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Adult celiac disease presented with celiac crisis: Report of two cases

Year 2024, , 103 - 107, 29.10.2024
https://doi.org/10.56016/dahudermj.1442873

Abstract

Two patients (case 1: 29 years old and case 2: 66 years old. female) with no known medical history of chronic diseases, including celiac disease, presented to the hospital with prolonged diarrhea, weight loss, and severe hypocalcemia at different times. They were admitted to the hospital for hemodynamic instability in the setting of severe dehydration and electrolyte disturbances. Physical examination revealed a positive trousseau sign in Case 1. The typical laboratory features of both cases were low magnesium, low potassium, low vitamin D, low ferritin, and prolonged coagulation tests. In addition to those labs, case 2 also has metabolic acidosis. In both cases, the titers of the tissue transglutaminase IgA and IgG and the anti-endomysium antibody were high, and the histopathology of the duodenal biopsy was consistent with villous atrophy, crypt hyperplasia, and an increase in intraepithelial lymphocytes, suggesting celiac disease. Both cases responded quickly to treatment with a gluten-free diet, fluid, electrolyte, vitamin D, and K replacements, and were discharged. Celiac crisis is a rare presentation of celiac disease characterized by acute, severe metabolic imbalances resulting in high mortality and morbidity, with severe diarrhea, hypoproteinemia, and metabolic and electrolyte disturbances. It is typically seen in children under 2 years of age but can also be encountered in adulthood. Most cases respond to gluten cessation, nutritional support, and rarely steroid treatment.

Ethical Statement

As the case is included in the word file, no ethics committee approval is required.

References

  • Lebwohl B, Rubio-Tapia A. Epidemiology, Presentation, and Diagnosis of Celiac Disease. Gastroenterology. 2021 Jan;160(1):63-75. doi: 10.1053/j.gastro.2020.06.098.
  • Rostom A, Murray JA, Kagnoff MF. American Gastroenterological Association (AGA) Institute technical review on the diagnosis and management of celiac disease. Gastroenterology. 2006 Dec;131(6):1981-2002. doi: 10.1053/j.gastro.2006.10.004.
  • Rubio-Tapia A, Hill ID, Semrad C, Kelly CP, Greer KB, Limketkai BN, Lebwohl B. American College of Gastroenterology Guidelines Update: Diagnosis and Management of Celiac Disease. Am J Gastroenterol. 2023 Jan 1;118(1):59-76. doi: 10.14309/ajg.0000000000002075. Epub 2022 Sep 21. Erratum in: Am J Gastroenterol. 2024 Jul 1;119(7):1441. doi: 10.14309/ajg.0000000000002210.
  • ANDERSEN DH, DI SANT'AGNESE PA. Idiopathic celiac disease. I. Mode of onset and diagnosis. Pediatrics. 1953 Mar;11(3):207-23.
  • Wolf I, Mouallem M, Farfel Z. Adult celiac disease presented with celiac crisis: severe diarrhea, hypokalemia, and acidosis. J Clin Gastroenterol. 2000 Apr;30(3):324-6. doi: 10.1097/00004836-200004000-00026.
  • Balaban DV, Dima A, Jurcut C, Popp A, Jinga M. Celiac crisis, a rare occurrence in adult celiac disease: A systematic review. World J Clin Cases. 2019 Feb 6;7(3):311-319. doi: 10.12998/wjcc.v7.i3.311.
  • Ferreira R, Pina R, Cunha N, Carvalho A. A Celiac Crisis in an Adult: Raising Awareness of a Life-threatening Condition. Eur J Case Rep Intern Med. 2016 Feb 4;3(2):000384. doi: 10.12890/2016_000384.
  • do Vale RR, Conci NDS, Santana AP, Pereira MB, Menezes NYH, Takayasu V, Laborda LS, da Silva ASF. Celiac Crisis: an unusual presentation of gluten-sensitive enteropathy. Autops Case Rep. 2018 Jul 30;8(3):e2018027. doi: 10.4322/acr.2018.027.
  • Jamma S, Rubio-Tapia A, Kelly CP, Murray J, Najarian R, Sheth S, Schuppan D, Dennis M, Leffler DA. Celiac crisis is a rare but serious complication of celiac disease in adults. Clin Gastroenterol Hepatol. 2010 Jul;8(7):587-90. doi: 10.1016/j.cgh.2010.04.009.
  • Bai J, Moran C, Martinez C, Niveloni S, Crosetti E, Sambuelli A, Boerr L. Celiac sprue after surgery of the upper gastrointestinal tract. Report of 10 patients with special attention to diagnosis, clinical behavior, and follow-up. J Clin Gastroenterol. 1991 Oct;13(5):521-4. doi: 10.1097/00004836-199110000-00009.
  • Malnick SD, Atali M, Lurie Y, Fraser G, Geltner D. Celiac sprue presenting during the puerperium: a report of three cases and a review of the literature. J Clin Gastroenterol. 1998 Apr;26(3):164-6. doi: 10.1097/00004836-199804000-00002.
  • Ciacci C, Maiuri L, Russo I, Tortora R, Bucci C, Cappello C, Santonicola A, Luciani A, Passananti V, Iovino P. Efficacy of budesonide therapy in the early phase of treatment of adult coeliac disease patients with malabsorption: an in vivo/in vitro pilot study. Clin Exp Pharmacol Physiol. 2009 Dec;36(12):1170-6. doi: 10.1111/j.1440-1681.2009.05211.x.
Year 2024, , 103 - 107, 29.10.2024
https://doi.org/10.56016/dahudermj.1442873

Abstract

References

  • Lebwohl B, Rubio-Tapia A. Epidemiology, Presentation, and Diagnosis of Celiac Disease. Gastroenterology. 2021 Jan;160(1):63-75. doi: 10.1053/j.gastro.2020.06.098.
  • Rostom A, Murray JA, Kagnoff MF. American Gastroenterological Association (AGA) Institute technical review on the diagnosis and management of celiac disease. Gastroenterology. 2006 Dec;131(6):1981-2002. doi: 10.1053/j.gastro.2006.10.004.
  • Rubio-Tapia A, Hill ID, Semrad C, Kelly CP, Greer KB, Limketkai BN, Lebwohl B. American College of Gastroenterology Guidelines Update: Diagnosis and Management of Celiac Disease. Am J Gastroenterol. 2023 Jan 1;118(1):59-76. doi: 10.14309/ajg.0000000000002075. Epub 2022 Sep 21. Erratum in: Am J Gastroenterol. 2024 Jul 1;119(7):1441. doi: 10.14309/ajg.0000000000002210.
  • ANDERSEN DH, DI SANT'AGNESE PA. Idiopathic celiac disease. I. Mode of onset and diagnosis. Pediatrics. 1953 Mar;11(3):207-23.
  • Wolf I, Mouallem M, Farfel Z. Adult celiac disease presented with celiac crisis: severe diarrhea, hypokalemia, and acidosis. J Clin Gastroenterol. 2000 Apr;30(3):324-6. doi: 10.1097/00004836-200004000-00026.
  • Balaban DV, Dima A, Jurcut C, Popp A, Jinga M. Celiac crisis, a rare occurrence in adult celiac disease: A systematic review. World J Clin Cases. 2019 Feb 6;7(3):311-319. doi: 10.12998/wjcc.v7.i3.311.
  • Ferreira R, Pina R, Cunha N, Carvalho A. A Celiac Crisis in an Adult: Raising Awareness of a Life-threatening Condition. Eur J Case Rep Intern Med. 2016 Feb 4;3(2):000384. doi: 10.12890/2016_000384.
  • do Vale RR, Conci NDS, Santana AP, Pereira MB, Menezes NYH, Takayasu V, Laborda LS, da Silva ASF. Celiac Crisis: an unusual presentation of gluten-sensitive enteropathy. Autops Case Rep. 2018 Jul 30;8(3):e2018027. doi: 10.4322/acr.2018.027.
  • Jamma S, Rubio-Tapia A, Kelly CP, Murray J, Najarian R, Sheth S, Schuppan D, Dennis M, Leffler DA. Celiac crisis is a rare but serious complication of celiac disease in adults. Clin Gastroenterol Hepatol. 2010 Jul;8(7):587-90. doi: 10.1016/j.cgh.2010.04.009.
  • Bai J, Moran C, Martinez C, Niveloni S, Crosetti E, Sambuelli A, Boerr L. Celiac sprue after surgery of the upper gastrointestinal tract. Report of 10 patients with special attention to diagnosis, clinical behavior, and follow-up. J Clin Gastroenterol. 1991 Oct;13(5):521-4. doi: 10.1097/00004836-199110000-00009.
  • Malnick SD, Atali M, Lurie Y, Fraser G, Geltner D. Celiac sprue presenting during the puerperium: a report of three cases and a review of the literature. J Clin Gastroenterol. 1998 Apr;26(3):164-6. doi: 10.1097/00004836-199804000-00002.
  • Ciacci C, Maiuri L, Russo I, Tortora R, Bucci C, Cappello C, Santonicola A, Luciani A, Passananti V, Iovino P. Efficacy of budesonide therapy in the early phase of treatment of adult coeliac disease patients with malabsorption: an in vivo/in vitro pilot study. Clin Exp Pharmacol Physiol. 2009 Dec;36(12):1170-6. doi: 10.1111/j.1440-1681.2009.05211.x.
There are 12 citations in total.

Details

Primary Language English
Subjects ​Internal Diseases
Journal Section Case Reports
Authors

Mehmet Uzunlulu 0000-0001-8754-1069

Erhan Eken 0000-0002-8602-5940

Elif Pala 0009-0009-4714-8022

Ender İğneci 0000-0002-8766-8382

Zeyneb Nehar Toprak 0009-0006-8793-5950

Publication Date October 29, 2024
Submission Date February 26, 2024
Acceptance Date March 26, 2024
Published in Issue Year 2024

Cite

EndNote Uzunlulu M, Eken E, Pala E, İğneci E, Toprak ZN (October 1, 2024) Adult celiac disease presented with celiac crisis: Report of two cases. DAHUDER Medical Journal 4 4 103–107.



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