Pediatric retrograde cricopharyngeal dysfunction diagnosed by high-resolution impedance manometry

JOURNAL OF PEDIATRIC GASTROENTEROLOGY AND NUTRITION(2024)

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摘要
ObjectivesThe inability to burp, known as retrograde cricopharyngeal dysfunction (R-CPD), was initially described in adults. The proposed clinical diagnostic criteria for R-CPD include belching inability, abdominal bloating and discomfort/nausea, postprandial chest pain, and involuntary noises. Botulinum toxin injection to the cricopharyngeal muscle has been reported to be beneficial. High-resolution esophageal impedance-manometry (HRIM) features in adolescent patients with R-CPD have not been described yet. The aim of our study was to describe the clinical and HRIM findings of pediatric patients with R-CPD.MethodsClinical and manometric features of five pediatric patients diagnosed with R-CPD were reviewed. HRIM study protocol was modified to include the consumption of carbonated drink to provoke symptoms and distinctive manometric features.ResultsWe report five female patients aged 15-20 years who presented with an inability to burp and involuntary throat sounds. HRIM revealed normal upper esophageal sphincter (UES) relaxation during swallowing, but abnormal UES relaxation with concurrent high esophageal impedance reflecting air entrapment and secondary peristalsis following the carbonated drink challenge. Four patients exhibited esophageal motility disorder. All patients reported improvement or resolution of symptoms after botulinum toxin injection to the cricopharyngeus muscle.ConclusionsAdolescents with an inability to burp, reflux-like symptoms, bloating, and involuntary throat noises should be assessed for R-CPD by pediatric gastroenterologists with HRIM. The relatively recent recognition of this novel condition is the likely reason for its under- and misdiagnosis in children. image What is Known Retrograde cricopharyngeal dysfunction (R-CPD) has been described in adults. Diagnosis is based mainly on clinical criteria. Esophageal high-resolution impedance manometry (HRIM) with modified carbonation protocol was proposed to support the diagnosis of R-CPD in adults.What is New Adolescent patients and youth with R-CPD present distinct features on HRIM. HRIM with a modified carbonation protocol is helpful in the diagnosis of R-CPD in adolescent patients, concomitant with the reproduction of their symptoms. Adolescent patients with an inability to burp and involuntary throat noises should be assessed for R-CPD by HRIM. Botulinum toxin injection of the cricopharyngeus muscle is a potential treatment that should be considered after confirmation of the diagnosis on HRIM or impedance.
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关键词
belch,burp,dysphagia,esophageal motility disorders,upper esophageal sphincter
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