Effects of the COVID-19 Pandemic on Brief Resolved Unexplained Events (BRUEs) in Children: A Comparative Analysis of Pre-Pandemic and Pandemic Periods.

BACKGROUND: Brief Resolved Unexplained Events (BRUEs), formerly known as Apparent Life-Threatening Events (ALTEs), are concerning episodes of short duration (typically <1 min) characterized by a change in breathing, consciousness, muscle tone, and/or skin color. In some cases, SARS-CoV-2 infection has been associated with episodes of BRUEs in previously healthy children. This study aimed to compare the demographic, respiratory, perinatal, and infectious characteristics in children affected by BRUEs before the COVID-19 pandemic and after the spread of SARS-CoV-2. METHODS: We conducted a retrospective observational study covering January 2018 to March 2020 (pre-COVID-19) and April 2023 (during the ongoing COVID-19 pandemic). Collected variables included clinical information during pregnancy and neonatal details of children with BRUEs. RESULTS: The number of children in the pre-COVID-19 period was 186 (41%); after the emergence and spread of SARS-CoV-2 this number was 268 (59%). The risk of infection at birth for children developing BRUEs was higher during the pandemic. Children were less likely to have ongoing symptomatic infection during BRUEs during the pandemic (coefficient B = 0.783; p = 0.009). Respiratory symptoms during BRUEs were more frequent during the pandemic (coefficient B = 0.654; p = 0.052). Fever during BRUEs was less likely during the pandemic (coefficient B = -0.465, p = 0.046). CONCLUSIONS: These findings could have significant clinical implications for managing children with BRUEs during the COVID-19 pandemic.

Congenital Long QT Syndrome (LQTS) in Infancy: A Challenging Case.

Long QT syndrome (LQTS), is an arrhythmia disorder, related to ventricular myocardial repolarization characterized by a prolonged QT interval on the electrocardiogram that can lead to symptomatic ventricular arrhythmias and increase the mortality rate. The prevalence of congenital LQTS is about 1 in 2000 live births. Here, we report the case of a two-month-old female, with a significant family history of early death, who was brought to our emergency with an episode of blueish discoloration. The initial workup was positive for COVID-19 in the respiratory panel, so she was admitted as a case of bronchiolitis. It was a challenge because of the overlapping presentation between a serious condition and other common pediatric illnesses. Furthermore, the paper aims to increase awareness of this condition among physicians and emphasizes the importance of obtaining a complete medical history, physical examination, and family screening in selected cases to facilitate early diagnosis and timely management.

Doença do refluxo gastroesofágico em lactentes que apresentaram Eventos Resolvidos Breves Não Explicados - Brief Resolved Unexplained Event (BRUE) / Gastroesophageal reflux disease in infants who presented brief resolved unexplained event (brue)

ABSTRACT BACKGROUND: The term brief resolved unexplained events (BRUE) is a description of the acute event occurring in infants less than 1-year-old that includes at least one of the following characteristics: cyanosis or pallor; absent, decreased, or irregular breathing; marked change in tone or altered level of responsiveness. An investigative proceeding is required to identify the triggering phenomenon in those who are at high risk of complications. Prolonged esophageal pHmetry has been used as a tool in searching for gastroesophageal reflux disease (GERD) as one of the underlying etiologies. OBJECTIVE: The study aims to verify the frequency of GERD in infants up to 1-year-old, when pHmetry has been performed for investigating high-risk BRUE (HR-BRUE) and to analyze if clinical characteristics or any particular symptom related by caregivers during BRUE could be correlated to GERD. METHODS: It was performed a cross-sectional study. The data was collected retrospectively of patients less than 1-year-old, who had performed pHmetry in a tertiary hospital for investigating HR-BRUE between October 2008 and January 2018. For the analysis of medical records, a data collection protocol included: gender, age at the first HR-BRUE episode, age at the time of the pHmetry, gestational age, type of delivery (normal or caesarean) and birth weight and symptoms associated to HR-BRUE related by caregivers. Relation between variables were assessed using Fisher's exact test and Mann-Whitney test. The significance level was set at 0.05. RESULTS: A total of 54 infants were included (preterm 25, term 29), 62.9% males, median age at the HR-BRUE was 36 days, 53.7% HR-BRUE episodes had occurred during or right after feeding. According to pHmetry results: nine pHmetry results were considered inconclusive, physiological reflux (n=30) and GERD (n=15). The frequency of GERD diagnosed by pHmetry was 33%. GERD was not statistically related to gender (P-value=0.757), age at first HR-BRUE episode (P-value=0.960), age at the time of the pHmetry (P-value=0.720), prematurity (P-value=0.120) or type of delivery (P-value=0.738). GERD was statistically related to low birth weight (P-value=0.023). There was no association between symptoms reported by caregivers during HR-BRUE and GERD. CONCLUSION: GERD diagnosed by the pHmetry was found in one third of infants that experiencing a HR-BRUE, showing the importance of properly investigation. In half of infants BRUE occurred during or right after feeding. Besides low birth weight, it was not possible to select other data from the clinical history that suggest that these patients would be more likely to have GERD.

RESUMO CONTEXTO: O termo Eventos Resolvidos Breves Não Explicados (Brief Resolved Unexplained Event - BRUE) é uma descrição do evento agudo que ocorre em lactentes menores de 1 ano de idade que inclui pelo menos uma das seguintes características: cianose ou palidez; respiração ausente, diminuída ou irregular, alteração acentuada no tônus ou nível alterado de responsividade. É necessário um procedimento investigativo para identificar o fenômeno desencadeante naqueles que apresentam alto risco de complicações. A pHmetria esofágica prolongada tem sido usada como uma ferramenta na pesquisa de doença do refluxo gastroesofágico (DRGE) como uma das etiologias subjacentes. OBJETIVO: Este estudo tem como objetivo verificar a frequência da DRGE em lactentes de até 1 ano de idade, quando a pHmetria foi realizada para investigação da BRUE de alto risco, e analisar se alguma característica clínica ou sintoma particular relatado pelos cuidadores durante a BRUE poderia estar correlacionado a DRGE. MÉTODOS: Foi realizado um estudo observacional, transversal, cujos dados foram coletados retrospectivamente de pacientes menores de 1 ano de idade, que realizaram pHmetria em hospital terciário para investigação de BRUE de alto risco de outubro de 2008 e janeiro de 2018. Para a análise dos prontuários, um protocolo de coleta de dados incluiu: sexo, idade no primeiro episódio de BRUE de alto risco, idade no momento da pHmetria, idade gestacional, tipo de parto (normal ou cesárea), peso ao nascer e sintomas associados a alto risco-BRUE relatado por cuidadores. A relação entre as variáveis foi avaliada por meio do teste exato de Fisher, qui-quadrado e teste de Mann-Whitney. O nível de significância foi estabelecido em 0,05. RESULTADOS: Foram incluídos 54 lactentes (pré-termo 25, termo 29), 62,9% do sexo masculino, idade mediana na BRUE de alto risco foi de 36 dias. De acordo com o relatório do cuidador, 53,7% dos episódios de BRUE de alto risco ocorreram durante ou logo após a alimentação. Resultados da pHmetria: nove resultados da pHmetria foram considerados inconclusivos, refluxo fisiológico (n=30) e DRGE (n=15). A frequência de DRGE diagnosticada por pHmetria foi de 33%. A DRGE não foi estatisticamente relacionada ao sexo (P=0,757), idade no primeiro episódio de BRUE de alto risco (P=0,96), idade no momento da pHmetria (P=0,72) prematuridade (P=0,321) ou tipo de parto (P=0,738). A DRGE foi estatisticamente relacionada ao baixo peso ao nascer (P=0,023). Não houve associação entre os sintomas relatados pelos cuidadores durante BRUE de alto risco e o diagnóstico de DRGE. CONCLUSÃO: A DRGE diagnosticada pela pHmetria foi encontrada em um terço dos lactentes que vivenciaram BRUE de alto risco, mostrando a importância da investigação adequada. Em metade das crianças, o evento ocorreu durante ou logo após a alimentação. Além do baixo peso ao nascer, não foi possível selecionar outros dados da história clínica que sugiram que esses pacientes terão maior probabilidade de apresentar DRGE.

Clinical Characteristics and Outcomes of Patients Admitted with Brief Resolved Unexplained Events to a Tertiary Care Pediatric Intensive Care Unit.

The aim of this study was to determine outcomes of patients admitted to a tertiary care pediatric intensive care unit (PICU) with brief, resolved, unexplained event (BRUE), and to review the diagnostic and treatment options utilized for such patients. A retrospective data analysis was conducted for infants and children who were admitted to the PICU at a tertiary hospital with a diagnosis of BRUE over a period of three years (2015-2017). The study included 30 infants, 15 males, and 15 females. All patients survived to hospital discharge. The most frequent presenting symptoms and signs were apnea (73.3%), cyanosis (60.0%), and cough (20.0%). The most frequent reported affected systems were respiratory (33.3%), gastrointestinal (20%), and infection-related illness (20.0%). We conclude that the careful history taking, complete physical examination, and the appropriate workup for patients with BRUE play an integral role in optimum health service and utilization of critical care beds. Survival to hospital discharge with no serious in-hospital events warrants the adaptation of evidence-based medicine guidelines to stratify such patients based on the risk of recurrence or a serious underlying condition. Prospective multicenter studies are recommended to explore the effectiveness of such guidelines implementation on outcomes and diagnostic testing in such patients to optimize the utilization of the limited critical care beds.

Implementing lower-risk brief resolved unexplained events guideline reduces admissions in a modelled population.

RATIONALE: American Academy of Pediatrics released a clinical practice guideline (CPG) in 2016 recommending the term apparent life-threatening events (ALTE) be replaced by brief resolved unexplained events (BRUE). The CPG provides recommendations for the clinical evaluation and management of infants with this condition based on the risk of a serious underlying disorder or repeat event. The lower-risk CPG was applied to a modelled population, studying predictors of hospital admission, defined as length of stay (LOS) ≥ 24 hours. METHODS: An algorithm was derived using a Pediatric Emergency Care Applied Research Network database. Propensity score weighting, based on probability of following the CPG, determined the adjusted odds ratio (aOR) and 95% confidence interval (CI) of hospital admission. Multiple imputation allowed any missing data problems be addressed and a sensitivity analysis of database robustness. RESULTS: Applying the modelling algorithm, 3116 observations were identified, among whom 1974 (63.4%) the CPG was followed and 1142 (36.6%) not followed. The CPG was followed for 60.1% of infants staying ≥24 hours compared with 76.6% of infants staying <24 hours (P < .001). After propensity score weighting and multiple imputation, the likelihood of hospital admission was significantly lower when the CPG was followed (aOR = 0.49; 95% CI, 0.39-0.62, P < .001). CONCLUSIONS: Results suggest that use of the CPG under strict conditions would lead to fewer hospital admissions among infants with a lower-risk BRUE. Implementation of CPGs in modelled populations may help clinicians identify unanticipated factors and address these issues beforehand. We noted differences in care based on race, necessitating further investigation.

Síndrome de enterocolitis inducida por proteínas alimentarias por ingesta de kiwi como causa de evento potencialmente grave inexplicado en una niña / Food Protein Induced Enterocolitis Syndrome due to ingestion of kiwi causing potentially severe unexplained event in a child

El síndrome de enterocolitis inducida por proteínas alimentarias es una alergia alimentaria no mediada por inmunoglobulina E que se manifiesta clínicamente con vómitos profusos y repetitivos, en ocasiones, asociados a diarrea, y puede llegar a asociar deshidratación y letargia, con riesgo de desarrollo de shock. A pesar de su potencial gravedad, el índice de sospecha de este síndrome es bajo, lo que demora su diagnóstico, especialmente, en aquellos casos que son desencadenados por alimentos sólidos. La presencia de vómitos y la duración de más de un minuto son los datos clave que pueden diferenciarlo de los episodios breves, resueltos e inexplicados. Se presenta el caso de una lactante de 6 meses de vida con diagnóstico final de síndrome de enterocolitis inducida por proteínas alimentarias por ingesta de kiwi.

Food protein-induced enterocolitis syndrome (FPIES) is a non-IgE food allergy manifesting as profuse, repetitive vomiting, sometimes with diarrhea, leading to dehydration and lethargy that can be severe and lead to shock. Despite the potential severity, awareness of FPIES is low and diagnosis is often delayed, especially in those triggered by solid foods. Presence of vomits and duration of more than 1 minute are the key differential factors to distinguish FPIES from brief resolved unexplained events. We report a case of a 6-month-old infant finally diagnosed as having kiwi induced FPIES.

[Food Protein Induced Enterocolitis Syndrome due to ingestion of kiwi causing potentially severe unexplained event in a child]. / Síndrome de enterocolitis inducida por proteínas alimentarias por ingesta de kiwi como causa de evento potencialmente grave inexplicado en una niña.

Food protein-induced enterocolitis syndrome (FPIES) is a non-IgE food allergy manifesting as profuse, repetitive vomiting, sometimes with diarrhea, leading to dehydration and lethargy that can be severe and lead to shock. Despite the potential severity, awareness of FPIES is low and diagnosis is often delayed, especially in those triggered by solid foods. Presence of vomits and duration of more than 1 minute are the key differential factors to distinguish FPIES from brief resolved unexplained events. We report a case of a 6-month-old infant finally diagnosed as having kiwi induced FPIES.

El síndrome de enterocolitis inducida por proteínas alimentarias es una alergia alimentaria no mediada por inmunoglobulina E que se manifiesta clínicamente con vómitos profusos y repetitivos, en ocasiones, asociados a diarrea, y puede llegar a asociar deshidratación y letargia, con riesgo de desarrollo de shock. A pesar de su potencial gravedad, el índice de sospecha de este síndrome es bajo, lo que demora su diagnóstico, especialmente, en aquellos casos que son desencadenados por alimentos sólidos. La presencia de vómitos y la duración de más de un minuto son los datos clave que pueden diferenciarlo de los episodios breves, resueltos e inexplicados. Se presenta el caso de una lactante de 6 meses de vida con diagnóstico final de síndrome de enterocolitis inducida por proteínas alimentarias por ingesta de kiwi.

Severe Brief Resolved Unexplained Event in a Newborn Infant in Association with Maternal Sertralin Treatment during Pregnancy.

Background: Selective serotonin reuptake inhibitors are a very common choice of antidepressive drug-therapy during pregnancy. In up to 30% of cases, they have been found to cause neonatal abstinence syndrome in newborn infants. Although often both time-limiting and self-limiting, severe symptoms of neonatal abstinence syndrome (NAS) can occur. Methods/Results: We report a term male infant suffering from a severe brief resolved unexplained event caused by his mother's sertraline intake during pregnancy. Conclusions: Newborn infants exposed to selective serotonine reuptake inhibitors (SSRIs) during pregnancy should be evaluated very carefully concerning NAS and monitored for NAS symptoms for a minimum of 72⁻96 h, or until symptoms have fully recovered using standardized protocols. There is a risk of severe NAS symptoms which might occur, and this circumstance should be discussed with the parents and taken into account before administering the drug.

The Characteristics of Esophageal Multichannel Intraluminal Impedance-PH Measurements in Infants Experiencing Brief Resolved Unexplained Events.

BACKGROUND: Brief Resolved Unexplained Events (BRUE) is defined as a sudden, brief and now resolved episode characterized by color change, altered respirations, change in tone, and altered level of responsiveness. This study aims to identify the characteristics of esophageal Multichannel Intraluminal Impedance-pH (MII-pH) monitoring in infants who have experienced a BRUE. METHODS: This study was a retrospective review of records of infants younger than 12 months who presented to the University of South Alabama Children's and Women's Hospital with an admission diagnosis of BRUE. Patients who underwent esophageal MII-pH monitoring between October 2015 and February 2017 and diagnosed with BRUE were initially included in this study. RESULTS: Fifty-three infants (preterm 25, term 28) who experienced a higher risk BRUE were included in our study. The mean age at diagnosis was 2.25 ± 2.07 months. Apnea (41/53; 77.4%) was the most common manifestation of BRUE. Non-acid reflux events were the most common findings in the MII-pH studies (66%). MII-pH results showed 6/53 (11%) acid reflux, 17/53 (32%) non-acid reflux and 12/53 (23%) both acid/nonacid reflux and 18/53 (34%) were normal. There were significant differences in the longest acid reflux episode and the Reflux Symptom Sensitivity Index (RSSI) of coughing/choking/gagging between preterm and term infants. The Reflux Symptom Index (RSI), RSSI and Reflux Symptom Association Probability (RSAP) were significantly correlated with each other in all symptoms (pain/fussiness, coughing/choking/gagging and vomiting). CONCLUSIONS: Among infants experiencing a higher risk BRUE, esophageal MII-pH monitoring revealed acid or nonacid reflux in 2/3 of patients.

Apparent Life-Threatening Events (ALTE): Italian guidelines.

Five years after the first edition, we have revised and updated the guidelines, re-examining the queries and relative recommendations, expanding the issues addressed with the introduction of a new entity, recently proposed by the American Academy of Pediatrics: BRUE, an acronym for Brief Resolved Unexplained Events. In this manuscript we will use the term BRUE only to refer to mild, idiopathic cases rather than simply replace the acronym ALTE per se.In our guidelines the acronym ALTE is used for severe cases that are unexplainable after the first and second level examinations.Although the term ALTE can be used to describe the common symptoms at the onset, whenever the aetiology is ascertained, the final diagnosis may be better specified as seizures, gastroesophageal reflux, infection, arrhythmia, etc. Lastly, we have addressed the emerging problem of the so-called Sudden Unexpected Postnatal Collapse (SUPC), that might be considered as a severe ALTE occurring in the first week of life.

Posterior Tracheopexy for Severe Tracheomalacia Associated with Esophageal Atresia (EA): Primary Treatment at the Time of Initial EA Repair versus Secondary Treatment.

PURPOSE: We review outcomes of posterior tracheopexy for tracheomalacia in esophageal atresia (EA) patients, comparing primary treatment at the time of initial EA repair versus secondary treatment. METHODS: All EA patients who underwent posterior tracheopexy from October 2012 to September 2016 were retrospectively reviewed. Clinical symptoms, tracheomalacia scores, and persistent airway intrusion were collected. Indication for posterior tracheopexy was the presence of clinical symptoms, in combination with severe tracheomalacia as identified on bronchoscopic evaluation, typically defined as coaptation in one or more regions of the trachea. Secondary cases were usually those with chronic respiratory symptoms who underwent bronchoscopic evaluation, whereas primary cases were those found to have severe tracheomalacia on routine preoperative dynamic tracheobronchoscopy at the time of initial EA repair. RESULTS: A total of 118 patients underwent posterior tracheopexy: 18 (15%) primary versus 100 (85%) secondary cases. Median (interquartile range) age was 2 months (1-4 months) for primary (22% type C) and 18 months (8-40 months) for secondary (87% type C) cases (p < 0.001). There were statistically significant improvements in most clinical symptoms postoperatively for primary and secondary cases, with no significant differences in any postoperative symptoms between the two groups (p > 0.1). Total tracheomalacia scores improved significantly in primary (p = 0.013) and secondary (p < 0.001) cases. Multivariable Cox regression analysis indicated no differences in persistent airway intrusion requiring reoperation between primary and secondary tracheopexy adjusting for imbalances in age and EA type (p = 0.67). CONCLUSION: Posterior tracheopexy is effective in treating severe tracheomalacia with significant improvements in clinical symptoms and degree of airway collapse on bronchoscopy. With no significant differences in outcomes between primary and secondary treatment, posterior tracheopexy should be selectively considered at the time of initial EA repair.