Prevalence of continuous positive airway pressure-related aerophagia in obstructive sleep apnea: an observational study of 753 cases undergoing CPAP/BiPAP treatment in a sleep clinic - part one of a two-part series.

PURPOSE: Aerophagia occurring during continuous positive airway pressure (C-aerophagia in CPAP) in patients with obstructive sleep apnea (OSA) disturbs CPAP therapy. However, the diagnostic criteria and exact prevalence of C-aerophagia are not well documented; therefore, this study aimed to evaluate them. METHODS: Newly developed criteria (flatulence, eructation, and abdominal bloating, with quantitative evaluation of these symptoms and a clear timing of onset) were used to diagnose C-aerophagia. The study included 753 adults with OSA who underwent follow-up visits for positive airway pressure treatment (including CPAP and bilevel positive airway pressure). The observation period ranged from 6 months to 3 years past the survey date (between May 1 and July 31, 2023). Medical records of patients were retrospectively analyzed. Discomfort associated with C-aerophagia was examined using a visual analog scale (VAS); a score ≥ 7 was associated with definite discomfort. Association of patient demographics and CPAP parameters with occurrence of C-aerophagia was analyzed using multivariate analysis. RESULTS: The prevalence of C-aerophagia was 7.2%. Although more than half of these patients reported discomfort associated with aerophagia, only 44.4% reported to their physician. The multivariate analysis showed that increased CPAP pressure level (odds ratio [OR] = 1.24) and comorbid gastroesophageal reflux disease (GERD; OR = 2.52) promote C-aerophagia, while increased age (OR = 0.76) and body mass index (BMI; OR = 0.88) inhibit it. CONCLUSION: The prevalence of C-aerophagia was 7.2%. Most patients with C-aerophagia experience discomfort, but may not report these symptoms. High CPAP pressure and GERD promoted C-aerophagia, while aging and increased BMI prevented it.

Fatal gastrointestinal complications in Pitt-Hopkins syndrome.

Pitt-Hopkins syndrome (PTHS) is a rare neurodevelopmental disorder caused by mutations of the transcription factor 4 (Tcf4) gene. Individuals with PTHS often suffer from severe abdominal bloating and constipation. In this short communication, we discuss two individuals with PTHS who died unexpectedly due to gastrointestinal complications. We aim to increase awareness among healthcare professionals who care for individuals with PTHS, to ensure adequate screening and management of gastrointestinal symptoms in this population. Moreover, we discuss how fatal gastrointestinal complications may be related to PTHS and provide an overview of the literature.

Teashirt 1 (Tshz1) is essential for the development, survival and function of hypoglossal and phrenic motor neurons in mouse.

Feeding and breathing are essential motor functions and rely on the activity of hypoglossal and phrenic motor neurons that innervate the tongue and diaphragm, respectively. Little is known about the genetic programs that control the development of these neuronal subtypes. The transcription factor Tshz1 is strongly and persistently expressed in developing hypoglossal and phrenic motor neurons. We used conditional mutation of Tshz1 in the progenitor zone of motor neurons (Tshz1MNΔ) to show that Tshz1 is essential for survival and function of hypoglossal and phrenic motor neurons. Hypoglossal and phrenic motor neurons are born in correct numbers, but many die between embryonic day 13.5 and 14.5 in Tshz1MNΔ mutant mice. In addition, innervation and electrophysiological properties of phrenic and hypoglossal motor neurons are altered. Severe feeding and breathing problems accompany this developmental deficit. Although motor neuron survival can be rescued by elimination of the pro-apoptotic factor Bax, innervation, feeding and breathing defects persist in Bax-/-; Tshz1MNΔ mutants. We conclude that Tshz1 is an essential transcription factor for the development and physiological function of phrenic and hypoglossal motor neurons.

Prevalence of Functional Gastrointestinal Disorders in Children and Adolescents.

OBJECTIVES: To determine the prevalence of functional gastrointestinal (GI) disorders (FGIDs) in children and adolescents in a representative community sample of the US. STUDY DESIGN: The study recruited a general population sample of mothers (n = 949) of children and adolescents aged 4-18 years. Child and adolescent GI symptoms were assessed using parental report through online questionnaires, including the Questionnaire on Pediatric Gastrointestinal Symptoms and the PedsQL4.0 Generic Core Scale. Parental GI symptoms, and demographic characteristics were also assessed. The data was used to determine prevalence of FGIDs. RESULTS: Using Rome III criteria by parental report, 23.1% of children and adolescents qualified for at least 1 FGID. Functional constipation and abdominal migraine were the most common FGIDs. All 10 child/adolescent FGIDs occurred, except rumination. Significant prevalence differences were not found between sexes, except in functional constipation, which was more prevalent in males than females (P = .022). There were no significant prevalence differences between racial or ethnic groups. Children who met criteria for an FGID had lower quality of life (median = 76.4) than children who did not (median = 89.6; P < .001). Children were more likely to qualify for a FGID if their parent also qualified for a FGID (P < .01). CONCLUSIONS: FGIDs are common in children and adolescents in the US. There are no significant differences in FGIDs between sex, race, or ethnic groups, except in functional constipation. There is overlap between parental and child FGID symptoms. Children with a FGID report a lower quality of life than healthy children.

Does severe and chronic esophageal distension by air (observed in SGB and GB) affect the esophageal mucosa integrity (MNBI).

BACKGROUND: Supragastric belching (SGB) and aerophagia are behavioral disorders characterized by air induced esophageal distension. SGB is known to be associated with Gastro Esophageal Reflux Disease (GERD). Low Mean Nocturnal Baseline Impedance (MNBI) values support GERD diagnosis. We aimed to assess if chronic esophageal distension by air affects the esophageal mucosa integrity by assessing changes in MNBI. METHODS: In a single-center database study, we searched retrospectively for patients with a diagnosis of pathological SGB (n = 146) or aerophagia (n = 34) based on impedance-pH reflux monitoring. During the examined period, patients with a conclusive negative diagnosis of SGB and no evidence of aerophagia were used as a control cohort (n = 191). MNBI at 3, 5, and 17 cm over Lower Esophageal Sphincter (LES) was evaluated. GERD was diagnosed if acid exposure time (AET) >6%. All impedance studies of included patients were prospectively reevaluated. RESULTS: GERD was diagnosed in 31.7% patients with SGB, a rate not different in comparison to patients without SGB (30.8%, p = 0.906). MNBI at 3 and 5 cm above the LES was significantly decreased among patients with SGB. SGB was not correlated with MNBI at 3 cm over the LES, (p: 0.086 OR: 1.000 95% CI: 0.999-1.001) when using multivariate analysis. Moreover no difference was spotted as far as MNBI at 3, 5, and 17 cm over the LES is concerned among patients with or without aerophagia. CONCLUSION: Even if patients with SGB do show lower MNBI values, esophageal distention due to excessive air movement does not directly lead to impairment of esophageal mucosa integrity.

Simple office-based behavioral approach to patients with chronic belching.

Chronic belching can be a difficult and socially disabling symptom often attributed to reflux with poor response to therapy. In patients where aerophagia is identified as a clear cause, treatment with baclofen may not be tolerated, and biofeedback therapy is time-intensive and may still not be effective. In this pilot study, an office-based easy-to-perform method based on sustained glottal opening was used in five patients with chronic belching, in whom reflux and other causes had been excluded. Treatment consisted of having the patient breathe slowly and diaphragmatically with his or her mouth open during supine, then sitting periods to prevent belching. When this was successful, patients were then counseled on continuing this breathing with mouth slightly ajar as an outpatient using this persistently. Wide mouth opening was used for rescue therapy of belching attacks. All five patients responded to the office-based therapy with complete cessation of belching during the visit. At 1-month follow up, four patients remained asymptomatic. One patient was asymptomatic but for two breakthrough attacks easily managed with the protocol. A simple office-based procedure based on complete glottal opening can be curative for a subset of patients with chronic eructation secondary to repetitive air swallowing.

Persistent Nausea and Gastrointestinal Distention: A Case Report of Aerophagia.

A 43-year-old woman experienced acute nausea, diarrhea, and abdominal pain, leading her to our hospital. No relevant medical history or physical abnormalities were noted. Symptoms persisted for a month, causing weight loss and abdominal bloating. CT scans revealed distension throughout the gastrointestinal tract without stenosis. Intestinal pseudo-obstruction and aerophagia were suspected. MR enterography confirmed normal gastric and intestinal motility, diagnosing the condition as aerophagia-induced gastrointestinal distention. This case underscores the value of MR enterography in assessing intestinal motility and differentiating between intestinal pseudo-obstruction and aerophagia.

National Prevalence of Functional Gastrointestinal Disorders in Jordanian Children.

INTRODUCTION: Functional gastrointestinal disorders are common in children. After the introduction of Rome criteria for diagnosis, assessment of prevalence of such disorders became an attainable goal. Since data from our part of the world are scarce, this study aimed at estimating the prevalence of functional gastrointestinal disorders in Jordanian children. PATIENTS AND METHODS: In a school-based cross-sectional study, recruited children between the age of 4 and 18 were asked to fill the Arabic version of the Questionnaire on Pediatric Gastrointestinal Symptoms-Rome III Version (QPGS-RIII). Patients were identified based on meeting the ROME III criteria. RESULTS: Of 2000 children that were recruited, 1587 (79.4%) returned completed questionnaires. Males accounted for 841 (53%) of participants. Mean age was 10.2 years (range, 4 to 18 years). A total of 815 (51.4%) of participants were younger group (4 to 10 years of age), mean ± SD age of the two age groups was 8.1 ± 1.4 and 14.0 ± 1.8 years, respectively. Overall, 514 (32.4%) children met the criteria for having at least one FGID. The most common FGIDs in Jordanian children were functional constipation, followed by Aerophagia, abdominal migraine, and irritable bowel syndrome (prevalence estimates of 16.0%, 7.0%, 5.1%, and 3.6%, respectively). FGIDs were more common in younger girls and older boys but were not statistically significant. Concomitant presence of more than one FGID was observed in almost 15% and 22% of younger and older age groups, respectively. CONCLUSION: Functional gastrointestinal disorders (FGIDs) are common in Jordanian school children. Functional constipation is the most common disorder. Further studies investigating the predisposing factors in our population are needed.

Gastric Necrosis and Perforation Following Massive Gastric Dilatation in an Adolescent Girl: A Rare Cause of Acute Abdomen.

Gastric necrosis with perforation is a rare and potentially life-threatening condition in childhood beyond the neonatal period. We report a case of gastric necrosis and perforation of a portion of the great curvature due to a massive gastric dilatation caused by pathological aerophagia in a 13-years-old, mentally impaired adolescent girl. Despite the successful surgical treatment, the patient's condition rapidly deteriorated post-operatively and she died due to the multisystem organ failure and multiple infections. In addition, we surveyed the literature on this rare condition and assessed the preventive actions to reduce this life-treating condition.

Unusual invasion of primary pulmonary adenocarcinoma in a cat.

CASE SUMMARY: An 11-year-old female spayed domestic shorthair cat was referred to the Foster Hospital for Small Animals, USA for suspected dysautonomia based on weight loss, vomiting and referral radiographs that showed severe dilation of the esophagus, stomach and entire gastrointestinal tract. After recheck radiographs revealed a decrease in gas compared with the referral images, persistently reduced gastrointestinal motility was deemed less likely; however, cardiomegaly and a soft tissue opacity overlying the esophagus were noted. Echocardiogram identified a pulmonary mass either impinging on or invading the local organs. At necropsy, gross pathology and histopathology showed an aggressive pulmonary carcinoma invading the heart, trachea and esophagus, resulting in a focal esophageal stricture. Gastrointestinal gas dilation was most likely secondary to aerophagia as a result of chronic partial esophageal obstruction. RELEVANCE AND NOVEL INFORMATION: To our knowledge, this is the first reported feline case of invasion of the esophagus and heart by a pulmonary adenocarcinoma. Other than weight loss, the clinical signs for this invasive neoplasm were not evident until the cat was diagnosed with hyperthyroidism and prescribed oral medications. This reinforces the fact that pulmonary adenocarcinoma is difficult to detect clinically until secondary problems from the primary or metastatic neoplasm arise.

A Randomized Crossover Trial Comparing Autotitrating and Continuous Positive Airway Pressure in Subjects With Symptoms of Aerophagia: Effects on Compliance and Subjective Symptoms.

STUDY OBJECTIVES: To assess the benefit and tolerance of autotitrating positive airway pressure (APAP) versus continuous positive airway pressure (CPAP) in subjects who experience aerophagia. METHODS: This is the report of a prospective, two-week, double-blinded, randomized crossover trial set in an Australian clinical sleep laboratory in a tertiary hospital. Fifty-six subjects who reported symptoms of aerophagia that they attributed to CPAP were recruited. Full face masks were used by 39 of the 56 subjects recruited. Subjects were randomly and blindly allocated to either CPAP at their treatment recommended pressure or APAP 6-20 cm H2O, in random order. Subjects spent two weeks on each therapy mode. Therapy usage hours, 95th centile pressure, maximum pressure, 95th centile leak, and residual apnea-hypopnea index (AHI) were reported at the end of each two-week treatment period. Functional Outcome of Sleepiness Questionnaire, Epworth Sleepiness Scale, and visual analog scale to measure symptoms of aerophagia were also completed at the end of each 2-week treatment arm. RESULTS: The median pressure (P < .001) and 95th centile pressure (P < .001) were reduced with APAP but no differences in compliance (P = .120) and residual AHI were observed. APAP reduced the symptoms of bloating (P = .011), worst episode of bloating (P = .040), flatulence (P = .010), and belching (P = .001) compared to CPAP. There were no differences in Epworth Sleepiness Scale or Functional Outcome of Sleepiness Questionnaire outcomes between CPAP and APAP. CONCLUSIONS: APAP therapy reduces the symptoms of aerophagia while not affecting compliance when compared with CPAP therapy. CLINICAL TRIAL REGISTRATION: Australian and New Zealand Clinical Trials Registry at https://www.anzctr.org.au, trial number ACTRN12611001250921. COMMENTARY: A commentary on this article appears in this issue on page 859.

Multichannel intraesophageal impedance pattern of children with aerophagia.

BACKGROUND: Childhood aerophagia is defined by the Rome III criteria as two or more of the following symptoms; air swallowing, abdominal distension due to intraluminal air, or repetitive belching and/or flatus. The aim of this study was to determine the multichannel intraesophageal impedance (MII) pattern in children suffering from aerophagia. METHODS: We compared the MII tracings of five children with aerophagia according to Rome III criteria (three girls, median age 7 years) to five controls (three girls, median age 8 years). Controls underwent MII for symptoms of gastro-esophageal reflux, and had normal findings. Liquid swallows (LS), air swallows (AS), mixed swallows (MS) and supragastric belching (SGB) were recorded. Meals were excluded from the analysis. All MII parameters were separately analyzed in the upright and recumbent position. All data are reported as median number of events per hour. KEY RESULTS: There was no difference in the total number of LS and MS between the two groups. However, the total number of AS in patients was significantly higher than in controls (26/h vs 5.5/h, p < 0.05) but only in the upright position (46/h vs 8.2 in control group, p < 0.05). SGB was noted only in patients with aerophagia (2.6/h vs 0/h, p < 0.01). CONCLUSIONS & INFERENCES: Children suffering from aerophagia have a specific MII pattern with an increased frequency of air swallows in the upright position and supragastric belching. MII may be used as a tool to confirm diagnosis of aerophagia in children.

Functional aerophagia in children: a frequent, atypical disorder.

Aerophagia is a functional gastrointestinal disorder characterized by repetitive air swallowing, abdominal distension, belching and flatulence. Pathologic aerophagia is a condition caused by the swallowing of excessive volumes of air with associated various gastrointestinal symptoms, such as burping, abdominal cramps, flatulence and a reduced appetite. It is a clinical entity that can simulate pediatric gastrointestinal motility disorders, such as gastroparesis, megacolon and intestinal pseudo-obstruction, and presents more frequently in children with mental retardation. Early recognition and diagnosis of functional aerophagia or pathologic aerophagia is required to avoid unnecessary, expensive diagnostic investigations or serious clinical complications. Functional aerophagia is frequent in the adult population, but rarely discussed in the pediatric literature. We present two pediatric clinical cases with a history of functional constipation in whom gaseous abdominal distension was the most important symptom. Mechanical intestinal obstruction, chronic intestinal pseudo-obstruction, malabsorption and congenital aganglionic megacolon were ruled out. Extensive gaseous abdominal distension was due to aerophagia, and treatment consisted of parents' reassurance and psychological counseling.

A Case Report of Aerophagia Successfully Treated with Bukuryotakushato-Based Kampo Medicine / 日本東洋医学雑誌

A 20-year-old woman had a tendency of belching from a young age. She began to realize that she might have excessive belching compared to other people for the past two years. She was prescribed some medicines to improve gastrointestinal function by some physicians, but her symptoms did not resolve. Aerophagia was suspected as forceful belching tends to occur soon after eating or drinking. Incidentally, ihon is a condition in which one vomits food half a day after the ingestion due to the malfunction of the digestive system. I considered her symptoms as ihon and prescribed bukuryotakushato, and her symptoms improved. This case report indicated that ihon could be defined as a symptom of forceful regurgitation of food or air, regardless of the timing after meals.

Stomach gas analyses in canine acute gastric dilatation with volvulus.

BACKGROUND: The origin of the gas in the stomachs of dogs with acute gastric dilatation or gastric dilatation with volvulus (GDV) often is disputed. HYPOTHESIS: We tested the hypothesis that gaseous distention resulted from aerophagia. ANIMALS: Ten cases of GDV that were submitted to an emergency clinic were sampled intraoperatively. METHODS: With the abdomen open, the needle of a vacutainer blood collection set was inserted into the distended stomach, and gas was collected into 10 mL glass vacutainer vials with rubber stoppers. These were stored at room temperature for 1-7 days and then analyzed by gas chromatography and mass spectroscopy. RESULTS: CO2 composition ranged from 13 to 20%. One dog had an H2 concentration of 29%. CONCLUSIONS: Because the CO2 content of atmospheric air is less than 1%, these findings suggest that the gaseous gastric distention in GDV is not the result of aerophagia.

The role of esophageal multichannel intraluminal impedance monitoring for 24 hours in diagnosis of aerophagia / 中华实用儿科临床杂志

Objective To investigate the clinical characteristics of aerophagia in children,and to evaluate the effect of 24 hours esophageal multichannel intraluminal impedance monitoring in diagnosis of aerophagia.Methods The clinical data of 30 children with aerophagia hospitalized in Department of Digestive Disease,Nanjing Children's Hospital Affiliated to Nanjing Medical University from July 2005 to June 2013 were reviewed.The 24 hours esophageal multichannel intraluminal impedance monitoring was performed in 8 cases of aerophagia.The treatment of these patients normally included psychological counseling,dietary adjustment,Iactulose,Domperidone and vitamins,while Simethicone was given to the patients if normal treatment was invalid.Results Of the 30 patients,aged 2 to 13 years,averaged (8.6 ± 4.5) years,the chief complaints were abdominal distention in 30 cases (100.00％),visible or audible air swallowing in 28 cases (93.33％),repetitive belching in 27 cases (90.00％),reduced appetite in 25 cases (83.33％),constipation in 22 cases (73.33％),psychological stresses in 15 cases (50.00％),recurrent abdominal pain syndrome in 10 cases(33.33％),chronic diarrhea in 3 cases(10.00％) and acute abdominal pain in 3 cases(10.00％).Among the 30 patients,24 hours esophageal multichannel intraluminal impedance monitoring was performed in 8 cases,averaging 149.63 (47-553)times of air swallowing were recorded.In the 30 patients,24 cases (80.00％) were relieved after being treated with psychological counseling,dietary adjustment,lactulose,domperidone and vitamins,and only 6 cases(20.00％) were invalid for normal treatment but well responeded to Simethicone.Conclusions Aerophagia is a kind of functional gastrointestinal disorders in children.Abdominal distention,air swallowing,repetitive belching,reduced appetite,constipation and pain are common symptoms.It can be cured through psychological counseling management,diet modification,laxatives,propulsives and vitamins.24 hours esophageal multichannel intraluminal impedance monitoring is a useful technology in the diagnosis of aerophagia children.

Hangekobokuto (Banxia-houpo-tang), a Kampo Medicine that Treats Functional Dyspepsia.

Although abdominal bloating is one of the most bothersome symptoms experienced by patients with functional dyspepsia (FD), therapeutic drugs to relieve abdominal bloating have not been established. We investigated the Kampo (Chinese herbal) medicine, Hangekobokuto (Banxia-houpo-tang, HKT) for patients with FD from the standpoint of bowel gas retention. The bowel gas volume calculated from a plain abdominal radiogram (gas volume score, GVS) in FD patients was significantly higher than that in healthy subjects. Two week administration of HKT in the FD patients showed a significant decrease of GVS. Furthermore, gastrointestinal symptoms, especially symptoms of abdominal pain, indigestion and constipation, all of which are closely related to abdominal bloating, improved significantly in FD patients after the administration of HKT. These results suggest that HKT improves abdominal bloating accompanied by the reduction of bowel gas in FD patients.

Foreign body granulamatous reaction to endoluminal gas in pneumatosis intestinalis - a case report.

Pneumatosis intestinalis is a rare condition occurs in a wide variety of patients, some of whom require urgent surgery, while others can be observed with resolution of symptoms and radiographic findings. Most often the need for surgery is intestinal obstruction. We report a case of pneumatosis intestinalis with an abdominal mass causing obstruction. The histological surprise and the rarity of the obstruction is highlighted in this case report.