Dose-dependent seizure control with MEK inhibitor therapy for progressive glioma in a child with neurofibromatosis type 1
Background Low-grade gliomas (LGGs) occurring in children can result in many different neurologic complications, including seizures. MEK inhibitors are increasingly being used to treat LGG, but their effect on associated neurologic symptoms has not been established. Results Here, we report a patient...
Saved in:
| Published in | Child's nervous system Vol. 38; no. 11; pp. 2245 - 2249 |
|---|---|
| Main Authors | , , , , |
| Format | Journal Article |
| Language | English |
| Published |
Berlin/Heidelberg
Springer Berlin Heidelberg
01.11.2022
|
| Subjects | |
| Online Access | Get full text |
Cover
Loading…
| Abstract | Background
Low-grade gliomas (LGGs) occurring in children can result in many different neurologic complications, including seizures. MEK inhibitors are increasingly being used to treat LGG, but their effect on associated neurologic symptoms has not been established.
Results
Here, we report a patient with neurofibromatosis type 1 (NF1), medically refractory epilepsy (MRE), and an extensive optic pathway glioma (OPG) who developed dose-dependent seizure control while being treated with selumetinib. Seizure frequency rebounded after dose reduction for cardiac toxicity, then improved, and finally ceased after restarting full dosing, allowing confidence in the cause of improvement.
Conclusion
Selumetinib may have promise in epilepsy management in other children with NF1 or LGG. |
|---|---|
| AbstractList | BACKGROUNDLow-grade gliomas (LGGs) occurring in children can result in many different neurologic complications, including seizures. MEK inhibitors are increasingly being used to treat LGG, but their effect on associated neurologic symptoms has not been established. RESULTSHere, we report a patient with neurofibromatosis type 1 (NF1), medically refractory epilepsy (MRE), and an extensive optic pathway glioma (OPG) who developed dose-dependent seizure control while being treated with selumetinib. Seizure frequency rebounded after dose reduction for cardiac toxicity, then improved, and finally ceased after restarting full dosing, allowing confidence in the cause of improvement. CONCLUSIONSelumetinib may have promise in epilepsy management in other children with NF1 or LGG. Abstract Background Low-grade gliomas (LGGs) occurring in children can result in many different neurologic complications, including seizures. MEK inhibitors are increasingly being used to treat LGG, but their effect on associated neurologic symptoms has not been established. Results Here, we report a patient with neurofibromatosis type 1 (NF1), medically refractory epilepsy (MRE), and an extensive optic pathway glioma (OPG) who developed dose-dependent seizure control while being treated with selumetinib. Seizure frequency rebounded after dose reduction for cardiac toxicity, then improved, and finally ceased after restarting full dosing, allowing confidence in the cause of improvement. Conclusion Selumetinib may have promise in epilepsy management in other children with NF1 or LGG. Background Low-grade gliomas (LGGs) occurring in children can result in many different neurologic complications, including seizures. MEK inhibitors are increasingly being used to treat LGG, but their effect on associated neurologic symptoms has not been established. Results Here, we report a patient with neurofibromatosis type 1 (NF1), medically refractory epilepsy (MRE), and an extensive optic pathway glioma (OPG) who developed dose-dependent seizure control while being treated with selumetinib. Seizure frequency rebounded after dose reduction for cardiac toxicity, then improved, and finally ceased after restarting full dosing, allowing confidence in the cause of improvement. Conclusion Selumetinib may have promise in epilepsy management in other children with NF1 or LGG. Low-grade gliomas (LGGs) occurring in children can result in many different neurologic complications, including seizures. MEK inhibitors are increasingly being used to treat LGG, but their effect on associated neurologic symptoms has not been established. Here, we report a patient with neurofibromatosis type 1 (NF1), medically refractory epilepsy (MRE), and an extensive optic pathway glioma (OPG) who developed dose-dependent seizure control while being treated with selumetinib. Seizure frequency rebounded after dose reduction for cardiac toxicity, then improved, and finally ceased after restarting full dosing, allowing confidence in the cause of improvement. Selumetinib may have promise in epilepsy management in other children with NF1 or LGG. |
| Author | Cantor, Evan Weisenberg, Judith L. Z. Meyer, Ashley Brossier, Nicole M. Morris, Stephanie M. |
| Author_xml | – sequence: 1 givenname: Evan surname: Cantor fullname: Cantor, Evan organization: Department of Pediatrics, Washington University School of Medicine – sequence: 2 givenname: Ashley surname: Meyer fullname: Meyer, Ashley organization: Department of Pediatrics, Washington University School of Medicine – sequence: 3 givenname: Stephanie M. surname: Morris fullname: Morris, Stephanie M. organization: Department of Neurology, Washington University School of Medicine – sequence: 4 givenname: Judith L. Z. surname: Weisenberg fullname: Weisenberg, Judith L. Z. organization: Department of Neurology, Washington University School of Medicine – sequence: 5 givenname: Nicole M. orcidid: 0000-0002-4197-6283 surname: Brossier fullname: Brossier, Nicole M. email: nbrossier@wustl.edu organization: Department of Pediatrics, Washington University School of Medicine |
| BackLink | https://www.ncbi.nlm.nih.gov/pubmed/35648241$$D View this record in MEDLINE/PubMed |
| BookMark | eNp9kc1u1TAQhS1URG8LL8ACeckmZSa287NBQqUURBEbWFuOM7nXVa4d7KQoPD2GlAo2rMbS-c7x2OeMnfjgibHnCBcIUL9KAKLBAsqyAKVqLNZHbIdSiAKEghO2g1JVRQ0STtlZSrcAqJqyfcJOhapkU0rcsfVtSFT0NJHvyc88kfuxROI2-DmGkX9384F_uvrInT-4zs0h8vlA0UwrH_J5imEfKSV3R3w_unA0GeSG24Mb-83saYlhcF3M4hySS3xeJ-L4lD0ezJjo2f08Z1_fXX25fF_cfL7-cPnmprBSVnNhJA2IBiozKCRBddv2sgKUvRhEi0LVeZbW9o1pVWcqsNTVEoRF1WPXGXHOXm-509Idqbf5ldGMeoruaOKqg3H6X8W7g96HO91WWDfY5oCX9wExfFsozfrokqVxNJ7CknRZ1aXAtpEyo-WG2hhSijQ8XIOgf3Wmt8507kz_7kyv2fTi7wUfLH9KyoDYgJQlv6eob8MSff60_8X-BFznqCk |
| CitedBy_id | crossref_primary_10_1002_pbc_30296 crossref_primary_10_1007_s40278_023_35855_3 crossref_primary_10_1016_j_eplepsyres_2024_107336 crossref_primary_10_1016_j_nbd_2023_106074 crossref_primary_10_1002_ajmg_a_63428 crossref_primary_10_1002_epd2_20180 |
| Cites_doi | 10.1016/j.eplepsyres.2022.106890 10.2217/cns-2018-0007 10.1111/ejn.13652 10.1016/j.neuroscience.2008.07.010 10.1136/adc.67.2.178 10.1016/S0022-3476(95)70159-1 10.1146/annurev.genom.6.080604.162305 10.1111/epi.12348 10.1111/jnc.12982 10.1093/neuonc/noab047 10.1212/WNL.0000000000009458 10.1016/j.pharep.2018.02.021 10.1212/WNL.0000000000003881 10.1038/s41591-018-0172-x 10.1007/s00381-020-04710-7 10.1111/j.1468-1331.2010.03151.x 10.1093/cercor/bhaa413 10.1111/neup.12242 10.1038/ng.2646 10.1016/S1474-4422(07)70103-5 10.1038/sj.emboj.7601911 10.1016/S1470-2045(19)30277-3 10.3390/brainsci10050311 |
| ContentType | Journal Article |
| Copyright | The Author(s) 2022 2022. The Author(s). |
| Copyright_xml | – notice: The Author(s) 2022 – notice: 2022. The Author(s). |
| DBID | C6C NPM AAYXX CITATION 7X8 5PM |
| DOI | 10.1007/s00381-022-05571-y |
| DatabaseName | Springer Nature OA Free Journals PubMed CrossRef MEDLINE - Academic PubMed Central (Full Participant titles) |
| DatabaseTitle | PubMed CrossRef MEDLINE - Academic |
| DatabaseTitleList | MEDLINE - Academic CrossRef PubMed |
| Database_xml | – sequence: 1 dbid: C6C name: Springer Nature OA Free Journals url: http://www.springeropen.com/ sourceTypes: Publisher – sequence: 2 dbid: NPM name: PubMed url: https://proxy.k.utb.cz/login?url=http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed sourceTypes: Index Database |
| DeliveryMethod | fulltext_linktorsrc |
| Discipline | Medicine |
| EISSN | 1433-0350 |
| EndPage | 2249 |
| ExternalDocumentID | 10_1007_s00381_022_05571_y 35648241 |
| Genre | Journal Article |
| GrantInformation_xml | – fundername: Hyundai Hope On Wheels grantid: DR-2019-672 funderid: http://dx.doi.org/10.13039/100008258 – fundername: Alex's Lemonade Stand Foundation for Childhood Cancer grantid: 18-12558 funderid: http://dx.doi.org/10.13039/100001445 – fundername: Eunice Kennedy Shriver National Institute of Child Health and Human Development grantid: K12HD076244 funderid: http://dx.doi.org/10.13039/100009633 – fundername: Neurofibromatosis Therapeutic Acceleration Pro grantid: 210112 funderid: http://dx.doi.org/10.13039/100014804 – fundername: Neurofibromatosis Therapeutic Acceleration Pro grantid: 210112 – fundername: Alex's Lemonade Stand Foundation for Childhood Cancer grantid: 18-12558 – fundername: Eunice Kennedy Shriver National Institute of Child Health and Human Development grantid: K12HD076244 – fundername: Hyundai Hope On Wheels grantid: DR-2019-672 – fundername: ; grantid: 210112 – fundername: ; grantid: K12HD076244 – fundername: ; grantid: 18-12558 – fundername: ; grantid: DR-2019-672 |
| GroupedDBID | --- -53 -5E -5G -BR -EM -Y2 -~C .86 .GJ .VR 06C 06D 0R~ 0VY 1N0 1SB 2.D 203 29B 29~ 2J2 2JN 2JY 2KG 2KM 2LR 2P1 2VQ 2~H 30V 4.4 406 408 409 40D 40E 53G 5QI 5RE 5VS 67Z 6NX 78A 8TC 8UJ 95- 95. 95~ 96X AAAVM AABHQ AABYN AAFGU AAHNG AAIAL AAJKR AAKSU AANXM AANZL AAPBV AARHV AARTL AATNV AATVU AAUYE AAWCG AAYFA AAYIU AAYQN AAYTO ABBBX ABBXA ABDZT ABECU ABFGW ABFTV ABHLI ABHQN ABIPD ABJNI ABJOX ABKAS ABKCH ABKTR ABMNI ABMQK ABNWP ABPLI ABPTK ABQBU ABSXP ABTEG ABTKH ABTMW ABULA ABUWZ ABWNU ABXPI ACBMV ACBRV ACBXY ACBYP ACGFS ACHSB ACHXU ACIGE ACIPQ ACKNC ACMDZ ACMLO ACOKC ACOMO ACTTH ACUDM ACVWB ACWMK ADHHG ADHIR ADIMF ADINQ ADJJI ADKNI ADKPE ADMDM ADOXG ADRFC ADTPH ADURQ ADYFF ADZKW AEBTG AEEQQ AEFIE AEFTE AEGAL AEGNC AEJHL AEJRE AEKMD AENEX AEOHA AEPYU AESKC AESTI AETLH AEVLU AEVTX AEXYK AFAFS AFEXP AFFNX AFLOW AFNRJ AFQWF AFWTZ AFZKB AGAYW AGDGC AGGBP AGGDS AGJBK AGKHE AGMZJ AGQMX AGWIL AGWZB AGYKE AHAVH AHBYD AHIZS AHKAY AHSBF AHYZX AIAKS AIIXL AILAN AIMYW AITGF AJBLW AJDOV AJRNO AJZVZ AKMHD AKQUC ALMA_UNASSIGNED_HOLDINGS ALWAN AMKLP AMXSW AMYLF AMYQR AOCGG ARMRJ ASPBG AVWKF AXYYD AZFZN B-. BA0 BDATZ BGNMA C6C CAG COF CSCUP DDRTE DL5 DNIVK DPUIP DU5 EBD EBLON EBS EIOEI EJD EMB EMOBN EN4 ESBYG F5P FEDTE FERAY FFXSO FIGPU FINBP FNLPD FRRFC FSGXE FWDCC G-Y G-Z GGCAI GGRSB GJIRD GNWQR GQ6 GQ7 GQ8 GRRUI GXS HF~ HG5 HG6 HMJXF HQYDN HRMNR HVGLF HZ~ I09 IHE IJ- IKXTQ IMOTQ IWAJR IXC IXD IXE IZIGR IZQ I~X I~Z J-C J0Z JBSCW JCJTX JZLTJ KDC KOV KOW KPH LAS LLZTM M4Y MA- N2Q N9A NB0 NDZJH NPVJJ NQJWS NU0 O9- O93 O9G O9I O9J OAM OVD P19 P2P P9S PF0 PT4 PT5 QOK QOR QOS R4E R89 R9I RHV RIG RNI ROL RPX RRX RSV RZK S16 S1Z S26 S27 S28 S37 S3B SAP SCLPG SDE SDH SDM SHX SISQX SJYHP SMD SNE SNPRN SNX SOHCF SOJ SPISZ SRMVM SSLCW SSXJD STPWE SV3 SZ9 SZN T13 T16 TEORI TSG TSK TSV TT1 TUC U2A U9L UG4 UNUBA UOJIU UTJUX UZXMN VC2 VFIZW W23 W48 WJK WK8 YLTOR Z45 Z5O Z7U Z81 Z82 Z86 Z87 Z8O Z8U Z8V Z91 ZGI ZMTXR ZOVNA ~EX AACDK AAJBT AASML AAYZH ABAKF ACAOD ACDTI ACZOJ AEFQL AEMSY AFBBN AGQEE AGRTI AIGIU NPM AAEOY AAYXX AJOOF CITATION H13 7X8 5PM |
| ID | FETCH-LOGICAL-c446t-a4ef11a06af51e3e799d46014d3f3913573f32ccd8a95ba60ceb7403c15d1bba3 |
| IEDL.DBID | C6C |
| ISSN | 0256-7040 |
| IngestDate | Tue Sep 17 21:31:03 EDT 2024 Sat Aug 17 04:22:14 EDT 2024 Thu Sep 12 16:42:04 EDT 2024 Wed Oct 16 00:40:56 EDT 2024 Sat Dec 16 12:06:13 EST 2023 |
| IsDoiOpenAccess | true |
| IsOpenAccess | true |
| IsPeerReviewed | true |
| IsScholarly | true |
| Issue | 11 |
| Keywords | NF1 Seizure MEK inhibitor Selumetinib Epilepsy Neurofibromatosis |
| Language | English |
| License | 2022. The Author(s). Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. |
| LinkModel | DirectLink |
| MergedId | FETCHMERGED-LOGICAL-c446t-a4ef11a06af51e3e799d46014d3f3913573f32ccd8a95ba60ceb7403c15d1bba3 |
| Notes | ObjectType-Case Study-2 SourceType-Scholarly Journals-1 ObjectType-Feature-4 content type line 23 ObjectType-Report-1 ObjectType-Article-3 |
| ORCID | 0000-0002-4197-6283 |
| OpenAccessLink | https://doi.org/10.1007/s00381-022-05571-y |
| PMID | 35648241 |
| PQID | 2672319844 |
| PQPubID | 23479 |
| PageCount | 5 |
| ParticipantIDs | pubmedcentral_primary_oai_pubmedcentral_nih_gov_9617819 proquest_miscellaneous_2672319844 crossref_primary_10_1007_s00381_022_05571_y pubmed_primary_35648241 springer_journals_10_1007_s00381_022_05571_y |
| PublicationCentury | 2000 |
| PublicationDate | 2022-11-01 |
| PublicationDateYYYYMMDD | 2022-11-01 |
| PublicationDate_xml | – month: 11 year: 2022 text: 2022-11-01 day: 01 |
| PublicationDecade | 2020 |
| PublicationPlace | Berlin/Heidelberg |
| PublicationPlace_xml | – name: Berlin/Heidelberg – name: Germany |
| PublicationTitle | Child's nervous system |
| PublicationTitleAbbrev | Childs Nerv Syst |
| PublicationTitleAlternate | Childs Nerv Syst |
| PublicationYear | 2022 |
| Publisher | Springer Berlin Heidelberg |
| Publisher_xml | – name: Springer Berlin Heidelberg |
| References | Mahdi, Shah, Sato, Morris, McKinstry, Listernick, Packer, Fisher, Gutmann (CR6) 2017; 88 Tartaglia, Gelb (CR9) 2005; 6 Holter, Hewitt, Nishimura, Knowles, Bjorklund, Shah, Fry, Rees, Gupta, Daniels, Li, Marsh, Treiman, Olive, Anderson, Sanabria, Snider, Newbern (CR20) 2021; 31 Houser, Huang, Peng (CR16) 2008; 156 CR18 Soffietti, Baumert, Bello, Von Deimling, Duffau, Frénay, Grisold, Grant, Graus, Hoang-Xuan, Klein, Melin, Rees, Siegal, Smits, Stupp, Wick (CR8) 2010; 17 Bernardo, Cinalli, Santoro (CR4) 2020; 36 Patil, Guzman, Carter, Rathore, Yoshor, Curry, Wilfong, Agadi, Swann, Adesina, Bhattacharjee, Anderson (CR10) 2016; 36 Fangusaro, Onar-Thomas, Poussaint, Wu, Ligon, Lindeman, Campagne, Banerjee, Gururangan, Kilburn, Goldman, Qaddoumi, Baxter, Vezina, Bregman, Patay, Jones, Stewart, Fisher, Doyle, Smith, Dunkel, Fouladi (CR21) 2021; 23 Nguyen, Leiser, Song, Brunner, Roberds, Wong, Bordey (CR11) 2022; 181 Nateri, Raivich, Gebhardt, Da Costa, Naumann, Vreugdenhil, Makwana, Brandner, Adams, Jefferys, Kann, Behrens (CR19) 2007; 26 Chukwueke, Wen (CR23) 2019; 8 Koh, Kim, Jang, Kim, Han, Lim, Son, Choi, Park, Heo, Han, Lee, Lee, Kang, Shong, Paik, Kim, Lee (CR2) 2018; 24 Mahdi, Goyal, Griffith, Morris, Gutmann (CR7) 2020; 95 Ostendorf, Gutmann, Weisenberg (CR3) 2013; 54 Kopanitsa, Gou, Afinowi, Bayés, Grant, Komiyama (CR12) 2018; 70 Fangusaro, Onar-Thomas, Young Poussaint, Wu, Ligon, Lindeman, Banerjee, Packer, Kilburn, Goldman, Pollack, Qaddoumi, Jakacki, Fisher, Dhall, Baxter, Kreissman, Stewart, Jones, Pfister, Vezina, Stern, Panigrahy, Patay, Tamrazi, Jones, Haque, Enterline, Cha, Fisher, Doyle, Smith, Dunkel, Fouladi (CR22) 2019; 20 van Breemen, Wilms, Vecht (CR1) 2007; 6 Listernick, Darling, Greenwald, Strauss, Charrow (CR5) 1995; 127 Carvill, Heavin, Yendle, McMahon, O'Roak, Cook, Khan, Dorschner, Weaver, Calvert, Malone, Wallace, Stanley, Bye, Bleasel, Howell, Kivity, Mackay, Rodriguez-Casero, Webster, Korczyn, Afawi, Zelnick, Lerman-Sagie, Lev, Møller, Gill, Andrade, Freeman, Sadleir, Shendure, Berkovic, Scheffer, Mefford (CR13) 2013; 45 Salman, Sheilabi, Bhattacharyya, Kitchen, Conner, Bill, Woodroofe, Conner, Princivalle (CR15) 2017; 46 Sharland, Burch, McKenna, Paton (CR14) 1992; 67 Glazova, Nikitina, Hudik, Kirillova, Dorofeeva, Korotkov, Chernigovskaya (CR17) 2015; 132 VV Patil (5571_CR10) 2016; 36 J Fangusaro (5571_CR22) 2019; 20 R Listernick (5571_CR5) 1995; 127 M Tartaglia (5571_CR9) 2005; 6 M Sharland (5571_CR14) 1992; 67 MM Salman (5571_CR15) 2017; 46 MC Holter (5571_CR20) 2021; 31 MV Glazova (5571_CR17) 2015; 132 P Bernardo (5571_CR4) 2020; 36 5571_CR18 MS van Breemen (5571_CR1) 2007; 6 MV Kopanitsa (5571_CR12) 2018; 70 UN Chukwueke (5571_CR23) 2019; 8 J Mahdi (5571_CR7) 2020; 95 GL Carvill (5571_CR13) 2013; 45 AS Nateri (5571_CR19) 2007; 26 J Mahdi (5571_CR6) 2017; 88 CR Houser (5571_CR16) 2008; 156 LH Nguyen (5571_CR11) 2022; 181 R Soffietti (5571_CR8) 2010; 17 AP Ostendorf (5571_CR3) 2013; 54 J Fangusaro (5571_CR21) 2021; 23 HY Koh (5571_CR2) 2018; 24 |
| References_xml | – volume: 181 start-page: 106890 year: 2022 ident: CR11 article-title: Inhibition of MEK-ERK signaling reduces seizures in two mouse models of tuberous sclerosis complex publication-title: Epilepsy Res doi: 10.1016/j.eplepsyres.2022.106890 contributor: fullname: Bordey – ident: CR18 – volume: 8 start-page: Cns28 year: 2019 ident: CR23 article-title: Use of the Response Assessment in Neuro-Oncology (RANO) criteria in clinical trials and clinical practice publication-title: CNS Oncol doi: 10.2217/cns-2018-0007 contributor: fullname: Wen – volume: 46 start-page: 2121 year: 2017 end-page: 2132 ident: CR15 article-title: Transcriptome analysis suggests a role for the differential expression of cerebral aquaporins and the MAPK signalling pathway in human temporal lobe epilepsy publication-title: Eur J Neurosci doi: 10.1111/ejn.13652 contributor: fullname: Princivalle – volume: 156 start-page: 222 year: 2008 end-page: 237 ident: CR16 article-title: Dynamic seizure-related changes in extracellular signal-regulated kinase activation in a mouse model of temporal lobe epilepsy publication-title: Neuroscience doi: 10.1016/j.neuroscience.2008.07.010 contributor: fullname: Peng – volume: 67 start-page: 178 year: 1992 end-page: 183 ident: CR14 article-title: A clinical study of Noonan syndrome publication-title: Arch Dis Child doi: 10.1136/adc.67.2.178 contributor: fullname: Paton – volume: 127 start-page: 718 year: 1995 end-page: 722 ident: CR5 article-title: Optic pathway tumors in children: the effect of neurofibromatosis type 1 on clinical manifestations and natural history publication-title: J Pediatr doi: 10.1016/S0022-3476(95)70159-1 contributor: fullname: Charrow – volume: 6 start-page: 45 year: 2005 end-page: 68 ident: CR9 article-title: Noonan syndrome and related disorders: genetics and pathogenesis publication-title: Annu Rev Genomics Hum Genet doi: 10.1146/annurev.genom.6.080604.162305 contributor: fullname: Gelb – volume: 54 start-page: 1810 year: 2013 end-page: 1814 ident: CR3 article-title: Epilepsy in individuals with neurofibromatosis type 1 publication-title: Epilepsia doi: 10.1111/epi.12348 contributor: fullname: Weisenberg – volume: 132 start-page: 218 year: 2015 end-page: 229 ident: CR17 article-title: Inhibition of ERK1/2 signaling prevents epileptiform behavior in rats prone to audiogenic seizures publication-title: J Neurochem doi: 10.1111/jnc.12982 contributor: fullname: Chernigovskaya – volume: 23 start-page: 1777 year: 2021 end-page: 1788 ident: CR21 article-title: A phase II trial of selumetinib in children with recurrent optic pathway and hypothalamic low-grade glioma without NF1: a Pediatric Brain Tumor Consortium study publication-title: Neuro Oncol doi: 10.1093/neuonc/noab047 contributor: fullname: Fouladi – volume: 95 start-page: e1052 year: 2020 end-page: e1059 ident: CR7 article-title: Nonoptic pathway tumors in children with neurofibromatosis type 1 publication-title: Neurology doi: 10.1212/WNL.0000000000009458 contributor: fullname: Gutmann – volume: 70 start-page: 777 year: 2018 end-page: 783 ident: CR12 article-title: Chronic treatment with a MEK inhibitor reverses enhanced excitatory field potentials in Syngap1(+/-) mice publication-title: Pharmacol Rep doi: 10.1016/j.pharep.2018.02.021 contributor: fullname: Komiyama – volume: 88 start-page: 1584 year: 2017 end-page: 1589 ident: CR6 article-title: A multi-institutional study of brainstem gliomas in children with neurofibromatosis type 1 publication-title: Neurology doi: 10.1212/WNL.0000000000003881 contributor: fullname: Gutmann – volume: 24 start-page: 1662 year: 2018 end-page: 1668 ident: CR2 article-title: BRAF somatic mutation contributes to intrinsic epileptogenicity in pediatric brain tumors publication-title: Nat Med doi: 10.1038/s41591-018-0172-x contributor: fullname: Lee – volume: 36 start-page: 2333 year: 2020 end-page: 2350 ident: CR4 article-title: Epilepsy in NF1: a systematic review of the literature publication-title: Childs Nerv Syst doi: 10.1007/s00381-020-04710-7 contributor: fullname: Santoro – volume: 17 start-page: 1124 year: 2010 end-page: 1133 ident: CR8 article-title: Guidelines on management of low-grade gliomas: report of an EFNS–EANO* Task Force publication-title: Eur J Neurol doi: 10.1111/j.1468-1331.2010.03151.x contributor: fullname: Wick – volume: 31 start-page: 3064 year: 2021 end-page: 3081 ident: CR20 article-title: Hyperactive MEK1 signaling in cortical GABAergic neurons promotes embryonic parvalbumin neuron loss and defects in behavioral inhibition publication-title: Cereb Cortex doi: 10.1093/cercor/bhaa413 contributor: fullname: Newbern – volume: 36 start-page: 146 year: 2016 end-page: 156 ident: CR10 article-title: Activation of extracellular regulated kinase and mechanistic target of rapamycin pathway in focal cortical dysplasia publication-title: Neuropathology doi: 10.1111/neup.12242 contributor: fullname: Anderson – volume: 45 start-page: 825 year: 2013 end-page: 830 ident: CR13 article-title: Targeted resequencing in epileptic encephalopathies identifies de novo mutations in CHD2 and SYNGAP1 publication-title: Nat Genet doi: 10.1038/ng.2646 contributor: fullname: Mefford – volume: 6 start-page: 421 year: 2007 end-page: 430 ident: CR1 article-title: Epilepsy in patients with brain tumours: epidemiology, mechanisms, and management publication-title: Lancet Neurol doi: 10.1016/S1474-4422(07)70103-5 contributor: fullname: Vecht – volume: 26 start-page: 4891 year: 2007 end-page: 4901 ident: CR19 article-title: ERK activation causes epilepsy by stimulating NMDA receptor activity publication-title: EMBO J doi: 10.1038/sj.emboj.7601911 contributor: fullname: Behrens – volume: 20 start-page: 1011 year: 2019 end-page: 1022 ident: CR22 article-title: Selumetinib in paediatric patients with BRAF-aberrant or neurofibromatosis type 1-associated recurrent, refractory, or progressive low-grade glioma: a multicentre, phase 2 trial publication-title: Lancet Oncol doi: 10.1016/S1470-2045(19)30277-3 contributor: fullname: Fouladi – volume: 36 start-page: 2333 year: 2020 ident: 5571_CR4 publication-title: Childs Nerv Syst doi: 10.1007/s00381-020-04710-7 contributor: fullname: P Bernardo – volume: 54 start-page: 1810 year: 2013 ident: 5571_CR3 publication-title: Epilepsia doi: 10.1111/epi.12348 contributor: fullname: AP Ostendorf – volume: 31 start-page: 3064 year: 2021 ident: 5571_CR20 publication-title: Cereb Cortex doi: 10.1093/cercor/bhaa413 contributor: fullname: MC Holter – volume: 24 start-page: 1662 year: 2018 ident: 5571_CR2 publication-title: Nat Med doi: 10.1038/s41591-018-0172-x contributor: fullname: HY Koh – volume: 6 start-page: 45 year: 2005 ident: 5571_CR9 publication-title: Annu Rev Genomics Hum Genet doi: 10.1146/annurev.genom.6.080604.162305 contributor: fullname: M Tartaglia – volume: 88 start-page: 1584 year: 2017 ident: 5571_CR6 publication-title: Neurology doi: 10.1212/WNL.0000000000003881 contributor: fullname: J Mahdi – volume: 132 start-page: 218 year: 2015 ident: 5571_CR17 publication-title: J Neurochem doi: 10.1111/jnc.12982 contributor: fullname: MV Glazova – volume: 127 start-page: 718 year: 1995 ident: 5571_CR5 publication-title: J Pediatr doi: 10.1016/S0022-3476(95)70159-1 contributor: fullname: R Listernick – volume: 67 start-page: 178 year: 1992 ident: 5571_CR14 publication-title: Arch Dis Child doi: 10.1136/adc.67.2.178 contributor: fullname: M Sharland – volume: 23 start-page: 1777 year: 2021 ident: 5571_CR21 publication-title: Neuro Oncol doi: 10.1093/neuonc/noab047 contributor: fullname: J Fangusaro – volume: 17 start-page: 1124 year: 2010 ident: 5571_CR8 publication-title: Eur J Neurol doi: 10.1111/j.1468-1331.2010.03151.x contributor: fullname: R Soffietti – volume: 45 start-page: 825 year: 2013 ident: 5571_CR13 publication-title: Nat Genet doi: 10.1038/ng.2646 contributor: fullname: GL Carvill – volume: 156 start-page: 222 year: 2008 ident: 5571_CR16 publication-title: Neuroscience doi: 10.1016/j.neuroscience.2008.07.010 contributor: fullname: CR Houser – volume: 8 start-page: Cns28 year: 2019 ident: 5571_CR23 publication-title: CNS Oncol doi: 10.2217/cns-2018-0007 contributor: fullname: UN Chukwueke – volume: 95 start-page: e1052 year: 2020 ident: 5571_CR7 publication-title: Neurology doi: 10.1212/WNL.0000000000009458 contributor: fullname: J Mahdi – volume: 181 start-page: 106890 year: 2022 ident: 5571_CR11 publication-title: Epilepsy Res doi: 10.1016/j.eplepsyres.2022.106890 contributor: fullname: LH Nguyen – volume: 26 start-page: 4891 year: 2007 ident: 5571_CR19 publication-title: EMBO J doi: 10.1038/sj.emboj.7601911 contributor: fullname: AS Nateri – ident: 5571_CR18 doi: 10.3390/brainsci10050311 – volume: 20 start-page: 1011 year: 2019 ident: 5571_CR22 publication-title: Lancet Oncol doi: 10.1016/S1470-2045(19)30277-3 contributor: fullname: J Fangusaro – volume: 70 start-page: 777 year: 2018 ident: 5571_CR12 publication-title: Pharmacol Rep doi: 10.1016/j.pharep.2018.02.021 contributor: fullname: MV Kopanitsa – volume: 6 start-page: 421 year: 2007 ident: 5571_CR1 publication-title: Lancet Neurol doi: 10.1016/S1474-4422(07)70103-5 contributor: fullname: MS van Breemen – volume: 36 start-page: 146 year: 2016 ident: 5571_CR10 publication-title: Neuropathology doi: 10.1111/neup.12242 contributor: fullname: VV Patil – volume: 46 start-page: 2121 year: 2017 ident: 5571_CR15 publication-title: Eur J Neurosci doi: 10.1111/ejn.13652 contributor: fullname: MM Salman |
| SSID | ssj0015829 |
| Score | 2.4185185 |
| Snippet | Background
Low-grade gliomas (LGGs) occurring in children can result in many different neurologic complications, including seizures. MEK inhibitors are... Low-grade gliomas (LGGs) occurring in children can result in many different neurologic complications, including seizures. MEK inhibitors are increasingly being... Abstract Background Low-grade gliomas (LGGs) occurring in children can result in many different neurologic complications, including seizures. MEK inhibitors... BACKGROUNDLow-grade gliomas (LGGs) occurring in children can result in many different neurologic complications, including seizures. MEK inhibitors are... |
| SourceID | pubmedcentral proquest crossref pubmed springer |
| SourceType | Open Access Repository Aggregation Database Index Database Publisher |
| StartPage | 2245 |
| SubjectTerms | Case Report Medicine Medicine & Public Health Neurosciences Neurosurgery |
| Title | Dose-dependent seizure control with MEK inhibitor therapy for progressive glioma in a child with neurofibromatosis type 1 |
| URI | https://link.springer.com/article/10.1007/s00381-022-05571-y https://www.ncbi.nlm.nih.gov/pubmed/35648241 https://search.proquest.com/docview/2672319844 https://pubmed.ncbi.nlm.nih.gov/PMC9617819 |
| Volume | 38 |
| hasFullText | 1 |
| inHoldings | 1 |
| isFullTextHit | |
| isPrint | |
| link | http://utb.summon.serialssolutions.com/2.0.0/link/0/eLvHCXMwlV3da9swED_WFkpfxj66zd0WNOjbKoisD9uPaZasrKRPDaRPRpblxbDZI3YG6V-_k-wEko7BXqwHfWD0O3F3urufAC5Fzk2i0MmJlLJUcCOoVjmnhXT0c56i3BUKz-7UzVx8W8hFT5PjamEO4veO7BN1CnU5544titHNEZxIpoZOgsdqvIsYyNi_SOZUOI1QMvsCmb-vsa-EnliWTxMkD6KkXvlMX8Dz3mokow7ml_DMVq_gdNbHxV_D5kvdWLp90LYljS0f1ytL-kR04m5byWxyS8pqWWZ4iFekK7zaEDRaiU_Scvmwvy35_qOsf2ocSDTxld7dZM97WaBvjZ1t3ZQNcZe3hJ3DfDq5H9_Q_lEFatDza6kWtmBMD5UuJLPcRkmSC_TKELSCJ4zLCNvQmDzWicy0GhqbRWLIDZM5yzLN38BxVVf2HRBtuCjQHJG5CIXR6AzhbMEtFzn6cToO4PN2l9NfHXdGumNJ9pikiEnqMUk3AXzaApGiiLu4ha5svW7SUEVohSaxEAG87YDZrcelEjFaIQFEe5DtBjj67P2eqlx6Gu3EVUeyJICrLbhpf36bf_zmxf8Nfw9noRM8X734AY7b1dp-RDOmzQZwMppeX9-59uvD7WTg5Rm_83D0B6jh7yI |
| link.rule.ids | 230,315,786,790,891,27955,27956,41114,41153,41556,42183,42222,42625,51609,52144,52267 |
| linkProvider | Springer Nature |
| linkToHtml | http://utb.summon.serialssolutions.com/2.0.0/link/0/eLvHCXMwlV1La9tAEB7aFNpcQl9plPSxhd6SBa_3IekYXAe3iXOyIbdltVo1glQKlhxwf31nV7LBTgn0pMM-WPTNMjM7M98AfBM5t6lCJydWylHBraBG5ZwW0tPPBYpyXyg8vVaTufh5I296mhxfC7MTv_dkn6hTqM8592xRjK6ewwv_bOG7FYzUaBMxkEnoSOZVOI1RMvsCmX_vsa2EHlmWjxMkd6KkQflcvIaD3mok5x3Mb-CZq97Cy2kfF38Hq-914-i6oW1LGlf-WS4c6RPRiX9tJdPxJSmr2zLDS7wgXeHViqDRSkKSls-HfXDk111Z_zY4kRgSKr27xYH3skDfGgfbuikb4h9vCXsP84vxbDShfVMFatHza6kRrmDMDJQpJHPcxWmaC_TKELSCp4zLGL9Da_PEpDIzamBdFosBt0zmLMsMP4S9qq7cERBjuSjQHJG5GApr0BnC1YI7LnL040wSwen6L-v7jjtDb1iSAyYaMdEBE72K4OsaCI0i7uMWpnL1stFDFaMVmiZCRPChA2azH5dKJGiFRBBvQbaZ4Omzt0eq8jbQaKe-OpKlEZytwdX9_W2eOObx_03_Aq8ms-mVvvpxfXkC-0MvhKGS8SPstYul-4QmTZt9DrL8F1Jo7go |
| linkToPdf | http://utb.summon.serialssolutions.com/2.0.0/link/0/eLvHCXMwlV1Lb9QwEB5BkSouVXmnLWAkbmB1HT-SHCvaVaFsxYGVerMcx6GRIKk2WaTl13fsPMRShMQpBz8U5RtrZjLzfQZ4KwpuM4VJTqKUo4JbQY0qOC2ll58LEuWeKLy4VOdL8elKXv3G4g_d7mNJsuc0eJWmuju-KcrjifgWClzUd6J7DSlGN_fhgXeN3saX8clUR5BpuKfMO3aaoL0OtJm_77Htmu7Em3fbJv-onQaXNN-HvSGWJCc9-I_gnqsfw-5iqJY_gc1p0zo6XnPbkdZVv9YrR4b2dOL_wZLF2QWp6usqx6O9Ij0da0MwlCWhdct3yf505Nv3qvlhcCIxJPC_-8VBDbPEjBsHu6atWuJ_6RL2FJbzs68fzulw1QK1mA921AhXMmZmypSSOe6SLCsE5moIZckzxmWCz9jaIjWZzI2aWZcnYsYtkwXLc8OfwU7d1O4FEGO5KDFIkYWIhTWYIuFqwR0XBWZ3Jo3g3fiV9U2vqKEn7eSAiUZMdMBEbyJ4MwKh0fB9NcPUrlm3OlYJxqZZKkQEz3tgpv24VCLF2CSCZAuyaYIX1d4eqavrIK6dec4kyyJ4P4Krh1Pd_uM1D_5v-mvY_XI6158_Xl4cwsPY22CgNx7BTrdau5cY53T5q2DKtxhS9nk |
| openUrl | ctx_ver=Z39.88-2004&ctx_enc=info%3Aofi%2Fenc%3AUTF-8&rfr_id=info%3Asid%2Fsummon.serialssolutions.com&rft_val_fmt=info%3Aofi%2Ffmt%3Akev%3Amtx%3Ajournal&rft.genre=article&rft.atitle=Dose-dependent+seizure+control+with+MEK+inhibitor+therapy+for+progressive+glioma+in+a+child+with+neurofibromatosis+type+1&rft.jtitle=Child%27s+nervous+system&rft.au=Cantor%2C+Evan&rft.au=Meyer%2C+Ashley&rft.au=Morris%2C+Stephanie+M.&rft.au=Weisenberg%2C+Judith+L.+Z.&rft.date=2022-11-01&rft.pub=Springer+Berlin+Heidelberg&rft.issn=0256-7040&rft.eissn=1433-0350&rft.volume=38&rft.issue=11&rft.spage=2245&rft.epage=2249&rft_id=info:doi/10.1007%2Fs00381-022-05571-y&rft.externalDocID=10_1007_s00381_022_05571_y |
| thumbnail_l | http://covers-cdn.summon.serialssolutions.com/index.aspx?isbn=/lc.gif&issn=0256-7040&client=summon |
| thumbnail_m | http://covers-cdn.summon.serialssolutions.com/index.aspx?isbn=/mc.gif&issn=0256-7040&client=summon |
| thumbnail_s | http://covers-cdn.summon.serialssolutions.com/index.aspx?isbn=/sc.gif&issn=0256-7040&client=summon |