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PubMedDate not supplied

Prevalence and clinical presentation of subclinical/silent celiac disease in adults: an analysis on a 12-year observation.

BACKGROUND/AIMS: In recent years, an increased incidence of subclinical/silent celiac disease has been reported. Thus, we herein describe the prevalence and the clinical presentation of subclinical/silent celiac disease in 252 consecutive diagnosed celiac patients. METHODOLOGY: From 1988 to 1999 we diagnosed 252 celiac patients (74M and 178F, mean age: 27.9 yrs; range: 15-65 yrs, F/M ratio: 2.4). 144 patients were referred to us due to gastrointestinal symptoms, while 108 were referred to us from other specialists due to unexplained or unresponsive disease. All patients underwent both total immunoglobulin A and antigliadin antibodies antiendomysium antibody and evaluation, followed by gastrointestinal endoscopy with duodenal histological examination. RESULTS: 144 (57.14%) and 108 (42.86%) patients showed classical and subclinical/silent celiac disease, respectively. The most frequent extraintestinal marker of subclinical celiac disease were iron-deficiency anemia (27.77%), alopecia and dermatitis herpetiformis (11.36%), osteoporosis (6.81%) and recurrent aphtous stomatitis (5.68%), while first-degree relatives (30%), Basedow's disease (25%) and insulin-dependent diabetes (20%) were the most frequent in silent celiac disease. CONCLUSIONS: This study confirms the extremely polymorphic nature of this condition that can affect several organs and apparatus without gastrointestinal symptoms. However, a more precise description of subclinical/silent celiac disease can only emerge from screening studies on general populations.

Adolescent↗
PubMedDate not supplied

Celiac disease: helping families adapt.

Lifelong dietary compliance prevents the long-term effects associated with celiac disease. However, this simple solution, diet, has many long-reaching effects on the children affected with celiac disease and their families. Assisting families in coping with this chronic illness is necessary for children and families to develop a sense of control. An overview of celiac disease is presented through the use of a case study. Guidelines are given to enable the healthcare provider to understand and help families through the process of adapting to a child with a chronic illness. Recommendations for nutritional management with helpful suggestions related to making celiac disease an everyday part of family life are discussed.

Adaptation, Psychological↗
PubMed1998

Gluten-free diet induces regression of T-cell activation in the rectal mucosa of patients with celiac disease.

OBJECTIVE: An increase in the number of intraepithelial lymphocytes (IEL) in the rectal epithelium of patients with active celiac disease has been described. No data are available about how they vary during a gluten-free diet. The aim of the study was to assess the effect of a gluten-free diet on T-cell activation in the rectal mucosa of adult patients with celiac disease. METHODS: Frozen duodenal and rectal biopsies were available in four celiac patients (one male, three female, mean age 39 yr) both before and after 7 to 24 months on a gluten-free diet. Biopsy samples were stained using monoclonal antibodies directed against CD3, betaF1, TcRdelta1, CD25, and HLADR. Numbers of IEL were estimated by counting the peroxidase-stained cells per 100 epithelial cells. Four patients without histological abnormalities were used as control subjects. RESULTS: In the four patients with active celiac disease but in none of the controls, CD25 was expressed by both duodenal and rectal lamina propria cells and HLADR was expressed by duodenal (4/4) and rectal (2/4) epithelial cells. In addition, two patients with active celiac disease had features of lymphocytic colitis, i.e., >20 IEL per 100 epithelial cells. After a gluten-free diet, the mean number of rectal CD3+ betaF1+ IEL decreased (9% vs 21%) and the expression of CD25 and HLADR was no longer present. These changes mirrored those found in the small intestinal biopsies. CONCLUSION: These results suggest that in celiac disease, gluten-driven T-cell activation is not restricted to the proximal part of the intestine but is present on the whole intestinal length. Assessment of the effectiveness of a gluten-free diet through rectal biopsies warrants investigation, as it could lessen discomfort for patients and prove more cost-effective.

Adult↗
PubMed2005

The prevalence of unrecognized adult celiac disease in Central Anatolia.

BACKGROUND: The aim of this study is to assess the prevalence of unrecognized adult celiac disease in Central Anatolia of Turkey and establish if prevalence figures are similar to other reports in the international literature. METHODS: Subjects were randomly selected from patients at the time of blood sampling because of a routine examination or suspicion of some disorder other than celiac diseases and were screened with anti-tissue transglutaminase IgA and serum IgA measurements. Duodenal biopsies were taken from the patients who were found positive for anti-tissue transglutaminase IgA and had low IgA levels. RESULTS: A total of 906 subjects between 20 and 59 years of age were included. Small bowel biopsies were performed for 55 of the 906 participants. Fifty-two of 55 participants taken biopsies had anti-tissue transglutaminase IgA levels greater than 15 IU/mL and 3 of them had low IgA levels. Celiac disease was diagnosed as 9 of 906 (0.99%). The majority of the patients with celiac disease had nonspecific gastrointestinal symptoms. There was no correlation between the titers of anti-tissue transglutaminase IgA and the severity of histopathologic findings. CONCLUSIONS: In this study, we found that unrecognized adult celiac disease in Central Anatolia affects approximately 1% of the population, and the major constellation of symptoms are nonspecific gastrointestinal related. Serologic data are not adequate for a definite diagnosis, but the anti-tissue transglutaminase IgA test has high diagnostic value and may be used as screening tool. Confirmation with intestinal biopsy is required for a definite diagnosis.

Adult↗
PubMed2003

[Celiac disease; a world in exploration].

Celiac disease (CD) or gluten-sensitive enteropathy is an autoimmune disorder triggered by gluten ingestion in genetically predisposed subjects. The presence of gluten in these patients leads to a self-perpetuating mucosal damage, while the elimination of gluten results in a full mucosal recovery. The prevalence of CD in the general population is between 0.3% and 1%. The clinical manifestation of CD is variable; in addition to the classical gastrointestinal form a variety of other clinical manifestation of the disease have been described, including atypical and asymptomatic form. The diagnosis of CD is still based on the small intestinal biopsy findings, but can be suspected using serological testing, e.g. the antigliadin antibody (AGA), the antiendomysial antibody (EMA) and the anti-tissue transglutaminase antibody (tTG). The keystone treatment of CD patients is a life-long gluten-free diet.

Biopsy↗
PubMed1983

Toxic shock syndrome in a patient with celiac disease.

A young woman with celiac disease presented with fever, headache, diffuse myalgia, vomiting, diarrhea and hypotension during a menstrual period in which tampons were used. The clinical picture and laboratory data were typical of toxic shock syndrome. This recently defined entity is rare outside of the United States, and has not been previously reported in a menstruating woman in Israel. The clinical and laboratory characteristics of the syndrome are discussed.

Adult↗
PubMed1997

Serodiagnosis of celiac disease.

The detection rate of celiac disease greatly underestimates its prevalence. This is the result of lack of awareness of the many manifestations of the condition and the requirement for at least one small intestinal biopsy for diagnosis. The advent of effective serologic tests for the condition heralds a new era of non-invasive testing. The clinical utility and acceptance of these tests is limited by a lack of standardization of both the techniques used and variability of reference ranges quoted.

Adult↗
PubMed2004

[Prevalence of celiac disease in patients with chronic diarrhea].

The objective of this research was to study the frequency of celiac disease in patients with chronic diarrhea. The biopsy materials of the small intestine and levels of antibodies to alpha-gliadin of class A immunoglobulins (IgA) and tissue transglutaminase were studied in 206 patients with chronic diarrhea. Morphologic celiac-specific symptoms were discovered in 35 (16.9%) patients. Symptoms of the total atrophy were discovered in 28 patients (13.5%); those of subtotal one were found in 7 (3.4%) patients. The increase of antibody levels to IgA alpha-gliadin and tissue transglutaminase was discovered in all 35 patients. Their average level made up 123.7 21.2 units per milliliter and 48.7 11.3 units per milliliter, respectively. It was possible to observe the typical celiac form only in 4 (11.4%) patients; the latent form was found in 30 (85.7%) patients, and the torpid (refractory) form was discovered in 1 (2.8%) patient. The frequency of celiac disease in patients with chronic diarrhea is equal to 16.9%. Patients with the latent form of the disease prevail among patients with celiac disease. Immunological screenings with the subsequent morphologic study of the mucous coat of the small intestine should be prescribed to all patients with the chronic diarrhea syndrome to enable the early diagnostics of celiac disease.

Adolescent↗
PubMed1998

Sideropenic anemia and celiac disease: one study, two points of view.

Recent studies have pointed to the relationship between iron deficiency anemia and celiac disease, although data on the prevalence of celiac disease in anemic patients have been conflicting, and there is no agreement on the best screening procedure for CD in these patients. Our aims were to evaluate the relationship between anemia and celiac disease (CD) from two different points of view--the hematology clinic and the pediatric gastroenterology department--and to evaluate the utility of anti-endomysial antibody determination in screening anemic patients for CD using human umbilical cord as substrate. We studied 130 patients with CD (58 males, 72 females; median age 18 months) diagnosed at a department of Pediatric Gastroenterology, and 85 patients with iron deficiency anemia (38 males, 47 females; median age 48 years) observed at a hematology outpatient clinic. From the 85 adult patients with iron deficiency anemia, we selected a subgroup of 25 subjects with no improvement in Hb after two months of iron therapy (80 mg/day orally). Routine hematochemical tests were performed in all 215 patients. All pediatric and adult subjects underwent immunological screening for celiac disease (AGA and EmA assay); intestinal biopsy was also performed on patients testing positive. In the adult anemic patients a serum sample was stored at -20 degrees C on first observation, and after 6-18 months EmA on human umbilical cord were assayed. In the pediatric patients with CD, anemia was observed in 91/130 patients (70% of cases, the most frequent symptom after poor growth); however, this was the only presenting symptom of CD in 2/130 patients (1.5% of cases). Anemia was sideropenic in 41/91 patients (iron <45 microg/dl, ferritin <15 microg/liter). In the adult patients with iron deficiency anemia, immunological screening (AGA and EmA) showed suspected CD in 5/85 cases (5.8%), with diagnosis confirmed on intestinal biopsy. These five patients were in the subgroup of iron supplementation therapy nonresponders. CD prevalence in the refractory anemia subgroup was, therefore, 5/25 (20%). On diagnosis the hematological indices of the anemia + CD patients were not different than those of the refractory anemia patients without CD. The median age of the CD + anemia patients was significantly lower than that of the whole group of anemic subjects, and there was also a prevalence of females (4/5 cases). The results of the EmA determination on human umbilical cord in the adult anemic patients showed a perfect concordance with those using a traditional kit that uses monkey esophagus as substrate. In the pediatric age group many cases of CD with anemia as the only sign of the disease are probably not diagnosed. In our adult patients with sideropenic anemia, CD prevalence was 5-6%; however, the observation of anemic patients not responding to oral iron therapy makes a diagnosis of CD much more probable. EmA determination on human umbilical cord is the most logical approach to screen anemic patients for suspected CD.

Anemia, Iron-Deficiency↗
PubMed1993

Extremely low incidence rates of celiac disease in the Danish population of children.

Incidence rates by birth of celiac disease of 0.09 per 1000 live births were described for the period 1960-88 in the entire Danish population. Broad diagnostic criteria were used in order to prevent the risk of under-diagnosing the disease. The observations support previous local reports from Copenhagen County of low incidence rates by birth of celiac disease. The study presents the lowest incidence rate by birth of celiac disease yet described.

Celiac Disease↗
PubMed2003

Plasma cytokine profiles in patients with celiac disease and selective IgA deficiency.

Celiac disease (CD) and selective IgA deficiency (IgAD) are frequently associated, and share the same genetic background. The aim of the present study was to evaluate both Type 1 and 2 plasma cytokine levels in CD and in CD-IgAD. IL-2, TNF-alpha, IL-10, IL-4 and IL-13 plasma levels were measured both at diagnosis and after a gluten-free diet (GFD) in 32 CD patients, in 27 CD-IgAD patients and in 30 healthy controls. IFN-gamma levels were significantly higher in CD and CD-IgAD than in controls, TNF-alpha displayed significantly higher levels in CD-IgAD when compared both with controls and with CD, and IL-2 was in CD-IgAD significantly increased respect to controls. Kinetics of the Type 1 cytokine plasma levels did not show a clear relationship with the GFD in both groups of CD patients, and particularly in those with IgAD. IL-4 and IL-13, both at diagnosis and after a GFD, were not significantly different in controls and in celiac patients (with and without IgAD). IL-10, whose production is stimulated by the TNF-alpha, had significantly higher plasma levels in CD-IgAD, but not in CD patients, with a significant decrease after a GFD. CD and especially CD-IgAD patients display persistently higher pro-inflammatory cytokine levels, suggesting a persistent state of activation of pro-phlogistic signals in CD, particularly when IgAD coexists. Serial measurement of serum IL-10 may be an adjunctive evaluating criterion in the follow-up of CD-IgAD patients.

Adolescent↗
PubMed2004

Celiac disease with various presentations.

BACKGROUND: Celiac disease (CD) has a wide clinical spectrum from malabsorption syndrome to extra intestinal presentations. A total of 45 children with CD presented with mainly chronic diarrhea (n :23), anemia (n: 12), and short stature (n: 10) were evaluated in this study. The aim was to find common parameters of CD with various presentations. METHODS: Basic anthropometric, biochemical and hematological parameters in cases with CD with various presentations were compared. RESULTS: It was found that children with CD presenting with chronic diarrhea were younger. There was no significant difference in hemoglobin levels in children with CD presenting with anemia. Children with CD with short stature had significantly lower serum vitamin B(12) levels and lower levels of height standard deviation scores, bone age delay, and alkaline phosphatase. CONCLUSIONS: It was concluded that children, especially infants with chronic diarrhea with CD, may not be affected with generalized malabsorption. Anemia and short stature are frequent findings in cases with CD whether they are main presenting symptoms or not. Children with CD presenting with short stature may have lower levels of vitamin B(12) than other presentations.

Age Distribution↗
PubMed2002

Genome-wide linkage analysis for celiac disease in North American families.

Celiac disease (CD) is an autoimmune disease caused by sensitivity to the dietary protein gluten. It has a prevalence of 1 in 250 in the United States. Multiple-case families are common with a risk to siblings from 10-12%. Previous linkage studies have found no significant evidence for linkage other than to HLA. In this study, we performed a genome-wide search on 62 families with at least two cases of CD to identify non-HLA loci for CD. Two-point and multipoint parametric and nonparametric analyses were performed on the entire set of families and on sets stratified by the HLA genotype. Accounting for multiple testing, we found genome-wide intermediate linkage evidence at 18q (heterogeneity LOD (HLOD) = 3.6) and at 3p (HLOD = 3.2) and suggestive evidence at 5p (HLOD = 2.7). Good consensus between two-point and multipoint evidence was not found, and after genotyping with new markers in these regions, our results were inconclusive. The 18q region had intermediate two-point evidence, but weak multipoint evidence. At 3p and 5p, the addition of follow-up markers added flanking support, yet multipoint evidence was still lacking. Our results indicate that multipoint analyses may be hindered by the complexity of CD. Multipoint analyses are not robust to model misspecification, and further development of models is needed. Additional study of these and other families is necessary to validate or rule out the regions implicated in this study.

Antibody Specificity↗
PubMed2000

[Influence of gluten free diet on bone mineral density (BMD) in children with celiac disease].

UNLABELLED: In children with celiac disease (CD) bone metabolism and mineralization are frequently disturbed. The present study aimed to assess the influence of gluten free diet (GFD) on bone mineral density (BMD) in 73 children with CD, mean age of 12.4 +/- 0.4 years and mean body mass index (BMI) 17.9 +/- 0.4 kg/m2 (mean +/- SEM). Diagnosis of CD was established according to ESPEGAN criteria. Compliance to the GFD was verified on the basis of interview and by estimation of antiendomysial antibodies (IgAEmA/IgGEmA) in blood serum. BMD was measured by dual energy X-ray absorptiometry (DEXA). Plasma calcium (Ca) and phosphorus (P) concentrations, alkaline phosphatase (AP) and its bone fraction (BAP) were estimated before BMD measurement. All children were divided into two groups. Group A consisted of 33 children where gluten free diet was strictly respected for 11.7 +/- 0.6 years. The second group (Group B) comprised 40 children without strictly respected GFD. Children who strictly followed GFD showed statistically higher BMI, AP-spine BMD and total body BMD in comparison with children without GFD (BMI 19 +/- 0.52 kg/m2 vs 17.3 +/- 0.4 kg/m2; p < 0.01, AP-spine BMD 0.951 +/- 0.04 g/cm2 vs 0.767 + 0.03 g/cm2; p < 0.005, Total Body BMD 1.013 +/- 0.02 g/cm2 vs 0.933 +/- 0.01 g/cm2; p < 0.05) respectively. No significant differences were found in plasma Ca, P, AP, BAP between both groups. A statistically significant positive correlation (p < 0.001) was found between duration of GFD and AP-spine BMD and total body BMD, respectively. A statistically significant positive correlation (p < 0.05) was also found between duration of GFD and BMI. CONCLUSION: Long-term GFD significantly improves BMD and BMI in children with CD.

Absorptiometry, Photon↗
PubMed2005

Effect of elemental diet on mucosal immunopathology and clinical symptoms in type 1 refractory celiac disease.

BACKGROUND & AIMS: Patients with celiac disease (CD) who do not improve or exhibit villous atrophy on a gluten-free diet may have type 1 refractory CD (RCD) with a polyclonal mucosal T-cell infiltrate, or type 2 RCD with a monoclonal infiltrate, also termed cryptic T-cell lymphoma. Both conditions are difficult to treat. Here we describe the effects of a nonimmunogenic elemental diet on clinical symptoms and mucosal immunopathology in type 1 RCD. METHODS: Ten CD patients on a strict gluten-free diet were diagnosed with type 1 RCD after extensive clinical evaluation in a tertiary referral hospital. A 4-week amino-acid-based liquid elemental diet regimen was given with no other treatment, except in 1 patient who also received methotrexate. Duodenal biopsy specimens were obtained before and after treatment for histologic assessment, immunophenotyping of intraepithelial lymphocytes, T-cell receptor clonality, mucosal interleukin (IL)-15 expression, flow-cytometric analysis of interferon (IFN)-gamma-secreting T cells, and whole biopsy specimen IFN-gamma messenger RNA determination. RESULTS: Nine patients completed the treatment; however, 1 patient did not tolerate the diet. Histologic improvement and reduced epithelial IL-15 were seen in 8 patients, whereas IFN-gamma-secreting mucosal T cells and IFN-gamma messenger RNA levels decreased in 4 and 7 patients, respectively. Clinical improvement was noted in 6 patients, with 1 patient showing normalization of hypoalbuminemia. Three patients could discontinue their total parenteral nutrition. CONCLUSIONS: Persistent mucosal IFN-gamma and IL-15 production often occurs in type 1 RCD despite conventional treatment. Elemental diet is a therapeutic option that can provide long-term immunopathologic and clinical improvement of this difficult condition.

Adult↗
PubMed2006

Hepatobiliary and pancreatic disorders in celiac disease.

A variety of hepatic and biliary tract disorders may complicate the clinical course of celiac disease. Some of these have been hypothesized to share common genetic factors or have a common immunopathogenesis, such as primary biliary cirrhosis, primary sclerosing cholangitis and autoimmune forms of hepatitis or cholangitis. Other hepatic changes in celiac disease may be associated with malnutrition resulting from impaired nutrient absorption, including hepatic steatosis. In addition, celiac disease may be associated with rare hepatic complications, such as hepatic T-cell lymphoma. Finally, pancreatic exocrine function may be impaired in celiac disease and represent a cause of treatment failure.

Celiac Disease↗
PubMed1975

The interrelationship of enterokinase and trypsin activities in intractable diarrhea of infancy, celiac disease, and intravenous alimentation.

Enterokinase initiates digestion of protein by conversion of trypsinogen into trypsin. The interactions between enterokinase and trysin were investigated in 6 patients with intractable diarrhea of infancy and 34 children with celiac disease. The six infants between 2 and 3 months with intractable diarrhea of infancy had reduced mucosal enterokinase activity (9.5 +/- 4.8muM per gram of protein per minute) and reduced intraluminal trypsin activity (2.9 +/- 0.7muM per gram of protein per minute) as compared with healthy controls (109 +/- 34.2muM per gram of protein per minute and 14.3 +/- 5.8muM per gram of protein per minute) respectively. The activities of all enzymes returned toward normal following treatment with intravenous alimentation. The mucosal morphology of all pretreatment biopsies in all cases showed Grade III atrophy which improved. These findings suggest that enterokinase deficiency and reduced intraluminal trypsin activity in intractable diarrhea of infancy may be one of the contributing factors to protein malabsorption and consequent malnutrition. Thirty-four children with celiac disease were between the age of 9 months and 13 years. The 11 newly diagnosed patients with celiac disease demonstrated Grade III to IV atrophy of the mucosa. The 23 patients with treated celiac disease on a gluten-free diet showed a normal to Grade II atrophy. In both treated and untreated celiac disease the enterokinase activities and the intraluminal trypsin activity were within normal limits. The enterokinase activity in celiac disease is near normal in contrast to the marked reduction noted in intractable diarrhea of infancy even though the intestinal mucosa shows marked morphological alteration and the disaccharidase activities are greatly reduced in celiac disease. After a prolonged alimentary fast of up to 26 days on intravenous alimentation, two patients with intractable diarrhea of infancy showed improvement in the activities of enterokinase and trypsin. These findings demonstrate that enterokinase and trypsin activities in the gut were present and improved in the absence of oral feeding.

Adolescent↗
PubMed2001

Commentary: advances in the laboratory diagnosis of celiac disease.

Anti-endomysial autoantibodies are very useful in the diagnosis and monitoring of celiac disease (gluten-sensitive enteropathy) due to their high sensitivity and specificity for that disorder. The recent discovery that the autoantigen responsible for the endomysial pattern is tissue transglutaminase (tTG) has led to the commercial development of automated enzyme immunoassays for quantitation of that autoantibody. These assays are standardized to provide highly accurate and comparable testing between laboratories for anti-tTG autoantibodies. Celiac disease is a common genetic disease in populations of Europe and the United States. It has a spectrum of expression ranging from silent or mild to severe, with resulting malabsorption that produces multiple-organ system effects due to malnutrition. Many cases miss the diagnosis because the symptoms are not classic or the clinical syndrome is not severe. Because the treatment of celiac disease (avoidance of wheat products) is so effective and inexpensive and because celiac disease is so common in selective populations, a highly reliable test for its detection such as anti-tTG should find wide application in clinical practice.

Antibodies↗