Dorfman Lev, El-Chammas Khalil, Mansi Sherief, Graham Kahleb, Kaul Ajay
Division of Gastroenterology, Hepatology and Nutrition, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA.
Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio, USA.
J Pediatr Gastroenterol Nutr. 2024 May;78(5):1098-1107. doi: 10.1002/jpn3.12193. Epub 2024 Mar 22.
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.
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.
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.
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.
无法打嗝，即逆行性环咽肌功能障碍（R-CPD），最初是在成人中被描述的。R-CPD的临床诊断标准包括无法打嗝、腹胀和不适/恶心、餐后胸痛以及不自主的声音。据报道，向环咽肌注射肉毒杆菌毒素是有益的。R-CPD青少年患者的高分辨率食管阻抗测压（HRIM）特征尚未被描述。我们研究的目的是描述R-CPD儿科患者的临床和HRIM检查结果。
回顾了5例被诊断为R-CPD的儿科患者的临床和测压特征。对HRIM研究方案进行了修改，包括饮用碳酸饮料以诱发症状和独特的测压特征。
我们报告了5例年龄在15至20岁之间的女性患者，她们表现为无法打嗝和不自主的喉咙声音。HRIM显示吞咽时食管上括约肌（UES）松弛正常，但在饮用碳酸饮料激发试验后，UES松弛异常，同时食管阻抗升高，反映有气体潴留和继发性蠕动。4例患者表现出食管运动障碍。所有患者在向环咽肌注射肉毒杆菌毒素后症状均有改善或缓解。
对于无法打嗝、有反流样症状、腹胀和不自主喉咙声音的青少年，儿科胃肠病学家应通过HRIM对其进行R-CPD评估。这种新疾病相对较新才被认识，可能是其在儿童中诊断不足和误诊的原因。
