MEK inhibitors for neurofibromatosis type 1 manifestations: Clinical evidence and consensus Article Swipe
Peter de Blank
,
Andrea M. Gross
,
Srivandana Akshintala
,
Jaishri O. Blakeley
,
Gideon Bollag
,
Ashley Cannon
,
Eva Dombi
,
Jason Fangusaro
,
Bruce D. Gelb
,
Darren Hargrave
,
AeRang Kim
,
Laura J. Klesse
,
Mignon L. Loh
,
Staci Martin
,
Christopher L. Moertel
,
Roger J. Packer
,
Jonathan M. Payne
,
Katherine A. Rauen
,
Jonathan J. Rios
,
Nathan Robison
,
Elizabeth K. Schorry
,
Kevin Shannon
,
David A. Stevenson
,
Elliot Stieglitz
,
Nicole J. Ullrich
,
Karin S. Walsh
,
Brian Weiss
,
Pamela L. Wolters
,
Kaleb Yohay
,
Marielle E. Yohe
,
Brigitte C. Widemann
,
Michael J. Fisher
·
YOU?
·
· 2022
· Open Access
·
· DOI: https://doi.org/10.1093/neuonc/noac165
YOU?
·
· 2022
· Open Access
·
· DOI: https://doi.org/10.1093/neuonc/noac165
The wide variety of clinical manifestations of the genetic syndrome neurofibromatosis type 1 (NF1) are driven by overactivation of the RAS pathway. Mitogen-activated protein kinase kinase inhibitors (MEKi) block downstream targets of RAS. The recent regulatory approvals of the MEKi selumetinib for inoperable symptomatic plexiform neurofibromas in children with NF1 have made it the first medical therapy approved for this indication in the United States, the European Union, and elsewhere. Several recently published and ongoing clinical trials have demonstrated that MEKi may have potential benefits for a variety of other NF1 manifestations, and there is broad interest in the field regarding the appropriate clinical use of these agents. In this review, we present the current evidence regarding the use of existing MEKi for a variety of NF1-related manifestations, including tumor (neurofibromas, malignant peripheral nerve sheath tumors, low-grade glioma, and juvenile myelomonocytic leukemia) and non-tumor (bone, pain, and neurocognitive) manifestations. We discuss the potential utility of MEKi in related genetic conditions characterized by overactivation of the RAS pathway (RASopathies). In addition, we review practical treatment considerations for the use of MEKi as well as provide consensus recommendations regarding their clinical use from a panel of experts.
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Metadata
- Type
- review
- Language
- en
- Landing Page
- https://doi.org/10.1093/neuonc/noac165
- https://academic.oup.com/neuro-oncology/advance-article-pdf/doi/10.1093/neuonc/noac165/45048084/noac165.pdf
- OA Status
- bronze
- Cited By
- 81
- References
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- Related Works
- 10
- OpenAlex ID
- https://openalex.org/W4283827625
All OpenAlex metadata
Raw OpenAlex JSON
- OpenAlex ID
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https://openalex.org/W4283827625Canonical identifier for this work in OpenAlex
- DOI
-
https://doi.org/10.1093/neuonc/noac165Digital Object Identifier
- Title
-
MEK inhibitors for neurofibromatosis type 1 manifestations: Clinical evidence and consensusWork title
- Type
-
reviewOpenAlex work type
- Language
-
enPrimary language
- Publication year
-
2022Year of publication
- Publication date
-
2022-07-05Full publication date if available
- Authors
-
Peter de Blank, Andrea M. Gross, Srivandana Akshintala, Jaishri O. Blakeley, Gideon Bollag, Ashley Cannon, Eva Dombi, Jason Fangusaro, Bruce D. Gelb, Darren Hargrave, AeRang Kim, Laura J. Klesse, Mignon L. Loh, Staci Martin, Christopher L. Moertel, Roger J. Packer, Jonathan M. Payne, Katherine A. Rauen, Jonathan J. Rios, Nathan Robison, Elizabeth K. Schorry, Kevin Shannon, David A. Stevenson, Elliot Stieglitz, Nicole J. Ullrich, Karin S. Walsh, Brian Weiss, Pamela L. Wolters, Kaleb Yohay, Marielle E. Yohe, Brigitte C. Widemann, Michael J. FisherList of authors in order
- Landing page
-
https://doi.org/10.1093/neuonc/noac165Publisher landing page
- PDF URL
-
https://academic.oup.com/neuro-oncology/advance-article-pdf/doi/10.1093/neuonc/noac165/45048084/noac165.pdfDirect link to full text PDF
- Open access
-
YesWhether a free full text is available
- OA status
-
bronzeOpen access status per OpenAlex
- OA URL
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https://academic.oup.com/neuro-oncology/advance-article-pdf/doi/10.1093/neuonc/noac165/45048084/noac165.pdfDirect OA link when available
- Concepts
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Medicine, Neurofibromatosis, Selumetinib, Neurocognitive, Clinical trial, Neurofibromin 1, Oncology, Bioinformatics, Internal medicine, Pathology, KRAS, Cancer, Psychiatry, Cognition, Colorectal cancer, BiologyTop concepts (fields/topics) attached by OpenAlex
- Cited by
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81Total citation count in OpenAlex
- Citations by year (recent)
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2025: 25, 2024: 26, 2023: 26, 2022: 4Per-year citation counts (last 5 years)
- References (count)
-
110Number of works referenced by this work
- Related works (count)
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10Other works algorithmically related by OpenAlex
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