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Leukoencephalopathy with vanishing white matter 5(VWM5)

MedGen UID:
1830483
Concept ID:
C5779973
Disease or Syndrome
Synonyms: Leukoencephalopathy with vanishing white matter 5, with or without ovarian failure; VWM5
 
Gene (location): EIF2B5 (3q27.1)
 
Monarch Initiative: MONDO:0957873
OMIM®: 620315

Definition

Leukoencephalopathy with vanishing white matter-5 (VWM5) is a chronic and progressive autosomal recessive leukoencephalopathy characterized by neurologic deterioration usually beginning in late infancy or early childhood; however, juvenile- and adult-onset cases have been reported. Neurologic signs include cerebellar ataxia, spasticity, and relatively preserved mental abilities. The disease is chronic and progressive with, in most individuals, additional episodes of rapid deterioration following febrile infections or minor head trauma. Death occurs after a variable period of a few years to a few decades, usually following an episode of fever and coma. Magnetic resonance imaging (MRI) and magnetic resonance spectroscopy are diagnostic and show a diffuse abnormality of the cerebral white matter beginning in the presymptomatic stage, with increasing amounts of the abnormal white matter vanishing and being replaced by cerebrospinal fluid; autopsy confirms these findings (summary by Leegwater et al., 2001). Ovarian dysgenesis may be present in affected females (Fogli et al., 2003). For a discussion of genetic heterogeneity of VWM, see 603896. Cree Leukoencephalopathy An infantile leukoencephalopathy among the native Cree and Chippewayan indigenous population in Northern Quebec and Manitoba results from homozygosity for an arg195-to-his (R195H; 603945.0005) mutation in the EIF2B5 gene. These patients have disease onset between 3 and 9 months of age, with death in 100% by 21 months of age. [from OMIM]

Clinical features

From HPO
Coma
MedGen UID:
1054
Concept ID:
C0009421
Disease or Syndrome
The complete absence of wakefulness and consciousness, which is evident through a lack of response to any form of external stimuli.
Megalencephaly
MedGen UID:
65141
Concept ID:
C0221355
Congenital Abnormality
Diffuse enlargement of the entire cerebral hemispheres leading to macrocephaly (with or without overlying cortical dysplasia).
Abnormal cerebral white matter morphology
MedGen UID:
181756
Concept ID:
C0948163
Pathologic Function
An abnormality of the cerebral white matter.
Loss of ambulation
MedGen UID:
332305
Concept ID:
C1836843
Finding
Inability to walk in a person who previous had the ability to walk.
Progressive neurologic deterioration
MedGen UID:
381506
Concept ID:
C1854838
Finding
Lateral ventricle dilatation
MedGen UID:
383904
Concept ID:
C1856409
Pathologic Function
Dilated third ventricle
MedGen UID:
870494
Concept ID:
C4024941
Finding
An increase in size of the third ventricle.
Decreased CSF asialotransferrin to transferrin ratio
MedGen UID:
1054129
Concept ID:
CN377049
Finding
The concentration of asialotransferrin relative to that of transferrin in the cerebrospinal fluid (CSF) is below the lower limit of normal.

Professional guidelines

PubMed

Triplett JD, Kutlubaev MA, Kermode AG, Hardy T
Pract Neurol 2022 Jun;22(3):183-189. Epub 2022 Jan 19 doi: 10.1136/practneurol-2021-003194. PMID: 35046115
Steck AJ
J Neuroimmunol 2021 Dec 15;361:577725. Epub 2021 Sep 28 doi: 10.1016/j.jneuroim.2021.577725. PMID: 34610502
Bernard-Valnet R, Koralnik IJ, Du Pasquier R
Ann Neurol 2021 Dec;90(6):865-873. Epub 2021 Sep 7 doi: 10.1002/ana.26198. PMID: 34405435Free PMC Article

Recent clinical studies

Etiology

Bandeira GA, Lucato LT
Handb Clin Neurol 2024;204:455-486. doi: 10.1016/B978-0-323-99209-1.00006-5. PMID: 39322394
Freund B, Kotchetkov IS, Kaplan PW
J Clin Neurophysiol 2021 Sep 1;38(5):359-361. doi: 10.1097/WNP.0000000000000745. PMID: 34155178
Sarret C
Rev Neurol (Paris) 2020 Jan-Feb;176(1-2):10-19. Epub 2019 Jun 4 doi: 10.1016/j.neurol.2019.04.003. PMID: 31174885
van der Knaap MS, Schiffmann R, Mochel F, Wolf NI
Lancet Neurol 2019 Oct;18(10):962-972. Epub 2019 Jul 12 doi: 10.1016/S1474-4422(19)30143-7. PMID: 31307818
Köhler W, Curiel J, Vanderver A
Nat Rev Neurol 2018 Feb;14(2):94-105. Epub 2018 Jan 5 doi: 10.1038/nrneurol.2017.175. PMID: 29302065Free PMC Article

Diagnosis

Bandeira GA, Lucato LT
Handb Clin Neurol 2024;204:455-486. doi: 10.1016/B978-0-323-99209-1.00006-5. PMID: 39322394
Williams T, John N, Doshi A, Chataway J
Handb Clin Neurol 2024;204:399-430. doi: 10.1016/B978-0-323-99209-1.00003-X. PMID: 39322392
Renaud DL
Continuum (Minneap Minn) 2016 Apr;22(2 Dementia):559-78. doi: 10.1212/CON.0000000000000303. PMID: 27042908
Kanekar S
Radiol Clin North Am 2014 Mar;52(2):xi-xii. doi: 10.1016/j.rcl.2013.12.005. PMID: 24582349
Bateman H, Rehman A, Valeriano-Marcet J
Curr Rheumatol Rep 2009 Dec;11(6):422-9. doi: 10.1007/s11926-009-0062-9. PMID: 19922732

Therapy

Yasiry Z, Obeda MS, Batty R, Sharrack B
Pract Neurol 2019 Jun;19(3):270-271. Epub 2019 Jan 5 doi: 10.1136/practneurol-2018-002121. PMID: 30612099
Yoshimura K, Tokunaga S, Daga H, Inoue M
Intern Med 2019 Feb 15;58(4):621-622. Epub 2018 Oct 17 doi: 10.2169/internalmedicine.0961-18. PMID: 30333392Free PMC Article
McCann MR, Albuja AC, Villamar MF, Lukins DE
Arq Neuropsiquiatr 2018 Dec;76(12):858-859. doi: 10.1590/0004-282X20180123. PMID: 30698212
Chen S, Hu J, Xu L, Brandon D, Yu J, Zhang J
Mol Neurobiol 2016 Dec;53(10):6897-6909. Epub 2015 Dec 14 doi: 10.1007/s12035-015-9560-0. PMID: 26666662
Helman G, Van Haren K, Escolar ML, Vanderver A
Pediatr Clin North Am 2015 Jun;62(3):649-66. Epub 2015 Apr 8 doi: 10.1016/j.pcl.2015.03.006. PMID: 26022168Free PMC Article

Prognosis

Bandeira GA, Lucato LT
Handb Clin Neurol 2024;204:455-486. doi: 10.1016/B978-0-323-99209-1.00006-5. PMID: 39322394
Mellemkjær T, Chandra RV, Speiser L, Ulhøi BP, Simonsen CZ
Neuroradiol J 2021 Aug;34(4):373-378. Epub 2021 Jul 5 doi: 10.1177/19714009211029172. PMID: 34224249Free PMC Article
van der Knaap MS, Schiffmann R, Mochel F, Wolf NI
Lancet Neurol 2019 Oct;18(10):962-972. Epub 2019 Jul 12 doi: 10.1016/S1474-4422(19)30143-7. PMID: 31307818
Köhler W, Curiel J, Vanderver A
Nat Rev Neurol 2018 Feb;14(2):94-105. Epub 2018 Jan 5 doi: 10.1038/nrneurol.2017.175. PMID: 29302065Free PMC Article
Donmez FY, Agildere AM
Neurol Sci 2015 Sep;36(9):1603-9. Epub 2015 Apr 18 doi: 10.1007/s10072-015-2212-3. PMID: 25894844

Clinical prediction guides

van der Knaap MS, Bugiani M, Abbink TEM
Handb Clin Neurol 2024;204:77-94. doi: 10.1016/B978-0-323-99209-1.00015-6. PMID: 39322396
Dulski J, Stanley ER, Chitu V, Wszolek ZK
Neurol Neurochir Pol 2023;57(5):444-449. Epub 2023 Oct 27 doi: 10.5603/pjnns.97373. PMID: 37889001
Minor KM, Letko A, Becker D, Drögemüller M, Mandigers PJJ, Bellekom SR, Leegwater PAJ, Stassen QEM, Putschbach K, Fischer A, Flegel T, Matiasek K, Ekenstedt KJ, Furrow E, Patterson EE, Platt SR, Kelly PA, Cassidy JP, Shelton GD, Lucot K, Bannasch DL, Martineau H, Muir CF, Priestnall SL, Henke D, Oevermann A, Jagannathan V, Mickelson JR, Drögemüller C
Sci Rep 2018 Apr 11;8(1):5818. doi: 10.1038/s41598-018-23938-7. PMID: 29643404Free PMC Article
Kang SJ, Choi SH, Lee BH, Jeong Y, Hahm DS, Han IW, Cummings JL, Na DL
J Geriatr Psychiatry Neurol 2004 Mar;17(1):32-5. doi: 10.1177/089198873258818. PMID: 15018695
Hénon H
Clin Exp Hypertens 2002 Oct-Nov;24(7-8):677-86. doi: 10.1081/ceh-120015344. PMID: 12450243

Recent systematic reviews

Iso-Markku P, Kujala UM, Knittle K, Polet J, Vuoksimaa E, Waller K
Br J Sports Med 2022 Jun;56(12):701-709. Epub 2022 Mar 17 doi: 10.1136/bjsports-2021-104981. PMID: 35301183Free PMC Article
Mühlbauer V, Möhler R, Dichter MN, Zuidema SU, Köpke S, Luijendijk HJ
Cochrane Database Syst Rev 2021 Dec 17;12(12):CD013304. doi: 10.1002/14651858.CD013304.pub2. PMID: 34918337Free PMC Article
Bosnjak Kuharic D, Markovic D, Brkovic T, Jeric Kegalj M, Rubic Z, Vuica Vukasovic A, Jeroncic A, Puljak L
Cochrane Database Syst Rev 2021 Sep 17;9(9):CD012820. doi: 10.1002/14651858.CD012820.pub2. PMID: 34532852Free PMC Article
Cao Q, Tan CC, Xu W, Hu H, Cao XP, Dong Q, Tan L, Yu JT
J Alzheimers Dis 2020;73(3):1157-1166. doi: 10.3233/JAD-191092. PMID: 31884487
Vitaliti G, Matin N, Tabatabaie O, Di Traglia M, Pavone P, Lubrano R, Falsaperla R
Expert Rev Neurother 2015;15(11):1321-41. Epub 2015 Oct 29 doi: 10.1586/14737175.2015.1102061. PMID: 26513633

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