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CASE REPORT Table of Contents   
Year : 2001  |  Volume : 7  |  Issue : 2  |  Page : 71-73
Adult celiac disease associated with type 1 diabetes


College of Medicine, King Khalid University, Abha, Saudi Arabia

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Date of Submission11-Mar-2000
Date of Acceptance01-May-2000
 

How to cite this article:
Al-Humaidi MA. Adult celiac disease associated with type 1 diabetes. Saudi J Gastroenterol 2001;7:71-3

How to cite this URL:
Al-Humaidi MA. Adult celiac disease associated with type 1 diabetes. Saudi J Gastroenterol [serial online] 2001 [cited 2020 Apr 1];7:71-3. Available from: http://www.saudijgastro.com/text.asp?2001/7/2/71/33406


Celiac disease was first described by Samuel Ghee in 1888 [1] . A similar description of a chronic malabsorptove disorder by Aretaeus from Turkey reaches as far back as the second century AD [2] . Epidemiological studies using serologic assays for IgA antibodies to gliadin and endomysium with biopsy verification established a prevalence of 1:300 to 1:500 in most countries [3] . The prevalence of celiac disease in Saudi Arabia is not known however, there are few reports on celiac disease patients seen in clinical practice. The frequent interfamilial occurrence and the close association with the HLA-DQ2 and/or DQ8 gene locus, which provided the basis of current understanding of celiac disease as an immune disorder that is triggered by the gliadin component of gluten in genetically predisposed individuals [5],[6],[7] . We report a rare case of celiac disease in young Saudi female from Asir region, Saudi Arabia, who presented with classic clinical features of the disease with predominance of hypocalcemic symptoms. The diagnosis of celiac disease was established by elevated IgG and IgA anti-gliadin antibodies and verified by duodenal biopsy. This patient has negative family history of celiac disease and here HLA phenotyping was HLADQ6, which may suggest the non-HLA locus of gene defect. She had good response to gluten free diet with weight increase of 15 kg and disappearance of malabsorpion features within three months. Later she developed Type I Diabetes Mellitus as part of the autoimmune process involved in both diseases.


   Case report Top


A 22-year Saudi female presented to the endocrinology clinic with a-year history of carpopedal spasm, weight loss of 15 kg and loose bowel motions became very frequent over the last three months within which she was seen by an internist who documented a low calcium and phosphate, high parathyroid hormone (PTH) level and calcium supplement was started. There were no cardiovascular, respiratory or central nervous system symptoms.

On examination, she was not pale or jaundiced, weight 37kg, height 149cm, with BMI of 16.8kg/m 2 . BP 100/70, pulse 90/m regular, chvostek's and trousseau sign were positive; skin cardiovascular, respiratory, central nervous system and abdominal examination were normal. Laboratory shows hemoglobin 14.2gm/dl, WBC 8000, MCV 82, platelet 200/000/ul, ESR 19mm in one hour, albumin 3.9gm/dl, calcium 7.8mg/dl, phosphate 2mg/Dl, alkaline phosphate 955u/l, sodium 135mmo;/l, potassium 3,2mmol; glucose, urea, creatinin, total bilirubin, total protein, AST, ALT were normal. PTH 483 pg/ml, (normal 9-55 pg/ml), vitamin D3 level 10 ng/ml (normal 17-54 ng/ml), gliadin IgG antibody > 88 ELA units positive). Gliadin IgA antibody > 80 EIA units (> 15 EIA units positive). Antipancreatic Islet cells antibody and anti human insulin antibody were negative. Duodenal biopsy revealed total villous atrophy with elongated crypts and increased inflammatory cell infiltrating the lamina propia [Figure - 1]a,b.

The diagnosis of celiac disease with vitamin D deficiency and secondary hyper parathyroidism were established and the patient was started on gluten-free diet, calcium and vitamin D3 supplement; the hypocalcaemic symptom and loose bowel motions disappeared and her weight increased by 15kg within 12 weeks of treatment. After six months of therapy for gliadin IgA antibody decreased to 30 EIA units while gliadin IgA antibody level remained unchanged.

Five months after the diagnosis of celiac disease, she was admitted to Asir Central Hospital with clinical and biochemical evidence of diabetic ketoacidosis (DKA). Serum blood glucose of 393 mg/dl, PH 7.15, PCO 2 24, PO,83, HCO, 8, SAT 94% and urine analysis showed 4+ glucose and 3+ ketones. She was managed with IV insulin, IV normal saline, potassium supplements and discharged on BID mixed insulin dose in addition to 1600 Kcal diabetic and gluten-free diet.


   Discussion Top


Celiac disease also called gluten enteropathy and non-tropical sprue occurs primarily in white of northern European ancestry. Reports in the 1950s suggested that the prevalence of celiac disease among Europeans ranged between 1:4000 and 1:8000. The situation changed in the 1970s with the increasing awareness of the often oligosymptomatic form of celiac disease and the advent of sensitive and specific serologic assays for IgA antibodies to gliadin and endomysium. Epidemiological studies using these tests with biopsy verification established higher prevalence of 1:300 to 1:500 in most countries [2],[3] . Studies in 2000 Americans blood donors, for example suggested a prevalence of 1:250 based upon endomysial antibody testing [4] . The prevalence of celiac disease in Saudi Arabia is not known, however, it is infrequent. The frequent interfamilial occurrence and the close association with the HLA-DQ2 and/or DQ8 gene locus provide the basis of current understanding of celiac disease as an immune disorder that is triggered by the gliadin component of gluten in genetically predisposed individuals. It has been estimated that the HLA contribution to the development of celiac disease among siblings is 36% [5] . Thus, another gene(s) at an HLA unlinked locus must also participate and is likely to be stronger determinant of disease susceptibility than the HLA locus [6],[7] . This patient has negative family history of celiac disease and here HLA phenotyping was HLA-DQ6, which may suggest the non-HLA locus of gene defect.

This patient presented with classic symptom of celiac disease including malabsorpion, weight loss in addition to clinical features of vitamin D deficiency, hypocalcemia and secondary hyperparathyroidism. Some patients of celiac disease can present mainly with hypocalcemia and bone disease, usually osteomalacia, with minimal or no gastrointestinal symptom. Among 15 cases in one report, four had no gastrointestinal symptoms and seven had only mild or intermittent gastrointestinal symptoms [8] .. Other non-gastrointestinal manifestations of celiac disease, which have been described include; neuropsychiatry symptoms such as ataxia, depression, anxiety or epilepsy [9],[10],[11] . The frequency of this association is not known. In one series, for example, antibodies to gliadin were present in 68% of the patients diagnosed with idiopathic ataxia [11] . Arthritis was presented in 26% of 200 adult with celiac disease, it was peripheral in 19 (36%) patients, axial in 15 (29%) and overlap of both in 18 (35%) [12] . Celiac disease may be a surprisingly frequent cause of iron deficiency anemia. 12% of patients presenting for evaluation of iron deficiency had small bowel biopsy findings compatible with celiac disease [13] .

The diagnosis of celiac disease in our case was established by the histological features of duodenal biopsy, elevated gliadin IgA and IgG antibodies and remarkable response to gluten-free diet.

During the course of her disease, she developed DM, which required insulin treatment, but the islet cell insulin antibodies were negative, this clearly reflected the major role for T cells in the pathogenesis of both DM and celiac disease. The observation in a cell culture system that autoantibodies to tissue trans glutaminase block intestinal epithelial differentiation suggests that the humoral autoimmune response also may be important in the pathogenesis of celiac disease [14],[15] . Celiac disease can be detected in up to 7% of patients with type 1 diabetes mellitus with antibody screening followed by small bowel biopsy. Most cases are asymptomatic and the therapeutic benefit of dietary therapy in these patients remains uncertain [16],[17] .

There is an increased incidence of autoimmune thyroid disease among patients with celiac disease, with more frequent hypothyroidism than hyperthyroidism [18] . Our patient was increased for thyroid disease, she was clinically euthyroid with normal T4 and TSH and autoantibodies to thyroglobulin, microsomal and TSH receptor was negative.

In conclusion, we report a case of celiac disease in young Saudi female from Asir region of Saudi Arabia, who presented with classic clinical features of the disease with predominance of hypocaicemic symptoms. She had good response to gluten-free diet with weight increase of 15kg and disappearance of malabsorpion features in three months. Then she developed Type I DM as part of the autoimmune process involved in both diseases.

 
   References Top

1.Booth CC History of celiac disease. BMJ 1989; 298-527.  Back to cited text no. 1    
2.Lebenthal E, Branski D. Childhood celiac Disease. A reappraisal. J Paediat 1981; 98: 681-90.  Back to cited text no. 2    
3.Fasanto A. where have all the American celiacs gone? Acta Pediatr 1996; 412 (Supp.): 20-4.  Back to cited text no. 3    
4.Not T. Horvath K. Hill ID et al. Endomysium antibodies in blood donors predicts a high prevalence of celiac disease in the USA (Abstract). Gastroenterology 1996; 110A: 351-2.  Back to cited text no. 4    
5.Petronzeli F, Bonamico M, Ferrante P, et al. Genetic contribution of the HLA region to the familial clustering of coeliac disease. Ann Hum genet 1997: 61: 307-17.  Back to cited text no. 5    
6.Houlston RS, Ford D. Genetics of coeliac disease. QJM 1996; 89: 737-43.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]
7.Zhong F, Mc Combs, Olson CC, et al. An autosomal screen for genes that predispose to celiac disease in the western countries of Ireland. Nat Genet 1996. 14:329-33.  Back to cited text no. 7    
8.Shaker JL. Brickner RC, Finding JW, et al. Hypocalcemia and skeletal disease as presenting features of celiac disease. Arch intern Med 1997; 157: 1013-6.  Back to cited text no. 8    
9.Addolorato G. Stefanini GF, Capristo E. et al. Anxiety and depression in adult untreated celiac subjects and in patients affected by inflammatory disease: A personality "trait" or a reactive illness? Hepatogastroenterology 1996: 43: 15 13-7.  Back to cited text no. 9    
10.Cronin CC, Jachson LM, Feighery C. et al. Coeliac disease and epilepsy. QJM 1998; 4: 303-8.  Back to cited text no. 10    
11.Hadjivassiliou M, Grunewald RA, Chattopadhyay AK, et al. Clinical radiological, neurophysiological and neuropathological characteristics of gluten ataxia. Lancet 1998; 352-5.  Back to cited text no. 11    
12.Lubrano E. Ciaci C, Ames PR. et al. The arthritis of coeliac disease prevalence and pattern in 200 adult patients. Br J Rheumatol 1996:35:1314-8.  Back to cited text no. 12    
13.Ackerman Z, Eliakim R, Stainikowica R, Rachmilewitz.. Role of small bowel biopsy in the endoscopic evaluation of adults with iron deficiency anemia. Am J Gastroenterol 1996; 91: 2099-102.  Back to cited text no. 13    
14.Schuppan D, Dieterich W, Ricken EO. Exposing gliadin as a tasty food for celiac disease Lymphocytes? Nat Med 1998: 4: 666-7.  Back to cited text no. 14    
15.Haltunen T, Maki M. Celiac disease autoantibodies and tissue transglutaminase antibodies interfere similarly with fibroblast-induced epithelial cell (T84) differentiation. Gastroenterology 1998; 114: A 377-9.  Back to cited text no. 15    
16.Acerini CL, Ahmed ML, Ross KM, et al. Coeliac disease in children and adolescents with IDDM: clinical characteristics and response to gluten-free diet. Diabetes Med 1998, 15: 38-44.  Back to cited text no. 16    
17.Cronin CC, Feighery A, Ferris JB, et al. High prevalence of celiac disease among patients with insulin-dependent (typel) diabetes mellitus. Am J Gastroenterol 1997: 92: 2210-2.  Back to cited text no. 17    
18.Counsel CE, Taha A, Ruddel WS. Celiac disease and autoimmune thyroid disease gut 1994; 35: 844-6.  Back to cited text no. 18    

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Correspondence Address:
Mohammed Awad Al-Humaidi
Associate professor of medicine, College of Medicine, King Khalid University, P. 0. Box 641, Abha
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


PMID: 19861773

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