Subependymal Giant Cell Astrocytomas in Tuberous Sclerosis Complex-Current Views on Their Pathogenesis and Management.
mTOR inhibitors
management
recommendations
subependymal giant cell astrocytoma (SEGA)
tuberous sclerosis complex (TSC)
Journal
Journal of clinical medicine
ISSN: 2077-0383
Titre abrégé: J Clin Med
Pays: Switzerland
ID NLM: 101606588
Informations de publication
Date de publication:
26 Jan 2023
26 Jan 2023
Historique:
received:
28
11
2022
revised:
21
01
2023
accepted:
22
01
2023
entrez:
11
2
2023
pubmed:
12
2
2023
medline:
12
2
2023
Statut:
epublish
Résumé
Introduction, Tuberous sclerosis complex (TSC) is an autosomal-dominant disorder caused by mutations inactivating TSC1 or TSC2 genes and characterized by the presence of tumors involving many organs, including the brain, heart, kidneys, and skin. Subependymal giant cell astrocytoma (SEGA) is a slow-growing brain tumor almost exclusively associated with TSC. Despite the fact that SEGAs are benign, they require well-considered decisions regarding the timing and modality of pharmacological or surgical treatment. In TSC children and adolescents, SEGA is the major cause of mortality and morbidity. Until recently, surgical resection has been the standard therapy for SEGAs but the discovery of the role of the mTOR pathway and the introduction of mTOR inhibitors to clinical practice changed the therapeutic landscape of these tumors. In the current paper, we discuss the pros and cons of mTOR inhibitors and surgical approaches in SEGA treatment. In 2021, the International Tuberous Sclerosis Complex Consensus Group proposed a new integrative strategy for SEGA management. In the following review, we discuss the proposed recommendations and report the results of the literature search for the latest treatment directions.
Identifiants
pubmed: 36769603
pii: jcm12030956
doi: 10.3390/jcm12030956
pmc: PMC9917805
pii:
doi:
Types de publication
Journal Article
Review
Langues
eng
Subventions
Organisme : National Agency of Academic Exchange
ID : PPN/BCN/2019/1/00066/U/00001
Organisme : International Cooperation Project of the Ministry of Science and Technology of China
ID : G2021026025L
Organisme : Open Project of Henan Clinical Medical Research Center of Childhood Diseases
ID : YJZX202205
Organisme : Special Key Project of Henan Province Traditional Chinese Medicine Scientific Research
ID : 20-21ZY1072
Déclaration de conflit d'intérêts
The authors declare no conflict of interest.
Références
Neurol Med Chir (Tokyo). 2002 May;42(5):224-7
pubmed: 12064158
J Eur Acad Dermatol Venereol. 2016 Apr;30(4):586-94
pubmed: 26403211
Front Neurosci. 2020 Aug 21;14:899
pubmed: 32973442
N Engl J Med. 1999 Mar 4;340(9):703-7
pubmed: 10053179
N Engl J Med. 2010 Nov 4;363(19):1801-11
pubmed: 21047224
Front Neurol. 2019 Aug 02;10:821
pubmed: 31428037
Ital J Pediatr. 2013 Sep 17;39:57
pubmed: 24044547
Pediatr Neurol. 2013 Dec;49(6):439-44
pubmed: 24138953
Orphanet J Rare Dis. 2017 Jan 5;12(1):2
pubmed: 28057044
Childs Nerv Syst. 1989 Feb;5(1):43-4
pubmed: 2702677
Childs Nerv Syst. 1995 Jun;11(6):335-9
pubmed: 7671268
Folia Neuropathol. 2011;49(1):39-46
pubmed: 21455842
Ann N Y Acad Sci. 1991;615:125-7
pubmed: 2039137
Lancet. 2008 Aug 23;372(9639):657-68
pubmed: 18722871
Surg Neurol Int. 2020 Feb 25;11:26
pubmed: 32123614
Folia Neuropathol. 2015;53(1):8-14
pubmed: 25909870
Cureus. 2020 Feb 8;12(2):e6920
pubmed: 32071825
Lancet Neurol. 2022 Sep;21(9):843-856
pubmed: 35963265
Mayo Clin Proc. 1991 Aug;66(8):792-6
pubmed: 1861550
Mod Pathol. 2021 Feb;34(2):264-279
pubmed: 33051600
Pediatr Neurol. 2014 Apr;50(4):307-12
pubmed: 24507694
Expert Opin Pharmacother. 2020 Aug;21(11):1329-1336
pubmed: 32338549
Childs Nerv Syst. 2012 Jul;28(7):963-8
pubmed: 22562196
Pediatr Neurol. 2017 Mar;68:59-63
pubmed: 28162859
Acta Neuropathol. 2007 Aug;114(2):97-109
pubmed: 17618441
Lancet. 2013 Jan 12;381(9861):125-32
pubmed: 23158522
Curr Neuropharmacol. 2012 Dec;10(4):404-15
pubmed: 23730262
J Child Neurol. 2013 May;28(5):602-7
pubmed: 22805244
Epilepsy Behav. 2021 Aug;121(Pt A):108032
pubmed: 34087679
Front Neurol. 2019 Jul 03;10:705
pubmed: 31333563
Neurol Med Chir (Tokyo). 1998 May;38(5):287-91
pubmed: 9640965
World Neurosurg. 2017 Nov;107:1053.e1-1053.e6
pubmed: 28866062
Eur J Neurol. 2009 Jun;16(6):691-6
pubmed: 19236458
J Neurol Neurosurg Psychiatry. 1999 Mar;66(3):370-5
pubmed: 10084537
PLoS One. 2016 Jun 28;11(6):e0158476
pubmed: 27351628
Eur J Paediatr Neurol. 2010 Mar;14(2):146-9
pubmed: 19369101
Oncotarget. 2017 Sep 8;8(56):95516-95529
pubmed: 29221145
Neurochirurgie. 2022 Dec;68(6):627-636
pubmed: 35907444
Childs Nerv Syst. 2014 Dec;30(12):2037-42
pubmed: 25227171
Am J Hum Genet. 2001 Jan;68(1):64-80
pubmed: 11112665
Eur J Paediatr Neurol. 2012 Jan;16(1):83-5
pubmed: 22000822
Pediatr Neurol. 2015 Apr;52(4):435-41
pubmed: 25771998
Eur J Paediatr Neurol. 2013 Jul;17(4):348-52
pubmed: 23391693
Pediatr Blood Cancer. 2015 Apr;62(4):616-21
pubmed: 25557360
Folia Neuropathol. 2010;48(3):139-49
pubmed: 20924998
MAGMA. 2017 Aug;30(4):397-405
pubmed: 28321524
Childs Nerv Syst. 2020 Oct;36(10):2527-2536
pubmed: 32978642
Brain. 2019 Jan 1;143(1):131-149
pubmed: 31834371
J Neurooncol. 2020 May;147(3):731-736
pubmed: 32285309
J Neurosurg Pediatr. 2015 Aug;16(2):134-7
pubmed: 25978531
Childs Nerv Syst. 2020 May;36(5):951-960
pubmed: 31853898
J Mol Diagn. 2005 Oct;7(4):544-9
pubmed: 16237225
Pediatr Neurol. 2021 Oct;123:50-66
pubmed: 34399110
Pediatr Res. 2021 May;89(6):1447-1451
pubmed: 32516799
Neurosurgery. 2007 Jan;60(1):83-89; discussion 89-90
pubmed: 17228255
Semin Pediatr Neurol. 2015 Dec;22(4):274-81
pubmed: 26706014
Eur J Paediatr Neurol. 2013 Sep;17(5):479-85
pubmed: 23567018
Pharmacol Rep. 2016 Jun;68(3):536-42
pubmed: 26891243
Epilepsia. 2022 Jan;63(1):6-21
pubmed: 34741464
Front Neurol. 2021 Apr 09;12:581102
pubmed: 33897576
Pediatr Neurol. 2015 Mar;52(3):281-9
pubmed: 25591831
Diabetes Res Clin Pract. 2015 Nov;110(2):101-8
pubmed: 26421362