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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...
Autores principales: | , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Frontiers Media S.A.
2022
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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. |
format | Online Article Text |
id | pubmed-9160658 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
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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