BRUEs in Boston –Two Punch Study

DR Duncan et al. J Pediatr 2024; 272: 114128. Brief Resolved Unexplained Events Symptoms Frequently Result in Inappropriate Gastrointestinal Diagnoses and Treatment

In this prospective cohort study from Boston Children’s (2017-2022, n=157), the authors examined diagnostic evaluations in children presenting with Brief Resolved Unexplained Events (BRUEs).

Key findings:

  • Only 28% (20% during the hospitalization, 8% afterwards) underwent VFSS with 71% abnormal.
  • 42% of infants had their BRUE attributed to GERD, and 33% were treated with acid suppression during follow-up
  • Provision of GERD diagnosis was associated with a delay in making an aspiration diagnosis. 10% (6/66) of patients with “GERD” diagnosis subsequently had swallow studies –all were abnormal. Mean diagnostic delay was 56 days.

Discussion points:

  • The approach of using symptoms to determine evaluation of BRUEs has been advised by AAP clinical practice guidelines (2016 & 2019); “however, our results suggest that reliance on these clinical characteristics may result in negative outcomes.”
  • Most aspiration in infants is silent aspiration and not detected by clinical feeding evaluation (CFE) in the absence of a VFSS. “It is concerning that 63% of patients had CFE alone without confirmatory VFSS in the present cohort, and it may be that this practice is even more common in other centers.” Silent aspiration can lead to repeat hospital visits and even long-lasting pulmonary damage including bronchiectasis.
  • Establishing a GERD diagnosis likely increases unnecessary (& potentially harmful) acid suppression

My take: The two punches in this study:

  1. Clinicians cannot diagnose aspiration based on history or bedside feeding evaluations. Objective testing (e.g. VFSS) is needed if there are concerns for dysphagia
  2. Inappropriate diagnosis of GERD may cause harm. GERD medications have been associated with increased infections and may increase risk for allergies.

The role of aspiration in causing BRUEs has been well-recognized since 2017 (see below) by the same group in Boston. It is likely that evaluation of dysphagia is even less frequent in other medical centers.

Related blog posts:

Double Arch at Arches National Park

Blaming Reflux for BRUEs -Not Changing Despite Guideline Recommendations

Briefly noted: DR Duncan et al. J Pediatr 2019; 211: 112-9.

In this retrospective cohort study of infants with brief resolved unexplained events (BRUEs) at Boston Children’s Hospital, the authors examined guideline implementation among 359 subjects in the year before and the year after AAP guidelines.

Key findings:

  • There were no significant changes in practice after guideline publication
  • Only 13% had videofluoroscopic swallow study performed; 72% of these showed aspiration/penetration
  • No subject had reflux testing, “yet reflux was implicated as the cause” for BRUE in 40%. Children continued to be “discharged on acid suppression despite lack of efficacy”

My take: The pendulum is (slowly) starting to swing back from blaming everything (including BRUEs) on reflux but this change is not evident in this study.

Related blog posts:

Something Useful for Apparent Life-Threatening Events (ALTEs)

In many cases of Apparent Life-Threatening Events (ALTEs) (also called Brief Resolved Unexplained Events, BRUEs) in infants, the exact reasons are unclear.  Sometimes these events are blamed on reflux despite studies indicating this is unlikely in the vast majority (see links at bottom of post).

A recent study (DR Duncan, J Amirault, PD Mitchell, K Larson, RL Rosen. JPGN 2017; 65: 168-73) finds that oropharyngeal dysphagia is correlated with ALTEs.

In this retrospective study which took place between 2012-15, the authors reviewed all patients admitted with ALTE.  They excluded infants with underlying diseases that included known neurologic impairment, congenital heart disease, and other congenital anomalies.

Demographics:

  • Median age 49 days
  • Color change: blue 65%, pale 8%, red 10%, none 17%
  • URI symptoms: 23%
  • Relationship to feeds: during 20%, after 35%, none 45%
  • Appeared well in ED 86%

Key findings:

  • Video fluoroscopic swallow study (VFSS) [also called oropharyngeal motility swallow study] was obtained in 29%.  In this group, 73% (n=40) had evidence of aspiration or penetration.
  • 26% of patients who had clinical feeding evaluation and VFSS were ascribed as having no oropharyngeal dysphagia prior to detecting aspiration on VFSS.
  • “Of all of the diagnostic tests ordered on patients with  ALTEs, the VFSS had the highest rate of abnormalities.”

Conclusion (from authors): “Oropharyngeal dysphagia with aspiration is the most common diagnosis identified in infants presenting with ALTEs.  The algorithm for ALTE should be revised to include an assessment of VFSS as clinical feeding evaluations are inadequate to assess for aspiration.”

Related blog post: What to do with ALTEs

Also, an except and link from NASPGHAN Consensus guidelines on GERD (2009)

  • In premature infants, a relationship between GER (i.e. reflux) and pathologic apnea and/or bradycardia has not been established. Despite a lack of convincing evidence, if pathological apnea occurs in the face of pre- existing reflux, then the following two statements are the most common features:
  • Although reflux causes physiologic apnea, it causes pathologic apneic episodes in only a very small number of newborns and infants.
  • When reflux causes pathological apnea, the infant is more likely to be awake and the apnea is more likely to be obstructive in nature.
  • A diagnosis of an acute life-threatening event (ALTE) warrants consideration of causes other than GER (i.e. reflux).Reflux of gastric acid seems to be related to ALTEs (episodes of combinations of apnea, color change, change in muscle tone, choking, and gagging) in < 5 % of infants with ALTE. 
  • Link: Gastroesophageal Reflux Disease in the Pediatric  – NASPGHAN.o

Dry Falls, Highlands, NC