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Retinitis pigmentosa 54(RP54)

MedGen UID:
462041
Concept ID:
C3150691
Disease or Syndrome
Synonym: RP54
 
Gene (location): PCARE (2p23.2)
 
Monarch Initiative: MONDO:0013263
OMIM®: 613428

Definition

Retinitis pigmentosa-54 (RP54) is characterized by typical signs of RP, including poor night vision and peripheral field loss, retinal bone spicule-type pigment deposits, pale optic discs, and markedly reduced or extinguished responses on electroretinography. Atypical features that have been observed include early degeneration of the cone photoreceptor system with macular abnormalities, and ring scotoma on the visual field (Collin et al., 2010). Patients may exhibit an early-onset form of cone-rod dystrophy (CORD23), with central vision loss and ring scotoma around the fovea that progresses to marked chorioretinal atrophy in the macular area (Serra et al., 2019). For a general phenotypic description and a discussion of genetic heterogeneity of retinitis pigmentosa, see 268000. For a general phenotypic description and discussion of genetic heterogeneity of cone-rod dystrophy, see CORD2 (120970). [from OMIM]

Clinical features

From HPO
Nyctalopia
MedGen UID:
10349
Concept ID:
C0028077
Disease or Syndrome
Inability to see well at night or in poor light.
Abnormal electroretinogram
MedGen UID:
96908
Concept ID:
C0476397
Finding
Any abnormality of the electrical responses of various cell types in the retina as measured by electroretinography.
Bone spicule pigmentation of the retina
MedGen UID:
323029
Concept ID:
C1836926
Finding
Pigment migration into the retina in a bone-spicule configuration (resembling the nucleated cells within the lacuna of bone).
Fundus atrophy
MedGen UID:
382226
Concept ID:
C2673929
Finding
Attenuation of retinal blood vessels
MedGen UID:
480605
Concept ID:
C3278975
Finding
Visual impairment
MedGen UID:
777085
Concept ID:
C3665347
Finding
Visual impairment (or vision impairment) is vision loss (of a person) to such a degree as to qualify as an additional support need through a significant limitation of visual capability resulting from either disease, trauma, or congenital or degenerative conditions that cannot be corrected by conventional means, such as refractive correction, medication, or surgery.
Rod-cone dystrophy
MedGen UID:
1632921
Concept ID:
C4551714
Disease or Syndrome
An inherited retinal disease subtype in which the rod photoreceptors appear to be more severely affected than the cone photoreceptors. Typical presentation is with nyctalopia (due to rod dysfunction) followed by loss of mid-peripheral field of vision, which gradually extends and leaves many patients with a small central island of vision due to the preservation of macular cones.

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  

Professional guidelines

PubMed

Kierdaszuk B, Kaliszewska M, Rusecka J, Kosińska J, Bartnik E, Tońska K, Kamińska AM, Kostera-Pruszczyk A
Genes (Basel) 2020 Dec 31;12(1) doi: 10.3390/genes12010054. PMID: 33396418Free PMC Article
Shah M, Shanks M, Packham E, Williams J, Haysmoore J, MacLaren RE, Németh AH, Clouston P, Downes SM
Ophthalmic Genet 2020 Aug;41(4):331-337. Epub 2020 Jun 16 doi: 10.1080/13816810.2020.1778736. PMID: 32543920
Xiao X, Sun W, Li S, Jia X, Zhang Q
Mol Vis 2019;25:821-833. Epub 2019 Dec 2 PMID: 31908400Free PMC Article

Recent clinical studies

Etiology

Antropoli A, Arrigo A, Bianco L, Cavallari E, Bandello F, Battaglia Parodi M
Invest Ophthalmol Vis Sci 2023 Sep 1;64(12):8. doi: 10.1167/iovs.64.12.8. PMID: 37669062Free PMC Article
Griffith J 3rd, Sioufi K, Wilbanks L, Magrath GN, Say EAT, Lyons MJ, Wilkes M, Pai GS, Peterseim MMW
Genes (Basel) 2022 Aug 20;13(8) doi: 10.3390/genes13081490. PMID: 36011402Free PMC Article
Marques JP, Neves E, Geada S, Carvalho AL, Murta J, Saraiva J, Silva R
Graefes Arch Clin Exp Ophthalmol 2022 Sep;260(9):2859-2866. Epub 2022 Apr 7 doi: 10.1007/s00417-022-05649-y. PMID: 35389060
Karali M, Testa F, Brunetti-Pierri R, Di Iorio V, Pizzo M, Melillo P, Barillari MR, Torella A, Musacchia F, D'Angelo L, Banfi S, Simonelli F
Int J Mol Sci 2019 Dec 20;21(1) doi: 10.3390/ijms21010086. PMID: 31877679Free PMC Article
Lin R, Shen M, Pan D, Xu SZ, Shen RJ, Shao Y, Shi C, Lu F, Jin ZB
Invest Ophthalmol Vis Sci 2019 Nov 1;60(14):4520-4531. doi: 10.1167/iovs.19-27114. PMID: 31675423

Diagnosis

Antropoli A, Arrigo A, Bianco L, Cavallari E, Bandello F, Battaglia Parodi M
Invest Ophthalmol Vis Sci 2023 Sep 1;64(12):8. doi: 10.1167/iovs.64.12.8. PMID: 37669062Free PMC Article
Wang DD, Gao FJ, Hu FY, Cao WJ, Xu P, Huang Y, Sun XH, Wu JH
Curr Eye Res 2022 Sep;47(9):1339-1345. Epub 2022 Aug 3 doi: 10.1080/02713683.2022.2085303. PMID: 35924323
Holtan JP, Selmer KK, Heimdal KR, Bragadóttir R
Acta Ophthalmol 2020 May;98(3):286-295. Epub 2019 Aug 19 doi: 10.1111/aos.14218. PMID: 31429209
Oishi A, Oishi M, Ogino K, Morooka S, Yoshimura N
Adv Exp Med Biol 2016;854:307-13. doi: 10.1007/978-3-319-17121-0_41. PMID: 26427426
Allard RE
J Am Optom Assoc 1983 Sep;54(9):793-800. PMID: 6604745

Therapy

Leung EH, Levie-Sprick A, Sharma S, Lee GD, Cho H, Yee DC, Mukkamala K
Ophthalmic Surg Lasers Imaging Retina 2023 Jul;54(7):388-394. Epub 2023 Jun 1 doi: 10.3928/23258160-20230522-02. PMID: 37310751
Miyata M, Oishi A, Oishi M, Hasegawa T, Ikeda HO, Tsujikawa A
BMC Ophthalmol 2018 Sep 14;18(1):248. doi: 10.1186/s12886-018-0914-z. PMID: 30217183Free PMC Article
Zong Y, Lin L, Yi C, Huang X, Fu Y, Dong Y, Qian X, Li Y, Gao Q
Graefes Arch Clin Exp Ophthalmol 2016 Feb;254(2):243-52. Epub 2015 May 8 doi: 10.1007/s00417-015-3039-6. PMID: 25952041
Lemos Reis RF, Moreira-Gonçalves N, Estrela Silva SE, Brandão EM, Falcão-Reis FM
Ophthalmologica 2015;233(1):43-50. Epub 2014 Nov 26 doi: 10.1159/000368052. PMID: 25428176
Cai CX, Locke KG, Ramachandran R, Birch DG, Hood DC
Invest Ophthalmol Vis Sci 2014 Oct 23;55(11):7417-22. doi: 10.1167/iovs.14-15013. PMID: 25342618Free PMC Article

Prognosis

Markova TG, Lalayants MR, Alekseeva NN, Ryzhkova OP, Shatokhina OL, Galeeva NM, Bliznetz EA, Weener ME, Belov OA, Chibisova SS, Polyakov AV, Tavartkiladze GA
Int J Pediatr Otorhinolaryngol 2022 Jun;157:111140. Epub 2022 Apr 15 doi: 10.1016/j.ijporl.2022.111140. PMID: 35452909
Marques JP, Neves E, Geada S, Carvalho AL, Murta J, Saraiva J, Silva R
Graefes Arch Clin Exp Ophthalmol 2022 Sep;260(9):2859-2866. Epub 2022 Apr 7 doi: 10.1007/s00417-022-05649-y. PMID: 35389060
Ma DJ, Lee HS, Kim K, Choi S, Jang I, Cho SH, Yoon CK, Lee EK, Yu HG
BMC Med Genomics 2021 Mar 10;14(1):74. doi: 10.1186/s12920-021-00874-6. PMID: 33691693Free PMC Article
Alnawaiseh M, Schubert F, Heiduschka P, Eter N
Retina 2019 Jan;39(1):210-217. doi: 10.1097/IAE.000000000000190. PMID: 30570620
Bittner AK, Haythornthwaite JA, Patel C, Smith MT
Optom Vis Sci 2018 Sep;95(9):837-843. doi: 10.1097/OPX.0000000000001265. PMID: 30169358Free PMC Article

Clinical prediction guides

Antropoli A, Arrigo A, Bianco L, Cavallari E, Bandello F, Battaglia Parodi M
Invest Ophthalmol Vis Sci 2023 Sep 1;64(12):8. doi: 10.1167/iovs.64.12.8. PMID: 37669062Free PMC Article
Ma DJ, Lee HS, Kim K, Choi S, Jang I, Cho SH, Yoon CK, Lee EK, Yu HG
BMC Med Genomics 2021 Mar 10;14(1):74. doi: 10.1186/s12920-021-00874-6. PMID: 33691693Free PMC Article
Karali M, Testa F, Brunetti-Pierri R, Di Iorio V, Pizzo M, Melillo P, Barillari MR, Torella A, Musacchia F, D'Angelo L, Banfi S, Simonelli F
Int J Mol Sci 2019 Dec 20;21(1) doi: 10.3390/ijms21010086. PMID: 31877679Free PMC Article
Bittner AK, Haythornthwaite JA, Patel C, Smith MT
Optom Vis Sci 2018 Sep;95(9):837-843. doi: 10.1097/OPX.0000000000001265. PMID: 30169358Free PMC Article
Allard RE
J Am Optom Assoc 1983 Sep;54(9):793-800. PMID: 6604745

Recent systematic reviews

Lee BJH, Tham YC, Tan TE, Bylstra Y, Lim WK, Jain K, Chan CM, Mathur R, Cheung CMG, Fenner BJ
Ophthalmic Genet 2023 Apr;44(2):109-118. Epub 2023 Mar 1 doi: 10.1080/13816810.2023.2182329. PMID: 36856324

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