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SAT-LB88 Recurrent Dysphagia Due to an Esophageal Stricture in a Patient With Autoimmune Polyendocrinopathy Candidiasis Ectodermal Dystrophy (APECED)
Background: Chronic Mucocutaneous Candidiasis is one of the hallmark features of Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy (APECED). Untreated esophageal candidiasis may lead to complications such as esophageal stricture, rupture and or fistula formation(1,2). Case Presentation:...
Autores principales: | , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Oxford University Press
2020
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7208786/ http://dx.doi.org/10.1210/jendso/bvaa046.2204 |
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author | Johnson, Timothy Gabriely, Ilan |
author_facet | Johnson, Timothy Gabriely, Ilan |
author_sort | Johnson, Timothy |
collection | PubMed |
description | Background: Chronic Mucocutaneous Candidiasis is one of the hallmark features of Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy (APECED). Untreated esophageal candidiasis may lead to complications such as esophageal stricture, rupture and or fistula formation(1,2). Case Presentation:A 31-year-old man with a past medical history significant for APECED with involvement including hypoparathyroidism, hypothyroidism and adrenal insufficiency presented to the Emergency Department with dysphagia and globus sensation after eating. This was the fourth time in the last 2 years that the patient had presented with this chief complaint. The patient underwent emergent esophagogastroduodenoscopy (EGD) where a large meat bolus was identified at approximately 20 cm and removed. A discrete esophageal tear at 19-20 cm was identified and clipped. The esophagus was noted to be diffusely narrowed with mild corrugation, and biopsies showed evidence of esophageal candidiasis. A chest CT scan demonstrated diffuse pneumomediastinum and soft tissue emphysema throughout the esophagus, suspicious for a tear. Fluoroscopic Esophagogram showed mild narrowing of the upper third of the esophagus without evidence of gross extravasation. The patient was placed on NPO (nothing by mouth) diet, started total parenteral nutrition, and treated with IV Fluconazole, administered for 10 days and then transitioned to oral Fluconazole for a total of 21 days, with improvement. A subsequent repeat fluoroscopic esophagogram showed absence of extravasation at the previously identified level of the esophagus. The patient is currently scheduled for a repeat EGD in 3 months. Conclusion:This case demonstrates a significant complication of APECED involving esophageal candidiasis. Timely diagnosis and treatment of esophageal candidiasis in APECED may prevent life-threatening esophageal pathology. References:1)Kim, Byung-Wook et al. Esophagomediastinal fistula and esophageal stricture as a complication of esophageal candidiasis: A case report. Gastrointestinal Endoscopy, Volume 52, Issue 6, 772 – 775.2)Hyun J, Chun H, Keum B et al. Candida esophagitis complicated by esophageal stricture. Endoscopy 2010; 42(S02): 180 - 181. doi:10.1055/s-0029-1244014. |
format | Online Article Text |
id | pubmed-7208786 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2020 |
publisher | Oxford University Press |
record_format | MEDLINE/PubMed |
spelling | pubmed-72087862020-05-13 SAT-LB88 Recurrent Dysphagia Due to an Esophageal Stricture in a Patient With Autoimmune Polyendocrinopathy Candidiasis Ectodermal Dystrophy (APECED) Johnson, Timothy Gabriely, Ilan J Endocr Soc Thyroid Background: Chronic Mucocutaneous Candidiasis is one of the hallmark features of Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy (APECED). Untreated esophageal candidiasis may lead to complications such as esophageal stricture, rupture and or fistula formation(1,2). Case Presentation:A 31-year-old man with a past medical history significant for APECED with involvement including hypoparathyroidism, hypothyroidism and adrenal insufficiency presented to the Emergency Department with dysphagia and globus sensation after eating. This was the fourth time in the last 2 years that the patient had presented with this chief complaint. The patient underwent emergent esophagogastroduodenoscopy (EGD) where a large meat bolus was identified at approximately 20 cm and removed. A discrete esophageal tear at 19-20 cm was identified and clipped. The esophagus was noted to be diffusely narrowed with mild corrugation, and biopsies showed evidence of esophageal candidiasis. A chest CT scan demonstrated diffuse pneumomediastinum and soft tissue emphysema throughout the esophagus, suspicious for a tear. Fluoroscopic Esophagogram showed mild narrowing of the upper third of the esophagus without evidence of gross extravasation. The patient was placed on NPO (nothing by mouth) diet, started total parenteral nutrition, and treated with IV Fluconazole, administered for 10 days and then transitioned to oral Fluconazole for a total of 21 days, with improvement. A subsequent repeat fluoroscopic esophagogram showed absence of extravasation at the previously identified level of the esophagus. The patient is currently scheduled for a repeat EGD in 3 months. Conclusion:This case demonstrates a significant complication of APECED involving esophageal candidiasis. Timely diagnosis and treatment of esophageal candidiasis in APECED may prevent life-threatening esophageal pathology. References:1)Kim, Byung-Wook et al. Esophagomediastinal fistula and esophageal stricture as a complication of esophageal candidiasis: A case report. Gastrointestinal Endoscopy, Volume 52, Issue 6, 772 – 775.2)Hyun J, Chun H, Keum B et al. Candida esophagitis complicated by esophageal stricture. Endoscopy 2010; 42(S02): 180 - 181. doi:10.1055/s-0029-1244014. Oxford University Press 2020-05-08 /pmc/articles/PMC7208786/ http://dx.doi.org/10.1210/jendso/bvaa046.2204 Text en © Endocrine Society 2020. http://creativecommons.org/licenses/by-nc-nd/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs licence (http://creativecommons.org/licenses/by-nc-nd/4.0/), which permits non-commercial reproduction and distribution of the work, in any medium, provided the original work is not altered or transformed in any way, and that the work is properly cited. For commercial re-use, please contact journals.permissions@oup.com |
spellingShingle | Thyroid Johnson, Timothy Gabriely, Ilan SAT-LB88 Recurrent Dysphagia Due to an Esophageal Stricture in a Patient With Autoimmune Polyendocrinopathy Candidiasis Ectodermal Dystrophy (APECED) |
title | SAT-LB88 Recurrent Dysphagia Due to an Esophageal Stricture in a Patient With Autoimmune Polyendocrinopathy Candidiasis Ectodermal Dystrophy (APECED) |
title_full | SAT-LB88 Recurrent Dysphagia Due to an Esophageal Stricture in a Patient With Autoimmune Polyendocrinopathy Candidiasis Ectodermal Dystrophy (APECED) |
title_fullStr | SAT-LB88 Recurrent Dysphagia Due to an Esophageal Stricture in a Patient With Autoimmune Polyendocrinopathy Candidiasis Ectodermal Dystrophy (APECED) |
title_full_unstemmed | SAT-LB88 Recurrent Dysphagia Due to an Esophageal Stricture in a Patient With Autoimmune Polyendocrinopathy Candidiasis Ectodermal Dystrophy (APECED) |
title_short | SAT-LB88 Recurrent Dysphagia Due to an Esophageal Stricture in a Patient With Autoimmune Polyendocrinopathy Candidiasis Ectodermal Dystrophy (APECED) |
title_sort | sat-lb88 recurrent dysphagia due to an esophageal stricture in a patient with autoimmune polyendocrinopathy candidiasis ectodermal dystrophy (apeced) |
topic | Thyroid |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7208786/ http://dx.doi.org/10.1210/jendso/bvaa046.2204 |
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