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Sirolimus in Refractory Cronkhite-Canada Syndrome and Focus on Standard Treatment.
J Investig Med High Impact Case Rep. 2018 Jan-Dec; 6:2324709618765893.JI

Abstract

Cronkhite-Canada syndrome is a rare syndrome consisting of extensive gastrointestinal polyposis and ectodermal changes including cutaneous hyperpigmentation, alopecia, and onychodystrophy. We report the case of a 45-year-old Caucasian male patient who failed multiple treatments over 2 years including steroids, azathioprine, adalimumab, and cyclosporine. He had recurrent and prolonged hospitalizations because of diarrhea, abdominal pain, weight loss, and malnutrition. Sirolimus was initiated with a significant clinical and endoscopic benefit apparent within, respectively, 2 and 8 weeks. An ongoing remission was achieved and maintained for over 6 months after prednisone tapering. We review the current evidence on treatment of Cronkhite-Canada syndrome and suggest the incorporation of sirolimus in that algorithm.

Authors+Show Affiliations

Centre Hospitalier de l'Université de Montréal, Montreal, Quebec, Canada.Centre Hospitalier de l'Université de Montréal, Montreal, Quebec, Canada. Montreal Clinical Research Institute, Montreal, Quebec, Canada.Chicoutimi Hospital, Chicoutimi, Quebec, Canada.Centre Hospitalier de l'Université de Montréal, Montreal, Quebec, Canada.Centre Hospitalier de l'Université de Montréal, Montreal, Quebec, Canada.

Pub Type(s)

Journal Article

Language

eng

PubMed ID

29619395

Citation

Langevin, Catherine, et al. "Sirolimus in Refractory Cronkhite-Canada Syndrome and Focus On Standard Treatment." Journal of Investigative Medicine High Impact Case Reports, vol. 6, 2018, p. 2324709618765893.
Langevin C, Chapdelaine H, Picard JM, et al. Sirolimus in Refractory Cronkhite-Canada Syndrome and Focus on Standard Treatment. J Investig Med High Impact Case Rep. 2018;6:2324709618765893.
Langevin, C., Chapdelaine, H., Picard, J. M., Poitras, P., & Leduc, R. (2018). Sirolimus in Refractory Cronkhite-Canada Syndrome and Focus on Standard Treatment. Journal of Investigative Medicine High Impact Case Reports, 6, 2324709618765893. https://doi.org/10.1177/2324709618765893
Langevin C, et al. Sirolimus in Refractory Cronkhite-Canada Syndrome and Focus On Standard Treatment. J Investig Med High Impact Case Rep. 2018 Jan-Dec;6:2324709618765893. PubMed PMID: 29619395.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Sirolimus in Refractory Cronkhite-Canada Syndrome and Focus on Standard Treatment. AU - Langevin,Catherine, AU - Chapdelaine,Hugo, AU - Picard,Jean-Maxime, AU - Poitras,Pierre, AU - Leduc,Raymond, Y1 - 2018/03/22/ PY - 2017/12/12/received PY - 2018/01/27/revised PY - 2018/02/03/accepted PY - 2018/4/6/entrez PY - 2018/4/6/pubmed PY - 2018/4/6/medline KW - Cronkhite-Canada syndrome KW - sirolimus KW - treatment SP - 2324709618765893 EP - 2324709618765893 JF - Journal of investigative medicine high impact case reports JO - J Investig Med High Impact Case Rep VL - 6 N2 - Cronkhite-Canada syndrome is a rare syndrome consisting of extensive gastrointestinal polyposis and ectodermal changes including cutaneous hyperpigmentation, alopecia, and onychodystrophy. We report the case of a 45-year-old Caucasian male patient who failed multiple treatments over 2 years including steroids, azathioprine, adalimumab, and cyclosporine. He had recurrent and prolonged hospitalizations because of diarrhea, abdominal pain, weight loss, and malnutrition. Sirolimus was initiated with a significant clinical and endoscopic benefit apparent within, respectively, 2 and 8 weeks. An ongoing remission was achieved and maintained for over 6 months after prednisone tapering. We review the current evidence on treatment of Cronkhite-Canada syndrome and suggest the incorporation of sirolimus in that algorithm. SN - 2324-7096 UR - https://www.unboundmedicine.com/medline/citation/29619395/Sirolimus_in_Refractory_Cronkhite_Canada_Syndrome_and_Focus_on_Standard_Treatment_ L2 - http://journals.sagepub.com/doi/full/10.1177/2324709618765893?url_ver=Z39.88-2003&rfr_id=ori:rid:crossref.org&rfr_dat=cr_pub=pubmed DB - PRIME DP - Unbound Medicine ER -
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