Raras
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Síndrome Usher
ORPHA:886CID-10 · H35.5CID-11 · LD2H.4DOENÇA RARA

É uma síndrome (um conjunto de características médicas) que combina surdez neurossensorial (um tipo de surdez que afeta o ouvido interno ou o nervo da audição, geralmente presente desde o nascimento) com retinite pigmentosa (uma doença que atinge os olhos) e perda progressiva da visão.

Mantido por Agente Raras·Colaborar como especialista →

Introdução

O que você precisa saber de cara

📋

É uma síndrome (um conjunto de características médicas) que combina surdez neurossensorial (um tipo de surdez que afeta o ouvido interno ou o nervo da audição, geralmente presente desde o nascimento) com retinite pigmentosa (uma doença que atinge os olhos) e perda progressiva da visão.

Pesquisas ativas
13 ensaios
35 total registrados no ClinicalTrials.gov
Publicações científicas
1.499 artigos
Último publicado: 2026 Apr 14

Escala de raridade

CLASSIFICAÇÃO ORPHANET · BRASIL 2024
1-9 / 100 000
Ultra-rara
<1/50k
Muito rara
1/20k
Rara
1/10k
Pouco freq.
1/5k
Incomum
1/2k
Prevalência
4.53
Worldwide
Início
Childhood
+ infancy, neonatal
🏥
SUS: Sem cobertura SUSScore: 0%
CID-10: H35.5
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Entender a doença

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Sinais e sintomas

O que aparece no corpo e com que frequência cada sintoma acontece

Partes do corpo afetadas

👁️
Olhos
24 sintomas
👂
Ouvidos
11 sintomas
🧠
Neurológico
7 sintomas
🦷
Dentes
3 sintomas
🦴
Ossos e articulações
2 sintomas
❤️
Coração
2 sintomas

+ 22 sintomas em outras categorias

Características mais comuns

90%prev.
Perda visual progressiva
Muito frequente (99-80%)
90%prev.
Areflexia vestibular
Muito frequente (99-80%)
90%prev.
Eletroretinograma anormal
Muito frequente (99-80%)
90%prev.
Nictalopia
Muito frequente (99-80%)
90%prev.
Função vestibular anormal
Muito frequente (99-80%)
90%prev.
Defeito do campo visual
Muito frequente (99-80%)
73sintomas
Muito frequente (10)
Frequente (4)
Ocasional (23)
Sem dados (36)

Os sintomas variam de pessoa para pessoa. Abaixo estão as 73 características clínicas mais associadas, ordenadas por frequência.

Perda visual progressivaProgressive visual loss
Muito frequente (99-80%)90%
Areflexia vestibularVestibular areflexia
Muito frequente (99-80%)90%
Eletroretinograma anormalAbnormal electroretinogram
Muito frequente (99-80%)90%
NictalopiaNyctalopia
Muito frequente (99-80%)90%
Função vestibular anormalAbnormal vestibular function
Muito frequente (99-80%)90%

Linha do tempo da pesquisa

Publicações por ano — veja quando o interesse científico cresceu
Anos de pesquisa1desde 2026
Total histórico1.499PubMed
Últimos 10 anos200publicações
Pico202594 papers
Linha do tempo
2026Hoje · 2026🧪 1993Primeiro ensaio clínico📈 2025Ano de pico
Publicações por ano (últimos 10 anos)

Encontrou um erro ou informação desatualizada? Sugira uma correção →

Genética e causas

O que está alterado no DNA e como passa nas famílias

Genes associados

16 genes identificados com associação a esta condição. Padrão de herança: Autosomal recessive.

HARS1Histidine--tRNA ligase, cytoplasmicDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Catalyzes the ATP-dependent ligation of histidine to the 3'-end of its cognate tRNA, via the formation of an aminoacyl-adenylate intermediate (His-AMP) (PubMed:29235198). Plays a role in axon guidance (PubMed:26072516)

LOCALIZAÇÃO

Cytoplasm

VIAS BIOLÓGICAS (1)
Cytosolic tRNA aminoacylation
MECANISMO DE DOENÇA

Usher syndrome 3B

A syndrome characterized by progressive vision and hearing loss during early childhood. Some patients have the so-called 'Charles Bonnet syndrome,' involving decreased visual acuity and vivid visual hallucinations. USH is a genetically heterogeneous condition characterized by the association of retinitis pigmentosa with sensorineural deafness. Age at onset and differences in auditory and vestibular function distinguish Usher syndrome type 1 (USH1), Usher syndrome type 2 (USH2) and Usher syndrome type 3 (USH3). USH3 is characterized by postlingual, progressive hearing loss, variable vestibular dysfunction, and onset of retinitis pigmentosa symptoms, including nyctalopia, constriction of the visual fields, and loss of central visual acuity, usually by the second decade of life.

OUTRAS DOENÇAS (3)
Usher syndrome type 3Bautosomal dominant Charcot-Marie-Tooth disease type 2WUsher syndrome type 3
HGNC:4816UniProt:P12081
ESPNEspinCandidate gene tested inTolerante
FUNÇÃO

Multifunctional actin-bundling protein. Plays a major role in regulating the organization, dimension, dynamics and signaling capacities of the actin filament-rich microvilli in the mechanosensory and chemosensory cells (PubMed:29572253). Required for the assembly and stabilization of the stereociliary parallel actin bundles. Plays a crucial role in the formation and maintenance of inner ear hair cell stereocilia (By similarity). Involved in the elongation of actin in stereocilia (PubMed:29572253

LOCALIZAÇÃO

Cytoplasm, cytoskeletonCell projection, stereociliumCell projection, microvillus

VIAS BIOLÓGICAS (2)
Sensory processing of sound by outer hair cells of the cochleaSensory processing of sound by inner hair cells of the cochlea
MECANISMO DE DOENÇA

Deafness, autosomal recessive, 36, with or without vestibular involvement

A form of non-syndromic sensorineural hearing loss. Sensorineural deafness results from damage to the neural receptors of the inner ear, the nerve pathways to the brain, or the area of the brain that receives sound information. DFNB36 is characterized by prelingual, profound hearing loss, and vestibular areflexia in some patients.

EXPRESSÃO TECIDUAL(Tecido-específico)
Testículo
79.6 TPM
Skin Not Sun Exposed Suprapubic
28.7 TPM
Skin Sun Exposed Lower leg
24.1 TPM
Fígado
19.1 TPM
Pituitária
18.6 TPM
OUTRAS DOENÇAS (4)
autosomal recessive nonsyndromic hearing loss 36Usher syndrome, type 1MUsher syndrome type 1hearing loss, autosomal recessive
HGNC:13281UniProt:B1AK53
MT-TS2Candidate gene tested inDesconhecido
LOCALIZAÇÃO

VIAS BIOLÓGICAS (3)
G alpha (i) signalling eventsFormyl peptide receptors bind formyl peptides and many other ligandsG alpha (q) signalling events
OUTRAS DOENÇAS (4)
mitochondrial diseaseMERRF syndromeUsher syndrome type 3MELAS syndrome
HGNC:7498
CIB2Calcium and integrin-binding family member 2Candidate gene tested inTolerante
FUNÇÃO

Calcium- and integrin-binding protein that plays a role in intracellular calcium homeostasis (By similarity). Acts as an auxiliary subunit of the sensory mechanoelectrical transduction (MET) channel in hair cells (By similarity). Essential for mechanoelectrical transduction (MET) currents in auditory hair cells and thereby required for hearing (By similarity). Regulates the function of hair cell mechanotransduction by controlling the distribution of transmembrane channel-like proteins TMC1 and T

LOCALIZAÇÃO

CytoplasmCell projection, stereociliumPhotoreceptor inner segmentCell projection, cilium, photoreceptor outer segmentCell membrane, sarcolemma

VIAS BIOLÓGICAS (2)
Sensory processing of sound by outer hair cells of the cochleaSensory processing of sound by inner hair cells of the cochlea
MECANISMO DE DOENÇA

Deafness, autosomal recessive, 48

A form of non-syndromic sensorineural hearing loss. Sensorineural deafness results from damage to the neural receptors of the inner ear, the nerve pathways to the brain, or the area of the brain that receives sound information. DFNB48 patients have prelingual onset of severe to profound sensorineural hearing loss affecting all frequencies.

OUTRAS DOENÇAS (3)
autosomal recessive nonsyndromic hearing loss 48Usher syndrome type 1hearing loss, autosomal recessive
HGNC:24579UniProt:O75838
CEP78Centrosomal protein of 78 kDaCandidate gene tested inTolerante
FUNÇÃO

Centriole wall protein that localizes to mature centrioles and regulates centriole and cilia biogenesis (PubMed:27246242, PubMed:27588451, PubMed:28242748, PubMed:34259627). Involved in centrosome duplication: required for efficient PLK4 centrosomal localization and PLK4-induced overduplication of centrioles (PubMed:27246242). Involved in cilium biogenesis and controls cilium length (PubMed:27588451). Acts as a regulator of protein stability by preventing ubiquitination of centrosomal proteins,

LOCALIZAÇÃO

Cytoplasm, cytoskeleton, microtubule organizing center, centrosomeCytoplasm, cytoskeleton, microtubule organizing center, centrosome, centrioleCytoplasm, cytoskeleton, cilium basal body

VIAS BIOLÓGICAS (7)
Recruitment of mitotic centrosome proteins and complexesLoss of proteins required for interphase microtubule organization from the centrosomeLoss of Nlp from mitotic centrosomesRegulation of PLK1 Activity at G2/M TransitionAURKA Activation by TPX2
MECANISMO DE DOENÇA

Cone-rod dystrophy and hearing loss 1

An autosomal recessive disease defined by the association of progressive cone-rod dystrophy with sensorineural hearing loss. Cone-rod dystrophy is characterized by retinal pigment deposits visible on fundus examination, predominantly in the macular region, and initial loss of cone photoreceptors followed by rod degeneration. This leads to decreased visual acuity and sensitivity in the central visual field, followed by loss of peripheral vision. Severe loss of vision occurs earlier than in retinitis pigmentosa, due to cone photoreceptors degenerating at a higher rate than rod photoreceptors.

INTERAÇÕES PROTEICAS (5)
OUTRAS DOENÇAS (2)
cone-rod dystrophy and hearing loss 1Usher syndrome type 3
HGNC:25740UniProt:Q5JTW2
CLRN1Clarin-1Disease-causing germline mutation(s) inTolerante
FUNÇÃO

May have a role in the excitatory ribbon synapse junctions between hair cells and cochlear ganglion cells and presumably also in analogous synapses within the retina

LOCALIZAÇÃO

Cell membrane

MECANISMO DE DOENÇA

Usher syndrome 3A

USH is a genetically heterogeneous condition characterized by the association of retinitis pigmentosa with sensorineural deafness. Age at onset and differences in auditory and vestibular function distinguish Usher syndrome type 1 (USH1), Usher syndrome type 2 (USH2) and Usher syndrome type 3 (USH3). USH3 is characterized by postlingual, progressive hearing loss, variable vestibular dysfunction, and onset of retinitis pigmentosa symptoms, including nyctalopia, constriction of the visual fields, and loss of central visual acuity, usually by the second decade of life.

OUTRAS DOENÇAS (4)
retinitis pigmentosa 61Usher syndrome type 3AUsher syndrome type 3retinitis pigmentosa
HGNC:12605UniProt:P58418
ADGRV1Adhesion G-protein coupled receptor V1Disease-causing germline mutation(s) inTolerante
FUNÇÃO

G-protein coupled receptor which has an essential role in the development of hearing and vision. Couples to G-alpha(i)-proteins, GNAI1/2/3, G-alpha(q)-proteins, GNAQ, as well as G-alpha(s)-proteins, GNAS, inhibiting adenylate cyclase (AC) activity and cAMP production. Required for the hair bundle ankle formation, which connects growing stereocilia in developing cochlear hair cells of the inner ear. In response to extracellular calcium, activates kinases PKA and PKC to regulate myelination by inh

LOCALIZAÇÃO

Cell membraneCell projection, stereocilium membranePhotoreceptor inner segment

VIAS BIOLÓGICAS (1)
EGR2 and SOX10-mediated initiation of Schwann cell myelination
MECANISMO DE DOENÇA

Usher syndrome 2C

USH is a genetically heterogeneous condition characterized by the association of retinitis pigmentosa with sensorineural deafness. Age at onset and differences in auditory and vestibular function distinguish Usher syndrome type 1 (USH1), Usher syndrome type 2 (USH2) and Usher syndrome type 3 (USH3). USH2 is characterized by congenital mild hearing impairment with normal vestibular responses.

OUTRAS DOENÇAS (4)
Usher syndrome type 2Cfebrile seizures, familial, 4Usher syndrome type 2generalized epilepsy with febrile seizures plus
HGNC:17416UniProt:Q8WXG9
USH1CHarmoninDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Anchoring/scaffolding protein that is a part of the functional network formed by USH1C, USH1G, CDH23 and MYO7A that mediates mechanotransduction in cochlear hair cells. Required for normal development and maintenance of cochlear hair cell bundles (By similarity). As part of the intermicrovillar adhesion complex/IMAC plays a role in brush border differentiation, controlling microvilli organization and length. Probably plays a central regulatory role in the assembly of the complex, recruiting CDHR

LOCALIZAÇÃO

Cytoplasm, cytosolCytoplasm, cytoskeletonCell projection, microvillus

VIAS BIOLÓGICAS (2)
Sensory processing of sound by outer hair cells of the cochleaSensory processing of sound by inner hair cells of the cochlea
MECANISMO DE DOENÇA

Usher syndrome 1C

USH is a genetically heterogeneous condition characterized by the association of retinitis pigmentosa with sensorineural deafness. Age at onset and differences in auditory and vestibular function distinguish Usher syndrome type 1 (USH1), Usher syndrome type 2 (USH2) and Usher syndrome type 3 (USH3). USH1 is characterized by profound congenital sensorineural deafness, absent vestibular function and prepubertal onset of progressive retinitis pigmentosa leading to blindness.

EXPRESSÃO TECIDUAL(Tecido-específico)
Intestino delgado
33.0 TPM
Brain Spinal cord cervical c-1
31.6 TPM
Cólon transverso
27.2 TPM
Rim - Córtex
23.4 TPM
Rim - Medula
9.9 TPM
OUTRAS DOENÇAS (4)
autosomal recessive nonsyndromic hearing loss 18AUsher syndrome type 1CUsher syndrome type 1hearing loss, autosomal recessive
HGNC:12597UniProt:Q9Y6N9
MYO7AUnconventional myosin-VIIaDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Myosins are actin-based motor molecules with ATPase activity. Unconventional myosins serve in intracellular movements. Their highly divergent tails bind to membranous compartments, which are then moved relative to actin filaments. In the retina, plays an important role in the renewal of the outer photoreceptor disks. Plays an important role in the distribution and migration of retinal pigment epithelial (RPE) melanosomes and phagosomes, and in the regulation of opsin transport in retinal photore

LOCALIZAÇÃO

CytoplasmCytoplasm, cell cortexCytoplasm, cytoskeletonSynapse

VIAS BIOLÓGICAS (3)
The canonical retinoid cycle in rods (twilight vision)Sensory processing of sound by outer hair cells of the cochleaSensory processing of sound by inner hair cells of the cochlea
MECANISMO DE DOENÇA

Usher syndrome 1B

USH is a genetically heterogeneous condition characterized by the association of retinitis pigmentosa with sensorineural deafness. Age at onset and differences in auditory and vestibular function distinguish Usher syndrome type 1 (USH1), Usher syndrome type 2 (USH2) and Usher syndrome type 3 (USH3). USH1 is characterized by profound congenital sensorineural deafness, absent vestibular function and prepubertal onset of progressive retinitis pigmentosa leading to blindness.

EXPRESSÃO TECIDUAL(Ubíquo)
Testículo
43.8 TPM
Glândula adrenal
40.7 TPM
Pituitária
27.6 TPM
Baço
20.4 TPM
Fígado
12.7 TPM
OUTRAS DOENÇAS (9)
autosomal dominant nonsyndromic hearing loss 11autosomal recessive nonsyndromic hearing loss 2Usher syndrome type 1Bnonsyndromic genetic hearing loss
HGNC:7606UniProt:Q13402
PDZD7PDZ domain-containing protein 7Disease-causing germline mutation(s) inTolerante
FUNÇÃO

In cochlear developing hair cells, essential in organizing the USH2 complex at stereocilia ankle links. Blocks inhibition of adenylate cyclase activity mediated by ADGRV1

LOCALIZAÇÃO

Cell projection, ciliumNucleusCell projection, stereocilium

MECANISMO DE DOENÇA

Deafness, autosomal recessive, 57

A form of non-syndromic, sensorineural deafness characterized by symmetric, bilateral hearing loss with onset in early childhood. Vestibular function is preserved. Sensorineural deafness results from damage to the neural receptors of the inner ear, the nerve pathways to the brain, or the area of the brain that receives sound information. DFNB57 severity ranges from moderate to severe.

EXPRESSÃO TECIDUAL(Tecido-específico)
Cerebelo
35.8 TPM
Cérebro - Hemisfério cerebelar
31.2 TPM
Pituitária
10.4 TPM
Brain Frontal Cortex BA9
10.3 TPM
Córtex cerebral
10.1 TPM
OUTRAS DOENÇAS (4)
hearing loss, autosomal recessive 57Usher syndrome type 2CUsher syndrome type 2Usher syndrome type 2A
HGNC:26257UniProt:Q9H5P4
USH1Gpre-mRNA splicing regulator USH1GDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Plays a role in pre-mRNA splicing by regulating the release and transfer of U4/U6.U5 tri-small nuclear ribonucleoprotein (tri-snRNP) complexes from their assembly site in Cajal bodies to nuclear speckles, thereby contributing to the assembly of the pre-catalytic spliceosome on target pre-mRNAs (PubMed:34023904). May also participate in recycling of snRNPs back to Cajal bodies during splicing (PubMed:34023904). Plays a role in regulating MAGI2-mediated endocytosis (PubMed:24608321). Anchoring/sca

LOCALIZAÇÃO

Cytoplasm, cytosolCytoplasm, cytoskeletonCell membraneCell projection, ciliumNucleus speckleNucleus, Cajal bodyCytoplasm, cytoskeleton, microtubule organizing center, centrosomePhotoreceptor inner segment

VIAS BIOLÓGICAS (2)
Sensory processing of sound by outer hair cells of the cochleaSensory processing of sound by inner hair cells of the cochlea
MECANISMO DE DOENÇA

Usher syndrome 1G

USH is a genetically heterogeneous condition characterized by the association of retinitis pigmentosa with sensorineural deafness. Age at onset and differences in auditory and vestibular function distinguish Usher syndrome type 1 (USH1), Usher syndrome type 2 (USH2) and Usher syndrome type 3 (USH3). USH1 is characterized by profound congenital sensorineural deafness, absent vestibular function and prepubertal onset of progressive retinitis pigmentosa leading to blindness.

EXPRESSÃO TECIDUAL(Tecido-específico)
Esôfago - Mucosa
11.1 TPM
Testículo
4.8 TPM
Skin Sun Exposed Lower leg
4.2 TPM
Skin Not Sun Exposed Suprapubic
2.2 TPM
Útero
1.8 TPM
OUTRAS DOENÇAS (2)
Usher syndrome type 1GUsher syndrome type 1
HGNC:16356UniProt:Q495M9
CDH23Cadherin-23Disease-causing germline mutation(s) inTolerante
FUNÇÃO

Cadherins are calcium-dependent cell adhesion proteins. They preferentially interact with themselves in a homophilic manner in connecting cells. CDH23 is required for establishing and/or maintaining the proper organization of the stereocilia bundle of hair cells in the cochlea and the vestibule during late embryonic/early postnatal development. It is part of the functional network formed by USH1C, USH1G, CDH23 and MYO7A that mediates mechanotransduction in cochlear hair cells. Required for norma

LOCALIZAÇÃO

Cell membrane

VIAS BIOLÓGICAS (2)
Sensory processing of sound by outer hair cells of the cochleaSensory processing of sound by inner hair cells of the cochlea
MECANISMO DE DOENÇA

Usher syndrome 1D

USH is a genetically heterogeneous condition characterized by the association of retinitis pigmentosa with sensorineural deafness. Age at onset and differences in auditory and vestibular function distinguish Usher syndrome type 1 (USH1), Usher syndrome type 2 (USH2) and Usher syndrome type 3 (USH3). USH1 is characterized by profound congenital sensorineural deafness, absent vestibular function and prepubertal onset of progressive retinitis pigmentosa leading to blindness.

OUTRAS DOENÇAS (12)
autosomal recessive nonsyndromic hearing loss 12Usher syndrome type 1Dnonsyndromic genetic hearing lossUsher syndrome
HGNC:13733UniProt:Q9H251
USH2AUsherinDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Involved in hearing and vision as member of the USH2 complex. In the inner ear, required for the maintenance of the hair bundle ankle formation, which connects growing stereocilia in developing cochlear hair cells. In retina photoreceptors, the USH2 complex is required for the maintenance of periciliary membrane complex that seems to play a role in regulating intracellular protein transport

LOCALIZAÇÃO

Cell projection, stereocilium membraneSecreted

MECANISMO DE DOENÇA

Usher syndrome 2A

USH is a genetically heterogeneous condition characterized by the association of retinitis pigmentosa with sensorineural deafness. Age at onset and differences in auditory and vestibular function distinguish Usher syndrome type 1 (USH1), Usher syndrome type 2 (USH2) and Usher syndrome type 3 (USH3). USH2 is characterized by congenital mild hearing impairment with normal vestibular responses.

EXPRESSÃO TECIDUAL(Baixa expressão)
Testículo
1.2 TPM
Fígado
0.8 TPM
Cérebro - Hemisfério cerebelar
0.2 TPM
Cerebelo
0.2 TPM
Coração - Átrio
0.2 TPM
OUTRAS DOENÇAS (5)
retinitis pigmentosa 39Usher syndrome type 2AUsher syndromeretinitis pigmentosa
HGNC:12601UniProt:O75445
PCDH15Protocadherin-15Disease-causing germline mutation(s) inTolerante
FUNÇÃO

Calcium-dependent cell-adhesion protein. Essential for maintenance of normal retinal and cochlear function

LOCALIZAÇÃO

Cell membraneSecreted

VIAS BIOLÓGICAS (2)
Sensory processing of sound by outer hair cells of the cochleaSensory processing of sound by inner hair cells of the cochlea
MECANISMO DE DOENÇA

Usher syndrome 1F

USH is a genetically heterogeneous condition characterized by the association of retinitis pigmentosa with sensorineural deafness. Age at onset and differences in auditory and vestibular function distinguish Usher syndrome type 1 (USH1), Usher syndrome type 2 (USH2) and Usher syndrome type 3 (USH3). USH1 is characterized by profound congenital sensorineural deafness, absent vestibular function and prepubertal onset of progressive retinitis pigmentosa leading to blindness.

EXPRESSÃO TECIDUAL(Baixa expressão)
Hipotálamo
2.4 TPM
Cérebro - Amígdala
2.0 TPM
Hipocampo
1.8 TPM
Brain Anterior cingulate cortex BA24
1.7 TPM
Substância negra
1.7 TPM
OUTRAS DOENÇAS (5)
Usher syndrome type 1Fautosomal recessive nonsyndromic hearing loss 23Usher syndrome type 1DUsher syndrome type 1
HGNC:14674UniProt:Q96QU1
WHRNWhirlinDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Involved in hearing and vision as member of the USH2 complex. Necessary for elongation and maintenance of inner and outer hair cell stereocilia in the organ of Corti in the inner ear. Involved in the maintenance of the hair bundle ankle region, which connects stereocilia in cochlear hair cells of the inner ear. In retina photoreceptors, required for the maintenance of periciliary membrane complex that seems to play a role in regulating intracellular protein transport

LOCALIZAÇÃO

CytoplasmCell projection, stereociliumCell projection, growth conePhotoreceptor inner segmentSynapse

VIAS BIOLÓGICAS (2)
Sensory processing of sound by outer hair cells of the cochleaSensory processing of sound by inner hair cells of the cochlea
MECANISMO DE DOENÇA

Deafness, autosomal recessive, 31

A form of non-syndromic sensorineural hearing loss. Sensorineural deafness results from damage to the neural receptors of the inner ear, the nerve pathways to the brain, or the area of the brain that receives sound information.

EXPRESSÃO TECIDUAL(Ubíquo)
Glândula adrenal
101.0 TPM
Testículo
78.7 TPM
Pituitária
61.5 TPM
Útero
52.7 TPM
Ovário
35.9 TPM
OUTRAS DOENÇAS (4)
Usher syndrome type 2Dautosomal recessive nonsyndromic hearing loss 31Usher syndrome type 2hearing loss, autosomal recessive
HGNC:16361UniProt:Q9P202
ARSGArylsulfatase GDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Displays arylsulfatase activity at acidic pH towards artificial substrates, such as p-nitrocatechol sulfate and also, but with a lower activity towards p-nitrophenyl sulfate and 4-methylumbelliferyl sulfate (PubMed:18283100, PubMed:29300381). Catalyzes the hydrolysis of the 3-sulfate groups of the N-sulfo-D-glucosamine 3-O-sulfate units of heparin (PubMed:22689975)

LOCALIZAÇÃO

Lysosome

VIAS BIOLÓGICAS (2)
The activation of arylsulfatasesGlycosphingolipid catabolism
MECANISMO DE DOENÇA

Usher syndrome 4

A form of Usher syndrome, a genetically heterogeneous condition characterized by the association of retinitis pigmentosa with sensorineural deafness. Age at onset and differences in auditory and vestibular function distinguish different types of Usher syndrome. USH4 is characterized by late onset of retinitis pigmentosa and usually late-onset of progressive sensorineural hearing loss without vestibular involvement. USH4 inheritance is autosomal recessive.

INTERAÇÕES PROTEICAS (4)
OUTRAS DOENÇAS (2)
Usher syndrome, type 4Usher syndrome type 3
HGNC:24102UniProt:Q96EG1

Variantes genéticas (ClinVar)

316 variantes patogênicas registradas no ClinVar.

🧬 HARS1: NM_002109.6(HARS1):c.490A>G (p.Thr164Ala) ()
🧬 HARS1: NM_002109.6(HARS1):c.166C>T (p.Leu56Phe) ()
🧬 HARS1: NM_002109.6(HARS1):c.519G>T (p.Gln173His) ()
🧬 HARS1: NM_002109.6(HARS1):c.732_733del (p.Ser245fs) ()
🧬 HARS1: NM_002109.6(HARS1):c.883G>T (p.Ala295Ser) ()
Ver todas no ClinVar

Classificação de variantes (ClinVar)

Distribuição de 8,716 variantes classificadas pelo ClinVar.

872
4358
3486
Patogênica (10.0%)
VUS (50.0%)
Benigna (40.0%)
VARIANTES MAIS SIGNIFICATIVAS
USH2A: NM_206933.4(USH2A):c.6486-1G>C [Pathogenic]
USH2A: NM_206933.4(USH2A):c.10637G>A (p.Gly3546Glu) [Likely pathogenic]
HARS1: NM_002109.6(HARS1):c.1109T>C (p.Phe370Ser) [Uncertain significance]
HARS1: NM_002109.6(HARS1):c.613G>C (p.Gly205Arg) [Uncertain significance]
HARS1: NM_002109.6(HARS1):c.20T>A (p.Leu7Gln) [Uncertain significance]

Diagnóstico

Os sinais que médicos procuram e os exames que confirmam

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Tratamento e manejo

Remédios, cuidados de apoio e o que precisa acompanhar

Pipeline de tratamentos
Pipeline regulatório — de medicamentos já aprovados a drogas em pesquisa exploratória.
3Fase 33
2Fase 27
1Fase 11
·Pré-clínico9
Medicamentos catalogadosEnsaios clínicos· 0 medicamentos · 20 ensaios
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Onde tratar no SUS

Hospitais de referência no Brasil e o protocolo oficial do SUS (PCDT)

🇧🇷 Atendimento SUS — Síndrome Usher

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Publicações mais relevantes

Timeline de publicações
740 papers (10 anos)

Mostrando amostra de 200 publicações de um total de 740

#1

Generation of the induced pluripotent stem cell line BTHBIOi002-A derived from a USH2 patient with c.2512C>T and c.2802T>G mutations in USH2A gene.

Stem cell research2026 Apr

Mutation in USH2A gene cause autosomal recessive retinitis pigmentosa (RP) and Usher syndrome type II (USH2), constituting over 50% of USH2 and approximately 7% of RP. Here, we report the establishment of a human induced pluripotent stem cell (iPSC) line, BTHBIOi002-A, derived from the peripheral blood mononuclear cells (PBMCs) of a USH2 patient with compound heterozygous mutation in USH2A (c.[2512C>T]; [2802 T>G]), using the non-integrating episomal plasmids delivered by electroporation with OCT4, SOX2, NANOG, LIN28, c-Myc, and KLF4. The established iPSC line was validated for the karyotype stability, pluripotency markers, and the ability to differentiate into all three germ layers.

#2

Nonsense Mutation in USH2A Exon-13 Activates the Innate Immune Response in Müller Glial Cells.

International journal of molecular sciences2026 Feb 07

Pathological USH2A mutations cause Usher syndrome type II, characterized by progressive retinitis pigmentosa and hearing and balance impairment. This study aims to investigate the cellular mechanisms underlying USH2A-related retinal degeneration using human induced pluripotent stem cell (hiPSC)-derived retinal organoids. The introduction of a homozygous nonsense mutation in the USH2A hotspot exon-13 resulted in normal photoreceptor development but loss of ciliary localization of usherin long form B and its interacting proteins, ADGRV1 and whirlin. Notably, single-cell RNA sequencing revealed unexpected significant transcriptional changes in Müller glial cells (MGCs), suggestive of disruptions in the translation, innate immune response, and endolysosomal system. These findings suggest that, while photoreceptor cells are mildly affected by the exon-13 USH2A mutation, MGCs exhibit major transcriptional changes, potentially contributing to the disease progression and therefore shedding light on potential alternative therapeutic targets.

#3

Characterization of Usher Syndrome Type 2-Associated Proteins in the Retina via Affinity Purification-Mass Spectrometry.

Molecular &amp; cellular proteomics : MCP2026 Feb 05

Usher syndrome is the leading cause of inherited deaf-blindness, with type 2 (Usher syndrome type 2, USH2) being the most common form. USH2A, ADGRV1, and WHRN are the three known USH2 causative genes, which are also linked to isolated retinal degeneration and hearing loss. These genes encode usherin, ADGRV1, and whirlin, respectively, collectively called USH2 proteins. These proteins form a multiprotein complex (USH2 complex) at the periciliary membrane in retinal photoreceptors and at the stereociliary ankle link in inner ear hair cells. The molecular function of the USH2 complex and its disease mechanisms are poorly understood. Currently, there is no cure for diseases caused by mutations in the three USH2 genes. In this study, we employed multiple affinity purification methods combined with mass spectrometry to systematically identify the interaction partners of USH2 proteins in the retina. The ADGRV1 intracellular bait pulled down proteins involved in actin-based cell projections, the chaperone-containing TCP-1 complex, and the Bardet-Biedl syndrome complex. The extracellular domains of ADGRV1 and usherin pulled down proteins related to peptidase regulation, collagen biosynthesis and modification, and elastic fiber formation. The EAR/EPTP repeats of ADGRV1 specifically pulled down TGFβ signaling proteins. Further immunoprecipitation experiments identified, with high confidence, Gαi and Gαq as ADGRV1-interacting proteins, and retinal degeneration and ciliary proteins as interaction partners of USH2 proteins. We also demonstrated that the usherin extracellular domains interact with each other and with ADGRV1. Overall, these findings suggest that the USH2 complex connects the extracellular matrix (ECM) to the intracellular actin network, signals through Gαi and Gαq, and participates in ECM remodeling, TGFβ signaling, cell adhesion, and ciliary function in photoreceptors.

#4

Compensatory Interplay Between Clarin-1 and Clarin-2 Deafness-Associated Proteins Governs Phenotypic Variability in Hearing.

Advanced science (Weinheim, Baden-Wurttemberg, Germany)2026 Jan 22

Usher syndrome type III (USH3) is a genetic disorder characterized by progressive, post-lingual hearing loss, variable vestibular dysfunction, and onset of retinitis pigmentosa. USH3 is caused by mutations in CLRN1, which encodes clarin-1, a tetraspanin-like protein. Mutations in CLRN2, which encodes the related protein clarin-2, are also implicated in progressive, non-syndromic hearing loss in both humans and mice. USH3 patients show considerable phenotypic variability, even among individuals with the same mutation. This variability may result from environmental factors or interactions with other inner ear genes, such as CLRN2. To investigate the functional interplay of these genes, we generated Clrn1- /-Clrn2-/- double knockout mice. RNA-sequencing and functional/physiological analyses revealed that clarin-1 and clarin-2 jointly regulate mechanoelectrical transduction, ionic homeostasis, and synaptic organization. Their combined loss leads to more severe hearing phenotype compared to Clrn1-/- and Clrn2-/- mice, which reveals a functional compensation between them. CLRN2 variants may exacerbate hearing loss in USH3 patients, supporting inclusion of CLRN2 in genetic screening. By revealing a functional, compensatory interplay between clarin-1 and clarin-2, this study reframes CLRN1-associated deafness as a network-dependent disorder and provides a mechanistic basis for genetic stratification and therapeutic directions in USH3 and related sensorineural hearing loss.

#5

Exploring extracellular vesicle MicroRNAs in Usher syndrome type 1B: Tear-Derived EVs as potential indicators of retinal health.

Cellular and molecular life sciences : CMLS2026 Jan 13

Usher syndrome type 1B (USH1B) is a rare inherited disorder characterized by congenital deafness and progressive retinitis pigmentosa, caused by biallelic pathogenic variants in the MYO7A gene. We explored extracellular vesicles (EVs) from two sources: human tears and iPSC-derived RPE cells from USH1B patients and controls. Tear EVs were assessed as a non-invasive biomarker source, while RPE-derived EVs provided insights into disease mechanisms in a controlled, cell-type-specific context. Although RPE differentiation was successful and MYO7A expression levels were similar between patients and controls, Myosin VIIA was not detected by western blot in the patient-derived cells. We examined the EV cargo by small non-coding RNAs (sncRNAs) sequencing from iPSC-RPE apical site and tears to identify molecular signatures of retinal degeneration. Tear EVs showed higher load and diversity of miRNAs than RPE-derived EVs, reflecting a broader ocular origin. Comparative analysis revealed shared retinal sncRNAs (hsa-miR-204, hsa-miR-211, hsa-miR-181a-5p) and group-specific differences. Notably, when comparing to controls, hsa-miR-200a-3p and hsa-miR-194-5p were upregulated in patient tear EVs, while let-7i/c-5p and hsa-miR-320a/b, were downregulated in-patient RPE-derived EVs. Pathway analysis linked these sncRNAs to retinal structure and function, including cytoskeletal remodeling and junctional integrity. Our findings highlight the potential of tear EVs as a non-invasive source of biomarkers that capture retinal molecular alterations in USH1B, with applications for diagnosis, monitoring, and therapeutic development. Although this is a pilot study focused on uncovering promising biomarkers rather than establishing definitive cause-effect mechanisms, it provides a foundation for future research with larger cohorts to validate and expand these findings.

Publicações recentes

Ver todas no PubMed

📚 EuropePMC796 artigos no totalmostrando 197

2025

Deciphering the genetic basis of inherited retinal dystrophies via whole-exome sequencing in a Turkish cohort.

Molecular vision
2026

Functional reassessment of extended splice region variants in MYO7A with hearing loss and Usher syndrome.

The Journal of pathology
2026

Retinal vasoproliferative tumors in pediatric retinal dystrophies.

Journal of AAPOS : the official publication of the American Association for Pediatric Ophthalmology and Strabismus
2026

Somatic mosaicism of a novel USH2A variant in Usher syndrome.

Ophthalmic genetics
2026

Spontaneous resolution of cystoid macular edema with concurrent axial elongation in a pediatric patient with Usher syndrome type 1B: a case report.

BMC ophthalmology
2026

Generation of the induced pluripotent stem cell line BTHBIOi002-A derived from a USH2 patient with c.2512C>T and c.2802T>G mutations in USH2A gene.

Stem cell research
2026

Nonsense Mutation in USH2A Exon-13 Activates the Innate Immune Response in Müller Glial Cells.

International journal of molecular sciences
2026

Pathology of the Human Temporal Bone in a Rare Case of Combined Usher Syndrome and Cystic Fibrosis.

Otology &amp; neurotology : official publication of the American Otological Society, American Neurotology Society [and] European Academy of Otology and Neurotology
2026

From Sound to Stability: Lessons Learned From the CRUSH Study on Hearing Loss Progression and Vestibular Phenotype in Usher Syndrome Type 2A.

Otology &amp; neurotology : official publication of the American Otological Society, American Neurotology Society [and] European Academy of Otology and Neurotology
2026

CDH23-associated Usher syndrome: genotype-phenotype correlations.

Ophthalmic genetics
2026

Characterization of Usher Syndrome Type 2-Associated Proteins in the Retina via Affinity Purification-Mass Spectrometry.

Molecular &amp; cellular proteomics : MCP
2025

Analysis of Cytosine Base Editors in Bovine Zygotes: Efficiency and Editing Window Characterization Through Targeting the MYO7A Gene.

Current issues in molecular biology
2026

Compensatory Interplay Between Clarin-1 and Clarin-2 Deafness-Associated Proteins Governs Phenotypic Variability in Hearing.

Advanced science (Weinheim, Baden-Wurttemberg, Germany)
2026

Exploring extracellular vesicle MicroRNAs in Usher syndrome type 1B: Tear-Derived EVs as potential indicators of retinal health.

Cellular and molecular life sciences : CMLS
2026

Inherited retinal disorders in Scotland: A 5 year assessment.

Eye (London, England)
2026

A Novel Intronic Variant Causes Aberrant Splicing of PCDH15 in a Family With Usher Syndrome Type 1F.

Molecular genetics &amp; genomic medicine
2026

Genetic diversity of Usher syndrome in Moroccan patients.

Human genetics
2025

Listening Effort and Its Relation to Spatial Localization, and Vestibular and Visual Impairment in Usher Syndrome-Our Experience.

Audiology research
2025

Atypical Case of Pemphigus Erythematosus in a Previously Healthy Young Woman: Diagnostic Delay and Response to Rituximab.

Cureus
2025

Genetics of prelingual isolated deafness and Usher syndrome in the Maghreb and Jordan: Harnessing the potential of homozygosity.

Proceedings of the National Academy of Sciences of the United States of America
2025

Cloud-Based Personalized sEMG Classification Using Lightweight CNNs for Long-Term Haptic Communication in Deaf-Blind Individuals.

Bioengineering (Basel, Switzerland)
2026

Clinical Findings and Molecular Genetics of USH1C-Associated Usher Syndrome.

JAMA ophthalmology
2025

Epidemiological and genetic insights of Usher syndrome in Turkish population: A cross-sectional preliminary study from University of Health Sciences, Turkey.

The Journal of international medical research
2026

Compound heterozygous variants in PCDH15 non-coding regions in an Usher Syndrome Type 1F patient: minigene assay reveals pathogenicity of c.3123-1G>C.

Ophthalmic genetics
2025

A sound vision: MYO7A gene therapy reaches the inner ear.

The Journal of physiology
2025

Identification of a variant in the USH1G gene in a family with Usher syndrome.

Biomedica : revista del Instituto Nacional de Salud
2025

Exploring exon excision as a therapeutic intervention strategy for the future treatment of ADGRV1-associated retinitis pigmentosa.

Molecular therapy. Nucleic acids
2025

Adeno-associated virus-based rescue of Myo7a expression restores hair-cell function and improves hearing thresholds in a USH1B mouse strain.

The Journal of physiology
2025

Structural Abnormalities of the Brain Detected by 7 Tesla MRI in Patients with Usher Syndrome.

Journal of clinical medicine
2026

AAV-mediated exon skipping therapy for Usher syndrome, type 2A.

Molecular therapy : the journal of the American Society of Gene Therapy
2025

Clinical Research for Inherited Retinal Disease Related Pediatric Blindness: A Preliminary Descriptive Analysis Based on ClinicalTrials.gov.

Journal of multidisciplinary healthcare
2025

Computational study of deleterious missense SNPs in the USH1G gene implicated in Usher syndrome.

Journal of biomolecular structure &amp; dynamics
2025

Inherited retinal diseases in Kentucky: diagnostic yield, gene variants, and novel mutations in a U.S. population.

BMC medical genomics
2025

Author Correction: Alterations of the CIB2 calcium- and integrin-binding protein cause Usher syndrome type 1J and nonsyndromic deafness DFNB48.

Nature genetics
2025

Bilateral Ring-Shaped Retinitis Pigmentosa in Usher Syndrome Type IV.

Ophthalmology. Retina
2025

Autoimmune encephalitis associated with antibodies against α-enolase sequestrated from degenerating retina in retinitis pigmentosa.

BMC ophthalmology
2025

NGS sequencing reveals the cause of hearing loss in a group of Polish patients with an isolated, non-DFNB1 hearing loss.

Journal of applied genetics
2025

[Genetic and clinical phenotypic analysis of Usher syndrome-associated gene variants].

Lin chuang er bi yan hou tou jing wai ke za zhi = Journal of clinical otorhinolaryngology head and neck surgery
2025

Ciliopathy: Usher Syndrome.

Advances in experimental medicine and biology
2025

Phenotypic and Genotypic Characterization of 171 Patients with Syndromic Inherited Retinal Diseases Highlights the Importance of Genetic Testing for Accurate Clinical Diagnosis.

Genes
2025

Exploring Concomitant Ophthalmic Comorbidities in Portuguese Patients with Inherited Retinal Diseases: A Comprehensive Clinical Study.

Genes
2025

A Hybrid Sequential Feature Selection Approach for Identifying New Potential mRNA Biomarkers for Usher Syndrome Using Machine Learning.

Biomolecules
2025

An Overview of Recent Advances and Clinical Applications of Exon Skipping and Splice Modulation for Muscular Dystrophy and Various Genetic Diseases.

Methods in molecular biology (Clifton, N.J.)
2025

[Inherited retinal disorders: Current therapeutic options and future perspectives].

Klinische Monatsblatter fur Augenheilkunde
2025

Identification of Novel USH2A Mutations in a Consanguineous Chinese Family With Usher Syndrome.

Human mutation
2025

Novel ADGRV1 pathogenic variant associated to sleep-related hypermotor epilepsy.

Epileptic disorders : international epilepsy journal with videotape
2024

Pediatric Usher Syndrome Type 2A with Coexisting Rheumatic Heart Disease and Upper Gastro-Intestinal Bleed: A Case Report.

JNMA; journal of the Nepal Medical Association
2025

Liquid-Liquid Phase Separation in Hereditary Hearing Loss.

Neuroscience bulletin
2025

Current approaches for Usher syndrome disease models and developing therapies.

Frontiers in cell and developmental biology
2025

Mutation of beta-tubulin 4B gene (TUBB4B) causes autosomal dominant retinitis pigmentosa with sensorineural hearing loss in a multigenerational family.

Molecular vision
2025

Characterisation and prevalence of inherited retinal diseases in the Finnish population reveals enrichment of population-specific phenotypes and causative variants.

The British journal of ophthalmology
2025

THE IMPACT OF CYSTOID MACULOPATHY IN USH2A RETINITIS PIGMENTOSA : A Retrospective 5-year Analysis.

Retina (Philadelphia, Pa.)
2025

Outcomes of genetic testing for Usher syndrome in a diverse population cohort from South Florida.

Human genomics
2025

PHARC (Polyneuropathy, Hearing Loss, Ataxia, Retinitis Pigmentosa and Cataract) - A Case Report and Clinical-Focused Literature Review.

Cerebellum (London, England)
2025

De Novo SLC12A2 Variant Presenting as Congenital Hearing Loss With Vestibular Areflexia.

American journal of medical genetics. Part A
2025

Alpelisib Therapy in 2 Patients With Congenital Hyperinsulinism.

JCEM case reports
2025

Outcomes of cochlear implants in patients with PCDH15 mutations: a clinical study.

Frontiers in genetics
2025

Nonsense Mutations in Rare and Ultra-Rare Human Disorders: An Overview.

IUBMB life
2025

Resolving the Diagnostic Odyssey in Inherited Retinal Dystrophies Through Long-Read Genome Sequencing.

American journal of medical genetics. Part A
2025

Gene Duplication in a Patient With Usher Syndrome Type 2C: A Case Report.

Cureus
2025

Zebrafish cdh23 Affects Rod Cell Phototransduction Through Regulating Ca2+ Transport and MAPK Signaling Pathway.

International journal of molecular sciences
2025

Machine Learning-Based Ensemble Feature Selection and Nested Cross-Validation for miRNA Biomarker Discovery in Usher Syndrome.

Bioengineering (Basel, Switzerland)
2025

Advancing Therapeutic Strategies for Nonsense-Related Diseases: From Small Molecules to Nucleic Acid-Based Innovations.

IUBMB life
2025

Patient reported outcomes in Usher Syndrome: a systematic review.

Ophthalmic genetics
2025

Vestibular Phenotype-Genotype Correlation in a Cohort of 35 European Usher Syndrome Patients.

American journal of audiology
2025

Computational study of the potential impact of WHRN protein missense SNPs on WHRN-MYO15A protein complex interaction and their association with Usher syndrome.

Journal of biomolecular structure &amp; dynamics
2025

Syndromic forms of inherited retinal dystrophies: a comprehensive molecular diagnosis of consanguineous Pakistani families using capture panel sequencing.

Molecular vision
2025

Haploinsufficiency of Whrn Contributes to Progressive Sensorineural Hearing Loss in C57BL6 Mice.

Journal of the Association for Research in Otolaryngology : JARO
2025

Ethnic disparities in inherited retinal degenerations.

Canadian journal of ophthalmology. Journal canadien d'ophtalmologie
2025

Natural History of Microperimetry and Optical Coherence Tomography in USH2A-Retinopathy: A Structure-Function Association Study.

American journal of ophthalmology
2025

Clinical manifestations of dual-gene variants involving ABCA4 in retinal dystrophies.

BMC ophthalmology
2025

Detailed Clinical, Ophthalmic, and Genetic Characterization of MYO7A-Associated Usher Syndrome.

Investigative ophthalmology &amp; visual science
2025

A Genomic Analysis of Usher Syndrome: Population-Scale Prevalence and Therapeutic Targets.

American journal of medical genetics. Part C, Seminars in medical genetics
2025

Bilateral Vasoproliferative Tumors in Usher Syndrome.

Case reports in ophthalmology
2025

A Tonotopic Regulatory Axis Governing Isoform-Specific MYO7A Expression in Cochlear Hair Cells.

bioRxiv : the preprint server for biology
2025

Tonotopic Specialization of MYO7A Isoforms in Auditory Hair Cells.

bioRxiv : the preprint server for biology
2025

Single-guide RNA Cas9 and enhanced-deletion Cas9 rescue a recurrent USH2A-related splicing defect.

Molecular therapy. Nucleic acids
2025

Optic Nerve Coloboma in a Child With Compound Heterozygous USH2A Variants.

Case reports in genetics
2025

Generation of two induced pluripotent stem cell lines carrying the CDH23 c.1515-12G > A variant.

Stem cell research
2025

Clozapine Use Among Individuals With Schizophrenia Occurring on the Background of Intellectual Disability and Rare Genetic Variation: A Retrospective Chart Review.

Journal of clinical psychopharmacology
2025

Usher Syndrome: New Insights into Classification, Genotype-Phenotype Correlation, and Management.

Genes
2025

Etiologies of Early-Onset Hearing Impairment in Rwanda.

Genes
2025

The BBS/CCT chaperonin complex ensures the localization of the adhesion G protein-coupled receptor ADGRV1 to the base of primary cilia.

Frontiers in cell and developmental biology
2025

The USH3A causative gene clarin1 functions in Müller glia to maintain retinal photoreceptors.

PLoS genetics
2025

Deciphering the largest disease-associated transcript isoforms in the human neural retina with advanced long-read sequencing approaches.

Genome research
2025

Adoption and usability of a braille communication assistive device (CAD) for face-to-face and remote communication in two users with deafblindness.

Disability and rehabilitation. Assistive technology
2026

Improvement of perceived cochlear implant sound quality through individualized psychoacoustic-based frequency fitting.

Zeitschrift fur medizinische Physik
2024

A Case of Autosomal Recessive Retinitis Pigmentosa with Vitelliform-Like Appearance at the Macula Associated with Novel MYO7A Variant P.Ser383TrpfsTer64.

Beyoglu eye journal
2025

The Role of Visual Electrophysiology in Systemic Hereditary Syndromes.

International journal of molecular sciences
2025

A Knockin Model with the Mouse Equivalent to the c.2299delG Mutation in Usherin Exhibits Early-Onset Hearing Loss and Progressive Retinal Degeneration.

Advances in experimental medicine and biology
2025

Loss of Usher II Proteins in Mice Does Not Affect Photoreceptor Ultrastructure.

Advances in experimental medicine and biology
2025

Identification of Unexpected Pathomechanisms Underlying the Human Usher Syndrome.

Advances in experimental medicine and biology
2025

Comparison of CRISPR-Cas13b RNA base editing approaches for USH2A-associated inherited retinal degeneration.

Communications biology
2025

A Novel Compound Heterozygous Variant in the ABHD12 Gene Cause PHARC Syndrome in a Chinese Family: The Proband Presenting New Genotype and Phenotype.

Molecular genetics &amp; genomic medicine
2025

From bench to bedside: Developing CRISPR/Cas-based therapy for ocular diseases.

Pharmacological research
2025

WITHDRAWN: Retinal Phenotypes and Single-cell Sequencing Analysis of Ush2a Knockout Mice.

Experimental eye research
2025

Systematic empirical evaluation of individual base editing targets: Validating therapeutic targets in USH2A and comparison of methods.

Molecular therapy : the journal of the American Society of Gene Therapy
2025

Long-Lasting Auditory and Vestibular Recovery Following Gene Replacement Therapy in a Novel Usher Syndrome Type 1c Mouse Model.

Advanced science (Weinheim, Baden-Wurttemberg, Germany)
2025

Non-Viral Delivery Systems to Transport Nucleic Acids for Inherited Retinal Disorders.

Pharmaceuticals (Basel, Switzerland)
2025

Senear-Usher Syndrome or Coexistence of SLE with Pemphigus Vulgaris-A Case Report with Literature Review.

Journal of clinical medicine
2025

Genotype-phenotype correlations for 17 Chinese families with inherited retinal dystrophies due to homozygous variants.

Scientific reports
2025

Unraveling the genetic spectrum of inherited deaf-blindness in Portugal.

Orphanet journal of rare diseases
2025

Actigraphy-based assessment of circadian rhythmicity and sleep in patients with Usher syndrome type 2a: A case-control study.

Journal of sleep research
2025

Syndromic retinitis pigmentosa.

Progress in retinal and eye research
2025

Variants in the AGBL5 gene are responsible for autosomal recessive Retinitis pigmentosa with hearing loss.

European journal of human genetics : EJHG
2024

Exploring non-coding variants and evaluation of antisense oligonucleotides for splicing redirection in Usher syndrome.

Molecular therapy. Nucleic acids
2025

The phenotypic spectrum of CEP250 gene variants.

Ophthalmic genetics
2024

A Comparative Evaluation of the Genetic Variant Spectrum in the USH2A Gene in Russian Patients with Isolated and Syndromic Forms of Retinitis Pigmentosa.

International journal of molecular sciences
2024

Nuclear-Cytoplasmic Shuttling of the Usher Syndrome 1G Protein SANS Differs from Its Paralog ANKS4B.

Cells
2025

Prevalence of Molecular Diagnoses for Usher Syndrome and the Need for Coordinated Care.

The Laryngoscope
2024

Type 2 Usher Syndrome - A Cause for Sensorineural Hearing Loss.

Indian journal of otolaryngology and head and neck surgery : official publication of the Association of Otolaryngologists of India
2024

Key challenges in developing a gene therapy for Usher syndrome: machine-assisted scoping review.

Journal of community genetics
2024

PHARC syndrome: an overview.

Orphanet journal of rare diseases
2024

Fine-scale genetic structure and rare variant frequencies.

PloS one
2024

PCDH15 dual-AAV gene therapy for deafness and blindness in Usher syndrome type 1F models.

The Journal of clinical investigation
2024

Intestinal Tuft Cells Are Enriched With Protocadherins.

The journal of histochemistry and cytochemistry : official journal of the Histochemistry Society
2024

Genotype Characterization and MiRNA Expression Profiling in Usher Syndrome Cell Lines.

International journal of molecular sciences
2024

A rare transcript homozygous variants in CLRN1(USH3A) causes Usher syndrome type 3 in a Chinese family.

Orphanet journal of rare diseases
2024

MultiBac System-based Purification and Biophysical Characterization of Human Myosin-7a.

Journal of visualized experiments : JoVE
2025

Cochlear implantation in syndromic patients: difficulties and lessons learnt.

European archives of oto-rhino-laryngology : official journal of the European Federation of Oto-Rhino-Laryngological Societies (EUFOS) : affiliated with the German Society for Oto-Rhino-Laryngology - Head and Neck Surgery
2024

Mucopolysaccharidosis Type IIIE: A Real Human Disease or a Diagnostic Pitfall?

Diagnostics (Basel, Switzerland)
2025

Expanding the genetic landscape of Usher syndrome type IV caused by pathogenic ARSG variants.

Clinical genetics
2024

Concomitant ulcerative colitis and Usher syndrome in a Pakistani patient: A novel case report.

SAGE open medical case reports
2024

Usher Syndrome Type 2 in An Iranian Family: A Novel Founder Variation in The USH2A Gene.

Cell journal
2024

A novel compound heterozygous variant of MYO7A in Usher syndrome type 1.

Experimental eye research
2024

Investigating Splice Defects in USH2A Using Targeted Long-Read Sequencing.

Cells
2025

Long-term natural history of ellipsoid zone width in USH2A-retinopathy.

The British journal of ophthalmology
2024

Mild retinitis pigmentosa, including sector retinitis pigmentosa associated with 2 pathogenic variants in CDH23.

Ophthalmic genetics
2025

Balance Control Impairments in Usher Syndrome.

Ear and hearing
2024

A large-scale screening identified in USH2A gene the P3272L founder pathogenic variant explaining familial Usher syndrome in Sardinia, Italy.

BMC ophthalmology
2024

CDH23-Associated Usher Syndrome: Clinical Features, Retinal Imaging, and Natural History.

Investigative ophthalmology &amp; visual science
2024

Usher syndrome in the United Arab Emirates.

Ophthalmic genetics
2024

Generation of two induced pluripotent stem cell lines from an Usher syndrome type 1B patient with the homozygous c.496del MYO7A variant.

Stem cell research
2024

Long-Term Outcomes of Cochlear Implantation in Usher Syndrome.

Ear and hearing
2024

Adaptive optics retinal imaging in patients with usher syndrome.

Frontiers in ophthalmology
2023

A Case Report on Senear-Usher Syndrome.

Cureus
2024

Elasticity and Thermal Stability are Key Determinants of Hearing Rescue by Mini-Protocadherin-15 Proteins.

bioRxiv : the preprint server for biology
2024

Multicentric Longitudinal Prospective Study in a European Cohort of MYO7A Patients: Disease Course and Implications for Gene Therapy.

Investigative ophthalmology &amp; visual science
2024

High prevalence of exon-13 variants in USH2A-related retinal dystrophies in Taiwanese population.

Orphanet journal of rare diseases
2024

Optical coherence tomography biomarkers in MYO7A-inherited retinal dystrophy: longitudinal study in pediatric patients.

Graefe's archive for clinical and experimental ophthalmology = Albrecht von Graefes Archiv fur klinische und experimentelle Ophthalmologie
2024

Mutation spectrum of hearing loss patients in Northwest China: Identification of 20 novel variants.

Molecular genetics &amp; genomic medicine
2024

Self-Reported Functional Vision in USH2A-Associated Retinal Degeneration as Measured by the Michigan Retinal Degeneration Questionnaire.

Investigative ophthalmology &amp; visual science
2024

Mice with deficiency in Pcdh15, a gene associated with bipolar disorders, exhibit significantly elevated diurnal amplitudes of locomotion and body temperature.

Translational psychiatry
2024

A novel homozygous missense variant identified in the myosin VIIA motor domain of a Moroccan patient with usher syndrome.

Molecular biology reports
2024

Senear-Usher syndrome in a 5-year-old girl.

Postepy dermatologii i alergologii
2024

A New Mouse Model for Usher Syndrome Crossing Kunming Mice with CBA/J Mice.

Gene
2024

Nationwide Prevalence of Inherited Retinal Diseases in the Israeli Population.

JAMA ophthalmology
2025

Current updates on genetic spectrum of usher syndrome.

Nucleosides, nucleotides &amp; nucleic acids
2024

Current phenotypic and genetic spectrum of syndromic deafness in Tunisia: paving the way for precision auditory health.

Frontiers in genetics
2024

Pendular Nystagmus Presenting in Usher Syndrome Type I: A Case Report.

Journal of the American Academy of Audiology
2024

Syndromic Retinitis Pigmentosa: A 15-Patient Study.

Genes
2024

PRPS1-associated retinopathy: a diagnostic odyssey.

Ophthalmic genetics
2025

Optical coherence tomography (OCT) and OCT-angiography in syndromic versus non-syndromic USH2A-associated retinopathy.

European journal of ophthalmology
2024

The prevalence and clinical features of MYO7A-related hearing loss including DFNA11, DFNB2 and USH1B.

Scientific reports
2024

Pathophysiology of human hearing loss associated with variants in myosins.

Frontiers in physiology
2024

Navigating the Usher Syndrome Genetic Landscape: An Evaluation of the Associations between Specific Genes and Quality Categories of Cochlear Implant Outcomes.

Audiology research
2024

Exploring the support needs of Australian parents of young children with Usher syndrome: a qualitative thematic analysis.

Orphanet journal of rare diseases
2024

Disease-specific variant interpretation highlighted the genetic findings in 2325 Japanese patients with retinitis pigmentosa and allied diseases.

Journal of medical genetics
2025

CO-OCCURRING USHER SYNDROME TYPE 1 AND RENAL FAILURE.

Retinal cases &amp; brief reports
2024

Expanding the Genotype-Phenotype Correlations and Mutational Spectrum in Inherited Retinal Diseases: Novel and Recurrent Mutations.

Cureus
2024

Bioinformatics characterization of variants of uncertain significance in pediatric sensorineural hearing loss.

Frontiers in pediatrics
2024

Vestibulo-ocular reflex dynamics with head-impulses discriminates Usher patients type 1 and 2.

Scientific reports
2024

Lived experiences of genetic diagnosis for rare disease patients: a qualitative interview study.

Orphanet journal of rare diseases
2024

Spectrum of variants associated with inherited retinal dystrophies in Northeast Mexico.

BMC ophthalmology
2024

[Genetic characteristic analysis of slight-to-moderate sensorineural hearing loss in children].

Lin chuang er bi yan hou tou jing wai ke za zhi = Journal of clinical otorhinolaryngology head and neck surgery
2024

Optical coherence tomography in children with inherited retinal disease.

Clinical &amp; experimental optometry
2024

Extended time frame for restoring inner ear function through gene therapy in Usher1G preclinical model.

JCI insight
2024

Genetic profile of syndromic retinitis pigmentosa in Portugal.

Graefe's archive for clinical and experimental ophthalmology = Albrecht von Graefes Archiv fur klinische und experimentelle Ophthalmologie
2024

Late-onset mucopolysaccharidosis type IIIA mimicking Usher syndrome.

American journal of medical genetics. Part A
2023

Identification of a novel compound heterozygous pathogenic variant in MYO7A causing Usher syndrome type IB in a Chinese patient: a case report.

The Journal of international medical research
2023

Pathogenic Variants in USH1G/SANS Alter Protein Interaction with Pre-RNA Processing Factors PRPF6 and PRPF31 of the Spliceosome.

International journal of molecular sciences
2024

Functional Vision in Patients With Biallelic USH2A Variants.

American journal of ophthalmology
2023

Which Came First? When Usher Syndrome Type 1 Couples with Neuropsychiatric Disorders.

Audiology research
2023

Multimodal photoacoustic microscopy, optical coherence tomography, and fluorescence imaging of USH2A knockout rabbits.

Scientific reports
2023

[Advances on gene therapy for USH2A exon 13 related inherited retinal dystrophy].

[Zhonghua yan ke za zhi] Chinese journal of ophthalmology
2024

Overlap between ophthalmology and psychiatry - A narrative review focused on congenital and inherited conditions.

Psychiatry research
2024

The p.C759F Variant in USH2A Is a Pathogenic Mutation: Systematic Literature Review and Meta-Analysis of 667 Genotypes.

Ophthalmic research
2023

PCDH15 Dual-AAV Gene Therapy for Deafness and Blindness in Usher Syndrome Type 1F.

bioRxiv : the preprint server for biology
2023

Allelic hierarchy for USH2A influences auditory and visual phenotypes in South Korean patients.

Scientific reports
2023

Hearing Loss with Vision Impairment: Usher Syndrome. A Case of the East Democratic Republic of Congo.

Indian journal of otolaryngology and head and neck surgery : official publication of the Association of Otolaryngologists of India
2024

Novel heterozygous USH1C mutation impacts hair cell mechanotransduction and causes progressive hearing loss.

Science bulletin
2023

A Recalcitrant Case of Senear-Usher Syndrome Treated With Rituximab.

Cureus
2024

Outcomes of cochlear implantation in Usher syndrome: a systematic review.

European archives of oto-rhino-laryngology : official journal of the European Federation of Oto-Rhino-Laryngological Societies (EUFOS) : affiliated with the German Society for Oto-Rhino-Laryngology - Head and Neck Surgery
2023

Novel pathogenic WHRN variant causing hearing loss in a moroccan family.

Molecular biology reports
2023

Third-generation lentiviral gene therapy rescues function in a mouse model of Usher 1B.

Molecular therapy : the journal of the American Society of Gene Therapy
2023

Combined Presence in Heterozygosis of Two Variant Usher Syndrome Genes in Two Siblings Affected by Isolated Profound Age-Related Hearing Loss.

Biomedicines
2023

Cochlear Implantation in Children with Additional Disabilities: A Systematic Review.

Children (Basel, Switzerland)
2023

Dual AAV-based PCDH15 gene therapy achieves sustained rescue of visual function in a mouse model of Usher syndrome 1F.

Molecular therapy : the journal of the American Society of Gene Therapy
2023

Prevalence of inherited retinal diseases in a large Egyptian cohort.

BMC ophthalmology
2024

The effects of ush2a gene knockout on vesicle transport in photoreceptors.

Gene
2023

Carriers of autosomal recessive conditions: are they really 'unaffected?'.

Journal of medical genetics
2023

Gene Therapy in Hereditary Retinal Dystrophies: The Usefulness of Diagnostic Tools in Candidate Patient Selections.

International journal of molecular sciences
2023

Clinical Presentation of Usher Syndrome Type 1B (USH1B) in a 10-Month-Old: A Case Report.

Cureus
2023

Autosomal dominant non-syndromic hearing loss caused by a novel mutation in MYO7A: A case report and review of the literature.

World journal of clinical cases
2023

Expression of the human usherin c.2299delG mutation leads to early-onset auditory loss and stereocilia disorganization.

Communications biology
2023

Dual-AAV vector-mediated expression of MYO7A improves vestibular function in a mouse model of Usher syndrome 1B.

Molecular therapy. Methods &amp; clinical development
2023

Molecular regulatory mechanism of human myosin-7a.

The Journal of biological chemistry
Ver todos os 796 no EuropePMC

Associações

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Comunidades

Grupos ativos de quem convive com esta doença aqui no Raras

Ainda não existe comunidade no Raras para Síndrome Usher

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Doenças relacionadas

Doenças com sintomas parecidos — ajudam quem ainda está buscando diagnóstico

Referências e fontes

Bases de dados externas citadas neste artigo

Publicações científicas

Artigos indexados no PubMed ligados a esta doença no grafo RarasNet — título, periódico e PMID direto da fonte, sem intermediação de IA.

  1. Generation of the induced pluripotent stem cell line BTHBIOi002-A derived from a USH2 patient with c.2512C&gt;T and c.2802T&gt;G mutations in USH2A gene.
    Stem cell research· 2026· PMID 41763034mais citado
  2. Nonsense Mutation in USH2A Exon-13 Activates the Innate Immune Response in M&#xfc;ller Glial Cells.
    International journal of molecular sciences· 2026· PMID 41751772mais citado
  3. Characterization of Usher Syndrome Type 2-Associated Proteins in the Retina via Affinity Purification-Mass Spectrometry.
    Molecular &amp; cellular proteomics : MCP· 2026· PMID 41654259mais citado
  4. Compensatory Interplay Between Clarin-1 and Clarin-2 Deafness-Associated Proteins Governs Phenotypic Variability in Hearing.
    Advanced science (Weinheim, Baden-Wurttemberg, Germany)· 2026· PMID 41572507mais citado
  5. Exploring extracellular vesicle MicroRNAs in Usher syndrome type 1B: Tear-Derived EVs as potential indicators of retinal health.
    Cellular and molecular life sciences : CMLS· 2026· PMID 41528484mais citado
  6. A regulatory axis for tonotopic MYO7A expression in cochlear hair cells.
    Proc Natl Acad Sci U S A· 2026· PMID 41945437recente
  7. From Usher syndrome to Bardet-Biedl syndrome: Diagnosis after an atypical presentation.
    Clin Nephrol Case Stud· 2026· PMID 41940113recente
  8. Identification of a recessive PCDH15 nonsense variant in purebred goats with vestibular dysfunction.
    Vet Anim Sci· 2026· PMID 41907450recente
  9. Clinical Details of Low-Frequency Hearing Loss Observed in Autosomal Dominant MYO7A-Associated Hearing Loss Patients.
    Genes (Basel)· 2026· PMID 41898848recente
  10. Deciphering the genetic basis of inherited retinal dystrophies via whole-exome sequencing in a Turkish cohort.
    Mol Vis· 2025· PMID 41867366recente

Bases de dados e fontes oficiais

Identificadores e referências canônicas usadas para montar este verbete.

  1. ORPHA:886(Orphanet)
  2. MONDO:0019501(MONDO)
  3. GARD:7843(GARD (NIH))
  4. Variantes catalogadas(ClinVar)
  5. Busca completa no PubMed(PubMed)
  6. Artigo Wikipedia(Wikipedia)
  7. Q917399(Wikidata)

Dados compilados pelo RarasNet a partir de fontes abertas (Orphanet, OMIM, MONDO, PubMed/EuropePMC, ClinicalTrials.gov, DATASUS, PCDT/MS). Este conteúdo é informativo e não substitui avaliação médica.

Conteúdo mantido por Agente Raras · Médicos e pesquisadores podem colaborar

Síndrome Usher
Compêndio · Raras BR

Síndrome Usher

ORPHA:886 · MONDO:0019501
Prevalência
1-9 / 100 000
Herança
Autosomal recessive
CID-10
H35.5 · Distrofias hereditárias da retina
CID-11
Ensaios
13 ativos
Início
Childhood, Infancy, Neonatal
Prevalência
4.53 (Worldwide)
MedGen
UMLS
C1568248
EuropePMC
Wikidata
Wikipedia
Papers 10a
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