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Trientine induced colitis during therapy for Wilson disease: a case report and review of the literature
BACKGROUND: Wilson disease (WD) is an autosomal recessive disorder of human copper metabolism characterized by copper accumulation in the liver due to impaired excretion of copper into the bile. Brain accumulation of copper may cause neuropsychiatric symptoms. Trientine (triethylenetetramine dihydro...
Autores principales: | , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
BioMed Central
2015
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4654910/ https://www.ncbi.nlm.nih.gov/pubmed/26589720 http://dx.doi.org/10.1186/s40360-015-0031-z |
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author | Boga, Salih Jain, Dhanpat Schilsky, Michael L. |
author_facet | Boga, Salih Jain, Dhanpat Schilsky, Michael L. |
author_sort | Boga, Salih |
collection | PubMed |
description | BACKGROUND: Wilson disease (WD) is an autosomal recessive disorder of human copper metabolism characterized by copper accumulation in the liver due to impaired excretion of copper into the bile. Brain accumulation of copper may cause neuropsychiatric symptoms. Trientine (triethylenetetramine dihydrochloride) is a copper-chelating agent used to treat patients with WD. Trientine has been considered an option for initial treatment and maintentance therapy of WD due to its safety profile. CASE PRESENTATION: A 40 year old female with a recent diagnosis of WD was started on treatment with trientine for her WD. Within one month she developed profound bloody diarrhea unresponsive to medical treatment. Trientine was discontinued and a colonoscopy with biopsy showed moderately active ileitis and moderate to severe pancolitis, consistent with a drug induced mucosal injury. The colitis improved immediately upon withdrawal of trientine, and recurred when medication was rechallenged because of worsened WD symptoms. After second compulsory discontinuation of trientine, she remained on zinc therapy for her WD and her colitis resolved by time. CONCLUSION: Drug induced colitis is a very rare side effect of trientine. Although trientine therapy is well tolerated and less side effects are reported with this medication than penicillamine, colitis can occur during trientine treatment. Zinc therapy may be an effective alternative for treatment of WD in patients experiencing side effects from chelation therapy. |
format | Online Article Text |
id | pubmed-4654910 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2015 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-46549102015-11-22 Trientine induced colitis during therapy for Wilson disease: a case report and review of the literature Boga, Salih Jain, Dhanpat Schilsky, Michael L. BMC Pharmacol Toxicol Case Report BACKGROUND: Wilson disease (WD) is an autosomal recessive disorder of human copper metabolism characterized by copper accumulation in the liver due to impaired excretion of copper into the bile. Brain accumulation of copper may cause neuropsychiatric symptoms. Trientine (triethylenetetramine dihydrochloride) is a copper-chelating agent used to treat patients with WD. Trientine has been considered an option for initial treatment and maintentance therapy of WD due to its safety profile. CASE PRESENTATION: A 40 year old female with a recent diagnosis of WD was started on treatment with trientine for her WD. Within one month she developed profound bloody diarrhea unresponsive to medical treatment. Trientine was discontinued and a colonoscopy with biopsy showed moderately active ileitis and moderate to severe pancolitis, consistent with a drug induced mucosal injury. The colitis improved immediately upon withdrawal of trientine, and recurred when medication was rechallenged because of worsened WD symptoms. After second compulsory discontinuation of trientine, she remained on zinc therapy for her WD and her colitis resolved by time. CONCLUSION: Drug induced colitis is a very rare side effect of trientine. Although trientine therapy is well tolerated and less side effects are reported with this medication than penicillamine, colitis can occur during trientine treatment. Zinc therapy may be an effective alternative for treatment of WD in patients experiencing side effects from chelation therapy. BioMed Central 2015-11-20 /pmc/articles/PMC4654910/ /pubmed/26589720 http://dx.doi.org/10.1186/s40360-015-0031-z Text en © Boga et al. 2015 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. 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. |
spellingShingle | Case Report Boga, Salih Jain, Dhanpat Schilsky, Michael L. Trientine induced colitis during therapy for Wilson disease: a case report and review of the literature |
title | Trientine induced colitis during therapy for Wilson disease: a case report and review of the literature |
title_full | Trientine induced colitis during therapy for Wilson disease: a case report and review of the literature |
title_fullStr | Trientine induced colitis during therapy for Wilson disease: a case report and review of the literature |
title_full_unstemmed | Trientine induced colitis during therapy for Wilson disease: a case report and review of the literature |
title_short | Trientine induced colitis during therapy for Wilson disease: a case report and review of the literature |
title_sort | trientine induced colitis during therapy for wilson disease: a case report and review of the literature |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4654910/ https://www.ncbi.nlm.nih.gov/pubmed/26589720 http://dx.doi.org/10.1186/s40360-015-0031-z |
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