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Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease

Adult-onset Still’s disease (AOSD) is a rare systemic autoinflammatory disease with a broad spectrum of clinical manifestations including fever, skin rash, arthralgia and neutrophilic leucocytosis. Small bowel inflammation in AOSD has been reported in association with Crohn’s disease, coeliac diseas...

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Autores principales: Manzotti, Cristina, Castellani, Massimo, Murgo, Antonella, Basilisco, Guido
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BMJ Publishing Group 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9923262/
https://www.ncbi.nlm.nih.gov/pubmed/36759043
http://dx.doi.org/10.1136/bcr-2022-252345
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author Manzotti, Cristina
Castellani, Massimo
Murgo, Antonella
Basilisco, Guido
author_facet Manzotti, Cristina
Castellani, Massimo
Murgo, Antonella
Basilisco, Guido
author_sort Manzotti, Cristina
collection PubMed
description Adult-onset Still’s disease (AOSD) is a rare systemic autoinflammatory disease with a broad spectrum of clinical manifestations including fever, skin rash, arthralgia and neutrophilic leucocytosis. Small bowel inflammation in AOSD has been reported in association with Crohn’s disease, coeliac disease and chronic intestinal pseudo-obstruction. We have here reported the first-time case of AOSD with small bowel involvement, presenting with chronic vomiting. Fluorodeoxyglucose total-body positron emission tomography/CT showed high uptake of the duodenum and first jejunal loop. Faecal calprotectin increased. Duodenal histology revealed neutrophilic infiltrate among duodenal enterocytes and severe chronic lymphoplasmacellular infiltrate of the lamina propria. Vomiting disappeared when duodenojejunal inflammation was controlled by immunomodulatory treatments for AOSD, suggesting a possible causal relation between duodenojejunal inflammation and the symptom.
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spelling pubmed-99232622023-02-14 Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease Manzotti, Cristina Castellani, Massimo Murgo, Antonella Basilisco, Guido BMJ Case Rep Case Reports: Unusual presentation of more common disease/injury Adult-onset Still’s disease (AOSD) is a rare systemic autoinflammatory disease with a broad spectrum of clinical manifestations including fever, skin rash, arthralgia and neutrophilic leucocytosis. Small bowel inflammation in AOSD has been reported in association with Crohn’s disease, coeliac disease and chronic intestinal pseudo-obstruction. We have here reported the first-time case of AOSD with small bowel involvement, presenting with chronic vomiting. Fluorodeoxyglucose total-body positron emission tomography/CT showed high uptake of the duodenum and first jejunal loop. Faecal calprotectin increased. Duodenal histology revealed neutrophilic infiltrate among duodenal enterocytes and severe chronic lymphoplasmacellular infiltrate of the lamina propria. Vomiting disappeared when duodenojejunal inflammation was controlled by immunomodulatory treatments for AOSD, suggesting a possible causal relation between duodenojejunal inflammation and the symptom. BMJ Publishing Group 2023-02-09 /pmc/articles/PMC9923262/ /pubmed/36759043 http://dx.doi.org/10.1136/bcr-2022-252345 Text en © BMJ Publishing Group Limited 2023. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ. https://creativecommons.org/licenses/by-nc/4.0/This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/ (https://creativecommons.org/licenses/by-nc/4.0/) .
spellingShingle Case Reports: Unusual presentation of more common disease/injury
Manzotti, Cristina
Castellani, Massimo
Murgo, Antonella
Basilisco, Guido
Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease
title Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease
title_full Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease
title_fullStr Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease
title_full_unstemmed Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease
title_short Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease
title_sort duodenojejunal inflammation causing chronic vomiting in adult-onset still’s disease
topic Case Reports: Unusual presentation of more common disease/injury
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9923262/
https://www.ncbi.nlm.nih.gov/pubmed/36759043
http://dx.doi.org/10.1136/bcr-2022-252345
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