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Keywords:

  • celiac disease;
  • urticaria

C eliac sprue is a chronic malabsorption disease that occurs in predisposed individuals secondary to ingestion of gluten. We report a case of chronic urticaria associated with celiac disease with a marked improvement of symptoms after adoption of a gluten-free diet.

A 24-year-old woman came to us in May 1997 with hay fever which had begun 2 weeks before. The patient was tested by skin prick test (SPT) with the main inhalant allergens (house-dust mites, Gramineae, Parietaria, molds, trees, and animal danders) and the following food allergens: milk, soybean, apple, fish, tomato, wheat, and white egg (Bayropharm DHS, Milan, Italy), and the results were evaluated by comparing the diameter of each wheal to that induced by 10 mg/ml of histamine. SPT was positive to grass pollen, confirmed by the presence of specific IgE (Poa pratensis: 26.9 IU/ml; Radim, Pomezia, Italy). The total serum IgE level was 134 IU/ml. She was treated with an H1-antihistamine (cetirizine), with complete remission of the symptoms. Three months later, this patient had generalized urticaria characterized by multiple areas of well-demarcated edematous, intensely pruritic plaques of the diameter of many centimeters, with surrounding erythema. A careful medical history revealed no indications of food, food-additive, or drug allergy. No other symptoms (weight loss, diarrhea, or malabsorption) were associated with the urticaria. She was treated with astemizole for 1 month without benefit.

In December 1997, the patient was admitted to hospital with worsening generalized urticaria. The complete diagnostic procedure comprised physical examination, chest radiography, complete blood count and differential sedimentation rate, antinuclear antibody test, C3 and C4 determination, stool analysis for ova and parasites, T3, T4, TSH, and antibodies to peroxidase or thyroglobulin evaluation, and skin biopsy. All were normal. We also investigated serum antigliadin, antireticulin, and antiendomysium antibodies. Antigliadin antibodies were positive for both IgG and IgA ( Table 1– A); antireticulin and antiendomysium serum IgA was also positive. No specific IgE antibodies to wheat flour or gluten were found. Small-bowel biopsy revealed atrophy of the microvilli and inflammation with a diffuse mononuclear infiltrate. Direct immunofluorescence performed on lesional skin failed to detect IgA deposition at papillary levels. A diagnosis of celiac disease was made; therefore, a gluten-free diet was prescribed. The urticaria improved after 1 month and totally disappeared after 3 months. No significant difference in total serum IgE level measurement during the course of the disease was observed (August 1997 during the first episode of urticarial lesions: 145 IU/ml; December 1997, when the urticaria worsened: 152 IU/ml; and, finally, March 1998, when the urticarial lesions had cleared up: 136 IU/ml). Avoiding gluten in her diet led to a decline of antigliadin titers, first the IgA isotype followed by the IgG isotype, within 3 months. Furthermore, a decrease of antiendomysium IgA titers and the disappearance of antireticulin IgA in the serum were observed, as shown in Table 1 (B and C). After 1 year of a gluten-free diet, she was still free from urticaria.

Table 1. 
 A December 1997 B January 1998 C March 1998
IgG – antigliadin117 IU/ml340 IU/ml35 IU/ml
IgA – antigliadin236 IU/ml170 IU/ml84 IU/ml
IgA – antiendomysiumPositive (1:80)Positive (1:40)Positive (1:10)
IgA – antireticulinPositive (1:80)NegativeNegative
 [UPWARDS ARROW][UPWARDS ARROW] 
 Gluten-free dietImprovement of symptoms 

Gluten-sensitive enteropathy is a disease of the small intestine characterized by villous atrophy, crypt hyperplasia, and malabsorption. It is caused by hypersensitivity to cereal grain storage proteins of wheat, oats, barley, and rye ( 1). The alcohol-soluble prolamin fraction of gluten (gliadin in wheat, secalin in rye, avenin in oats, and hordein in barley) was identified as the offending substance. However, patients with celiac disease generally tolerate at least a small amounts of oats ( 2). An increase of atopic or immunologic disorders (such as asthma, eczema, and hay fever) in adults with celiac disease has been reported ( 3, 4). On the other hand, the association between chronic urticaria and inflammatory diseases is well known.

Hautekeete et al. first described a case of chronic urticaria associated with celiac disease ( 5). Here we report another case of chronic urticaria associated with celiac disease, in which a gluten-free diet resulted in both a decline of antigliadin antibodies and the disappearance of urticarial skin lesions. IgA antibodies, in comparison with mucosal tissue components (reticulin and endomysium), also decline after a gluten-free diet. As widely reported, an increased mucosal permeability may facilitate the passage of antigens. Theoretically, it is possible that immune-complex-dependent sequelae might cause urticarial lesions also when concomitant celiac disease occurs. Consequently, the restoration of mucosal integrity after a gluten-free diet may explain the improvement of urticaria in our patient, but this requires further investigation.

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Footnotes
  1. A gluten-free diet eliminated symptoms in patients with concomitant celiac disease and chronic urticaria.