Introduction

Disorders of Gut-Brain Interactions (DGBIs), such as functional dyspepsia (FD) or Irritable Bowel Syndrome (IBS) are common in patients with Hypermobile Ehlers-Danlos Syndrome/Hypermobile Spectrum Disorder (hEDS/HSD)¹. Healthcare utilisation (HCU) is high in DGBIs but no study to date has looked at HCU or healthcare costs (HCC) in patients with hEDS/HSD and these DGBIs.

Aims & Methods

To calculate the healthcare costs associated with patients with hEDS/HSD and FD and/or IBS.
A cross-sectional study was performed. Patients completed a ROME IV, SF6 (quality of life questionnaire) and HCU questionnaire. The latter was adapted from the IBD Boost study² and included consultations, admissions, clinically assisted nutrition and hydration (CANH), medications and personal expenses over 6 months. Costs were calculated based on the National Schedule of NHS Costs³ and NHS Business Services Authority Prescription Cost Analysis⁴. Patients were grouped based on ROME IV diagnoses of FD and IBS. Chi-squared was used for categorical data. The Kruskal-Wallis test was used for continuous variables. A multivariate regression analysis was performed to see what healthcare characteristics were the strongest predictors of rising HCCs. In view of the multiple comparisons p<0.005 was considered significant.

Results

660 hEDS/HSD patients completed the study; 92.9% were female, median age 39 years. 90(13.2%) had IBS only, 197(29.0%) had FD only, 212(31.2%) had both FD and IBS and 161(23.7%) had neither IBS nor FD. Table 1 describes and compares HCU across the 4 groups. There were significantly more outpatient visits, admissions and CANH use in patients with FD±IBS. Total HCC per patient was significantly higher in patients with both FD and IBS (£3622.15) and FD only (£3782.82) compared to IBS only (£1610.12,p<0.001) and neither IBS nor FD (£1376.34,p<0.001). Consultations and admissions contributed to the majority of total HCC (31.3% and 27.4% respectively). Rheumatology and Gastroenterology were the commonest secondary/tertiary services used. CANH was used in 37.6% of patients with FD only and 29.2% of patients with both FD and IBS and accounted for 11% and 7% of total HCC in these DGBI groups respectively. The 10 highest HCCs (£21,653.80-£99,983.90) were in patients with FD±IBS, and they all required an inpatient admission.
The average SF6 in patients with FD±IBS (0.253) was significantly lower than in patients with IBS only (0.511,p<0.001) or those with neither IBS nor FD (0.519,p<0.001). 22.0% of the entire cohort had a score less than 0, which is a quality of life worse than death.
The adjusted R² value of the regression model used to determine what factors independently correlated with rising HCCs was 0.527. The variables significantly predicted rising HCCs, F(31,557)=3.775, p<0.001. 'Polypharmacy' and 'Employment Status' were the greatest predictors of rising healthcare costs (β=-0.209, p<0.001 and β=-0.121,p=0.003 respectively).

Table 1: Comparison of HCU in hEDS/HSD with and without FD and IBS over 6 months.

Conclusion

In hEDS/HSD patients, those with FD±IBS had the highest HCU and HCC highlighting the need to prioritise the development of cost-effective services for those with foregut disorders.

References

¹Lam et al. Clinical Gastroenterology and Hepatology 2021;19(2)
²Roukas et al. Frontline Gastroenterology 2022;14(1)
³England N. 2019/20 National Cost Collection Data Publication 2022
⁴NHSBSA. Prescription Cost Analysis – England 2020/21 2021

Introduction

Patients with Hypermobile Ehlers Danlos Syndrome (hEDS) have a high prevalence of Disorders of Gut-Brain Interaction (DGBI) and pose complex nutritional challenges in gastroenterology clinic. Despite this, little is known regarding nutrient intake and adequacy in hEDS.

Aims & Methods

Our aim was to assess nutrient intake and adequacy of patients with hEDS, as well as to determine which factors, including co-existent DGBI, are associated with poor intake. We conducted a cross sectional survey of patients with hEDS from two tertiary neurogastroenterology clinics as well as the Ehlers-Danlos Support UK. Inclusion criteria were adults with hEDS, exclusion criteria were those currently or previously using artificial nutrition. Patients completed the validated semi-quantitative Scottish Collaboration Group Food Frequency Questionnaire (FFQ) alongside questionnaires characterising: demographics, DGBIs (Rome IV), gastrointestinal, mood, somatic and autonomic symptoms, past history of eating disorders and screen for Avoidant Restrictive Food Intake Disorder (ARFID) using the Nine Item Avoidant-Restrictive Scale (NIAS). Nutrient intake was calculated from the FFQ and nutrient adequacy was calculated by comparison with UK daily reference values (DRVs) for age and sex. Unpaired t-tests were performed to compare nutrient intakes among patients with and without comorbidities, including the most common DGBIs.

Results

425 participants were included in the final analysis (mean: 41 years, 96% female). Only 48.5% reported BMI in the normal range, whereas 5.2% were underweight (BMI <18.5 kg/m²)and46.4% were overweight/obese (BMI >25 kg/m²). Only one third met their estimated average requirements (EAR) for energy (kcal/d). The majority met their reference nutrient intake (RNI) for protein (87.3%) and fat (72.5%), in contrast to only 21.2% for carbohydrate. Fibre intakes were very low (AOAC mean 23.7g/day (SD: 12.7) with only 24.7% meeting the RNI of 30 g/d. More than 58% of participants met/exceeded their DRV for micronutrients (zinc, magnesium, copper, manganese and selenium) and >95% for vitamins. Patients with functional nausea (n=157) were less likely to meet their protein requirements compared to those without (82.2% vs. 90.3%, p=0.015) and also had lower intakes for sodium (p=0.02), magnesium (p=0.02), calcium (p=0.01), zinc (p=0.002), selenium (p=0.03), folic acid (p=0.02) and vitamin C (p=0.05). Patients with previous eating disorders were less likely to meet nutrient requirements for thiamine (p=0.01) and vitamin B12 (p=0.04). Patients with ARFID (n=110) were less likely to meet requirements for all nutrients (except fat, calcium, vitamin D, vitamin B12) compared to patients without.

Conclusion

Patients with hEDS are rarely underweight and tend to consume high protein, high fat diets that are low in fibre. Functional nausea is the single most important DGBI impacting nutrient intake, as well as previous eating disorders and disordered eating (ARFID). These factors should be considered by clinicians and dietitians when assessing nutrient intake in this complex patient group.

Disclosure

RT, AF and QA have received a grant from EDS-UK to conduct this study. AF undergoes consulting work for Abbvie.
KW has received research funding from Almond Board of California, Danone, International Nut & Dried Fruit Council, and has a patent for volatile organic compounds in the detection and management of IBS.
QA undergoes consulting work for Takeda Pharmacueticals, and Classado Biosciences Ltd.

Introduction

Hypermobile Ehlers-Danlos syndrome (hEDS) is a hereditary non-inflammatory disorder of connective tissue laxity characterized by joint hyperextensibility in association with musculoskeletal symptoms. The prevalence of gastrointestinal (GI) symptoms, such as abdominal pain, nausea, and constipation is high in hEDS. Research on the relationship between EDS and GI symptoms has mostly been restricted to disorders of the gut-brain axis, whereas the relationship between hEDS and GI dysmotility remains to be clarified.

Aims & Methods

This study aimed to explore antro-duodenal manometry (ADM) patterns in patients with hEDS presenting with GI symptoms.

In a prospective open-label study, patients with hEDS and GI symptoms presumed to be the results of GI dysmotility underwent a 6-hour high resolution ADM. The manometry tracings were analyzed by two investigators (KS and JC) independently. Antral contraction frequency (contractions per minute) < 1.0 was considered as antral hypomotility. Pylorospasm was defined as isolated pyloric pressure waves within 60 minutes postprandial. The following neuropathic enteric patterns were scored: presence of retrograde peristalsis, bursts, clustered contractions, and absence of phase III contractions. Abnormal ADM was defined as having antral hypomotility and/ or pylorospasm and/ or neuropathic enteric patterns.

Results

Fifty patients (48 women, mean age 30.4 ± 11.1 yrs.) were included. At baseline, 74% of patients were dependent on enteral feeding and the mean BMI was 20.4 ± 4.4 kg/m².
Overall, ADM tracings with one or more abnormal patterns were identified in 53% of patients. Antral hypomotility and pylorospasm were detected in 29% and 15% of patients, respectively. Neuropathic enteric patterns were only seen in 17% of patients. In all cases, no more than one neuropathic pattern was identified. Among the observed neuropathic enteric patterns, 15% of patients showed retrograde peristalsis, 13% absence of phase III contractions, and 2% bursts. Clustered contractions were not identified in this cohort.

Conclusion

Hypermobile Ehlers-Danlos patients presenting with gastrointestinal symptoms showed low numbers of abnormal ADM patterns. In particular, enteric dysmotility was observed in only a small minority of cases. Antroduodenal dysmotility therefore does not appear to be a principle pathomechanism in the majority of hEDS cases studied. Further research is needed to understand the mechanisms that underlie GI symptom generation in this population.

Introduction

Patients with Hypermobile Ehlers Danlos Syndrome (hEDS) have a high prevalence of Disorders of Gut-Brain Interaction (DGBI) and pose complex nutritional challenges in gastroenterology clinic. Despite this, little is known about their dietary patterns and whether this is associated with relevant clinical characteristics.

Aims & Methods

Our aim was to identify dietary patterns in hEDS, phenotype patients according to their dietary patterns and determine whether these patterns are associated with gastrointestinal or other factors. We conducted a cross sectional survey of patients with hEDS from two tertiary neurogastroenterology clinics as well as the Ehlers-Danlos Support UK. Patients completed the validated semi-quantitative Scottish Collaboration Group Food Frequency Questionnaire (FFQ) alongside questionnaires characterising: demographics, DGBIs (Rome IV), past history of Postural orthostatic Tachycardia Syndrome (PoTS) or Mast Cell Activation Syndrome (MCAS), somatic and autonomic symptoms, anxiety and depression, gastrointestinal specific anxiety, Restrictive Food Intake Disorder (ARFID) screen, artificial nutrition use, nutritional supplement use and previous dietetic consultation. The 169 food items on the FFQ were consolidated into 49 food groups and principal components analysis (PCA) was used to derive dietary patterns. Cluster analysis was then used to classify patients into dietary groups. Clinical characteristics were compared between clusters using the Chi-square test, analysis of variance (ANOVA) and Kruskal-Wallis test as appropriate.

Results

470 patients were included in the final analysis, of which four clusters were identified: cluster 1 (n=169) - low energy intake; cluster 2 (n=62) - vegetarian / health conscious; cluster 3 (n=133) - prudent / low residue and cluster 4 (n=106) - refined / highly processed food. Patients in the low energy intake group (cluster 1) were most likely to attend the tertiary neurogastroenterology clinic (p=0.002), have a dietetic consultation (p=0.02) and report previous or current use of artificial nutrition (p=0.03) compared to the other clusters. Patients in the vegetarian / health conscious group (cluster 2) had the lowest depression scores (p=0.03), lowest GI specific anxiety (p=0.005) and interestingly, were most likely to take nutritional supplements (p=0.04). Patients in the prudent / low residue group (cluster 3) and the refined / highly processed food group (cluster 4) had the highest body mass index (p=0.003). Patients in the refined / highly processed food group (cluster 4) were youngest (p<0.001), had higher scores for depression (p=0.03), higher scores for GI specific anxiety (p=0.005) and higher scores for ARFID (p=0.007). They were also least likely to take nutritional supplements (p=0.04) and least likely to attend the tertiary neurogastroenterology clinic (p=0.002). Interestingly, there was no significant association between dietary patterns and PoTS, MCAS or DGBIs.

Conclusion

This is the first study to identify dietary patterns in patients with hEDS. The most common dietary cluster was lower energy intake and this was most likely to be seen in those attending tertiary clinics and utilising artificial nutrition. Whereas those who are on a refined / highly processed food diet tend to be younger and have more psychopathology. Further research is needed to establish how these associations could best be utilised in clinical practice.

Disclosure

RT, AF and QA have received a grant from EDS-UK to conduct this study. AF undergoes consulting work for Abbvie.
KW has received research funding from Almond Board of California, Danone, International Nut & Dried Fruit Council, and has a patent for volatile organic compounds in the detection and management of IBS.
QA undergoes consulting work for Takeda Pharmacueticals, and Classado Biosciences Ltd.

Introduction

Exposure to COVID-19 has been shown previously associated with a higher risk for long-term gastrointestinal symptoms and disorders of gut-brain interaction (DGBI) occurrence, including irritable bowel syndrome (IBS) (1). However, it is still unclear whether SARS-CoV-2's effects on the gastrointestinal tract are more severe in patients already diagnosed with a DGBI and symptoms fluctuations in these patients over time after COVID-19 exposure.

Aims & Methods

To explore hospitalization outcomes and gastrointestinal (GI) symptoms fluctuation over a 1-year follow-up of COVID-19 patients with IBS or functional dyspepsia (FD) at enrollment compared to COVID-19 patients without any previous GI symptoms/disorders/diseases/surgeries.
In this retrospective multicenter cohort study, patient charts were reviewed for demographic data, medical history including GI chronic diseases, and hospitalization outcomes. All participants completed the Gastrointestinal Symptom Rating Scale (GSRS) over 4 time points: at COVID-19 onset, at 1, 6, and 12 months, and the Hospital Anxiety and Depression Scale (HADS) over 2 time points: at 6 and 12 months. The cohort was divided into two groups: (1) IBS/FD group (2) control group.

Results

A total of 599 patients were eligible for the study (n=61 IBS/FD group, n=538 control group). There were no differences between the groups regarding hospitalization outcomes. There was a significant decrease in both IBS/FD and control groups in depression [6.17±4.45 at 6 months and 3.17±4.14 at 12 months for IBS/FD group (p=0.007) and 3.55±4.02 at 6 months and 3.05±3.74 at 12 months for the control groups (p=0.019), respectively] and anxiety levels over time [5.94±4.32 at 6 months and 4.10±4.58 at 12 months for IBS/FD group (p=0.019) and 3.58±3.71 at 6 months and 3.26±3.65 at 12 months for the control groups (p<0.001), respectively]. The pooled Odds Ratios (OR) of the effect of IBS/FD over 4 time points ranged from 1.93 for diarrhea to 5.10 for increased distention, and were statistically significant for all GI symptoms, even after adjustments for gender, depression, and anxiety. While there was a significant improvement over time in the severity of abdominal pain (p=0.008), hunger pains (p=0.011), nausea (p=0.001), diarrhea (p=0.005) and loose stool (p=0.007), there was also a significant deterioration in constipation (p=0.002), hard stool (p<0.001) and feeling of incomplete evacuation (p=0.007) among the IBS/FD group.

Conclusion

Gastrointestinal symptoms within and between the control group and the IBS/FD group changed over time after COVID-19 onset. Patients with IBS or FD at enrollment showed increased complaints of constipation, hard stool, and incomplete evacuation after COVID-19 exposure.

References

1. Marasco G, Cremon C, Barbaro MR the GI-COVID19 study group, et al Post COVID-19 irritable bowel syndrome. Gut 2023;72:484-492.

Introduction

Among patients with functional gastrointestinal disorders (FGID), food intake is often identified as a symptom trigger. Common foods known to cause gastrointestinal (GI) symptoms include fatty foods (1) and fermentable carbohydrates (FODMAPs) (2), but the evidence is limited and may vary among different FGID.

Aims & Methods

The aim of this study was to explore if certain foods are considered more tolerable, and which specific foods are identified as triggers of GI symptoms among participants with irritable bowel syndrome (IBS) and functional dyspepsia (FD). An online survey was conducted including n=537 participants from the Swedish general population with symptoms compatible with a functional bowel disorder. The survey included questions on demographics and an adapted food frequency questionnaire that assessed intakes of 38 different food items or aggregated food groups. For each food, participants had to answer whether or not they considered the specific food to be a trigger for GI symptoms.

Results

Among the respondents, a total of 220 participants with IBS and/or FD (mean age 34.7 years, body mass index 26.8 kg/m², women: 78.2%) were included in the analyses, of whom n=128 fulfilled criteria for FD and n=136 for IBS. Foods that were least commonly reported to cause GI symptoms included hard bread, fish and shellfish, berries, nuts, and seeds (Figure). The most frequently reported food triggers included pizza, crisps, confectionery, candy, ice cream, white bread, dairy, french fries, pie, and sausages. White bread, more often than whole grain bread, was reported to cause GI symptoms. Most vegetables and fruits were well tolerated in FD. A few FODMAP-rich vegetables, such as onion, garlic, beans, lentils, cauliflower and cabbage were more often reported as food triggers in IBS compared with FD. Having overlapping IBS and FD increased the prevalence of foods that were considered to cause symptoms.

Conclusion

In individuals with IBS and/or FD, foods traditionally associated with a healthy dietary pattern were less commonly reported to cause GI symptoms, whereas foods associated with western food/junk food were more commonly reported to cause GI symptoms. Promoting healthy food choices seem compatible with dietary treatment of patients with IBS and FD.

References

1, Feinle-Bisset C, Azpiroz F. Dietary and lifestyle factors in functional dyspepsia. Nature reviews Gastroenterology & hepatology. 2013 Mar;10(3):150-7.
2. van Lanen AS, de Bree A, Greyling A. Efficacy of a low-FODMAP diet in adult irritable bowel syndrome: a systematic review and meta-analysis. European journal of nutrition. 2021 Sep;60:3505-22.