with unilateral FNP (88%) compared to GBS patients who presented predominantly with bilateral FNP (74%). Among non‐GBS patients, majority (70%) of FNP presented a median of 8 [IQR 10] days after the onset of initial COVID‐19 symptom(s). Treatment for non‐GBS patients consisted of steroids (60%), antivirals (29%), antibiotics (21%), and no treatment (21%). Complete FNP recovery in non‐GBS patients was achieved in 67% patients within a median of 11 [IQR 24] days. Conclusion FNP is a possible presentation post COVID‐19 infections, associated with both GBS and non‐GBS patients. Although no causation can be assumed, the clinical course of isolated FNP associated with COVID‐19 raises the possibility of a unique presentation differing from Bell's palsy, seen with higher proportion of patients developing bilateral FNP and a shorter duration to complete recovery. Laryngoscope , 2022

IMPORTANCEAlthough various clinical prediction models (CPMs) have been described for diagnosing pediatric foreign body aspiration (FBA), to our knowledge, there is still no consensus regarding indications for bronchoscopy, the criterion standard for identifying airway foreign bodies.OBJECTIVE To evaluate currently available CPMs for diagnosing FBA in children.DATA SOURCES Performed in Ovid MEDLINE, Ovid Embase, PubMed, Web of Science, and CINAHL database with citation searching of retrieved studies.STUDY SELECTION Prediction model derivation and validation studies for diagnosing FBA in children were included. Exclusion criteria included adult studies; studies that included variables that were not available in routine clinical practice and outcomes for FBA were not separate or extractable.DATA EXTRACTION AND SYNTHESIS We followed the Critical Appraisal and Data Extraction for Systematic Reviews of Prediction Modeling Studies and the Prediction Model Risk of Bias Assessment Tool framework. Data were pooled using a random-effects model. MAIN OUTCOMES AND MEASURESThe primary outcome was the diagnosis of FBA as confirmed by bronchoscopy. Characteristics of CPMs and individual predictors were evaluated. The final model presentation with available measures of performance was provided by narrative synthesis. A meta-analysis of individual predictor variables and prediction models was performed.RESULTS After screening 4233 articles, 7 studies (0.2%; 1577 patients) were included in the final analysis. There were 6 model derivation studies and 1 validation study. Air trapping (odds ratio [OR], 8.3; 95% CI, 4.4-15.5), unilateral reduced air entry (OR, 4.8; 95% CI, 3.5-6.5), witnessed choking (OR, 3.1; 95% CI, 1.0-9.6), wheezing (OR, 2.5; 95% CI, 1.2-5.2), and suspicious findings suggestive of FBA on radiography (OR, 18.5; 95% CI, 5.0-67.7) were the most commonly used predictor variables. Model performance varied, with discrimination scores (C statistic) ranging from 0.74 to 0.88. The pooled weighted C statistic score of all models was 0.86 (95% CI, 0.80-0.92). All studies were deemed to be at high risk of bias, with overfitting of models and lack of validation as the most pertinent concerns.CONCLUSIONS AND RELEVANCE This systematic review and meta-analysis suggests that existing CPMs for FBA in children are at a high risk of bias and have not been adequately validated. No current models can be recommended to guide clinical decision-making. Future CPM studies that adhere to recognized standards for development and validation are required.

Background Isomerism or heterotaxy syndrome is the loss of normal asymmetry of the internal thoraco‐abdominal organs in the left‐right axis and is associated with cardiovascular malformations. Mutations within DNAH11 can be associated with primary ciliary dyskinesia and heterotaxy syndromes. Methods We report a family of healthy, nonconsanguinous parents with subsequent pregnancies demonstrating a novel likely pathogenic variant in DNAH11 segregating in a sibship with varied presentations. Result The first affected pregnancy presented with right atrial isomerism. Further DNA testing identified three variants in DNAH11 related to primary ciliary dyskinesia: a maternally inherited heterozygous variant of unknown significance (VUS) c.2772G>A (p.Met924Ile), a maternally inherited novel likely pathogenic variant c.11662C>T (p.Arg3888Cys) as well as a paternally inherited pathogenic c.1648delA variant (p.Arg550GlyfsX16). The second pregnancy inherited the same variants including the pathogenic and likely pathogenic DNAH11 variants and presented with left isomerism and extracardiac abnormalities. Conclusion We present a novel likely pathogenic variant (c.11662C>T) in DNAH11 that has manifested in heterotaxy with variability in phenotypes for subsequent pregnancies of common parents. This report demonstrates that sibship illustrates potential variability in phenotypes associated with the same pathogenic variants within a family and highlights the difficulty in genetic counseling due to the variation in clinical presentation.