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Concomitant Celiac Disease and Wheat Allergy

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Concomitant Celiac Disease and Wheat Allergy 454 Practitioner's Corner reason, the protein bands detected in IgE immunoblotting (11- 14 kDa) could be indicative of sensitization to S wallisii, since Concomitant Celiac Disease and Wheat Allergy: we cannot ensure that profilin or a profilin with a low cross- 2 Case Reports reactivity pattern is the protein responsible for hypersensitivity in this case, as suggested elsewhere. Lombardi C1, Savi E2, Passalacqua G3 It is important to consider ornamental houseplants that do 1Departmental Unit of Allergology, Clinical Immunology & not produce pollen as possible sources of indoor allergens, Pneumology, Fondazione Poliambulanza, Brescia, Italy since other parts of these plants may cause immediate 2Departimental Unit of Allergology, G.Da Saliceto Hospital, hypersensitivity and may be more likely to do so than common Piacenza, Italy molds, house dust mites, and pet hair or dander. 3Allergy and Respiratory Diseases, Policlinico San Martino- University of Genoa, Genoa, Italy Funding The authors declare that no funding was received for the J Investig Allergol Clin Immunol 2019; Vol. 29(6): 454-456 doi: 10.18176/jiaci.0421 present study. Conflicts of Interest Key words: Celiac disease. Wheat flour allergy. Diagnosis. Molecular diagnosis. Autoimmune disease. The authors declare that they have no conflicts of interest. Palabras clave: Enfermedad celíaca. Alergia a la harina de trigo. Diagnóstico. Diagnóstico molecular. Enfermedad autoimmune. References 1. Kanerva L, Estlander T, Aalto-Korte K. Occupational protein contact dermatitis and rhinoconjunctivitis caused by Wheat flour (Triticum aestivum) is a major food source spathe (Spathiphyllum) flowers. Contact Dermatitis. 2000 throughout the world. It is used in the manufacture of bread, Jun;42(6):369-70. pasta, and cereals. Wheat can cause various diseases, including 2. Cahen YD, Lundberg M, Wüthrich B. Indoor allergy to IgE-mediated food allergy, wheat-dependent exercise-induced spathe flower (Spathiphyllum floribundum). Allergy. 1997 anaphylaxis, respiratory allergy (baker’s asthma), celiac Jan;52(1):114-5. disease, and nonceliac gluten sensitivity. Wheat allergy is 3. Mitchell J, et al. Plants and plant products injurious to the skin. an IgE-mediated response to any of the proteins present in Vancouver, British Columbia, Canada: Greengrass Ltd, 1979 wheat, including gluten. Its prevalence varies depending 4. Kanerva L, Mäkinen-Kiljunen S, Kiistala R, Granlund H. on age and region from 0.4% to 4% [1]. Around 65% of Occupational allergy caused by spathe flower (Spathiphyllum children outgrow this allergy by the age of 12 months. Celiac wallisii). Allergy. 1995 Feb;50(2):174-8. disease is an autoimmune disorder with an aberrant response 5. Kanerva L, Estlander T, Petman L, Mäkinen-Kiljunen S. to gluten proteins (present in wheat, barley, and rye) with Occupational allergic contact urticaria to yucca (Yucca subsequent atrophy of intestinal villi, impaired intestinal aloifolia), weeping fig (Ficus benjamina), and spathe flower absorption, and malnutrition. The symptoms of wheat allergy (Spathiphyllum wallisii). Allergy. 2001 Oct;56(10):1008-11. can range from mild to life-threatening and include skin 6. Bradford MM. A rapid and sensitive method for the quantitation rash, nausea, abdominal pain, vomiting/diarrhea, respiratory of microgram quantities of protein utilizing the principle of symptoms, and even anaphylaxis. Onset is usually within protein-dye binding. Anal Biochem. 1976 May 7;72:248-54. minutes, more rarely within 1-2 hours. A tentative diagnosis 7. Purillä P, Keskinen H, Leino T, Tupasela O, Tuppurainen M. can be made using skin prick testing or a specific serum IgE Occupational asthma caused by decorative flowers: review and assay. Nonetheless, the assessment of IgE with the whole case reports. Int Arch Occup Environ Health. 1994;66(2):131-6. wheat extract is poorly sensitive and specific owing to cross- 8. Jordan-Wagner DL, Whisman BA, Goetz DW. Cross- reactivity with other allergenic molecules. Wheat proteins are allergenicity among celery, cucumber, carrot, and watermelon. classified into albumins, salt-soluble globulins, and insoluble Ann Allergy. 1993 Jul;71(1):70-9. prolamins (gliadins and glutenins). Molecular tests can identify 9. Mäkinen-Kiljunen S, Turjanmaa K, Palosuo T, Reunala T. the presence of specific IgE against individual components: Characterization of latex antigens and allergens in surgical glutenins, gliadin, ω-5 gliadin, α-amylase inhibitors, lipid gloves and natural rubber by immunoelectrophoretic methods. transfer proteins, and Tri a 14 [2,3]. We report what could be J Allergy Clin Immunol. 1992 Aug;90(2):230-5. the first 2 cases of concomitant celiac disease and IgE-mediated allergy to wheat proteins. Manuscript received November 13, 2018; accepted for The first patient was a 15-year-old girl who was diagnosed publication May 20, 2019. with celiac disease at the age of 6 years based on symptoms, positive antitransglutaminase IgA level (240 IU/mL, [normal Valeria Herrera-Lasso Regás value <7 IU/mL], Thermo Fisher Scientific), antiendomysial Allergy Department IgA (1:256), and duodenal biopsy findings. She remained University Hospital Joan XXIII on a gluten-free diet, with clinical benefits and normal Tarragona, Spain E-mail: [email protected] immunological test results. She came to our emergency J Investig Allergol Clin Immunol 2019; Vol. 29(6): 451-471 © 2019 Esmon Publicidad Practitioner's Corner 455 department with generalized urticaria, lip swelling, abdominal although the diagnosis was not based on molecular data [5]. pain, diarrhea, vomiting, and respiratory distress (PaO2, The relationship between allergy and autoimmune disorders 89 mmHg). She promptly recovered after intravenous is complex and poorly understood, especially in wheat allergy methylprednisolone and intramuscular epinephrine. Symptoms and celiac disease, although some hypotheses can be put had occurred thirty minutes after eating flat bread during a party. forward [6]. No consumption of alcohol or medications could be documented. Kreiner et al [7] identified shared susceptibility loci and Tryptase serum levels were increased (14 µg/mL) during the similarities in pathways between allergy and autoimmune acute phase, although they returned to normal (7.4 µg/mL) diseases, suggesting partially shared mechanisms. IgE after 48 hours, thus supporting the diagnosis of anaphylaxis. autoantibodies have been known to be present in patients IgE to wheat was 1.5 kU/L, with gluten-specific IgE at with autoimmune disease for more than 40 years, although 2.4 kU/L. The molecular diagnosis revealed sensitization to autoantibodies are not associated with a higher rate of atopy. gliadin (4.9 kU/L) and rTri a 19.0101 (ω-5 gliadin) (5.6 kU/L). Nevertheless, IgE was recently suggested to be an active Findings for Tri a 14 were negative. The skin prick test, which trigger of autoimmunity through mechanisms involving the was performed 2 weeks after the episode, was positive for secretion of type-I interferons by plasmacytoid dendritic cells, gliadin (8 mm) (ALK-Abelló) but negative for the whole recruitment of basophils to lymph nodes, and activation of wheat extract. We deduced that the anaphylactic reaction was adaptive immune responses through B and T cells. There is elicited by ω-5 gliadin. The detailed clinical history revealed also evidence supporting the role of IgE receptors in dendritic that, after years following a strict diet the patient started to eat cell function. The activation of these cells by the immune wheat occasionally. The occasional consumption of wheat- complexes of DNA-specific IgE antibodies can also induce containing food after a long period with a gluten-free diet B-cell differentiation and plasma cell formation [8]. The may have induced sensitization to ω-5 gliadin. The patient involvement of B cells has also been hypothesized. CD5+ was again instructed to avoid the culprit allergens and received B cells can have a negative regulatory function, and their the epinephrine auto-injector. Nevertheless, she continued to deficiency can worsen both allergic and autoimmune diseases experience wheat-induced anaphylaxis. No exercise-induced (such as experimental autoimmune encephalomyelitis, chronic condition was documented. colitis, and lupus-like models of autoimmunity) [9]. The second patient was a 54-year-old woman who was first In conclusion, the 2 cases we describe support the possible diagnosed with celiac disease at the age of 26. She followed a coexistence of autoimmune and IgE-mediated diseases. The gluten-free diet, although her clinical history revealed episodes of presence of IgE specific for Tri a 14, gluten, gliadin, and ω-5 anaphylaxis after the ingestion of wheat-containing foods, with gliadin should be considered a diagnostic marker in patients raised serum tryptase (15.8 ng/mL [baseline, 4.3 ng/mL]). She with confirmed celiac disease who experience anaphylaxis [10] was referred to our emergency department with hypotension, after ingestion of food containing wheat. It is reasonable to generalized urticaria, lip swelling, abdominal pain, diarrhea/ hypothesize that the occasional consumption of wheat flour– vomiting, and dyspnea. The episode was rapidly controlled containing proteins could trigger IgE-mediated sensitization with intramuscular epinephrine, intravenous corticosteroids, overlapping with a pre-existing autoimmune disease. and antihistamines combined with rehydration. The patient was taking nebivolol, which was temporarily withdrawn. She had Funding
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