Cargando…

A case of sarcoidosis with isolated hepatosplenic onset and development of inflammatory bowel disease during recovery stage

Sarcoidosis is a systemic disease characterized by an immune-mediated disorder, which leads to the development of non-caseating granulomas in the involved organs. More than 90% of patients with sarcoidosis present lungs and lymphatic system involvement at onset, while less than 10% has an isolated e...

Descripción completa

Detalles Bibliográficos
Autores principales: Sangineto, Moris, Luglio, Chiara Valentina, Suppressa, Patrizia, Sabbà, Carlo, Napoli, Nicola
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer International Publishing 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5408327/
https://www.ncbi.nlm.nih.gov/pubmed/28455816
http://dx.doi.org/10.1007/s13317-017-0094-5
_version_ 1783232282715226112
author Sangineto, Moris
Luglio, Chiara Valentina
Suppressa, Patrizia
Sabbà, Carlo
Napoli, Nicola
author_facet Sangineto, Moris
Luglio, Chiara Valentina
Suppressa, Patrizia
Sabbà, Carlo
Napoli, Nicola
author_sort Sangineto, Moris
collection PubMed
description Sarcoidosis is a systemic disease characterized by an immune-mediated disorder, which leads to the development of non-caseating granulomas in the involved organs. More than 90% of patients with sarcoidosis present lungs and lymphatic system involvement at onset, while less than 10% has an isolated extrapulmonary localization. Here, we describe the case of an elderly patient with isolated hepato-splenic onset (multiple splenic lesions at imaging and cholestasis), and subsequent pulmonary involvement. The liver biopsy showed the presence of non-caseating granulomas, suggesting sarcoidosis. Despite the complete recovery was obtained with steroid therapy, after dosage reduction the patient presented watery diarrhea. Endoscopic investigations with biopsies were performed, describing the presence of an important lympho-plasmacytic infiltrate of terminal ileum mucosa with typical aspects of inflammatory bowel disease. The symptomatology completely disappeared after steroid dosage increase. This case confirms that sarcoidosis could present in a very atypical way, involving several organs in a different manner at the same time and that every symptom should not be underestimated, despite the rare presentation.
format Online
Article
Text
id pubmed-5408327
institution National Center for Biotechnology Information
language English
publishDate 2017
publisher Springer International Publishing
record_format MEDLINE/PubMed
spelling pubmed-54083272017-05-12 A case of sarcoidosis with isolated hepatosplenic onset and development of inflammatory bowel disease during recovery stage Sangineto, Moris Luglio, Chiara Valentina Suppressa, Patrizia Sabbà, Carlo Napoli, Nicola Auto Immun Highlights Case Report Sarcoidosis is a systemic disease characterized by an immune-mediated disorder, which leads to the development of non-caseating granulomas in the involved organs. More than 90% of patients with sarcoidosis present lungs and lymphatic system involvement at onset, while less than 10% has an isolated extrapulmonary localization. Here, we describe the case of an elderly patient with isolated hepato-splenic onset (multiple splenic lesions at imaging and cholestasis), and subsequent pulmonary involvement. The liver biopsy showed the presence of non-caseating granulomas, suggesting sarcoidosis. Despite the complete recovery was obtained with steroid therapy, after dosage reduction the patient presented watery diarrhea. Endoscopic investigations with biopsies were performed, describing the presence of an important lympho-plasmacytic infiltrate of terminal ileum mucosa with typical aspects of inflammatory bowel disease. The symptomatology completely disappeared after steroid dosage increase. This case confirms that sarcoidosis could present in a very atypical way, involving several organs in a different manner at the same time and that every symptom should not be underestimated, despite the rare presentation. Springer International Publishing 2017-04-28 /pmc/articles/PMC5408327/ /pubmed/28455816 http://dx.doi.org/10.1007/s13317-017-0094-5 Text en © The Author(s) 2017 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made.
spellingShingle Case Report
Sangineto, Moris
Luglio, Chiara Valentina
Suppressa, Patrizia
Sabbà, Carlo
Napoli, Nicola
A case of sarcoidosis with isolated hepatosplenic onset and development of inflammatory bowel disease during recovery stage
title A case of sarcoidosis with isolated hepatosplenic onset and development of inflammatory bowel disease during recovery stage
title_full A case of sarcoidosis with isolated hepatosplenic onset and development of inflammatory bowel disease during recovery stage
title_fullStr A case of sarcoidosis with isolated hepatosplenic onset and development of inflammatory bowel disease during recovery stage
title_full_unstemmed A case of sarcoidosis with isolated hepatosplenic onset and development of inflammatory bowel disease during recovery stage
title_short A case of sarcoidosis with isolated hepatosplenic onset and development of inflammatory bowel disease during recovery stage
title_sort case of sarcoidosis with isolated hepatosplenic onset and development of inflammatory bowel disease during recovery stage
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5408327/
https://www.ncbi.nlm.nih.gov/pubmed/28455816
http://dx.doi.org/10.1007/s13317-017-0094-5
work_keys_str_mv AT sanginetomoris acaseofsarcoidosiswithisolatedhepatospleniconsetanddevelopmentofinflammatoryboweldiseaseduringrecoverystage
AT lugliochiaravalentina acaseofsarcoidosiswithisolatedhepatospleniconsetanddevelopmentofinflammatoryboweldiseaseduringrecoverystage
AT suppressapatrizia acaseofsarcoidosiswithisolatedhepatospleniconsetanddevelopmentofinflammatoryboweldiseaseduringrecoverystage
AT sabbacarlo acaseofsarcoidosiswithisolatedhepatospleniconsetanddevelopmentofinflammatoryboweldiseaseduringrecoverystage
AT napolinicola acaseofsarcoidosiswithisolatedhepatospleniconsetanddevelopmentofinflammatoryboweldiseaseduringrecoverystage
AT sanginetomoris caseofsarcoidosiswithisolatedhepatospleniconsetanddevelopmentofinflammatoryboweldiseaseduringrecoverystage
AT lugliochiaravalentina caseofsarcoidosiswithisolatedhepatospleniconsetanddevelopmentofinflammatoryboweldiseaseduringrecoverystage
AT suppressapatrizia caseofsarcoidosiswithisolatedhepatospleniconsetanddevelopmentofinflammatoryboweldiseaseduringrecoverystage
AT sabbacarlo caseofsarcoidosiswithisolatedhepatospleniconsetanddevelopmentofinflammatoryboweldiseaseduringrecoverystage
AT napolinicola caseofsarcoidosiswithisolatedhepatospleniconsetanddevelopmentofinflammatoryboweldiseaseduringrecoverystage