|Year : 2020 | Volume
| Issue : 1 | Page : 26-31
|Health related quality of life among Saudi children and adolescents with celiac disease
Norah D Al Nofaie1, Jawaher R Al Ahmadi2, Omar I Saadah3
1 Joint Program of Family and Community Medicine, Minstry of Health, Jeddah, Saudi Arabia
2 Department of Family Medicine, Faculty of Medicine, King Abdulaziz University, Jeddah, Saudi Arabia
3 Department of Paediatrics, Paediatric Gastroenterology Unit, Faculty of Medicine, King Abdulaziz University, Jeddah, Saudi Arabia
Click here for correspondence address and email
|Date of Web Publication||18-Feb-2020|
| Abstract|| |
Background/Aims: Celiac disease (CD) is an immune-mediated enteropathy triggered by gluten ingestion in genetically susceptible individuals. This study reports on the quality of life (QOL) of Saudi children and adolescents with CD.
Patients and Methods: This is a case control study that included Saudi patients with CD, aged 9-18 years, who attended CD Clinic at KAU between February 2017 and July 2018. The study was conducted using the Short-Form (SF-36) questionnaire for all candidates, CD-specific QOL questionnaire for the CD patients group, and CD screen questionnaire for the control group.
Results: Overall, 354 subjects were studied (111 CD patients and 243 control). Female subjects constituted 56.8% of both patient and control groups. In the generic SF-36 questionnaire, QOL was comparable between patients and controls in all domains except for the general health domain, which showed difference in favor of the controls (55.01 ± 26.41 and 62.96 ± 18.16, P = 0.005). We also found that males have lower QOL scores in the domains comprising health change (P = 0.02), physical functioning (P = 0.04, role functioning/emotional and emotional well-being (P = 0.049). The CD-specific QOL showed excellent and good scores for 79.3% of patients overall in the tested domains. Patients with poor adherence showed poor QOL in all generic (SF-36) domains but not in CD-specific domains.
Conclusion: The HRQOL for Saudi CD children on GFD is generally comparable to the healthy control with exception of the general health domain. Adherence to the GFD improves the generic (SF-36) QOL domains.
Keywords: Adherence, celiac, children, quality of life, Saudi Arabia
|How to cite this article:|
Al Nofaie ND, Al Ahmadi JR, Saadah OI. Health related quality of life among Saudi children and adolescents with celiac disease. Saudi J Gastroenterol 2020;26:26-31
|How to cite this URL:|
Al Nofaie ND, Al Ahmadi JR, Saadah OI. Health related quality of life among Saudi children and adolescents with celiac disease. Saudi J Gastroenterol [serial online] 2020 [cited 2021 May 15];26:26-31. Available from: https://www.saudijgastro.com/text.asp?2020/26/1/26/274540
| Introduction|| |
Celiac disease (CD) is an immune mediated enteropathy, triggered by gluten ingestion in genetically susceptible individuals. Both human leukocyte antigen (HLA) and non-HLA genetics have been implicated in its susceptibility.,,, The global prevalence of CD in children has been estimated to be approximately 0.9%. Seroprevalence rates of 2.8% and 2.2%, respectively, have been reported for school-aged children and adolescents in the Kingdom of Saudi Arabia., CD is a chronic disease that requires lifelong restriction of gluten intake, and consumption of a gluten-free diet (GFD), which may have an impact on the quality of life in children and adolescents with CD. Untreated or poorly controlled CD patients may have different gastrointestinal and non-gastrointestinal symptoms that may impair the quality of life (QOL)., Strict adherence to the GFD is the recommended therapeutic approach that can lead to mucosal healing and resolve symptoms resulting in overall improvement of the QOL. However, some patients, especially adolescents, may have difficulty coping with the dietary restriction of a gluten-free diet, which may adversely affect their QOL., Such emotional and social effects of the diet may counteract the positive effect of mucosal healing.
Several tools have been used to assess QOL. The most widely used instrument is the RAND Short-Form survey (SF-36) which has been used in several clinical studies.,, The SF-36 was developed from a larger survey in the Medical Outcomes Study (MOS)., 2471 participants older than 14 years have been included from the MOS to develop the scoring system and to determine the reference standard values. The result of the SF-36 has been used as a control reference (RAND control) in several studies.,
To the best of our knowledge, only one study has been published from Saudi Arabia that looks at health-related quality of life (HRQOL) using a SF-36 questionnaire in patients with CD. This study reports the HRQOL of Saudi children and adolescents with CD in comparison with age- and sex-matched healthy controls, utilizing both the SF-36 questionnaire and the celiac specific quality of life questionnaire (CD-QOL). The control subjects were chosen from the local population rather than RAND control.
| Patients and Methods|| |
This is a case control study that included Saudi patients with biopsy proven CD, aged 9-18 years, who attended CD Clinic at King Abdulaziz University in the period between February 2017 and July 2018. Patients with other chronic illnesses were excluded to ensure the accuracy and validity of the results.
The control groups of children and adolescents were chosen from the general school by a random selection technique. Consent and assent were taken from these participants. We used our controls from the same population rather than use the RAND controls. Before participation, controls were assessed by using a locally validated CD screening questionnaire.
Adherence to the GFD was assessed by combined utilization of self-reported rating of adherence from KINDL ® questionnaire (yes/no) and demonstration of a decline in the level of tissue transglutaminase (tTG) antibody to normal or near-normal level.
Instrument and measures
The study was conducted using the interviewing Short-Form survey (SF-36) questionnaire for all candidates, the CD-QOL questionnaire for CD patients, and the celiac screen questionnaire for controls.
The SF-36 is a 36-question survey used for both patients and controls. It comprises of 2 primary domains, physical and mental, which measure the presence and the severity of symptoms and their limitation on daily activities. It is sub-divided into various secondary domains, including physical functioning, role functioning due to physical health problems, role functioning due to emotional health problems, energy and fatigue, emotional well-being, social functioning, bodily pain, general health, and health changes. Scores range between 0 and 100, with a higher score indicating a better HRQOL. SF-36 was tested and showed reliability and validity as a measurement of quality of life. An Arabic translated form of SF-36 showed validity, reliability, and equivalence to the original version.
The CD quality of life survey (CD-QOL) is a reliable and valid CD specific instrument. It comprises of 20 questions across 4 clinically relevant domains (limitations, dysphoria, health concerns, and inadequate treatment). The instrument assesses the presence of CD-related symptoms in the last 30 days. The questions were expressed in a 5-point Likert scale and the response labeled as poor, good, and excellent after reverse coding. This instrument has high reliability and validity.
Celiac screen questionnaire is a locally validated instrument that includes 15 questions about general health, usually used for the controls to identify individuals with symptoms suggestive of CD. Individuals scoring 25 or above must be excluded from the study.
The study was approved by the Research Committee at the Unit for Biomedical Ethics at Faculty of Medicine, King Abdulaziz University (Reference NO 531-18). Informed consent was obtained from all the participants' parents or legal guardians.
The statistical analysis was performed using the Statistical Package for the Social Science (SPSS) software, Version 21 for Windows, Descriptive statistics were reported as proportions for qualitative variables such as frequencies and percentages of QOL scales among controls and cases. Group comparison (CD patients vs. controls and CD patients with good adherence vs. poor adherence) was performed using an independent t-test for numerical data. The results were considered significant when the P value was less than 0.05.
| Results|| |
A total of 354 subjects were included in this study (111 CD patients and 243 controls). Out of the initial number of controls of 284 individuals, 41 were excluded through the CD screening questionnaire and referred to the hospital for further investigations, and 243 individuals were included in the study. Female subjects constituted 56.8% of both cases and controls.
The CD cases and controls were matching in the baseline general characteristics (age, gender, and level of education) with no statistically significant differences between both groups [Table 1].
Comparing our study controls with the RAND control regarding QOL, our study controls had good quality of life and a higher score in more sub-classes than RAND control. The difference was more obvious in the following domains: Energy/fatigue, role functioning and physical, general health, health change, and pain. This makes our study controls more ideal to compare with than RAND control.
Only the general health domain showed significant differences between CD patients and controls in the mean score ± SD (55.01 ± 26.41 vs. 62.96 ± 18.16, P= 0.005), indicating poor QOL of CD patients in this domain. In the domains emotional well-being and energy/fatigue, though the mean score was lower than that of the control group, the difference was not statistically significant [Table 2].
|Table 2: Comparison between the QOL domains in CD cases and control group|
Click here to view
Concerning the effect of gender on QOL for CD patients, we found that males have lower QOL scores in the following domains: health change (P = 0.02), physical functioning (P = 0.04), role functioning/emotional (P = 0.04) and emotional well-being (P = 0.049) [Table 3].
When general QOL domains were analyzed based on the level of adherence to the GFD in CD patients, children with good adherence showed significantly better quality of life compared to children with poor adherence in all domains (P < 0.001) [Table 4].
|Table 4: QOL of CD patients categorized according to the degree of adherence to the GFD|
Click here to view
Only 20.7% of the CD patients reported a poor response in all CD-specific domains, while 79.3% reported a good or excellent response in overall sum of the four tested domains [Table 5]. Stratifying responses by the level of adherence to the GFD showed no difference. Furthermore, there was no gender difference in CD patients regarding the level of adherence (good adherence was documented in 38 [69%] males vs. 42 [75%] females, P = 0.49).
|Table 5: Comparison between CD-QOL domains in children with CD according to the degree of adherence to the GFD|
Click here to view
| Discussion|| |
In this study, the effect of CD on the QOL of children and adolescents in Saudi Arabia was addressed against healthy controls. The HRQOL is a multidimensional concept that covers various domains including physical, emotional, social, and cognitive aspects. What matters about HRQOL is the patient's perception about their functioning. We used both, a generic instrument for assessment of HRQOL (SF-36) and a CD-specific instrument. The generic instrument allows for comparison of patients with healthy controls, whereas a CD-specific instrument measures various problems relevant to the disease. The control group was carefully selected among healthy children and adolescents using a locally validated questionnaire to exclude individuals with potential or undiagnosed CD.
Few studies have investigated HRQOL in children with CD and they report conflicting results.,,,, The discrepancy in these reported results may be attributed to the different instruments used for assessment of HRQOL, along with the different age groups and ethnicity of the study population.
When comparing the HRQOL between children and adolescent with CD and the controls, we found comparable results in all domains with exception of general health, and a trend for lower scores in the role functioning/emotional and emotional well-being domains. However, the study cohort included all patients regardless of their adherence level to the GFD. Further categorization of patients according to the adherence level showed significant difference in QOL domains between both groups, indicating better QOL in CD patients with good adherence as compared to patients with poor adherence. These findings are consistent with previous reports indicating better HRQOL (SF-36) in patients with better adherence to the GFD.,,, Other studies reported disagreement on the effect of adherence on the QOL.,,
Unlike other studies that reported lower perception of QOL in female CD patients for both, children , and adults,, our result showed the opposite, which may be related to cultural differences between the Saudi and the Western community, since young females in the Saudi community are less likely to spend as much of their time outdoors with their peers in the Western community. Therefore, they may have less impact on the quality of life in relation to dining outside or keeping self-esteem, compared to their male counterparts.
Some studies reported no difference according to the adherence level. The reasons for such differences may be related to different methods used to assess the dietary adherence, which included questionnaires, self-reporting, CD serology, and biopsy. These methods are considered relatively inaccurate to assess gluten ingestion.
Dietary adherence to GFD in older children and adolescents was related to the presence of gastrointestinal symptoms, age of the child, and their ethnic and social background, rather than overall perception of HRQOL.
The CD-specific QOL analysis results demonstrated that more than 80% of children and adolescents in this study showed excellent and good scores in all domains of QOL. Only less than 20% reported poor QOL for all domains. This suggests that most children and adolescents with CD have good QOL as measured by the CD-specific QOL questionnaire.
Comparing the CD-specific QOL domains according to the adherence level to the GFD, no differences were found [Table 5], unlike the result obtained when analyzing the domains of the generic HRQOL instrument [Table 5]. This may be attributed to the composition of the CD-specific questionnaire that relies mainly on perception of the CD as a chronic illness and various limitations related to the GFD and the disease complications. These concerns will be shared by both symptomatic and asymptomatic patients. Studies showed that screening-identified asymptomatic CD may be less willing to adhere to a GFD., However, in another study by Mahadev et al., using a CD-specific QOL questionnaire has shown no difference in QOL and adherence level between symptomatic and screen-detected asymptomatic patients.
The study is limited by its design and lack of QOL assessment before starting the GFD for comparison.
In conclusion, HRQOL for Saudi CD children on the GFD is generally comparable to the healthy controls with exception of the general health domain, when assessed by the generic SF-36 and CD-specific instruments. Children with better adherence to the GFD have better QOL in all SF-36 domains, but not in the CD-specific domains. Further studies are required to minimize the difference in QOL scores related to adherence level between assessment, using SF-36 or CD-specific questionnaires.
The authors would like to acknowledge Dr. Trevor Rawbone for reviewing the manuscript.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Al-Aama JY, Shaik NA, Banaganapalli B, Salama MA, Rashidi O, Sahly AN, et al
. Whole exome sequencing of a consanguineous family identifies the possible modifying effect of a globally rare AK5 allelic variant in celiac disease development among Saudi patients. PLoS One 2017;12:e0176664.
Al-Hussaini A, Alharthi H, Osman A, Eltayeb-Elsheikh N, Chentoufi A. Genetic susceptibility for celiac disease is highly prevalent in the Saudi population. Saudi J Gastroenterol 2018;24:268-73.
] [Full text]
Banaganapalli B, Rashidi O, Saadah OI, Wang J, Khan IA, Al-Aama JY, et al
. Comprehensive computational analysis of GWAS loci identifies CCR2 as a candidate gene for celiac disease pathogenesis. J Cell Biochem 2017;118:2193-207.
Saadah OI, Shaik NA, Banaganapalli B, Salama MA, Al-Harthi SE, Wang J, et al
. Replication of GWAS coding SNPs implicates MMEL1 as a potential susceptibility locus among saudi arabian celiac disease patients. Dis Markers 2015;2015:351673.
Singh P, Arora A, Strand TA, Leffler DA, Catassi C, Green PH, et al
. Global prevalence of celiac disease: Systematic review and meta-analysis. Clin Gastroenterol Hepatol 2018;16:823-36.e2.
Al-Hussaini A, Troncone R, Khormi M, AlTuraiki M, Alkhamis W, Alrajhi M, et al
. Mass screening for celiac disease among school-aged children: Toward exploring celiac iceberg in Saudi Arabia. J Pediatr Gastroenterol Nutr 2017;65:646-51.
Aljebreen AM, Almadi MA, Alhammad A, Al Faleh FZ. Seroprevalence of celiac disease among healthy adolescents in Saudi Arabia. World J Gastroenterol 2013;19:2374-8.
Green PH, Cellier C. Celiac disease. N Engl J Med 2007;357:1731-43.
Usai P, Minerba L, Marini B, Cossu R, Spada S, Carpiniello B, et al
. Case control study on health-related quality of life in adult coeliac disease. Dig Liver Dis 2002;34:547-52.
Tontini GE, Rondonotti E, Saladino V, Saibeni S, de Franchis R, Vecchi M. Impact of gluten withdrawal on health-related quality of life in celiac subjects: An observational case-control study. Digestion 2010;82:221-8.
Fabiani E, Taccari LM, Ratsch IM, Di Giuseppe S, Coppa GV, Catassi C. Compliance with gluten-free diet in adolescents with screening-detected celiac disease: A 5-year follow-up study. J Pediatr 2000;136:841-3.
Wolf RL, Lebwohl B, Lee AR, Zybert P, Reilly NR, Cadenhead J, et al
. Hypervigilance to a gluten-free diet and decreased quality of life in teenagers and adults with celiac disease. Dig Dis Sci 2018;63:1438-48.
Hallert C, Granno C, Grant C, Hulten S, Midhagen G, Strom M, et al
. Quality of life of adult coeliac patients treated for 10 years. Scand J Gastroenterol 1998;33:933-8.
McHorney CA, Ware JE Jr, Lu JF, Sherbourne CD. The MOS 36-item short-form health survey (SF-36): III. Tests of data quality, scaling assumptions, and reliability across diverse patient groups. Med Care 1994;32:40-66.
O'Leary C, Wieneke P, Healy M, Cronin C, O'Regan P, Shanahan F. Celiac disease and the transition from childhood to adulthood: A 28-year follow-up. Am J Gastroenterol 2004;99:2437-41.
Hays RD, Shapiro MF. An overview of generic health-related quality of life measures for HIV research. Qual Life Res 1992;1:91-7.
Ware JE, Jr., Sherbourne CD. The MOS 36-item short-form health survey (SF-36). I. Conceptual framework and item selection. Med Care 1992;30:473-83.
McHorney CA, Ware JE, Jr., Raczek AE. The MOS 36-item short-form health survey (SF-36): II. Psychometric and clinical tests of validity in measuring physical and mental health constructs. Med Care 1993;31:247-63.
Al-Qefari SN, Altwijri AW, Al-Adhadh AM, Al-Rashed OA, Al-Jarallah B. Health-related quality of life among patients with celiac disease in Saudi Arabia. Ann Med Health Sci Res 2018;8:74-7.
Bellini A, Zanchi C, Martelossi S, Di Leo G, Not T, Ventura A. Compliance with the gluten-free diet: The role of locus of control in celiac disease. J Pediatr 2011;158:463-6.e5.
Coons SJ, Alabdulmohsin SA, Draugalis JR, Hays RD. Reliability of an Arabic version of the RAND-36 Health Survey and its equivalence to the US-English version. Med Care 1998;36:428-32.
Dorn SD, Hernandez L, Minaya MT, Morris CB, Hu Y, Lewis S, et al
. Psychosocial factors are more important than disease activity in determining gastrointestinal symptoms and health status in adults at a celiac disease referral center. Dig Dis Sci 2010;55:3154-63.
Gill TM, Feinstein AR. A critical appraisal of the quality of quality-of-life measurements. JAMA 1994;272:619-26.
Yacavone RF, Locke GR 3rd
, Provenzale DT, Eisen GM. Quality of life measurement in gastroenterology: What is available? Am J Gastroenterol 2001;96:285-97.
Altobelli E, Paduano R, Gentile T, Caloisi C, Marziliano C, Necozione S, et al
. Health-related quality of life in children and adolescents with celiac disease: Survey of a population from central Italy. Health Qual Life Outcomes 2013;11:204.
Barrio J, Cilleruelo ML, Roman E, Fernandez C. Health-related quality of life in Spanish coeliac children using the generic KIDSCREEN-52 questionnaire. Eur J Pediatr 2018;177:1515-22.
Kolsteren MM, Koopman HM, Schalekamp G, Mearin ML. Health-related quality of life in children with celiac disease. J Pediatr 2001;138:593-5.
Myleus A, Petersen S, Carlsson A, Hammarroth S, Hogberg L, Ivarsson A. Health-related quality of life is not impaired in children with undetected as well as diagnosed celiac disease: A large population based cross-sectional study. BMC Public Health 2014;14:425.
van Doorn RK, Winkler LM, Zwinderman KH, Mearin ML, Koopman HM. CDDUX: A disease-specific health-related quality-of-life questionnaire for children with celiac disease. J Pediatr Gastroenterol Nutr 2008;47:147-52.
Barratt SM, Leeds JS, Sanders DS. Quality of life in coeliac disease is determined by perceived degree of difficulty adhering to a gluten-free diet, not the level of dietary adherence ultimately achieved. J Gastrointestin Liver Dis 2011;20:241-5.
Nachman F, del Campo MP, Gonzalez A, Corzo L, Vazquez H, Sfoggia C, et al
. Long-term deterioration of quality of life in adult patients with celiac disease is associated with treatment noncompliance. Dig Liver Dis 2010;42:685-91.
Nachman F, Maurino E, Vazquez H, Sfoggia C, Gonzalez A, Gonzalez V, et al
. Quality of life in celiac disease patients: Prospective analysis on the importance of clinical severity at diagnosis and the impact of treatment. Dig Liver Dis 2009;41:15-25.
Usai P, Manca R, Cuomo R, Lai MA, Boi MF. Effect of gluten-free diet and co-morbidity of irritable bowel syndrome-type symptoms on health-related quality of life in adult coeliac patients. Dig Liver Dis 2007;39:824-8.
Hopman EG, Koopman HM, Wit JM, Mearin ML. Dietary compliance and health-related quality of life in patients with coeliac disease. Eur J Gastroenterol Hepatol 2009;21:1056-61.
Viljamaa M, Collin P, Huhtala H, Sievanen H, Maki M, Kaukinen K. Is coeliac disease screening in risk groups justified? A fourteen-year follow-up with special focus on compliance and quality of life. Aliment Pharmacol Ther 2005;22:317-24.
Casellas F, Rodrigo L, Vivancos JL, Riestra S, Pantiga C, Baudet JS, et al
. Factors that impact health-related quality of life in adults with celiac disease: A multicenter study. World J Gastroenterol 2008;14:46-52.
Burger JPW, de Brouwer B, IntHout J, Wahab PJ, Tummers M, Drenth JPH. Systematic review with meta-analysis: Dietary adherence influences normalization of health-related quality of life in coeliac disease. Clin Nutr 2016;36:399-406.
Mager DR, Marcon M, Brill H, Liu A, Radmanovich K, Mileski H, et al
. Adherence to the gluten-free diet and health-related quality of life in an ethnically diverse pediatric population with celiac disease. J Pediatr Gastroenterol Nutr 2018;66:941-8.
Shamir R, Yehezkely-Schildkraut V, Hartman C, Eliakim R. Population screening for celiac disease: Follow up of patients identified by positive serology. J Gastroenterol Hepatol 2007;22:532-5.
Mahadev S, Gardner R, Lewis SK, Lebwohl B, Green PH. Quality of life in screen-detected celiac disease patients in the United States. J Clin Gastroenterol 2016;50:393-7.
Prof. Omar I Saadah
Paediatric Gastroenterology Unit/Department of Paediatrics/Faculty of Medicine, King Abdulaziz University, P.O. Box 80215 Jeddah - 21589
Source of Support: None, Conflict of Interest: None
[Table 1], [Table 2], [Table 3], [Table 4], [Table 5]
| Article Access Statistics|
| Viewed||1555 |
| Printed||24 |
| Emailed||0 |
| PDF Downloaded||169 |
| Comments ||[Add] |