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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...

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Autores principales: Boga, Salih, Jain, Dhanpat, Schilsky, Michael L.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2015
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.
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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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