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Case Report: Rare Systemic and Aggressive ALK-Positive Histiocytosis With Recurrent Pancreatitis Treating by Alectinib

ALK-positive histiocytosis (APH) is a rare and recently described, solitary or generalized, histiocytic proliferative disorder with a characteristic gene translocation involving the fusion of the ALK gene at chromosome 2p23. To date, only 25 cases of APH have been reported. The patient presented wit...

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Autores principales: Li, Yanchu, Shi, Changle, Wu, Yu, He, Mingmin, Xia, Xueming, Liu, Jie, Jiang, Yu
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9160658/
https://www.ncbi.nlm.nih.gov/pubmed/35665359
http://dx.doi.org/10.3389/fmed.2022.840407
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author Li, Yanchu
Shi, Changle
Wu, Yu
He, Mingmin
Xia, Xueming
Liu, Jie
Jiang, Yu
author_facet Li, Yanchu
Shi, Changle
Wu, Yu
He, Mingmin
Xia, Xueming
Liu, Jie
Jiang, Yu
author_sort Li, Yanchu
collection PubMed
description ALK-positive histiocytosis (APH) is a rare and recently described, solitary or generalized, histiocytic proliferative disorder with a characteristic gene translocation involving the fusion of the ALK gene at chromosome 2p23. To date, only 25 cases of APH have been reported. The patient presented with multiple nodules in the lung, liver, gallbladder, pancreas, kidney, and skin rashes, along with recurrent pancreatitis and cholecystitis. The histiocytes from the lesion were positive for CD68 and ALK and negative for S100 and CD1α. A reduced dose of the ALK inhibitor alectinib was administered rather than the standard dose of alectinib or chemotherapy because of recurrent pancreatitis, which has not been previously reported in APH cases. After 18 months of follow-up, the patient was maintained on alectinib, and a partial response (PR) was achieved.
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spelling pubmed-91606582022-06-03 Case Report: Rare Systemic and Aggressive ALK-Positive Histiocytosis With Recurrent Pancreatitis Treating by Alectinib Li, Yanchu Shi, Changle Wu, Yu He, Mingmin Xia, Xueming Liu, Jie Jiang, Yu Front Med (Lausanne) Medicine ALK-positive histiocytosis (APH) is a rare and recently described, solitary or generalized, histiocytic proliferative disorder with a characteristic gene translocation involving the fusion of the ALK gene at chromosome 2p23. To date, only 25 cases of APH have been reported. The patient presented with multiple nodules in the lung, liver, gallbladder, pancreas, kidney, and skin rashes, along with recurrent pancreatitis and cholecystitis. The histiocytes from the lesion were positive for CD68 and ALK and negative for S100 and CD1α. A reduced dose of the ALK inhibitor alectinib was administered rather than the standard dose of alectinib or chemotherapy because of recurrent pancreatitis, which has not been previously reported in APH cases. After 18 months of follow-up, the patient was maintained on alectinib, and a partial response (PR) was achieved. Frontiers Media S.A. 2022-05-19 /pmc/articles/PMC9160658/ /pubmed/35665359 http://dx.doi.org/10.3389/fmed.2022.840407 Text en Copyright © 2022 Li, Shi, Wu, He, Xia, Liu and Jiang. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Medicine
Li, Yanchu
Shi, Changle
Wu, Yu
He, Mingmin
Xia, Xueming
Liu, Jie
Jiang, Yu
Case Report: Rare Systemic and Aggressive ALK-Positive Histiocytosis With Recurrent Pancreatitis Treating by Alectinib
title Case Report: Rare Systemic and Aggressive ALK-Positive Histiocytosis With Recurrent Pancreatitis Treating by Alectinib
title_full Case Report: Rare Systemic and Aggressive ALK-Positive Histiocytosis With Recurrent Pancreatitis Treating by Alectinib
title_fullStr Case Report: Rare Systemic and Aggressive ALK-Positive Histiocytosis With Recurrent Pancreatitis Treating by Alectinib
title_full_unstemmed Case Report: Rare Systemic and Aggressive ALK-Positive Histiocytosis With Recurrent Pancreatitis Treating by Alectinib
title_short Case Report: Rare Systemic and Aggressive ALK-Positive Histiocytosis With Recurrent Pancreatitis Treating by Alectinib
title_sort case report: rare systemic and aggressive alk-positive histiocytosis with recurrent pancreatitis treating by alectinib
topic Medicine
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9160658/
https://www.ncbi.nlm.nih.gov/pubmed/35665359
http://dx.doi.org/10.3389/fmed.2022.840407
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