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PubMed1983

"Pseudoascites" as a presenting physical sign of celiac disease.

Two children initially referred for evaluation of ascites were diagnosed as suffering from celiac disease. Positive physical signs of ascites were present in the absence of free fluid in the peritioneal cavity. Physical findings of ascites were caused solely by dilated small intestinal loops filled with copious fluid. "Pseudoascites" should be recognized as a physical sign and possible mode of presentation of celiac disease.

Ascites↗
PubMed2004

Range of neurologic disorders in patients with celiac disease.

OBJECTIVE: During the past 2 decades, celiac disease (CD) has been recognized as a multisystem autoimmune disorder. A growing body of distinct neurologic conditions such as cerebellar ataxia, epilepsy, myoclonic ataxia, chronic neuropathies, and dementia have been reported, mainly in middle-aged adults. There still are insufficient data on the association of CD with various neurologic disorders in children, adolescents, and young adults, including more common and "soft" neurologic conditions, such as headache, learning disorders, attention-deficit/hyperactivity disorder (ADHD), and tic disorders. The aim of the present study is to look for a broader spectrum of neurologic disorders in CD patients, most of them children or young adults. METHODS: Patients with CD were asked to fill in a questionnaire regarding the presence of neurologic disorders or symptoms. Their medical charts were reviewed, and those who were reported as having neurologic manifestations underwent neurologic examination and brain imaging or electroencephalogram if required. Their neurologic data were compared with that of a control group matched for age and gender. RESULTS: Patients with CD were more prone to develop neurologic disorders (51.4%) in comparison with control subjects (19.9%). These disorders include hypotonia, developmental delay, learning disorders and ADHD, headache, and cerebellar ataxia. Epileptic disorders were only marginally more common in CD. In contrast, no difference was found in the prevalence of tic disorders in both groups. Therapeutic benefit, with gluten-free diet, was demonstrated only in patients with transient infantile hypotonia and migraine headache. CONCLUSION: This study suggests that the variability of neurologic disorders that occur in CD is broader than previously reported and includes "softer" and more common neurologic disorders, such as chronic headache, developmental delay, hypotonia, and learning disorders or ADHD. Future longitudinal prospective studies might better define the full range of these neurologic disorders and their clinical response to a gluten-free diet.

Adolescent↗
PubMed2004

Longitudinal changes in bone metabolism and bone mineral content in children with celiac disease during consumption of a gluten-free diet.

BACKGROUND: A gluten-free diet (GFD) rapidly corrects the bone mineral deficit of children with untreated celiac disease. The mechanisms underlying such changes are still poorly understood. OBJECTIVE: In a longitudinal study, we monitored changes in bone metabolism during consumption of a GFD. DESIGN: We studied 22 white patients with celiac disease (11 girls) aged 10.5 +/- 1.0 y at the time of diagnosis. We compared bone metabolism and bone mass values in these patients with those in 428 healthy white children aged 11.3 +/- 0.2 y. Bone-specific alkaline phosphatase (a bone formation index) and N-terminal telopeptide of type I collagen (NTx; a bone resorption marker) were measured at the time of diagnosis and after 2, 6, and 12 mo of the GFD. Bone mineral content was measured at the lumbar spine and for the whole skeleton. RESULTS: The bone mineral content of patients was significantly lower than that of control subjects at the time of diagnosis but not after 1 y of the GFD. Serum bone-specific alkaline phosphatase concentrations of patients were significantly lower than those of control subjects at the time of diagnosis (P = 0.0064) and increased gradually and significantly during the GFD (ANOVA F = 4.71; P = 0.024). Conversely, patients with untreated disease had significantly higher urinary concentrations of NTx than did healthy control subjects (P < 0.0001). Urinary concentrations of NTx were not significantly affected by treatment (P = 0.37). CONCLUSIONS: The rate of bone metabolism is altered in children with untreated celiac disease, and these alterations may be the cause of osteopathy. Remarkable changes occur after the initiation of a GFD, and they result in a more balanced equilibrium.

Alkaline Phosphatase↗
PubMedDate not supplied

[Myocardial depression in a patient with celiac disease. A clinical case report and literature review].

We report an autoinmune myocarditis case as a complication of celiac disease in a 28 year old woman. She had a 15 month history or diarrhea. She suffered pelvic trauma due to an episode of syncope and was admitted with refractory shock to fluid repletion. At laparotomy, two moderate hematomas were found in the subcutaneous space and retroperitoneum. Later she developed hemodynamic instability requiring positioning of a Swan-Ganz pulmonary artery catheter that demonstrated pattern of cardiogenic shock. Echocardiography demonstrated dilation of all four cavities and 35% ejection fraction. Dobutamine and milrinone infusion was begun. Later, a new echocardiographic study show improvement in eyection fraction. She was discharged without complications.

Adult↗
PubMed2005

Advances in celiac disease.

PURPOSE OF REVIEW: This article aims to summarize recent critical research in celiac disease. RECENT FINDINGS: The crucial epitopes that confer toxicity to gliadin and related prolamins continue to be defined, as do methods of assessing their toxicity. New approaches to making the gluten-free diet more palatable are being studied. SUMMARY: The position of proline residues is critical to the toxicity of cereal proteins to patients with celiac disease. Other genetic factors, apart from HLA status, remain elusive. Exciting advances in altering the toxicity of cereal proteins are being made.

Animals↗
PubMedDate not supplied

[IgA-class antigliadin antibodies in the screening and follow-up of celiac disease patients].

IgA antigliadin antibodies (IgA-AGA) have been determined with an enzyme immunoassay in 2.141 pediatric patients. High levels of IgA were found in 98% of 53 celiac patients (1st biopsy), in 81% of 16 celiac patients after gluten challenge, while high levels of these antibodies were not found in 200 patients on gluten-free diet. Moreover high levels of IgA-AGA were found in 29% of 48 patients with normal jejunal biopsy and in 4% of 1.824 patients with gastrointestinal problems other than celiac disease. Our results confirm the data report in literature about the sensibility and the specificity of the IgA-AGA dosage as a screening test for celiac disease, but the possibility of false pathological and false normal values confirms the intestinal biopsy, as the main procedure for the diagnosis of celiac disease.

Antibodies↗
PubMed2001

Comparative analysis of organ-specific autoantibodies and celiac disease--associated antibodies in type 1 diabetic patients, their first-degree relatives, and healthy control subjects.

OBJECTIVE: In type 1 diabetes the coexistence with other endocrine diseases and organ-specific autoantibodies has been frequently reported leading to the concept of autoimmune polyendocrine syndrome (APS). In addition, an association of type 1 diabetes with celiac disease has been described. These disorders share a similar genetic background, and first-degree relatives of type 1 diabetic patients may also be affected significantly. Screening for specific antibodies allows early diagnosis of these disorders. RESEARCH DESIGN AND METHODS: In the present cross-sectional study, we analyzed sera from 197 recent-onset type 1 diabetic patients at the time of diagnosis, 882 first-degree relatives, and sera of 150 healthy control subjects for prevalence and co-occurence of the following antibodies (method): insulin autoantibodies (radioimmunoassay); GAD and IA-2 antibodies (radioligand assay); islet cell antibody, anti-adrenal cortex antibodies, and anti-gastric parietal cell antibodies (indirect immunofluorescence); anti-thyroglobulin and anti-thyroid peroxidase antibodies; and gliadin IgG/A and tissue-transglutaminase IgA (enzyme-linked immunosorbent assay). RESULTS: The overall frequency of gastric patietal cell antibodies and adrenal antibodies did not differ significantly among groups. In contrast, type 1 diabetes-associated antibodies and thyroid antibodies were significantly more frequent both in recent-onset type 1 diabetic patients and in the group of first-degree relatives (P < 0.05). The prevalence of gliadin IgG/IgA and transglutaminase IgA was significantly higher in the group of recent-onset type 1 diabetic patients (P < 0.05), but the difference between first-degree relatives and control subjects did not reach statistical significance. Focusing on the coexistence of antibodies, the group of recentonset type 1 diabetic patients presented with 27.4% of the subjects testing antibody-positive-specific for two or more of the envisaged disorders (i.e., type 1 diabetes, autoimmune thyroiditis, and celiac disease) compared with 3.1% in the group of first-degree relatives and 0 of 150 in the control population (P < 0.05). CONCLUSIONS: We conclude that, in an active case-finding strategy, recent-onset type 1 diabetic patients should be routinely screened at least for concomitant autoimmune thyroid disease and additionally for celiac disease. Screening in their first-degree relatives should include at a minimum the search for thyroid autoimmunity in addition to screening for pre-type 1 diabetes.

Adolescent↗
PubMed1992

Determination of hair trace elements in childhood celiac disease and in cystic fibrosis.

Applying proton-induced X-ray emission authors investigated the hair trace element contents in 10 children with acute celiac disease after 3 to 6 and 12 months long gluten-free diet; in 9 children with cystic fibrosis and in a control group (6 children) of the same age. There was no difference in Cu, Fe, Ca, Cl values between the examined groups. The Zn contents of the hair are significantly low in the group with acute celiac disease after a short-term diet and also in the group with cystic fibrosis, the data approach the normal range only after a year's diet. The significant rise of hair potassium contents is well indicated in patients with acute celiac disease and this rise may be due to the destruction of cell cuticles. In case of cystic fibrosis there is no significant rise of hair potassium value.

Calcium↗
PubMed1996

Hydrophobic interactions between gliadin and proteins and celiac disease.

Gliadin-protein interaction and its relationship to the pathogenesis hypotheses of celiac disease was investigated. Wheat germ agglutinin was not immunodetected in gliadin preparations. Peptic-tryptic gliadin digest was used to study the gliadin-protein interactions by crossed immunoelectrophoresis and affinity blotting. Biotinylated gliadin digest interacted with IgG and bovine serum albumin but not with several glycoproteins. Since albumin and IgG light chains are not glycosylated, this interaction is not lectin-like, neither completely immunological because of recognition of the IgG Fc fraction. Immobilized and boiled IgG was not recognized by gliadin digest as a lectin. Gliadin digest fractions from T-gel chromatography reduced the fluorescence intensity of cis-parinaric acid bound to albumin. The gliadin-protein interaction is not lectin-like or completely immunological but hydrophobic. Hydrophobicity of gliadins may contribute to the pathogenic events that result in celiac disease.

Celiac Disease↗
PubMed1999

Birth outcomes of women with celiac disease: a nationwide historical cohort study.

OBJECTIVE: We aimed to examine birthweight, low birthweight (<2500 g), and intrauterine growth retardation in offspring of women with celiac disease in relation to their first hospitalization for the disease. METHODS: This was a historical cohort study based on The Danish Medical Birth Registry data of celiac women discharged from Danish hospitals from 1977-1992. The study included 211 newborns to 127 mothers with celiac disease, and 1260 control deliveries. RESULTS: Before celiac women were first hospitalized the mean birthweight of their newborns was 238 g (95% confidence interval [95% CI] = 150, 325 g) lower than that of the control women, after adjustment for potential confounders. After the first hospitalization the mean birthweight for newborns of diseased women was higher than that of controls, by 67 g (95% CI = -88, 223 g) after adjustment for potential confounders. Before celiac women were first hospitalized we found an increased risk of low birthweight (odds ratio [OR] = 2.6, 95% CI = 1.3-5.5) and intrauterine growth retardation (OR = 3.4, 95% CI = 1.6-7.2). After celiac women were first hospitalized we found no increased risk of low birthweight and no babies with intrauterine growth retardation. CONCLUSIONS: Offspring of mothers with celiac disease had lower birthweight than expected and more than a three-fold higher risk of intrauterine growth retardation when birth occurred before the first hospitalization for the disease. After the mother's first hospitalization the birthweight was similar to controls and no increased risk of low birthweight was seen. Our study indicates that treatment of celiac women is important in the prevention of fetal growth retardation.

Adult↗
PubMed2001

[Takayasu's disease associated with autoimmune thyroiditis and celiac disease. Clinical course and limitations of treatment].

Takayasu's arteritis is a non-specific form of vasculitis involving the aorta, its main branches and pulmonary arteries. It is a rare disease in our country, contrasting with the high prevalence in Southeast Asia, Africa and South America. We discuss the course of the disease in our patient who was a young women who developed Takayasu's arteritis associated with autoimmune thyroiditis and malabsorption syndrome due to celiac sprue. Long-term immunosuppressive therapy contributed to stabilizing the associated diseases, but did not stop the progression of the vascular lesions. The main adverse outcome in our patient was the development of severe renovascular hypertension. Bilateral renal artery stenosis was treated by angioplasty with stent implantation. Based on literature reports, the association of Takayasu's disease with multiple autoimmune disorders is a rare event. However, it would appear that the arteritis was the limiting disease for prognosis in our patient.

Celiac Disease↗
PubMedDate not supplied

Tight junctions of jejunal surface and crypt cells in celiac disease: a freeze-fracture study.

We studied mucosal biopsies of five children with celiac disease and five controls of similar age by freeze-fracture electron microscopy. Morphometric parameters of membrane structure (such as apical tight junctions, triple tight junctions, intramembrane particles) and of the brush border were obtained from a large number of micrographs. The quantitative comparison of celiac patients and control subjects showed similar values for the depth of tight junctions at the level of the crypts. At the surface level, the tight junctions of celiac patients showed reduced depth as well as a reduced number of strands. The number of intramembrane particles of microvillus membranes increases in control subjects and in celiac patients as the cells migrate from the crypts to the mucosal surface. The specimens from celiac disease patients possessed a reduced density of intramembrane particles. Differences in brush border morphology between celiac patients and control subjects were similar to earlier findings.

Celiac Disease↗
PubMed1998

Meningo-cortical calcifying angiomatosis and celiac disease.

A woman with ophthalmic migraine was found to have bilateral cerebellar and cerebral calcifications. She progressively developed severe intracranial hypertension, with swelling of the brain and downward transtentorial and tonsillar herniation. Because steroid treatment was ineffective, the right occipital pole was resected. Histological study demonstrated meningo-cortical calcifying angiomatosis. Within 2 months, brain swelling and papilledema disappeared. Subtle signs of malabsorption led to the hypothesis of celiac disease, confirmed by jejunal biopsy. Similar cerebral histological findings have been reported in the brain of two young patients affected by epilepsy and celiac disease. The association between cerebral calcifications and celiac disease is peculiar; the pathogenetic relationship is unknown.

Adult↗
PubMed2002

Even small amounts of gluten cause relapse in children with celiac disease.

BACKGROUND: Previously, a gluten challenge was customary to establish the diagnosis of celiac disease in children. There are no clear recommendations on how to perform this challenge or what markers to rely on for timing the biopsy after the challenge. The authors' aim was to monitor gluten intake, clinical symptoms, and antibody kinetics to evaluate the influence of gluten exposure during the challenge. METHODS: Twenty-five children under investigation for suspected celiac disease were challenged. One child was excluded because blood samples, food records, or biopsy was lacking. Median age at the postchallenge biopsy was 3.8 (2.7-8.8) years. The families kept daily records of the children's gluten intake and of symptoms that occurred. Blood samples were taken monthly for analysis of antigliadin and endomysium antibodies and total immunoglobulin A (IgA). A third biopsy was performed when clinical symptoms suggested a relapse. RESULTS: All 24 children showed deterioration of the mucosa or elevated antibodies during gluten challenge. Median duration of the challenge was 13 (5-51) weeks, and mean gluten intake was 1.7 (0.2-4.3) g/d and 0.1 (0.02-0.26) g/kg daily. CONCLUSIONS: Gluten intake during the challenge varied widely, and the parents were unable to give their children the recommended amount. Despite the small amounts given, all children showed signs of relapse at a clinical, laboratory, or histologic level. Much smaller amounts of gluten than previously suggested seem sufficient to cause relapse during gluten challenge in children.

Biopsy↗
PubMed2003

Abnormalities of Th1 function in non-IgE food allergy, celiac disease, and ileal lymphonodular hyperplasia: a new relationship?

BACKGROUND: Non-IgE mechanisms may also be involved food allergy (FA). Our group has been studying the immunopathogenesis clinical entities in children with gastro-intestinal symptoms and in whom biopsies of the terminal ileum show lymphoid tissue masses referred to as ileal lymphonodular hyperplasia. Our more recent studies have demonstrated Th1/Th2 cytokine profiles associated with non-IgE FA and other clinical entities. OBJECTIVE: We investigated 12 subjects with non-IgE FA (group 1) and 4 subjects with celiac disease (group 2). Cytokine profiles and immunologic studies of lymphocytes in peripheral blood and from gastro-intestinal biopsy tissues from patients in groups 1 and 2 were also evaluated. METHODS: Group 1 consisted of 12 children with clinical symptoms of anorexia, diarrhea, and abdominal pain. The diagnosis of non-IgE FA was established by positive double-blind, placebo-controlled food challenge and reduced or negative immediate-type skin testing and negative IgE radioallergosorbent tests. Group 2 consisted of four patients with celiac disease and three adult females with biopsy-proven clinical symptoms of celiac disease. RESULTS: In group 1, peripheral blood CD4 and CD8 lymphocyte distributions were normal, with a predominance of CD4+ cells with a decreased intracellular Th1 cytokine pattern and a normal Th2 intracellular cytokine pattern. In contrast, all four patients in group 2 not only displayed abnormal CD4 and CD8 peripheral blood lymphocyte distributions (CD8 > CD4), but also an abnormal predominance of CD4+ cells with an increased Th1 and a normal Th2 cytokine pattern. A similar abnormal pattern of CD4 > CD8 ratio was observed in intestinal biopsies. All 12 patients in group 1 showed lymphonodular hyperplasia in each of the biopsies and by ileoscopy. CONCLUSIONS: These studies suggest that abnormalities in Th1 function may not only play a role in some patients with non--IgE-mediated FA in whom decreased Th1 function is seen, but also in patients with celiac disease in whom an increased Th1 function is seen. The studies also suggest that lymphonodular hyperplasia may be a hallmark histologic lesion in patients with non--IgE-mediated FA.

Biopsy↗
PubMed2004

Gut transit in celiac disease: delay of small bowel transit and acceleration after dietary treatment.

OBJECTIVES: New studies indicate that body mass index (BMI) may influence gut transit. We studied gut transit and BMI in patients with celiac disease before and after treatment. METHODS: Twenty-seven (16 female) patients with untreated celiac disease were included for detailed gastrointestinal transit measurement and calculation of BMI. Ten patients (5 female) were also studied after dietary treatment. A newly developed radiological transit procedure was used. Eighty-three healthy subjects were used as controls. RESULTS: In untreated male patients BMI was significantly reduced compared to healthy males, and small bowel transit time (SBTT) was significantly longer compared with healthy males, 3.9 (1.2-5.5) versus 2.5 h (1.4-4.3), median and percentile 10 and 90, respectively, and p= 0.04. In the 10 men and women with repeated measurements, BMI increased significantly and small bowel transit accelerated after treatment and was 3.6 h (1.7-5.5) before treatment and 2.3 h (0.7-4.0) after treatment, and p= 0.007. In women, BMI did not differ significantly between untreated patients and healthy subjects, and 31% of the female patients were overweight. Small bowel transit was significantly faster in untreated female patients who were overweight compared with lean female patients. CONCLUSION: Small bowel transit seems to be delayed in lean patients with untreated celiac disease. BMI may have some influence on the variations of small bowel transit before and after treatment.

Adult↗
PubMed1998

Autoantibodies in celiac disease: importance of fibroblasts.

BACKGROUND: Serum reticulin and endomysium autoantibodies are highly celiac disease-specific, and the autoantigens have been shown to be derived from human fibroblasts. Among human tissues, the umbilical cord also expresses these antigens. This study was conducted to compare different autoantibody tests and especially to elucidate whether human umbilical cord is a suitable substrate in tests and whether the cord jelly-derived fibroblasts express the antigens. METHODS: The indirect immunofluorescence method was used to detect the tissue and Wharton's jelly-derived fibroblast antibodies in 334 celiac disease and control sera samples. Affinity chromatography studies were used to show the correlation between human fibroblast-derived autoantigens and tissue and gliadin antibodies. The jelly-derived fibroblasts were used as antigen in a whole-cell enzyme-linked immunosorbent assay. RESULTS: Celiac disease patient sera showed IgA-class human umbilical cord antibody with high sensitivity (100%) and specificity (99%). All celiac disease patient sera tested showed in indirect immunofluorescence the molecules expressed by Wharton's jelly-derived fibroblasts. The whole-cell fibroblast autoantibody enzyme-linked immunosorbent assay had a sensitivity of 100% and a specificity of 81%. Human fibroblast-derived celiac disease autoantigens absorbed most of the IgA responsible for human umbilical cord antibodies but not the IgA responsible for gliadin antibodies in the same sera. CONCLUSIONS: Wharton's jelly-derived fibroblast autoantibodies tested in a novel whole-cell enzyme-linked immunosorbent assay correlated well with the human umbilical cord but not with gliadin antibodies.

Adolescent↗
PubMedDate not supplied

Disease specificity and dynamics of changes in IgA class anti-endomysial antibodies in celiac disease.

We evaluated the sensitivity and kinetics of serum IgA class anti-endomysial antibodies in the diagnosis of celiac disease (CD) as established by the criteria of the European Society for Pediatric Gastroenterology and Nutrition (ESPGAN). Eighty-four cases that satisfied the ESPGAN criteria for CD were evaluated for IgA-EmA titers during various phases of establishing the diagnosis. Thirty-three cases were infants and children less than 5 years of age undergoing intestinal biopsies for symptoms of CD and 51 were previously diagnosed adults. Of the 33 children, 11 were untreated and symptomatic and were IgA-EmA positive at initial presentation. Twenty-two children previously controlled on a gluten-free diet (GFD) exhibited IgA-EmA titers during gluten challenge. Furthermore, the antibody levels declined in all cases (usually to negative) when the patients were again placed on a GFD for 6-12 months. Changes in intestinal histopathology paralleled the changes in antibody titers in six cases undergoing serial biopsies. Of the 51 adult patients with proven CD who were prescribed a GFD for at least 12 months, IgA-EmA were detected in 10 cases who were noncompliant to their GFD, whereas the antibodies was found in only 1 of the remaining 41 patients strictly adhering to their diet. The sera of 140 aged-matched children with various intestinal problems, 87 healthy adults, and 67 patients with dermatological diseases served as controls and were also IgA-EmA negative. On the basis of these findings, we suggest a role for the IgA-EmA as a serological screening test for active CD. It further offers the potential for monitoring compliance to diet in established cases of CD and also indicates the proper timing for biopsy in patients undergoing evaluation of CD.

Adolescent↗