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Vitelliform macular dystrophy 5(VMD5)

MedGen UID:
863780
Concept ID:
C4015343
Disease or Syndrome
Synonyms: MACULAR DYSTROPHY, VITELLIFORM, 5; VMD5
 
Gene (location): IMPG2 (3q12.3)
 
Monarch Initiative: MONDO:0014509
OMIM®: 616152

Definition

Macular dystrophies are inherited retinal dystrophies in which various forms of deposits, pigmentary changes, and atrophic lesions are observed in the macula lutea, the cone-rich region of the central retina. Vitelliform macular dystrophies (VMDs) form a subset of macular dystrophies characterized by round yellow deposits, usually at the center of the macula and containing lipofuscin, a chemically heterogeneous pigment visualized by autofluorescence imaging of the fundus (summary by Manes et al., 2013). Vitelliform macular dystrophy-5 (VMD5) is characterized by late-onset moderate visual impairment, preservation of retinal pigment epithelium (RPE) reflectivity, deposits above the RPE between the ellipsoid and outer segment interdigitation lines on spectral-domain optical coherence tomography (SD-OCT), and normal or borderline results on electrooculopathy (EOG) (Meunier et al., 2014). Brandl et al. (2017) examined patients with IMPG2- and IMPG1 (602870)-associated VMD (see VMD4; 616151) and observed strikingly similar phenotypic characteristics. They noted that retinal lesions progressed in consecutive stages, with the initial development of a single vitelliform lesion in the central macula, with detachment of the neurosensory retina and hyperreflective material located above the seemingly preserved Bruch membrane/RPE seen on SD-OCT. Next, resorption of the hyperreflective material occurs, leaving behind a dome-shaped, optically empty cavity; alternatively, the foveal cavity formed by retinal detachment may become successively filled with material. Finally, there is collapse of the cavity with central retinal atrophy and loss of RPE, resulting in the most pronounced loss of visual acuity. The authors also noted that symptoms tended to be more severe in patients with IMPG1 mutations. For a discussion of genetic heterogeneity of vitelliform macular dystrophy, see VMD1 (153840). [from OMIM]

Clinical features

From HPO
Central scotoma
MedGen UID:
57750
Concept ID:
C0152191
Finding
An area of depressed vision located at the point of fixation and that interferes with central vision.
Reduced visual acuity
MedGen UID:
65889
Concept ID:
C0234632
Finding
Diminished clarity of vision.
Macular dystrophy
MedGen UID:
196451
Concept ID:
C0730292
Disease or Syndrome
Macular dystrophy is a nonspecific term for premature retinal cell aging and cell death, generally confied to the macula in which no clear extrinsic cause is evident.
Moderately reduced visual acuity
MedGen UID:
723196
Concept ID:
C1301510
Finding
Moderate reduction of the ability to see. On the 6m visual acuity scale, moderate reduction is defined as less than 6/18 but at least 6/60. On the 20ft visual acuity scale, moderate reduction is defined as less than 20/70 but at least 20/200. On the decimal visual acuity scale, moderate reduction is defined as less than 0.3 but at least 0.1.
Vitelliform-like macular lesions
MedGen UID:
892486
Concept ID:
C4024817
Finding
Vitelliform maculopathy is a sharply demarcated lesion caused by the accumulation of material, often lipofuscin in the subretinal space underlying the macula.

Professional guidelines

PubMed

Bitner H, Schatz P, Mizrahi-Meissonnier L, Sharon D, Rosenberg T
Am J Ophthalmol 2012 Aug;154(2):403-412.e4. Epub 2012 May 24 doi: 10.1016/j.ajo.2012.02.036. PMID: 22633354
Booij JC, Boon CJ, van Schooneveld MJ, ten Brink JB, Bakker A, de Jong PT, Hoyng CB, Bergen AA, Klaver CC
Ophthalmology 2010 Jul;117(7):1415-22. Epub 2010 Apr 9 doi: 10.1016/j.ophtha.2009.11.044. PMID: 20381869
Wabbels B, Preising MN, Kretschmann U, Demmler A, Lorenz B
Graefes Arch Clin Exp Ophthalmol 2006 Nov;244(11):1453-66. Epub 2006 Apr 13 doi: 10.1007/s00417-006-0286-6. PMID: 16612637

Recent clinical studies

Etiology

Laich Y, Georgiou M, Fujinami K, Daich Varela M, Fujinami-Yokokawa Y, Hashem SA, Cabral de Guimaraes TA, Mahroo OA, Webster AR, Michaelides M
Ophthalmology 2024 Jul;131(7):845-854. Epub 2024 Jan 24 doi: 10.1016/j.ophtha.2024.01.027. PMID: 38278445
Wightman AJ, Guymer RH
Clin Exp Optom 2019 Sep;102(5):455-462. Epub 2018 Oct 8 doi: 10.1111/cxo.12842. PMID: 30298528
Parodi MB, Romano F, Sacconi R, Casati S, Marchini G, Bandello F, Iacono P
Retina 2018 Dec;38(12):2379-2386. doi: 10.1097/IAE.0000000000001893. PMID: 29065010
Battaglia Parodi M, Castellino N, Iacono P, Chowers I, Empeslidis T, Goldstein M, Bandello F
Retina 2018 Apr;38(4):841-848. doi: 10.1097/IAE.0000000000001600. PMID: 28301340
Battaglia Parodi M, Iacono P, Romano F, Bolognesi G, Fasce F, Bandello F
Eur J Ophthalmol 2017 Mar 10;27(2):201-204. Epub 2017 Feb 22 doi: 10.5301/ejo.5000878. PMID: 28233888

Diagnosis

Laich Y, Georgiou M, Fujinami K, Daich Varela M, Fujinami-Yokokawa Y, Hashem SA, Cabral de Guimaraes TA, Mahroo OA, Webster AR, Michaelides M
Ophthalmology 2024 Jul;131(7):845-854. Epub 2024 Jan 24 doi: 10.1016/j.ophtha.2024.01.027. PMID: 38278445
Šulavíková Z, Krásnik V
Cesk Slov Oftalmol 2023 Fall;79(5):268-272. doi: 10.31348/2023/31. PMID: 37993275
Cideciyan AV, Jacobson SG, Swider M, Sumaroka A, Sheplock R, Krishnan AK, Garafalo AV, Guziewicz KE, Aguirre GD, Beltran WA, Heon E
Invest Ophthalmol Vis Sci 2022 Dec 1;63(13):12. doi: 10.1167/iovs.63.13.12. PMID: 36512348Free PMC Article
Parodi MB, Romano F, Sacconi R, Casati S, Marchini G, Bandello F, Iacono P
Retina 2018 Dec;38(12):2379-2386. doi: 10.1097/IAE.0000000000001893. PMID: 29065010
Battaglia Parodi M, Castellino N, Iacono P, Chowers I, Empeslidis T, Goldstein M, Bandello F
Retina 2018 Apr;38(4):841-848. doi: 10.1097/IAE.0000000000001600. PMID: 28301340

Therapy

Jain N, Liao A, Garg SJ, Patel SN, Wykoff CC, Yu HJ, London NJS, Khurana RN, Zacks DN; Macula Society Pentosan Polysulfate Maculopathy Study Group
Ophthalmol Retina 2022 Mar;6(3):219-227. Epub 2021 Jul 21 doi: 10.1016/j.oret.2021.07.004. PMID: 34298229
Coleman DJ, Lee W, Chang S, Silverman RH, Lloyd HO, Daly S, Tsang SH
Ophthalmologica 2018;240(1):45-54. Epub 2018 Apr 25 doi: 10.1159/000486105. PMID: 29694963Free PMC Article
Padhi TR, Anderson BJ, Abbey AM, Yonekawa Y, Stem M, Alam D, Modi RR, Savla LP, Trese MT, Capone A, Drenser KA, Besirli CG
Br J Ophthalmol 2018 Sep;102(9):1232-1237. Epub 2017 Dec 7 doi: 10.1136/bjophthalmol-2017-310497. PMID: 29217515
Ozkaya A, Garip R, Nur Tarakcioglu H, Alkin Z, Taskapili M
J Fr Ophtalmol 2018 Jan;41(1):21-29. Epub 2017 Nov 28 doi: 10.1016/j.jfo.2017.06.009. PMID: 29195727
Iannaccone A, Kerr NC, Kinnick TR, Calzada JI, Stone EM
Arch Ophthalmol 2011 Feb;129(2):211-7. doi: 10.1001/archophthalmol.2010.367. PMID: 21320969

Prognosis

Laich Y, Georgiou M, Fujinami K, Daich Varela M, Fujinami-Yokokawa Y, Hashem SA, Cabral de Guimaraes TA, Mahroo OA, Webster AR, Michaelides M
Ophthalmology 2024 Jul;131(7):845-854. Epub 2024 Jan 24 doi: 10.1016/j.ophtha.2024.01.027. PMID: 38278445
Vázquez-Domínguez I, Li CHZ, Fadaie Z, Haer-Wigman L, Cremers FPM, Garanto A, Hoyng CB, Roosing S
Invest Ophthalmol Vis Sci 2022 May 2;63(5):27. doi: 10.1167/iovs.63.5.27. PMID: 35608844Free PMC Article
Romano F, Arrigo A, Leone PP, Bandello F, Battaglia Parodi M
Retina 2021 May 1;41(5):1010-1017. doi: 10.1097/IAE.0000000000002977. PMID: 32925562
Parodi MB, Romano F, Sacconi R, Casati S, Marchini G, Bandello F, Iacono P
Retina 2018 Dec;38(12):2379-2386. doi: 10.1097/IAE.0000000000001893. PMID: 29065010
Parodi MB, Iacono P, Campa C, Del Turco C, Bandello F
Am J Ophthalmol 2014 Nov;158(5):1086-92. Epub 2014 Jul 25 doi: 10.1016/j.ajo.2014.07.026. PMID: 25068640

Clinical prediction guides

Heath Jeffery RC, Thompson JA, Lo J, Chelva ES, Armstrong S, Pulido JS, Procopio R, Vincent AL, Bianco L, Battaglia Parodi M, Ziccardi L, Antonelli G, Barbano L, Marques JP, Geada S, Carvalho AL, Tang WC, Chan CM, Boon CJF, Hensman J, Chen TC, Lin CY, Chen PL, Vincent A, Tumber A, Heon E, Grigg JR, Jamieson RV, Cornish EE, Nash BM, Borooah S, Ayton LN, Britten-Jones AC, Edwards TL, Ruddle JB, Sharma A, Porter RG, Lamey TM, McLaren TL, McLenachan S, Roshandel D, Chen FK
Invest Ophthalmol Vis Sci 2024 May 1;65(5):22. doi: 10.1167/iovs.65.5.22. PMID: 38743414Free PMC Article
Laich Y, Georgiou M, Fujinami K, Daich Varela M, Fujinami-Yokokawa Y, Hashem SA, Cabral de Guimaraes TA, Mahroo OA, Webster AR, Michaelides M
Ophthalmology 2024 Jul;131(7):845-854. Epub 2024 Jan 24 doi: 10.1016/j.ophtha.2024.01.027. PMID: 38278445
Romano F, Arrigo A, Leone PP, Bandello F, Battaglia Parodi M
Retina 2021 May 1;41(5):1010-1017. doi: 10.1097/IAE.0000000000002977. PMID: 32925562
Shin DH, Kong M, Han G, Han JC, Ham DI
Sci Rep 2020 Jul 10;10(1):11469. doi: 10.1038/s41598-020-68493-2. PMID: 32651454Free PMC Article
Scoles D, Sulai YN, Cooper RF, Higgins BP, Johnson RD, Carroll J, Dubra A, Stepien KE
Retina 2017 Apr;37(4):741-748. doi: 10.1097/IAE.0000000000001203. PMID: 27467379Free PMC Article

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