Impact of a Child's Celiac Disease Diagnosis and Management on the Family.

BACKGROUND: Little attention has been paid to family-wide repercussions of a child's celiac disease diagnosis and concomitant gluten-free diet management. AIMS: We quantitatively and qualitatively describe positive and negative family-wide effects of a child's celiac disease diagnosis and disease management. METHODS: We interviewed 16 families with at least one child currently following a gluten-free diet, with a biopsy-confirmed celiac disease diagnosis ≥ 1 year prior. Mothers and fathers independently rated child's dietary adherence, concern about child's health status, burden in caring for child's dietary needs, and level of change in various aspects of life post- diagnosis. Children rated their own celiac-specific quality of life through a validated scale. Seventy-one in-depth semi-structured interviews were conducted with 16 children with celiac disease, 31 parents, and 24 siblings. RESULTS: Mothers and fathers rated the effects of their child's celiac disease differently, with mothers reporting more lifestyle changes and heavier burden. Negative and positive themes emerged from the interviews. Mothers felt the burden of managing a gluten-free diet. Fathers felt guilty for carrying a celiac disease-associated gene and both fathers and siblings regretted limited food choices at restaurants and home. The need to be a more creative cook was seen as a positive effect by mothers. Fathers appreciated new family traditions. Siblings felt they had developed empathy for others. A framework is proposed to illustrate these family-wide interactions. CONCLUSIONS: A child's celiac disease diagnosis and disease management affects the entire family. Our results will inform family-centered interventions that maximize quality of life for families.

Costs and Use of Health Care in Patients With Celiac Disease: A Population-Based Longitudinal Study.

INTRODUCTION: Celiac disease (CD) affects 1% of the population. Its effect on healthcare cost, however, is barely understood. We estimated healthcare use and cost in CD, including their temporal relationship to diagnosis. METHODS: Through biopsy reports from Sweden's 28 pathology departments, we identified 40,951 prevalent patients with CD (villous atrophy) as of January 1, 2015, and 15,086 incident patients with CD diagnosed in 2008-2015, including 2,663 who underwent a follow-up biopsy to document mucosal healing. Each patient was compared with age- and sex-matched general population comparators (n = 187,542). Using nationwide health registers, we retrieved data on all inpatient and nonprimary outpatient care, prescribed diets, and drugs. RESULTS: Compared with comparators, healthcare costs in 2015 were, on average, $1,075 (95% confidence interval, $864-1,278) higher in prevalent patients with CD aged <18 years, $715 ($632-803) in ages 18-64 years, and $1,010 ($799-1,230) in ages ≥65 years. Half of all costs were attributed to 5% of the prevalent patients. Annual healthcare costs were $391 higher 5 years before diagnosis and increased until 1 year after diagnosis; costs then declined but remained 75% higher than those of comparators 5 years postdiagnosis (annual difference = $1,044). Although hospitalizations, nonprimary outpatient visits, and medication use were all more common with CD, excess costs were largely unrelated to the prescription of gluten-free staples and follow-up visits for CD. Mucosal healing in CD did not reduce the healthcare costs. DISCUSSION: The use and costs of health care are increased in CD, not only before, but for years after diagnosis. Mucosal healing does not seem to lower the healthcare costs.

Persistent Economic Burden of the Gluten Free Diet.

Gluten free (GF) products have been reported to be more expensive and less available than their gluten containing counterparts. We examined the current U.S. cost and availability of GF products and made comparisons to the marketplace over a decade ago. Cost, determined by price per ounce and availability of a "market basket" of regular and GF products across four venues and five geographic regions was compared using a student's t test. GF products were more expensive (overall 183%), and in all regions and venues (p < 0.001). GF products from mass-market producers were 139% more expensive than the wheat-based version of the same product. Availability of GF products was greatest (66%) in the health food and upscale venues. In contrast to the results of the 2006 study, the cost of GF products has declined from 240% to 183% (adjusted for inflation). The introduction of mass-market production of GF products may have influenced the increase in availability and overall reduction of cost since 2006. The extent to which the cost of GF products impacts dietary adherence and quality of life for those on a GFD warrants exploration.

Hypervigilance to a Gluten-Free Diet and Decreased Quality of Life in Teenagers and Adults with Celiac Disease.

BACKGROUND AND AIMS: Avoidance of gluten is critical for individuals with celiac disease (CD), but there is also concern that "extreme vigilance" to a strict gluten-free diet may increase symptoms such as anxiety and fatigue, and therefore, lower quality of life (QOL). We examined the associations of QOL with energy levels and adherence to, and knowledge about, a gluten-free diet. METHODS: This is a cross-sectional prospective study of 80 teenagers and adults, all with biopsy-confirmed CD, living in a major metropolitan area. QOL was assessed with CD-specific measures. Dietary vigilance was based on 24-h recalls and an interview. Knowledge was based on a food label quiz. Open-ended questions described facilitators and barriers to maintaining a gluten-free diet. RESULTS: The extremely vigilant adults in our sample had significantly lower QOL scores than their less vigilant counterparts [(mean (SD): 64.2 (16.0) vs 77.2 (12.2), p = 0.004]. Extreme vigilance was also associated with greater knowledge [5.7 (0.7) vs 5.1 (0.8), p = 0.035]. Adults with lower energy levels had significantly lower overall QOL scores than adults with higher energy levels [68.0 (13.6) vs 78.9 (13.0), p = 0.006]. Patterns were similar for teenagers. Cooking at home and using internet sites and apps were prevalent strategies used by the hypervigilant to maintain a strict gluten-free diet. Eating out was particularly problematic. CONCLUSION: There are potential negative consequences of hypervigilance to a strict gluten-free diet. Clinicians must consider the importance of concurrently promoting both dietary adherence and social and emotional well-being for individuals with CD.

Anxiety and depression in caregivers of individuals with celiac disease - A population-based study.

BACKGROUND & AIMS: Partner burden is common in celiac disease (CD), but it is unclear if parents of children with CD have increased burden, and if this may translate into depression and anxiety meriting healthcare. METHODS: Nationwide population-based study of 41,753 parents and spouses ("caregivers") to 29,096 celiac patients and 215,752 caregivers to 144,522 matched controls. Caregivers were identified from the Swedish Total Population Register, and linked to data on psychiatric disease in the National Patient Registry. Hazard ratios (HRs) for depression, anxiety, and (as a reference outcome measure) bipolar disorder were examined in a lifetime fashion but also in temporal relationship to date of CD diagnosis using Cox regression. A priori, we focused on parents of individuals diagnosed ≤19 years of age (children at the age of disease onset) and spouses of individuals diagnosed in adulthood, as such parents and spouses ("high-risk caregivers") were most likely to live together with the patient at time of disease onset. RESULTS: On Cox analysis, depression was 11% more common in high-risk caregivers (HR=1.11: 95%CI=1.03-1.19) than in control caregivers while anxiety was 7% more common (HR=1.07: 95%CI=0.98-1.16). Combining anxiety and depression into a composite outcome measure, there was an 8% statistically significant risk increase (95%CI=1.02-1.14). The highest excess risks for both depression and anxiety were seen just before and 4-8 years after the CD diagnosis. In contrast, bipolar disorder was not more common in caregivers to CD patients. CONCLUSION: Caregivers to patients with CD may be at increased risk of severe burden.

Partner Burden: A Common Entity in Celiac Disease.

BACKGROUND: Caregiver burden is documented in several chronic diseases, but it has not been investigated in celiac disease (CD). AIMS: We aim to quantify the burden to partners of CD patients and identify factors that affect the perceived burden. METHODS: We surveyed patients with biopsy-proven CD and their partners. Patients completed CD-specific questions, including the validated Celiac Symptom Index (CSI) survey. Partners completed the validated Zarit Burden Interview (ZBI) and questions regarding sexual and relationship satisfaction. Univariable and multivariable analyses were used to assess the association between demographics, CD characteristics, and partner burden. RESULTS: In total, 94 patient/partner pairs were studied. Fifteen patients (16 %) reported a CSI score associated with a poor quality of life, and 34 partners (37 %) reported a ZBI score corresponding to mild-to-moderate burden. Twenty-two partners (23 %) reported moderate-to-low overall relationship satisfaction, and 12 (14 %) reported moderate-to-low sexual satisfaction. The degree of partner burden was directly correlated with patient CSI score (r = 0.27; p = 0.008), and there were moderate-to-strong inverse relationships between partners' burden and relationship quality (r = -0.70; p < 0.001) and sexual satisfaction (r = -0.42; p < 0.001). On multivariable logistic regression, predictors of mild-to-moderate partner burden were low partner relationship satisfaction (OR 17.06, 95 % CI 2.88-101.09, p = 0.002) and relationship duration ≥10 years (OR 14.42, 95 % CI 1.69-123.84, p = 0.02). CONCLUSIONS: Partner burden is common in CD, with more than one-third of partners experiencing mild-to-moderate burden. Partner burden is directly correlated with patient symptom severity, and it increases with poorer sexual and relationship satisfaction. Healthcare providers should address relationship factors in their care of patients with CD.

Cost Effectiveness of Routine Duodenal Biopsy Analysis for Celiac Disease During Endoscopy for Gastroesophageal Reflux.

BACKGROUND & AIMS: Some patients with refractory gastroesophageal reflux disease (GERD) actually have undiagnosed celiac disease. These patients often undergo an esophagogastroduodenoscopy (EGD) to determine the etiology and severity of GERD. Performing routine duodenal biopsy analysis during an EGD could identify patients with celiac disease. We evaluated the cost effectiveness of this approach. METHODS: We performed a systematic search of the MEDLINE database to identify publications through March 2014 on patients who underwent a duodenal biopsy analysis during an EGD for GERD. Data collected were used to construct a decision tree to calculate the cost effectiveness of an EGD with and without celiac disease tests. RESULTS: Among 10,000 patients with refractory GERD who underwent an EGD, we predicted a biopsy strategy would detect 70% of patients with celiac disease if the prevalence of celiac disease was 1% in this cohort. Biopsy analysis at the start of the EGD procedure would increase the remaining quality-adjusted life years (QALYs) by 0.0032, and increase the lifetime cost by $389/patient. Compared with no biopsy, the biopsy strategy cost $55,692.86/case of celiac disease detected, and $121,875/QALY gained. The incremental cost-effectiveness ratio for the biopsy strategy met the threshold of less than $50,000/QALY when 1 of the following parameters was met: when the utility of living with GERD was less than 0.88, when the prevalence of celiac disease in patients with refractory GERD was greater than 1.8%, when biopsy analysis detected celiac disease with more than 98.1% specificity, when the cost of a gluten-free diet was less than $645.85/y, or if the cost of proton pump inhibitor therapy was more than $5874.01/y. CONCLUSIONS: Based on base-case values, it is not cost effective to perform a biopsy analysis to detect celiac disease in patients undergoing an EGD for refractory GERD. However, the approach becomes cost effective when the prevalence of celiac disease in this population is 1.8% or greater.

Quantitative assessment of endoscopic images for degree of villous atrophy in celiac disease.

BACKGROUND: The degree of villous atrophy in celiac disease is difficult to assess at endoscopy. We sought to develop a quantitative technique for the evaluation of villous atrophy in endoscopic images. METHOD: In ten celiac patients as identified by standard endoscopy with biopsy, and ten control patients, standard and videocapsule endoscopic images of the duodenum were digitized. Subimages 7.5 × 7.5 mm(2) in area from random locations within each image were assessed by measuring the length of mucosal fissures per unit area (L), and correlating L with the histologic grade of villous atrophy as determined by modified Marsh criteria. RESULTS: Mean L values for standard endoscopic images were 37.8, 43.3, 64.1, and 83.5 mm for Marsh grades II, IIIa, IIIb, and IIIc, respectively. Mean L values for videocapsule images were 49.1, 50.0, 64.7, and 72.4 mm for Marsh grades II, IIIa, IIIb, and IIIc, respectively. Significant differences in the means existed between celiac images (Marsh scores II-IIIc) versus controls (p < 0.001) for both endoscopic and videocapsule images. There were no significant differences between measurements obtained from endoscopic versus videocapsule images. CONCLUSIONS: Quantified image analysis correlates with the histologic grade of villous atrophy, is automated, and lacks observer bias, thus lending itself to standardization.

Genetic testing before serologic screening in relatives of patients with celiac disease as a cost containment method.

GOALS AND BACKGROUND: Relatives of patients with celiac disease have an increased lifetime risk of developing celiac disease. Repeat screening of relatives would improve diagnosis rates, but at significant cost. Genetic testing before screening would potentially reduce costs by eliminating HLA-DQ2 and DQ8 negative patients who are at extremely low risk for developing celiac disease. STUDY: A decision tree was developed incorporating 3 diagnostic branches: initial screening with anti-tissue transglutaminase at time t0, repeat screening at time t1, and genetic testing before repeat screening. Costs were estimated using Medicare reimbursement fees. Modeling and sensitivity analyses were performed using Tree Age Pro 2006. RESULTS: The cost of an initial screening with anti-tissue transglutaminase is approximately $434 per person. Repeat screening would cost $683, but would diagnosis an additional 4.4% cases. Genetic testing before screening would cost $750, but would decrease the lower endoscopy workload by nearly 25%. Genetic testing would have to decrease from $301 to $234, a difference of $67, to justify its use before serologic testing. As the specificity of anti-tissue transglutaminase approaches 100%, the cost of genetic testing would have to continue to decrease to less than $200 in order for it to be an affordable option. CONCLUSIONS: Repeat screening of relatives with celiac disease results in a significant increase in cost, but also an associated increase in cases diagnosed. Genetic testing would potentially eliminate up to 60% of the population to be screened and, if available at a lower cost, would partially offset costs of repeat serologic screening.

Economic benefits of increased diagnosis of celiac disease in a national managed care population in the United States.

OBJECTIVES: To estimate the rate of celiac disease diagnosis and evaluate the economic benefits of diagnosis by analyzing retrospective cohorts from a national managed-care-population database. METHODS: We identified patients who received a new diagnosis of celiac disease. We also identified 3 control groups, persons without a diagnosis of celiac disease but who exhibited 1, 2, or 3 or more symptoms associated with the disease. Using claims, encounter, and eligibility data of approximately 10.2 million managed care members across the United States between January 1999 and December 2003, we measured and compared direct standardized relative value based (RVU) medical costs and utilization of selected health care services among the 4 study cohorts. RESULTS: The rate of new diagnosis for celiac disease more than doubled over the 4-year period. The celiac disease cohort had a significant trend reduction in direct standardized medical costs relative to the three control groups. RVU-based medical costs in the celiac cohort were 24%, 33%, and 27% lower than cohort 1 (p<0.05), 29.0%, 38%, and 24% lower than cohort 2 (p<0.05), and 38%, 33%, and 31% lower than cohort 3 (p<0.01) for the 12-month, 24-month and 36-month post-diagnosis periods, respectively. The reductions in costs were attributable to decreasing trends in utilization of office visits, lab, diagnostic, imaging, and endoscopy procedures relative to the 3 comparative cohorts over the 3-year follow-up period. CONCLUSIONS: There was an increase in the rate of celiac disease diagnosis, which was associated with significant reduction in direct standardized RVU-based medical costs and utilization of selected health care services over time.

The many faces of celiac disease: clinical presentation of celiac disease in the adult population.

The major modes of presentation of patients with celiac disease are the classic diarrhea-predominant form and silent celiac disease. Those with silent celiac disease lack diarrhea, although they may present with manifestations of celiac disease that include an irritable bowel syndrome, anemia, osteoporosis, neurologic diseases, or malignancy. A significant proportion of patients are diagnosed through screening at-risk groups including relatives of patients and insulin-dependant diabetics. Nondiarrheal presentations now are seen more commonly than those with diarrhea. Patients with celiac disease have a greater burden of disease than the general population because of autoimmune diseases and malignancies. There is a need for screening studies of patients with conditions associated with celiac disease to determine whether the large numbers of people with undiagnosed celiac disease currently are seeking health care.