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Near-complete response to low-dose ceritinib in recurrent infantile inflammatory myofibroblastic tumour

BACKGROUND: Infantile inflammatory myofibroblastic tumour (IMT) is rare and the majority are driven by anaplastic lymphoma kinase (ALK) rearrangements. Previous literature on the use of ALK inhibitors in paediatric IMTs is extremely limited with no published literature on the use in infants. Crizoti...

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Autores principales: Mittal, Abhenil, Gupta, Aarushi, Rastogi, Sameer, Barwad, Adarsh, Sharma, Swati
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Cancer Intelligence 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8057779/
https://www.ncbi.nlm.nih.gov/pubmed/33912240
http://dx.doi.org/10.3332/ecancer.2021.1215
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author Mittal, Abhenil
Gupta, Aarushi
Rastogi, Sameer
Barwad, Adarsh
Sharma, Swati
author_facet Mittal, Abhenil
Gupta, Aarushi
Rastogi, Sameer
Barwad, Adarsh
Sharma, Swati
author_sort Mittal, Abhenil
collection PubMed
description BACKGROUND: Infantile inflammatory myofibroblastic tumour (IMT) is rare and the majority are driven by anaplastic lymphoma kinase (ALK) rearrangements. Previous literature on the use of ALK inhibitors in paediatric IMTs is extremely limited with no published literature on the use in infants. Crizotinib and ceritinib are two ALK inhibitors which are available and have been used in IMTs; however, ceritinib is much more affordable in the low- and middle-income country (LMIC) setting than crizotinib. CASE: An 11-month-old child, who had undergone surgery for mesenteric IMT at the age of 3 months, had an unresectable recurrence with soft tissue deposits in the subdiaphragmatic location abutting the spleen and paravesical location. As surgery would have entailed splenectomy and partial cystectomy, she was treated with low-dose ceritinib (300 mg/m2/day) with which she had a near-complete response without any toxicity. DISCUSSION AND CONCLUSION: This is the first report of the use of ceritinib at a lower dose for infantile IMT having immense practical applications for the low- and middle-income setting.
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spelling pubmed-80577792021-04-27 Near-complete response to low-dose ceritinib in recurrent infantile inflammatory myofibroblastic tumour Mittal, Abhenil Gupta, Aarushi Rastogi, Sameer Barwad, Adarsh Sharma, Swati Ecancermedicalscience Case Report BACKGROUND: Infantile inflammatory myofibroblastic tumour (IMT) is rare and the majority are driven by anaplastic lymphoma kinase (ALK) rearrangements. Previous literature on the use of ALK inhibitors in paediatric IMTs is extremely limited with no published literature on the use in infants. Crizotinib and ceritinib are two ALK inhibitors which are available and have been used in IMTs; however, ceritinib is much more affordable in the low- and middle-income country (LMIC) setting than crizotinib. CASE: An 11-month-old child, who had undergone surgery for mesenteric IMT at the age of 3 months, had an unresectable recurrence with soft tissue deposits in the subdiaphragmatic location abutting the spleen and paravesical location. As surgery would have entailed splenectomy and partial cystectomy, she was treated with low-dose ceritinib (300 mg/m2/day) with which she had a near-complete response without any toxicity. DISCUSSION AND CONCLUSION: This is the first report of the use of ceritinib at a lower dose for infantile IMT having immense practical applications for the low- and middle-income setting. Cancer Intelligence 2021-04-01 /pmc/articles/PMC8057779/ /pubmed/33912240 http://dx.doi.org/10.3332/ecancer.2021.1215 Text en © the authors; licensee ecancermedicalscience. https://creativecommons.org/licenses/by/3.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0 (https://creativecommons.org/licenses/by/3.0/) ), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Mittal, Abhenil
Gupta, Aarushi
Rastogi, Sameer
Barwad, Adarsh
Sharma, Swati
Near-complete response to low-dose ceritinib in recurrent infantile inflammatory myofibroblastic tumour
title Near-complete response to low-dose ceritinib in recurrent infantile inflammatory myofibroblastic tumour
title_full Near-complete response to low-dose ceritinib in recurrent infantile inflammatory myofibroblastic tumour
title_fullStr Near-complete response to low-dose ceritinib in recurrent infantile inflammatory myofibroblastic tumour
title_full_unstemmed Near-complete response to low-dose ceritinib in recurrent infantile inflammatory myofibroblastic tumour
title_short Near-complete response to low-dose ceritinib in recurrent infantile inflammatory myofibroblastic tumour
title_sort near-complete response to low-dose ceritinib in recurrent infantile inflammatory myofibroblastic tumour
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8057779/
https://www.ncbi.nlm.nih.gov/pubmed/33912240
http://dx.doi.org/10.3332/ecancer.2021.1215
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