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Cachexia, chorea, and pain in chronic nonbacterial osteitis and inflammatory bowel disease: a case report

BACKGROUND: Inflammatory bowel disease is an inflammatory disorder that primarily impacts the gastrointestinal tract, leading to malnutrition and chronic microscopic intestinal blood loss. Uncontrolled systemic inflammation can impact other parts of the body, known as extraintestinal manifestations....

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Autores principales: Agharokh, Ladan, Mamola, Katherine, Yu, Andrew G., Medina, Annette L., Gurram, Bhaskar, Fuller, Julie, Park, Jason Y., Chen, Weina, Rajaram, Veena, Hammer, Matthew R., Waugh, Jeff L.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10230771/
https://www.ncbi.nlm.nih.gov/pubmed/37254165
http://dx.doi.org/10.1186/s13256-023-03894-1
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author Agharokh, Ladan
Mamola, Katherine
Yu, Andrew G.
Medina, Annette L.
Gurram, Bhaskar
Fuller, Julie
Park, Jason Y.
Chen, Weina
Rajaram, Veena
Hammer, Matthew R.
Waugh, Jeff L.
author_facet Agharokh, Ladan
Mamola, Katherine
Yu, Andrew G.
Medina, Annette L.
Gurram, Bhaskar
Fuller, Julie
Park, Jason Y.
Chen, Weina
Rajaram, Veena
Hammer, Matthew R.
Waugh, Jeff L.
author_sort Agharokh, Ladan
collection PubMed
description BACKGROUND: Inflammatory bowel disease is an inflammatory disorder that primarily impacts the gastrointestinal tract, leading to malnutrition and chronic microscopic intestinal blood loss. Uncontrolled systemic inflammation can impact other parts of the body, known as extraintestinal manifestations. Up to 25% of patients with inflammatory bowel disease are reported to have these complications in their skin, joints, bones, eyes, liver, lung, and pancreas (Rogler et al. in Gastroenterology 161(4):1118–1132, 2021). Neurologic involvement as extraintestinal manifestations are less common, reported at 3–19%, including neuropathies, demyelination, and cerebrovascular events (Morís in World J Gastroenterol. 20(5):1228–1237, 2014). CASE PRESENTATION: A 13-year-old Caucasian boy presented with 1 month of progressive lower-extremity pain, weakness, and weight loss. His physical examination was notable for cachexia, lower-extremity weakness, and chorea. Labs revealed normocytic anemia and systemic inflammation. Imaging revealed symmetric abnormal marrow signal in the pelvis and upper femurs. Pathologic examination of the bone revealed chronic inflammation consistent with chronic nonbacterial osteitis. Endoscopy revealed colonic inflammation consistent with inflammatory bowel disease. CONCLUSIONS: Children and adolescents with musculoskeletal pain lasting more than 2 weeks with systemic signs or symptoms like weight loss should prompt evaluation for systemic inflammatory disorders such as chronic nonbacterial osteitis, which can occur in isolation or associated with inflammatory bowel disease. This patient also had a nonspecific neurologic abnormality, chorea, which resolved with treatment of underlying inflammatory disorder. These extraintestinal manifestations may be concurrent with or precede intestinal inflammation, requiring a high index of suspicion when investigating nonspecific systemic inflammation. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s13256-023-03894-1.
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spelling pubmed-102307712023-06-01 Cachexia, chorea, and pain in chronic nonbacterial osteitis and inflammatory bowel disease: a case report Agharokh, Ladan Mamola, Katherine Yu, Andrew G. Medina, Annette L. Gurram, Bhaskar Fuller, Julie Park, Jason Y. Chen, Weina Rajaram, Veena Hammer, Matthew R. Waugh, Jeff L. J Med Case Rep Case Report BACKGROUND: Inflammatory bowel disease is an inflammatory disorder that primarily impacts the gastrointestinal tract, leading to malnutrition and chronic microscopic intestinal blood loss. Uncontrolled systemic inflammation can impact other parts of the body, known as extraintestinal manifestations. Up to 25% of patients with inflammatory bowel disease are reported to have these complications in their skin, joints, bones, eyes, liver, lung, and pancreas (Rogler et al. in Gastroenterology 161(4):1118–1132, 2021). Neurologic involvement as extraintestinal manifestations are less common, reported at 3–19%, including neuropathies, demyelination, and cerebrovascular events (Morís in World J Gastroenterol. 20(5):1228–1237, 2014). CASE PRESENTATION: A 13-year-old Caucasian boy presented with 1 month of progressive lower-extremity pain, weakness, and weight loss. His physical examination was notable for cachexia, lower-extremity weakness, and chorea. Labs revealed normocytic anemia and systemic inflammation. Imaging revealed symmetric abnormal marrow signal in the pelvis and upper femurs. Pathologic examination of the bone revealed chronic inflammation consistent with chronic nonbacterial osteitis. Endoscopy revealed colonic inflammation consistent with inflammatory bowel disease. CONCLUSIONS: Children and adolescents with musculoskeletal pain lasting more than 2 weeks with systemic signs or symptoms like weight loss should prompt evaluation for systemic inflammatory disorders such as chronic nonbacterial osteitis, which can occur in isolation or associated with inflammatory bowel disease. This patient also had a nonspecific neurologic abnormality, chorea, which resolved with treatment of underlying inflammatory disorder. These extraintestinal manifestations may be concurrent with or precede intestinal inflammation, requiring a high index of suspicion when investigating nonspecific systemic inflammation. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s13256-023-03894-1. BioMed Central 2023-05-31 /pmc/articles/PMC10230771/ /pubmed/37254165 http://dx.doi.org/10.1186/s13256-023-03894-1 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/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/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://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
Agharokh, Ladan
Mamola, Katherine
Yu, Andrew G.
Medina, Annette L.
Gurram, Bhaskar
Fuller, Julie
Park, Jason Y.
Chen, Weina
Rajaram, Veena
Hammer, Matthew R.
Waugh, Jeff L.
Cachexia, chorea, and pain in chronic nonbacterial osteitis and inflammatory bowel disease: a case report
title Cachexia, chorea, and pain in chronic nonbacterial osteitis and inflammatory bowel disease: a case report
title_full Cachexia, chorea, and pain in chronic nonbacterial osteitis and inflammatory bowel disease: a case report
title_fullStr Cachexia, chorea, and pain in chronic nonbacterial osteitis and inflammatory bowel disease: a case report
title_full_unstemmed Cachexia, chorea, and pain in chronic nonbacterial osteitis and inflammatory bowel disease: a case report
title_short Cachexia, chorea, and pain in chronic nonbacterial osteitis and inflammatory bowel disease: a case report
title_sort cachexia, chorea, and pain in chronic nonbacterial osteitis and inflammatory bowel disease: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10230771/
https://www.ncbi.nlm.nih.gov/pubmed/37254165
http://dx.doi.org/10.1186/s13256-023-03894-1
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