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Pediatric retrograde cricopharyngeal dysfunction diagnosed by high-resolution impedance manometry
Lev Dorfman1, Khalil El-Chammas1,2, Sherief Mansi1,2
1Division of Gastroenterology, Hepatology and Nutrition, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA.
Insights
Retrograde cricopharyngeal dysfunction (R-CPD) in adolescents causes inability to burp and throat noises. High-resolution esophageal impedance-manometry (HRIM) revealed abnormal upper esophageal sphincter relaxation and air entrapment, with symptom improvement after botulinum toxin injection.
Area of Science:
- Gastroenterology
- Pediatric Medicine
- Otolaryngology
Background:
- Retrograde cricopharyngeal dysfunction (R-CPD) is a condition characterized by the inability to burp, often presenting with symptoms like abdominal bloating, nausea, and chest pain.
- While R-CPD has been described in adults, its clinical and diagnostic features, particularly using high-resolution esophageal impedance-manometry (HRIM), have not been well-documented in pediatric populations.
- Botulinum toxin injection into the cricopharyngeal muscle has shown promise in treating R-CPD in adults.
Purpose of the Study:
- To describe the clinical presentation and HRIM findings in pediatric patients diagnosed with R-CPD.
- To characterize the specific manometric abnormalities associated with R-CPD in adolescents.
- To evaluate the efficacy of botulinum toxin treatment in this age group.
Main Methods:
- A retrospective review of five pediatric patients diagnosed with R-CPD was conducted.
- Patients' clinical and manometric data were analyzed.
- A modified HRIM protocol, including the ingestion of a carbonated beverage, was used to elicit symptoms and identify characteristic manometric patterns.
Main Results:
- Five female patients aged 15-20 years presented with inability to burp and involuntary throat sounds.
- HRIM revealed normal upper esophageal sphincter (UES) relaxation during swallowing but abnormal relaxation with high esophageal impedance after a carbonated drink, indicating air entrapment and secondary peristalsis.
- Four patients had esophageal motility disorders, and all experienced symptom improvement or resolution following botulinum toxin injection.
Conclusions:
- Adolescents presenting with inability to burp, reflux-like symptoms, bloating, and throat noises should be evaluated for R-CPD by pediatric gastroenterologists using HRIM.
- The findings highlight the utility of HRIM in diagnosing R-CPD in adolescents.
- Underdiagnosis of R-CPD in children may be attributed to its recent recognition and lack of established diagnostic criteria in this population.
Objectives:
The 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.
Methods:
Clinical 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.
Results:
We 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.
Conclusions:
Adolescents 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.
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