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Fibromuscular dysplasia of mesenteric arteries: a rare cause of multiple bowel resections—a case report and literature review

BACKGROUND: Fibromuscular dysplasia (FMD) is a type of unexplained nonatherosclerotic vascular disease that usually involves the renal and internal carotid arteries and rarely involves the mesenteric artery. Mesenteric artery FMD is difficult to distinguish from Crohn’s disease (CD) and Behcet’s dis...

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Autores principales: Du, Shuwen, Yang, Shanbing, Jia, Kangmei, Du, Peng, Zhang, Limin, Wang, Jiheng
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7986496/
https://www.ncbi.nlm.nih.gov/pubmed/33752607
http://dx.doi.org/10.1186/s12876-021-01702-y
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author Du, Shuwen
Yang, Shanbing
Jia, Kangmei
Du, Peng
Zhang, Limin
Wang, Jiheng
author_facet Du, Shuwen
Yang, Shanbing
Jia, Kangmei
Du, Peng
Zhang, Limin
Wang, Jiheng
author_sort Du, Shuwen
collection PubMed
description BACKGROUND: Fibromuscular dysplasia (FMD) is a type of unexplained nonatherosclerotic vascular disease that usually involves the renal and internal carotid arteries and rarely involves the mesenteric artery. Mesenteric artery FMD is difficult to distinguish from Crohn’s disease (CD) and Behcet’s disease (BD) solely based on symptoms. Patients with mesenteric artery FMD can present with an acute abdomen, but case reports of patients who have a long medical history and undergo multiple bowel resections are extremely rare. CASE PRESENTATION: The patient was a 45-year-old woman with an 11-year history of intermittent lower abdominal pain and fever. At the age of 34 years, she underwent right hemicolectomy and appendectomy due to an acute abdomen. She suffered from oral ulcers between 34 and 36 years old. A clinical diagnosis of presumed CD was made by the age of 41, and she was treated with mesalazine; however, the effect was poor. At the age of 42, she came to our centre, and based on her atypical symptoms and examination results, we thought she had CD. Hence, she was treated with glucocorticoids for 3 years. However, when she was 45, due to steroid dependence, thalidomide tablets were added. Unfortunately, she suffered from another episode of intestinal obstruction. Therefore, she underwent enterectomy. The postoperative histopathological diagnosis was mesenteric artery FMD. She no longer underwent pharmacotherapy after the surgery. Although she did not have any of her previous symptoms and postoperative colonoscopy showed no signs of recurrence, splenomegaly and abnormal routine blood results were still present. CONCLUSIONS: Patients with mesenteric artery FMD can present with an acute abdomen. In addition, the symptoms and endoscopic manifestations of mesenteric artery FMD may appear similar to CD and BD. Hence, it is difficult to make a clear clinical diagnosis and proceed with treatment. Mesenteric artery FMD often requires surgical pathology to confirm its diagnosis. For patients who suffer from this disorder, surgery may be the best choice to improve the patient’s quality of life. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s12876-021-01702-y.
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spelling pubmed-79864962021-03-24 Fibromuscular dysplasia of mesenteric arteries: a rare cause of multiple bowel resections—a case report and literature review Du, Shuwen Yang, Shanbing Jia, Kangmei Du, Peng Zhang, Limin Wang, Jiheng BMC Gastroenterol Case Report BACKGROUND: Fibromuscular dysplasia (FMD) is a type of unexplained nonatherosclerotic vascular disease that usually involves the renal and internal carotid arteries and rarely involves the mesenteric artery. Mesenteric artery FMD is difficult to distinguish from Crohn’s disease (CD) and Behcet’s disease (BD) solely based on symptoms. Patients with mesenteric artery FMD can present with an acute abdomen, but case reports of patients who have a long medical history and undergo multiple bowel resections are extremely rare. CASE PRESENTATION: The patient was a 45-year-old woman with an 11-year history of intermittent lower abdominal pain and fever. At the age of 34 years, she underwent right hemicolectomy and appendectomy due to an acute abdomen. She suffered from oral ulcers between 34 and 36 years old. A clinical diagnosis of presumed CD was made by the age of 41, and she was treated with mesalazine; however, the effect was poor. At the age of 42, she came to our centre, and based on her atypical symptoms and examination results, we thought she had CD. Hence, she was treated with glucocorticoids for 3 years. However, when she was 45, due to steroid dependence, thalidomide tablets were added. Unfortunately, she suffered from another episode of intestinal obstruction. Therefore, she underwent enterectomy. The postoperative histopathological diagnosis was mesenteric artery FMD. She no longer underwent pharmacotherapy after the surgery. Although she did not have any of her previous symptoms and postoperative colonoscopy showed no signs of recurrence, splenomegaly and abnormal routine blood results were still present. CONCLUSIONS: Patients with mesenteric artery FMD can present with an acute abdomen. In addition, the symptoms and endoscopic manifestations of mesenteric artery FMD may appear similar to CD and BD. Hence, it is difficult to make a clear clinical diagnosis and proceed with treatment. Mesenteric artery FMD often requires surgical pathology to confirm its diagnosis. For patients who suffer from this disorder, surgery may be the best choice to improve the patient’s quality of life. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s12876-021-01702-y. BioMed Central 2021-03-22 /pmc/articles/PMC7986496/ /pubmed/33752607 http://dx.doi.org/10.1186/s12876-021-01702-y Text en © The Author(s) 2021 Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Du, Shuwen
Yang, Shanbing
Jia, Kangmei
Du, Peng
Zhang, Limin
Wang, Jiheng
Fibromuscular dysplasia of mesenteric arteries: a rare cause of multiple bowel resections—a case report and literature review
title Fibromuscular dysplasia of mesenteric arteries: a rare cause of multiple bowel resections—a case report and literature review
title_full Fibromuscular dysplasia of mesenteric arteries: a rare cause of multiple bowel resections—a case report and literature review
title_fullStr Fibromuscular dysplasia of mesenteric arteries: a rare cause of multiple bowel resections—a case report and literature review
title_full_unstemmed Fibromuscular dysplasia of mesenteric arteries: a rare cause of multiple bowel resections—a case report and literature review
title_short Fibromuscular dysplasia of mesenteric arteries: a rare cause of multiple bowel resections—a case report and literature review
title_sort fibromuscular dysplasia of mesenteric arteries: a rare cause of multiple bowel resections—a case report and literature review
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7986496/
https://www.ncbi.nlm.nih.gov/pubmed/33752607
http://dx.doi.org/10.1186/s12876-021-01702-y
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