Raras
Buscar doenças, sintomas, genes...
Artrogripose distal
ORPHA:97120DOENÇA RARA

Doença do tecido muscular caracterizada por contraturas articulares congênitas das mãos e dos pés.

Mantido por Agente Raras·Colaborar como especialista →

Introdução

O que você precisa saber de cara

📋

Doença do tecido muscular caracterizada por contraturas articulares congênitas das mãos e dos pés.

Publicações científicas
368 artigos
Último publicado: 2026 Feb 21
🏥
SUS: Sem cobertura SUSScore: 0%
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Sinais e sintomas

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

Partes do corpo afetadas

🦴
Ossos e articulações
70 sintomas
😀
Face
46 sintomas
💪
Músculos
32 sintomas
🧠
Neurológico
24 sintomas
👁️
Olhos
14 sintomas
❤️
Coração
13 sintomas

+ 132 sintomas em outras categorias

Características mais comuns

Assimetria facial
Artralgia
Olho profundamente inserido
Desvio fibular dos dedos do pé
Disestesia
Sinostose tarsal
383sintomas
Sem dados (383)

Os sintomas variam de pessoa para pessoa. Abaixo estão as 383 características clínicas mais associadas, ordenadas por frequência.

Assimetria facialFacial asymmetry
ArtralgiaArthralgia
Olho profundamente inseridoDeeply set eye
Desvio fibular dos dedos do péFibular deviation of toes
DisestesiaDysesthesia

Linha do tempo da pesquisa

Publicações por ano — veja quando o interesse científico cresceu
Anos de pesquisa1desde 2026
Total histórico368PubMed
Últimos 10 anos200publicações
Pico202124 papers
Linha do tempo
2026Hoje · 2026🧪 2010Primeiro ensaio clínico📈 2021Ano 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.

ECEL1Endothelin-converting enzyme-like 1Disease-causing germline mutation(s) inTolerante
FUNÇÃO

May contribute to the degradation of peptide hormones and be involved in the inactivation of neuronal peptides

LOCALIZAÇÃO

Membrane

MECANISMO DE DOENÇA

Arthrogryposis, distal, 5D

An autosomal recessive form of distal arthrogryposis, a disease characterized by congenital joint contractures that mainly involve two or more distal parts of the limbs, in the absence of a primary neurological or muscle disease. DA5D is characterized by severe camptodactyly of the hands, mild camptodactyly of the toes, clubfoot and/or a calcaneovalgus deformity, extension contractures of the knee, unilateral ptosis or ptosis that is more severe on one side, a round-shaped face, arched eyebrows, a bulbous upturned nose, and micrognathia. Patients do not have ophthalmoplegia.

EXPRESSÃO TECIDUAL(Tecido-específico)
Ovário
134.1 TPM
Hipotálamo
47.3 TPM
Pituitária
31.0 TPM
Glândula adrenal
8.1 TPM
Brain Caudate basal ganglia
6.6 TPM
OUTRAS DOENÇAS (1)
distal arthrogryposis type 5D
HGNC:3147UniProt:O95672
TNNT3Troponin T, fast skeletal muscleDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Troponin T is the tropomyosin-binding subunit of troponin, the thin filament regulatory complex which confers calcium-sensitivity to striated muscle actomyosin ATPase activity

LOCALIZAÇÃO

VIAS BIOLÓGICAS (1)
Striated Muscle Contraction
MECANISMO DE DOENÇA

Arthrogryposis, distal, 2B2

A form of distal arthrogryposis, a disease characterized by congenital joint contractures that mainly involve two or more distal parts of the limbs, in the absence of a primary neurological or muscle disease. Distal arthrogryposis type 2 is characterized by contractures of the hands and feet, and a distinctive face characterized by prominent nasolabial folds, small mouth and downslanting palpebral fissures. DA2B2 inheritance is autosomal dominant.

EXPRESSÃO TECIDUAL(Ubíquo)
Músculo esquelético
2354.3 TPM
Coração - Ventrículo esquerdo
33.7 TPM
Tecido adiposo
25.6 TPM
Coração - Átrio
23.6 TPM
Fallopian Tube
19.1 TPM
OUTRAS DOENÇAS (3)
arthrogryposis, distal, type 2B2digitotalar dysmorphismSheldon-hall syndrome
HGNC:11950UniProt:P45378
PIEZO2Piezo-type mechanosensitive ion channel component 2Disease-causing germline mutation(s) inRestrito
FUNÇÃO

Pore-forming subunit of the mechanosensitive non-specific cation Piezo channel required for rapidly adapting mechanically activated (MA) currents and has a key role in sensing touch and tactile pain (PubMed:37590348). Piezo channels are homotrimeric three-blade propeller-shaped structures that utilize a cap-motion and plug-and-latch mechanism to gate their ion-conducting pathways (PubMed:37590348). Expressed in sensory neurons, is essential for diverse physiological processes, including respirat

LOCALIZAÇÃO

Cell membrane

VIAS BIOLÓGICAS (1)
Turbulent (oscillatory, disturbed) flow shear stress activates signaling by PIEZO1 and integrins in endothelial cells
MECANISMO DE DOENÇA

Arthrogryposis, distal, 5

A form of distal arthrogryposis, a disease characterized by congenital joint contractures that mainly involve two or more distal parts of the limbs, in the absence of a primary neurological or muscle disease. DA5 features include ocular abnormalities, typically ptosis, ophthalmoplegia and/or strabismus, in addition to contractures of the skeletal muscles. Some patients have pulmonary hypertension as a result of restrictive lung disease.

EXPRESSÃO TECIDUAL(Tecido-específico)
Pulmão
8.0 TPM
Brain Spinal cord cervical c-1
7.6 TPM
Nervo tibial
5.2 TPM
Cólon sigmoide
5.0 TPM
Próstata
3.4 TPM
OUTRAS DOENÇAS (4)
Gordon syndromearthrogryposis- oculomotor limitation-electroretinal anomalies syndromearthrogryposis, distal, with impaired proprioception and touchMarden-Walker syndrome
HGNC:26270UniProt:Q9H5I5
TPM2Tropomyosin beta chainDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Binds to actin filaments in muscle and non-muscle cells. Plays a central role, in association with the troponin complex, in the calcium dependent regulation of vertebrate striated muscle contraction. Smooth muscle contraction is regulated by interaction with caldesmon. In non-muscle cells is implicated in stabilizing cytoskeleton actin filaments. The non-muscle isoform may have a role in agonist-mediated receptor internalization

LOCALIZAÇÃO

Cytoplasm, cytoskeleton

VIAS BIOLÓGICAS (2)
Smooth Muscle ContractionStriated Muscle Contraction
MECANISMO DE DOENÇA

Congenital myopathy 23

An autosomal dominant muscular disorder characterized clinically by hypotonia and muscle weakness, and a static or slowly progressive clinical course. Disease onset ranges from birth to childhood. Histologic examination of muscle fibers shows various anomalies including fiber type disproportion, an irregular myofibrillar network, abnormal thread-like or rod-shaped structures, and cap-like structures which are well demarcated and peripherally located under the sarcolemma with abnormal accumulation of sarcomeric proteins.

EXPRESSÃO TECIDUAL(Ubíquo)
Artéria tibial
4226.1 TPM
Músculo esquelético
4057.1 TPM
Esôfago - Muscular
4024.0 TPM
Esôfago - Junção
3694.6 TPM
Aorta
3154.9 TPM
OUTRAS DOENÇAS (8)
congenital myopathy 23arthrogryposis, distal, type 1ASheldon-hall syndromecap myopathy
HGNC:12011UniProt:P07951
DSEDermatan-sulfate epimeraseDisease-causing germline mutation(s) inRestrito
FUNÇÃO

Converts D-glucuronic acid to L-iduronic acid (IdoUA) residues. Plays an important role in the biosynthesis of the glycosaminoglycan/mucopolysaccharide dermatan sulfate

LOCALIZAÇÃO

Endoplasmic reticulum membraneGolgi apparatus membraneCytoplasmic vesicle membraneMicrosome membrane

VIAS BIOLÓGICAS (1)
DS-GAG biosynthesis
MECANISMO DE DOENÇA

Ehlers-Danlos syndrome, musculocontractural type 2

A form of Ehlers-Danlos syndrome characterized by progressive multisystem manifestations, including joint dislocations and deformities, skin hyperextensibility, skin bruisability and fragility with recurrent large subcutaneous hematomas, cardiac valvular, respiratory, gastrointestinal, and ophthalmologic complications. Motor developmental delay is associated with muscle hypoplasia, muscle weakness, and an abnormal muscle fiber pattern in histology in adulthood.

VIAS REACTOME (1)
EXPRESSÃO TECIDUAL(Ubíquo)
Fibroblastos
45.3 TPM
Nervo tibial
27.5 TPM
Tecido adiposo
25.0 TPM
Adipose Visceral Omentum
22.4 TPM
Linfócitos
19.8 TPM
INTERAÇÕES PROTEICAS (4)
OUTRAS DOENÇAS (2)
Ehlers-Danlos syndrome, musculocontractural type 2Ehlers-Danlos syndrome, musculocontractural type
HGNC:21144UniProt:Q9UL01
CHST14Carbohydrate sulfotransferase 14Disease-causing germline mutation(s) inTolerante
FUNÇÃO

Catalyzes the transfer of sulfate to position 4 of the N-acetylgalactosamine (GalNAc) residue of dermatan sulfate. Plays a pivotal role in the formation of 4-0-sulfated IdoA blocks in dermatan sulfate. Transfers sulfate to the C-4 hydroxyl of beta1,4-linked GalNAc that is substituted with an alpha-linked iduronic acid (IdoUA) at the C-3 hydroxyl. Transfers sulfate more efficiently to GalNAc residues in -IdoUA-GalNAc-IdoUA- than in -GlcUA-GalNAc-GlcUA-sequences. Has preference for partially desul

LOCALIZAÇÃO

Golgi apparatus membrane

VIAS BIOLÓGICAS (1)
DS-GAG biosynthesis
MECANISMO DE DOENÇA

Ehlers-Danlos syndrome, musculocontractural type 1

A form of Ehlers-Danlos syndrome characterized by distinctive craniofacial dysmorphism, congenital contractures of thumbs and fingers, clubfeet, severe kyphoscoliosis, muscular hypotonia, hyperextensible thin skin with easy bruisability and atrophic scarring, wrinkled palms, joint hypermobility, and ocular involvement.

OUTRAS DOENÇAS (2)
Ehlers-Danlos syndrome, musculocontractural type 1Ehlers-Danlos syndrome, musculocontractural type
HGNC:24464UniProt:Q8NCH0
MYH3Myosin-3Disease-causing germline mutation(s) inTolerante
FUNÇÃO

Muscle contraction

LOCALIZAÇÃO

Cytoplasm, myofibril

VIAS BIOLÓGICAS (1)
Striated Muscle Contraction
MECANISMO DE DOENÇA

Arthrogryposis, distal, 2A

A form of distal arthrogryposis, a disease characterized by congenital joint contractures that mainly involve two or more distal parts of the limbs, in the absence of a primary neurological or muscle disease. DA2A is characterized by contractures of the hands and feet, oropharyngeal abnormalities, scoliosis, and a distinctive face that includes a very small oral orifice, puckered lips, and a H-shaped dimple of the chin.

EXPRESSÃO TECIDUAL(Ubíquo)
Testículo
17.2 TPM
Próstata
9.7 TPM
Cervix Ectocervix
9.2 TPM
Tireoide
8.6 TPM
Cervix Endocervix
8.5 TPM
OUTRAS DOENÇAS (8)
contractures, pterygia, and variable skeletal fusions syndrome 1Bcontractures, pterygia, and spondylocarpotarsal fusion syndrome 1Aarthrogryposis, distal, type 2B3Freeman-Sheldon syndrome
HGNC:7573UniProt:P11055
FBN2Fibrillin-2Disease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Fibrillins are structural components of 10-12 nm extracellular calcium-binding microfibrils, which occur either in association with elastin or in elastin-free bundles. Fibrillin-2-containing microfibrils regulate the early process of elastic fiber assembly. Regulates osteoblast maturation by controlling TGF-beta bioavailability and calibrating TGF-beta and BMP levels, respectively Hormone secreted by trophoblasts that promotes trophoblast invasiveness (PubMed:32329225). Has glucogenic activity:

LOCALIZAÇÃO

SecretedSecreted, extracellular space, extracellular matrix

VIAS BIOLÓGICAS (1)
Elastic fibre formation
MECANISMO DE DOENÇA

Contractural arachnodactyly, congenital

An autosomal dominant connective tissue disorder characterized by contractures, arachnodactyly, scoliosis, and crumpled ears.

EXPRESSÃO TECIDUAL(Tecido-específico)
Fibroblastos
126.7 TPM
Testículo
6.0 TPM
Glândula adrenal
5.0 TPM
Esôfago - Muscular
2.9 TPM
Coração - Ventrículo esquerdo
2.7 TPM
OUTRAS DOENÇAS (2)
macular degeneration, early-onsetcongenital contractural arachnodactyly
HGNC:3604UniProt:P35556
SLC35A3UDP-N-acetylglucosamine transporterDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Transports diphosphate-N-acetylglucosamine (UDP-GlcNAc) from the cytosol into the lumen of the Golgi apparatus, functioning as an antiporter that exchanges UDP-N-acetyl-alpha-D-glucosamine for UMP (PubMed:10393322). May supply UDP-GlcNAc as substrate for Golgi-resident glycosyltransferases that generate highly branched, multiantennary complex N-glycans and keratan sulfate (PubMed:23766508, PubMed:34981577). However, the exact role of SLC35A3 still needs to be elucidated, it could be a member of

LOCALIZAÇÃO

Golgi apparatus membrane

VIAS BIOLÓGICAS (1)
Transport of nucleotide sugars
MECANISMO DE DOENÇA

Arthrogryposis, impaired intellectual development, and seizures

A disease characterized by arthrogryposis, intellectual disability, autism spectrum disorder, and epilepsy. Additional features include limb malformations, distal joint involvement, microcephaly, retromicrognathia, and general muscle hypotonia.

EXPRESSÃO TECIDUAL(Ubíquo)
Cólon transverso
9.7 TPM
Glândula salivar
8.7 TPM
Intestino delgado
8.6 TPM
Tireoide
7.4 TPM
Vagina
7.3 TPM
OUTRAS DOENÇAS (1)
autism spectrum disorder - epilepsy - arthrogryposis syndrome
HGNC:11023UniProt:Q9Y2D2
ADAMTS15A disintegrin and metalloproteinase with thrombospondin motifs 15Disease-causing germline mutation(s) inRestrito
FUNÇÃO

Metalloprotease which has proteolytic activity against the proteoglycan VCAN, cleaving it at the 'Glu-1428-|-1429-Ala' site. Cleaves VCAN in the pericellular matrix surrounding myoblasts, facilitating myoblast contact and fusion which is required for skeletal muscle development and regeneration

LOCALIZAÇÃO

Secreted, extracellular space, extracellular matrixCell surface

VIAS BIOLÓGICAS (1)
O-glycosylation of TSR domain-containing proteins
MECANISMO DE DOENÇA

Arthrogryposis, distal, 12

A form of distal arthrogryposis, a disease characterized by congenital joint contractures that mainly involve two or more distal parts of the limbs, in the absence of a primary neurological or muscle disease. DA12 is an autosomal recessive form characterized by congenital contractures, primarily affecting the small joints of the fingers and toes. Additional features include knee, Achilles tendon, and toe contractures, spinal stiffness, scoliosis, and orthodontic abnormalities.

OUTRAS DOENÇAS (1)
arthrogryposis, distal, type 12
HGNC:HGNC:16305UniProt:Q8TE58
METHepatocyte growth factor receptorDisease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Receptor tyrosine kinase that transduces signals from the extracellular matrix into the cytoplasm by binding to hepatocyte growth factor/HGF ligand. Regulates many physiological processes including proliferation, scattering, morphogenesis and survival. Ligand binding at the cell surface induces autophosphorylation of MET on its intracellular domain that provides docking sites for downstream signaling molecules. Following activation by ligand, interacts with the PI3-kinase subunit PIK3R1, PLCG1,

LOCALIZAÇÃO

MembraneSecreted

VIAS BIOLÓGICAS (7)
MET Receptor ActivationDrug-mediated inhibition of MET activationSema4D mediated inhibition of cell attachment and migrationNegative regulation of MET activityMECP2 regulates neuronal receptors and channels
EXPRESSÃO TECIDUAL(Ubíquo)
Nervo tibial
21.3 TPM
Fibroblastos
18.5 TPM
Tireoide
16.7 TPM
Adipose Visceral Omentum
16.2 TPM
Mama
15.4 TPM
OUTRAS DOENÇAS (8)
hepatocellular carcinomahereditary papillary renal cell carcinomaarthrogryposis, distal, IIa 11autosomal recessive nonsyndromic hearing loss 97
HGNC:7029UniProt:P08581
MYL11Myosin regulatory light chain 11Disease-causing germline mutation(s) inModerado
FUNÇÃO

Myosin regulatory subunit that plays an essential role to maintain muscle integrity during early development (By similarity). Plays a role in muscle contraction (By similarity)

LOCALIZAÇÃO

VIAS BIOLÓGICAS (1)
Smooth Muscle Contraction
MECANISMO DE DOENÇA

Arthrogryposis, distal, 1C

A form of distal arthrogryposis, a disease characterized by congenital joint contractures that mainly involve two or more distal parts of the limbs, in the absence of a primary neurological or muscle disease. DA1C patients show multiple congenital contractures, scoliosis, short stature, and segmental amyoplasia. DA1C inheritance can be autosomal recessive or autosomal dominant.

OUTRAS DOENÇAS (1)
arthrogryposis, distal, type 1C
HGNC:HGNC:29824UniProt:Q96A32
TNNI2Troponin I, fast skeletal muscleDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Troponin I is the inhibitory subunit of troponin, the thin filament regulatory complex which confers calcium-sensitivity to striated muscle actomyosin ATPase activity

LOCALIZAÇÃO

VIAS BIOLÓGICAS (1)
Striated Muscle Contraction
MECANISMO DE DOENÇA

Arthrogryposis, distal, 2B1

A form of distal arthrogryposis, a disease characterized by congenital joint contractures that mainly involve two or more distal parts of the limbs, in the absence of a primary neurological or muscle disease. DA2B is characterized by contractures of the hands and feet, and a distinctive face characterized by prominent nasolabial folds, small mouth and downslanting palpebral fissures. DA2B1 inheritance is autosomal dominant.

EXPRESSÃO TECIDUAL(Ubíquo)
Músculo esquelético
3067.4 TPM
Skin Sun Exposed Lower leg
41.9 TPM
Skin Not Sun Exposed Suprapubic
36.3 TPM
Glândula salivar
30.8 TPM
Sangue
17.6 TPM
OUTRAS DOENÇAS (3)
distal arthrogryposis type 2B1Sheldon-hall syndromedigitotalar dysmorphism
HGNC:11946UniProt:P48788
MYH8Myosin-8Disease-causing germline mutation(s) inTolerante
FUNÇÃO

Muscle contraction

LOCALIZAÇÃO

Cytoplasm, myofibril

VIAS BIOLÓGICAS (1)
Striated Muscle Contraction
MECANISMO DE DOENÇA

Carney complex variant

Carney complex is a multiple neoplasia syndrome characterized by spotty skin pigmentation, cardiac and other myxomas, endocrine tumors, and psammomatous melanotic schwannomas. Familial cardiac myxomas are associated with spotty pigmentation of the skin and other phenotypes, including primary pigmented nodular adrenocortical dysplasia, extracardiac (frequently cutaneous) myxomas, schwannomas, and pituitary, thyroid, testicular, bone, ovarian, and breast tumors. Cardiac myxomas do not develop in all patients with the Carney complex, but affected patients have at least two features of the complex or one feature and a clinically significant family history.

EXPRESSÃO TECIDUAL(Baixa expressão)
Músculo esquelético
3.9 TPM
Rim - Medula
1.2 TPM
Testículo
0.5 TPM
Rim - Córtex
0.1 TPM
Baço
0.1 TPM
OUTRAS DOENÇAS (2)
trismus-pseudocamptodactyly syndromeCarney complex - trismus - pseudocamptodactyly syndrome
HGNC:7578UniProt:P13535
NALCNSodium leak channel NALCNCandidate gene tested inRestrito
FUNÇÃO

Voltage-gated ion channel responsible for the resting Na(+) permeability that controls neuronal excitability (PubMed:17448995, PubMed:31409833). NALCN channel functions as a multi-protein complex, which consists at least of NALCN, NALF1, UNC79 and UNC80 (PubMed:32494638, PubMed:33203861). NALCN is the voltage-sensing, pore-forming subunit of the NALCN channel complex (PubMed:17448995). NALCN channel complex is constitutively active and conducts monovalent cations but is blocked by physiological

LOCALIZAÇÃO

Cell membrane

VIAS BIOLÓGICAS (1)
Stimuli-sensing channels
MECANISMO DE DOENÇA

Hypotonia, infantile, with psychomotor retardation and characteristic facies 1

A neurodegenerative disease characterized by variable degrees of hypotonia, speech impairment, intellectual disability, pyramidal signs, subtle facial dysmorphism, and chronic constipation. Some patients manifest neuroaxonal dystrophy, optic atrophy, unmyelinated axons and spheroid bodies in tissue biopsies.

VIAS REACTOME (1)
EXPRESSÃO TECIDUAL(Tecido-específico)
Cérebro - Hemisfério cerebelar
17.0 TPM
Cerebelo
14.6 TPM
Brain Spinal cord cervical c-1
12.0 TPM
Brain Frontal Cortex BA9
11.4 TPM
Pituitária
10.8 TPM
OUTRAS DOENÇAS (5)
hypotonia, infantile, with psychomotor retardation and characteristic facies 1congenital contractures of the limbs and face, hypotonia, and developmental delaydigitotalar dysmorphismSheldon-hall syndrome
HGNC:19082UniProt:Q8IZF0
MYBPC1Myosin-binding protein C, slow-typeDisease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Thick filament-associated protein located in the crossbridge region of vertebrate striated muscle a bands. Slow skeletal protein that binds to both myosin and actin (PubMed:31025394, PubMed:31264822). In vitro, binds to native thin filaments and modifies the activity of actin-activated myosin ATPase. May modulate muscle contraction or may play a more structural role

LOCALIZAÇÃO

VIAS BIOLÓGICAS (1)
Striated Muscle Contraction
MECANISMO DE DOENÇA

Arthrogryposis, distal, 1B

A form of distal arthrogryposis, a disease characterized by congenital joint contractures that mainly involve two or more distal parts of the limbs, in the absence of a primary neurological or muscle disease. Distal arthrogryposis type 1 is characterized largely by camptodactyly and clubfoot. Hypoplasia and/or absence of some interphalangeal creases is common. The shoulders and hips are less frequently affected.

EXPRESSÃO TECIDUAL(Tecido-específico)
Músculo esquelético
3635.5 TPM
Próstata
54.5 TPM
Substância negra
28.0 TPM
Hipotálamo
19.2 TPM
Brain Nucleus accumbens basal ganglia
16.2 TPM
OUTRAS DOENÇAS (6)
lethal congenital contracture syndrome 4myopathy, congenital, with tremorarthrogryposis, distal, type 1Bdigitotalar dysmorphism
HGNC:7549UniProt:Q00872

Variantes genéticas (ClinVar)

585 variantes patogênicas registradas no ClinVar.

🧬 ECEL1: NM_004826.4(ECEL1):c.2152-15C>A ()
🧬 ECEL1: GRCh37/hg19 2q33.3-37.3(chr2:206965837-242783384)x3 ()
🧬 ECEL1: NM_004826.4(ECEL1):c.1185-12C>G ()
🧬 ECEL1: NM_004826.4(ECEL1):c.270_276dup (p.Gly93fs) ()
🧬 ECEL1: NM_004826.4(ECEL1):c.835C>T (p.Gln279Ter) ()
Ver todas no ClinVar

Classificação de variantes (ClinVar)

Distribuição de 313 variantes classificadas pelo ClinVar.

188
125
Patogênica (60.1%)
VUS (39.9%)
VARIANTES MAIS SIGNIFICATIVAS
ECEL1: NM_004826.4(ECEL1):c.2152-15C>A [Likely pathogenic]
TNNI2: NM_003282.4(TNNI2):c.486G>C (p.Arg162Ser) [Likely pathogenic]
ECEL1: NM_004826.4(ECEL1):c.835C>T (p.Gln279Ter) [Likely pathogenic]
ECEL1: NM_004826.4(ECEL1):c.1933C>T (p.Arg645Ter) [Likely pathogenic]
ECEL1: NM_004826.4(ECEL1):c.1303C>T (p.Gln435Ter) [Likely pathogenic]

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.
·Pré-clínico3
Medicamentos catalogadosEnsaios clínicos· 0 medicamentos · 3 ensaios
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Onde tratar no SUS

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

🇧🇷 Atendimento SUS — Artrogripose distal

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Selecione um estado ou use sua localização para ver resultados.

Dados de DATASUS/CNES, SBGM, ABNeuro e Ministério da Saúde. Sempre confirme a disponibilidade diretamente com o estabelecimento.

Pesquisa ativa

Ensaios clínicos abertos e novidades científicas recentes

Pesquisa e ensaios clínicos

4 ensaios clínicos encontrados.

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Publicações mais relevantes

Timeline de publicações
208 papers (10 anos)
#1

Distal arthrogryposis with impaired proprioception and touch: description of 9 additional cases harbouring novel PIEZO2 variants and literature review.

European journal of paediatric neurology : EJPN : official journal of the European Paediatric Neurology Society2026 Feb 21

Mechanosensation is the ability to detect dynamic mechanical stimuli and is essential for many processes, including sense of touch on the skin. PIEZO2 is a functional ion channel assembled by three monomers and is an essential mechanotransducer for touch, proprioception, and interoception. Heterozygous pathogenic variants in PIEZO2 gene are associated with distal arthrogryposis type 3 (MIM:114300) and type 5 (MIM:108145), and with Marden-Walker Syndrome (MIM:248700). Recessive pathogenic variants in PIEZO2 are associated with distal arthrogryposis with impaired proprioception and touch (DAIPT) (MIM:617146). Papers describing patient cohorts in literature are few. Here we described 9 patients from 8 families with a very similar clinical picture characterized by neonatal respiratory distress, hypotonia, delayed motor development, sensory ataxia, foot deformities, hyperlaxity, progressive scoliosis, and skeletal contractures. We also carried out a review of patients reported in literature with recessive PIEZO2 variants. We retrospectively analyzed clinical and genetic results of 5 patients followed at Bambino Gesù Children Hospital and 4 patients followed at Neuropediatric Unit, Clinica Meds, Santiago, Chile. In our cohort, we identified nine patients harbouring PIEZO2 variants. Among them, eight patients carried single nucleotide variants (SNVs): three were compound heterozygous, two were homozygous, and two harboured two heterozygous variants of unknown allelic phase. Additionally, one other patient, who presented with a highly suggestive clinical phenotype, was found to harbour only a single heterozygous maternally inherited, frameshift variant. Only one patient was found to have a large homozygous copy number variant (CNV). Our cohort clinical phenotype, even though of variable severity, is highly concordant between the patients and literature data. To our knowledge this is one of the largest cohort of patients with recessive PIEZO2 variants so far.

#2

Biallelic MYH3 Variants Cause Distal Arthrogryposis in Compound Heterozygosity and a Subclinical Phenotype in Simple Heterozygosity. Codominance or Recessive Inheritance?

Clinical genetics2026 Jan 21

Distal arthrogryposis constitutes a highly heterogeneous group of disorders with a critical need for clear classification. Phenotypes have traditionally been characterized using the classification system proposed by Bamshad or Hall for distal arthrogryposis. Recessive MYH3 inheritance has been described in contractures, pterygia and spondylocarpotarsal fusion syndrome, and, more recently, in distal arthrogryposis without skeletal fusion. We hereby report a nuclear family affected by distal arthrogryposis with biallelic MYH3-related disorder, identifying two novel variants, which in the heterozygous state yield a subclinical phenotype. Beyond refining the molecular diagnosis and guiding genetic counseling, our study emphasizes that the classification of MYH3-related disorders and their inheritance modes is still evolving, underscoring the need to integrate knowledge gained from careful analyses.

#3

Expanding the Genotype-Phenotype Correlation of Marden-Walker Syndrome due to PIEZO2 Gene Variants: A Case Report From Brazil.

American journal of medical genetics. Part A2026 Jan 22

Marden-Walker syndrome (MWS; OMIM 248700) is an extremely rare congenital disorder characterized by multiple joint contractures, craniofacial dysmorphism, neurological abnormalities, and multisystem involvement. Although historically diagnosed on clinical grounds, only a few cases have been molecularly confirmed. Here, we describe a Brazilian female infant with classic manifestations of MWS, carrying a heterozygous pathogenic variant in the PIEZO2 gene not previously reported in MWS. To our knowledge, this is the first molecularly confirmed MWS case from Brazil, thus expanding both the genotype-phenotype spectrum and geographic distribution of PIEZO2-related disorders. Comparative analysis of previously reported molecularly confirmed cases reveals shared core features and highlights the prominent neurological involvement observed in our patient. A review of individuals with the same PIEZO2 variant demonstrates marked phenotypic variability-from Gordon syndrome to distal arthrogryposis type 5-underscoring allelic heterogeneity and variable expressivity. This case refines the phenotypic spectrum of PIEZO2-related disorders and illustrates how allelic heterogeneity contributes to wide clinical variability, while also underscoring the importance of including underrepresented populations in variant interpretation.

#4

Impact of growth hormone treatment on a 12-year-old female with newly diagnosed panhypopituitarism and distal arthrogryposis.

Endocrinology, diabetes & metabolism case reports2026 Jan 01

Panhypopituitarism, characterized by multiple pituitary hormone deficiencies, is most often diagnosed in infancy or early childhood with adolescent presentation being uncommon. We present a 12-year-old female with late-onset panhypopituitarism presenting with short stature and concomitant bilateral distal arthrogryposis. Her height at presentation was 140.7 cm (6th percentile) with a growth velocity of 0.75 cm/year, Tanner stage 1, bone age consistent with chronological age, and predicted adult height of approximately 152.4 cm despite a mid-parental target near 167.6 cm. Laboratory testing supported growth hormone (GH) deficiency, central hypothyroidism, and adrenal insufficiency, with stimulation tests being subnormal. MRI showed hypoplasia of the anterior pituitary, an absent infundibulum, and an ectopic posterior pituitary, consistent with pituitary stalk interruption syndrome. Hydrocortisone (9 mg/m2/day) and levothyroxine (37.5 μg daily) were first initiated. Although GH was initially deferred due to concerns about worsening preexisting distal hand contractures, the family elected to begin weekly subcutaneous lonapegsomatropin-tcgd (Skytrofa, 7.6 mg, 0.24 mg/kg/week). At 3 months, the growth velocity increased to 8.7 cm/year with early breast development, and at 6 months, it reached 17.4 cm/year, bone age remained concordant with chronological age, and predicted adult height improved to approximately 162.6 cm. By 10 months, height percentile rose to the 12th percentile and Tanner stage 2 breast development was observed. Throughout treatment, there were no reported or observed changes in distal arthrogryposis hand contractures. This case report highlights that initiation of GH therapy may lead to a significant growth improvement without aggravating arthrogryposis-related contractures. To report the rare co-occurrence of arthrogryposis and panhypopituitarism and discuss management. Distal arthrogryposis coexisting with GH deficiency did not worsen with GH therapy in this patient, supporting individualized risk-benefit analysis. Shared decision making is key when theoretical risks (e.g. joint symptoms) are weighed against the consequences of untreated GH deficiency.

#5

Psychometric properties of functional mobility outcome measures in children with arthrogryposis multiplex congenita.

Developmental medicine and child neurology2025 Oct 22

To establish the construct validity, agreement, and minimal important difference (MID) of widely used mobility measures in arthrogryposis multiplex congenita (AMC). Participants (n = 248, 126 males, mean age 10 years 10 months, standard deviation 3 years 11 months) with AMC were assessed using the Functional Mobility Scale (FMS), Gillette Functional Assessment Questionnaire (FAQ), Functional Independence Measure for Children (WeeFIM), and Patient-Reported Outcomes Measurement Information System (PROMIS). Convergent and discriminant validity were evaluated using Spearman's rank correlations, while known-groups validity was examined using analysis of variance. Cohen's kappa and distribution-based methods were used to estimate agreement and MIDs respectively. Robust convergent (ρ = 0.66-0.82, 95% confidence interval [CI] 0.54-0.86) and discriminant (ρ = 0.06-0.31, 95% CI -0.11 to 0.43) validity were found for all four mobility measures. Known-groups validity was supported by significant mean differences across AMC subtypes (amyoplasia, distal arthrogryposis, central nervous system/syndromic; p < 0.001). The measures also showed weak to good agreement in classifying mobility. A difference of one and two levels on the FMS and FAQ respectively, was found to be minimally important. For the PROMIS and WeeFIM, estimated MID values were 3.19 to 4.34 and 14.24 respectively. The robust construct validity, agreement, and MIDs provide clinicians and researchers with evidence-based benchmarks for assessing mobility in children with AMC.

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2026

Distal arthrogryposis with impaired proprioception and touch: description of 9 additional cases harbouring novel PIEZO2 variants and literature review.

European journal of paediatric neurology : EJPN : official journal of the European Paediatric Neurology Society
2026

Expanding the Genotype-Phenotype Correlation of Marden-Walker Syndrome due to PIEZO2 Gene Variants: A Case Report From Brazil.

American journal of medical genetics. Part A
2026

Biallelic MYH3 Variants Cause Distal Arthrogryposis in Compound Heterozygosity and a Subclinical Phenotype in Simple Heterozygosity. Codominance or Recessive Inheritance?

Clinical genetics
2026

Impact of growth hormone treatment on a 12-year-old female with newly diagnosed panhypopituitarism and distal arthrogryposis.

Endocrinology, diabetes &amp; metabolism case reports
2025

Freeman-Sheldon Syndrome: A Rare Case Report with Dental Perspective.

Prague medical report
2025

Factors Associated With Functional Mobility in 256 Children With Arthrogryposis Multiplex Congenita: A Multicentric Cross-Sectional Study.

Archives of physical medicine and rehabilitation
2025

The Emerging TNNT3 Spectrum: From Distal Arthrogryposis to Congenital Myopathy.

Human mutation
2025

Novel Homozygous PIEZO2 Splice Site Mutation Causing Distal Arthrogryposis with Impaired Proprioception.

Indian journal of pediatrics
2025

Homozygous Pathogenic MYH3 Variants Associated With Arthrogryposis and Lingual Dystonia.

Tremor and other hyperkinetic movements (New York, N.Y.)
2025

Psychometric properties of functional mobility outcome measures in children with arthrogryposis multiplex congenita.

Developmental medicine and child neurology
2025

[Expert consensus on the clinical diagnosis and management of Arthrogryposis multiplex congenita (2025 Edition)].

Zhonghua yi xue yi chuan xue za zhi = Zhonghua yixue yichuanxue zazhi = Chinese journal of medical genetics
2025

Novel heterozygous mutation in MYH3 causes contractures, pterygia, and spondylocarpostarsal fusion syndrome 1: A case report.

Medicine
2025

Functional Characterization of a Novel Intronic Variant in PIEZO2 in a Recessive Form of Distal Arthrogryposis With Impaired Proprioception and Touch (DAIPT).

Molecular genetics &amp; genomic medicine
2025

ADGRG6-related disorder: a novel mutation resulting in distal arthrogryposis and a patchy neuropathy.

Neuromuscular disorders : NMD
2025

Genetic and clinical spectrum of PIEZO2-related disorders: insights from a multicenter study of 26 patients.

Neuromuscular disorders : NMD
2025

Multi-omics approach identifies a novel recessive pathogenic variant in the TNNT3 gene in two siblings with congenital myopathy.

Neuromuscular disorders : NMD
2025

[Clinical and genetic analysis of a pedigree affected with Distal arthrogryposis type 5D due to compound heterozygous variants of ECEL1 gene].

Zhonghua yi xue yi chuan xue za zhi = Zhonghua yixue yichuanxue zazhi = Chinese journal of medical genetics
2025

The evolving genetic landscape of neuromuscular fetal akinesias.

Journal of neuromuscular diseases
2025

Multidisciplinary treatment of severe spinal deformity complicated with severe cardiopulmonary dysfunction: a case report.

Italian journal of pediatrics
2025

Maternal, fetal and neonatal outcomes among pregnant women with arthrogryposis multiplex congenita: a scoping review.

Orphanet journal of rare diseases
2025

Genotype-Phenotype Landscape of NALCN and UNC80-Related Disorders.

Neurology
2025

Delineating the Clinical and Brain Imaging Characteristics of the Neonatal Form of CSTB -Related Neurodevelopmental Disorders.

Clinical genetics
2025

Unraveling the Genetic Landscape of Foot Arch Morphology: A Systematic Review of Single Nucleotide Polymorphisms.

Clinical genetics
2024

A Case of Freeman-Sheldon Syndrome With Glaucoma Successfully Treated by Trabeculotomy Using the Kahook Dual Blade.

Cureus
2025

Unsafe Care and Fake News in Freeman-Burian Syndrome.

Clinical case reports
2025

ECEL1 mutation in distal arthrogryposis type 5D: A case report.

European journal of obstetrics, gynecology, and reproductive biology
2024

Considerations in Correction of Wrist Deformity in Arthrogryposis.

The journal of hand surgery Asian-Pacific volume
2024

Effects of Osteopathic Manipulative Treatment on Rare Diseases: A Case Report on Gordon Syndrome.

Cureus
2024

Heterozygous loss-of-function variants in SPTAN1 cause a novel early childhood onset distal myopathy with chronic neurogenic features.

medRxiv : the preprint server for health sciences
2024

Fast-twitch myofibrils grow in proportion to Mylpf dosage in the zebrafish embryo.

bioRxiv : the preprint server for biology
2024

Midterm clinical and radiological outcomes of arthrogryposis-associated clubfoot treated with the Ponseti method: a retrospective observational study and comprehensive literature review.

Journal of orthopaedic surgery and research
2024

Heterogenic Genetic Background of Distal Arthrogryposis-Review of the Literature and Case Report.

Children (Basel, Switzerland)
2024

Musculocontractural type of Ehlers-Danlos syndrome with novel CHST14 pathogenic variant in two siblings.

Pediatric dermatology
2024

Bi-allelic variants in MYH3 cause recessively-inherited arthrogryposis.

Clinical genetics
2024

Minimally Invasive Method for Treatment of Syndromic Congenital Vertical Talus Deformity in Children.

Foot &amp; ankle international
2024

A retrospective study on the correction of distal arthrogryposis with a progressive extension brace.

Frontiers in pediatrics
2024

Outcomes of Ponseti Method for the Treatment of Clubfeet in Children With Arthrogryposis.

Journal of pediatric orthopedics
2024

The Intrarater and Interrater Reliability of the OMT Classification Among Physicians With a Different Background.

Journal of pediatric orthopedics
2024

Bone mineral density in adults with arthrogryposis multiplex congenita: a retrospective cohort analysis.

Scientific reports
2024

ECEL1 related distal arthrogryposis 5D in an Indian cohort-Report of recognizable musculoskeletal phenotype and a possible founder variant.

American journal of medical genetics. Part A
2024

New insights into the clinical and molecular spectrum of the MADD-related neurodevelopmental disorder.

Journal of human genetics
2024

Functional assessment of a novel biallelic MYH3 variation causing CPSKF1B (contractures, pterygia, and spondylocarpotarsal fusion syndrome1B).

Molecular genetics &amp; genomic medicine
2024

Compound heterozygous variants in MYBPC1 lead to severe distal arthrogryposis type-1 manifestations.

Gene
2024

MET is a new confirmed gene responsible for familial distal arthrogryposis.

EMBO molecular medicine
2023

Prenatal diagnosis of Freeman-Sheldon syndrome using ultrasound and genetic testing. Case report.

Revista colombiana de obstetricia y ginecologia
2024

Homologous mutations in human β, embryonic, and perinatal muscle myosins have divergent effects on molecular power generation.

Proceedings of the National Academy of Sciences of the United States of America
2024

A mutation in F-actin polymerization factor suppresses the distal arthrogryposis type 5 PIEZO2 pathogenic variant in Caenorhabditis elegans.

Development (Cambridge, England)
2024

A novel compound heterozygous variant of ECEL1 induced joint dysfunction and cartilage degradation: a case report and literature review.

Frontiers in neurology
2024

Clinical and molecular characteristics of 26 fetuses with lethal multiple congenital contractures.

Clinical genetics
2023

Unlocking the Role of sMyBP-C: A Key Player in Skeletal Muscle Development and Growth.

bioRxiv : the preprint server for biology
2023

Identification of two novel MYH3 variants causing different phenotypes in prenatal diagnosis.

Prenatal diagnosis
2023

Mutation in F-actin Polymerization Factor Suppresses Distal Arthrogryposis Type 5 (DA5) PIEZO2 Pathogenic Variant in Caenorhabditis elegans.

bioRxiv : the preprint server for biology
2023

New presentation of CLIFAHDD syndrome with a novel variant in NALCN gene: A report of a rare case.

Clinical case reports
2023

Variants in ACTC1 underlie distal arthrogryposis accompanied by congenital heart defects.

HGG advances
2023

Maternally inherited deletion encompassing the RTL1as and MEG8 genes of the human 14q32 imprinted region in a patient with a mild Kagami-Ogata syndrome phenotype.

American journal of medical genetics. Part A
2023

The range of publications on arthrogryposis multiplex congenita from 1995 to 2022-A scoping review.

American journal of medical genetics. Part A
2022

Diagnostic work-up and phenotypic characteristics of a family with variable severity of distal arthrogryposis type 2B (Sheldon-Hall syndrome) and TNNT3 pathogenic variant.

Frontiers in genetics
2023

Clinical and functional characterization of a long survivor congenital titinopathy patient with a novel metatranscript-only titin variant.

Acta neuropathologica communications
2023

Management and survival of foetuses with trisomy 18 in a French retrospective cohort.

Journal of gynecology obstetrics and human reproduction
2023

Biallelic variants in ADAMTS15 cause a novel form of distal arthrogryposis.

Genetics in medicine : official journal of the American College of Medical Genetics
2024

Distal Arthrogryposis with Impaired Proprioception and Touch: A Novel Variant in PIEZO2 Gene in Omani Patients and a Genotype-Phenotype Review from a Single-Center Experience.

Journal of pediatric genetics
2023

ECEL1 novel mutation in arthrogryposis type 5D: A molecular dynamic simulation study.

Molecular genetics &amp; genomic medicine
2023

Excessive mechanotransduction in sensory neurons causes joint contractures.

Science (New York, N.Y.)
2023

Distal arthrogryposis in a girl arising from a novel TNNI2 variant inherited from paternal somatic mosaicism.

Journal of human genetics
2023

Congenital vertical talus deformity in children with distal arthrogryposis: good clinical outcomes despite high rate of residual radiographic deformity.

Journal of pediatric orthopedics. Part B
2022

Further Evidence of a Continuum in the Clinical Spectrum of Dominant PIEZO2-Related Disorders and Implications in Cerebellar Anomalies.

Molecular syndromology
2023

Long term ophthalmic complications of distal arthrogryposis type 5D.

Ophthalmic genetics
2023

Lethal respiratory course and additional features expand the phenotypic spectrum of PIEZO2-related distal arthrogryposis type 5.

American journal of medical genetics. Part A
2022

A TNNI2 variant c.525G>T causes distal arthrogryposis in a Chinese family.

Molecular genetics &amp; genomic medicine
2022

TTN as a candidate gene for distal arthrogryposis type 10 pathogenesis.

Journal, genetic engineering &amp; biotechnology
2022

Distal Arthrogryposis type 5 in an Italian family due to an autosomal dominant gain-of-function mutation of the PIEZO2 gene.

Italian journal of pediatrics
2022

Characterizing the lipid fingerprint of the mechanosensitive channel Piezo2.

The Journal of general physiology
2022

Heterogeneous Distribution of Genetic Mutations in Myosin Binding Protein-C Paralogs.

Frontiers in genetics
2022

Lethal Congenital Contracture Syndrome 11: A Case Report and Literature Review.

Journal of clinical medicine
2022

PIEZO2-related distal arthrogryposis type 5: Longitudinal follow-up of a three-generation family broadens phenotypic spectrum, complications, and health surveillance recommendations for this patient group.

American journal of medical genetics. Part A
2022

A pathogenic mechanism associated with myopathies and structural birth defects involves TPM2-directed myogenesis.

JCI insight
2022

Oculoplastic surgery, diagnosis, and other matters in Freeman-Burian syndrome.

Ophthalmic genetics
2022

Bi-allelic MYH3 loss-of-function variants cause a lethal form of contractures, pterygia, and spondylocarpotarsal fusion syndrome 1B.

Neuromuscular disorders : NMD
2021

Freeman-Sheldon Syndrome with Stiff Knee Gait - A Case Report.

Journal of orthopaedic case reports
2022

Expanding the mutation and phenotype spectrum of MYH3-associated skeletal disorders.

NPJ genomic medicine
2022

The Hand in Distal Arthrogryposis.

The Journal of hand surgery
2022

Recessive variants in COL25A1 gene as novel cause of arthrogryposis multiplex congenita with ocular congenital cranial dysinnervation disorder.

Human mutation
2022

COG6-CDG: Novel variants and novel malformation.

Birth defects research
2022

Functional loss of ubiquitin-specific protease 14 may lead to a novel distal arthrogryposis phenotype.

Clinical genetics
2021

Clinical and Genetic Findings in a Series of Eight Families with Arthrogryposis.

Genes
2022

TNNI1 Mutated in Autosomal Dominant Proximal Arthrogryposis.

Neurology. Genetics
2023

Diagnostic workup in children with arthrogryposis: description of practices from a single reference centre, comparison with literature and suggestion of recommendations.

Journal of medical genetics
2021

The distal arthrogryposis-linked p.R63C variant promotes the stability and nuclear accumulation of TNNT3.

Journal of clinical laboratory analysis
2021

Expanding the Phenotypic Spectrum of ECEL1-Associated Distal Arthrogryposis.

Children (Basel, Switzerland)
2022

Arthrogryposis multiplex congenita: a 28-year retrospective study.

Developmental medicine and child neurology
2021

Troponin Variants in Congenital Myopathies: How They Affect Skeletal Muscle Mechanics.

International journal of molecular sciences
2021

The Clinical and Genotypic Spectrum of Scoliosis in Multiple Pterygium Syndrome: A Case Series on 12 Children.

Genes
2021

Patient-Reported Outcome Measurement Information System (PROMIS) Scores in Pediatric Patients With Arthrogryposis.

Journal of pediatric orthopedics
2021

A Genomic Approach to Delineating the Occurrence of Scoliosis in Arthrogryposis Multiplex Congenita.

Genes
2021

Models of Distal Arthrogryposis and Lethal Congenital Contracture Syndrome.

Genes
2021

Gordon syndrome: Dental implications and a case report.

Special care in dentistry : official publication of the American Association of Hospital Dentists, the Academy of Dentistry for the Handicapped, and the American Society for Geriatric Dentistry
2021

Case Report: Further Delineation of Neurological Symptoms in Young Children Caused by Compound Heterozygous Mutation in the PIEZO2 Gene.

Frontiers in genetics
2021

Biallelic Pathogenic Variants in TNNT3 Associated With Congenital Myopathy.

Neurology. Genetics
2021

Fetal akinesia: The need for clinical vigilance in first trimester with decreased fetal movements.

Taiwanese journal of obstetrics &amp; gynecology
2021

Trends in Piezo Channel Research Over the Past Decade: A Bibliometric Analysis.

Frontiers in pharmacology
2021

Mutations Q93H and E97K in TPM2 Disrupt Ca-Dependent Regulation of Actin Filaments.

International journal of molecular sciences
2021

Fast skeletal myosin-binding protein-C regulates fast skeletal muscle contraction.

Proceedings of the National Academy of Sciences of the United States of America
2021

Homozygous variant in MADD, encoding a Rab guanine nucleotide exchange factor, results in pleiotropic effects and a multisystemic disorder.

European journal of human genetics : EJHG
2021

A New Intronic Variant in ECEL1 in Two Patients with Distal Arthrogryposis Type 5D.

International journal of molecular sciences
2021

Homozygous intronic variants in TPM2 cause recessively inherited Escobar variant of multiple pterygium syndrome and congenital myopathy.

Neuromuscular disorders : NMD
2021

Distal arthrogryposis type 5D in a South Indian family caused by novel deletion in ECEL1 gene.

Clinical dysmorphology
2021

Confirming the involvement of PIEZO2 in the etiology of Marden-Walker syndrome.

American journal of medical genetics. Part A
2021

Amyoplasia and distal arthrogryposis.

Orthopaedics &amp; traumatology, surgery &amp; research : OTSR
2021

Further delineation of MYO18B-related autosomal recessive Klippel-Feil syndrome with myopathy and facial dysmorphism.

American journal of medical genetics. Part A
2020

A review of the orthopedic interventions and functional outcomes among a cohort of 114 children with arthrogryposis multiplex congenita.

Journal of pediatric rehabilitation medicine
2021

Neurogenetic fetal akinesia and arthrogryposis: genetics, expanding genotype-phenotypes and functional genomics.

Journal of medical genetics
2020

MYH3-associated distal arthrogryposis zebrafish model is normalized with para-aminoblebbistatin.

EMBO molecular medicine
2020

[Genetic analysis and prenatal diagnosis of a pregnant woman with Sheldon-Hall syndrome].

Zhonghua yi xue yi chuan xue za zhi = Zhonghua yixue yichuanxue zazhi = Chinese journal of medical genetics
2020

Talectomy by Medial Surgical Approach for Congenital Vertical Talus in Arthrogryposis Multiplex Congenita.

Orthopedics
2020

Drosophila myosin mutants model the disparate severity of type 1 and type 2B distal arthrogryposis and indicate an enhanced actin affinity mechanism.

Skeletal muscle
2020

Identification of a novel pathogenic variant in the MYH3 gene in a five-generation family with CPSFS1A (Contractures, Pterygia, and Spondylocarpotarsal Fusion Syndrome 1A).

Molecular genetics &amp; genomic medicine
2020

Identification and Recent Approaches for Evaluation and Management of Rehabilitation Concerns for Patients with Freeman-Burian Syndrome: Principles for Global Treatment.

Journal of pediatric genetics
2020

Mutations in MYLPF Cause a Novel Segmental Amyoplasia that Manifests as Distal Arthrogryposis.

American journal of human genetics
2020

Distal Arthrogryposis and Lethal Congenital Contracture Syndrome - An Overview.

Frontiers in physiology
2020

The Novel Compound Heterozygous Mutations of ECEL1 Identified in a Family with Distal Arthrogryposis Type 5D.

BioMed research international
2020

Bone densities and bone geometry in children and adolescents with arthrogryposis.

Bone
2020

Precise Pulmonary Function Evaluation and Management of a Patient With Freeman-Sheldon Syndrome Associated With Recurrent Pneumonia and Chronic Respiratory Insufficiency.

Annals of rehabilitation medicine
2020

Distal Arthrogryposis: A Clue to the Etiology of Neonatal Cholestasis.

Indian journal of pediatrics
2020

Findings, Phenotypes, Diagnostic Accuracy, and Treatment in Freeman-Burian Syndrome.

The Journal of craniofacial surgery
2020

A recurrent pathogenic variant in TPM2 reveals further phenotypic and genetic heterogeneity in multiple pterygium syndrome-related disorders.

Clinical genetics
2020

Patient-reported Outcomes in Arthrogryposis.

Journal of pediatric orthopedics
2020

Identification of a novel pathogenic mutation of the MYH3 gene in a family with distal arthrogryposis type 2B.

Molecular medicine reports
2019

Biallelic Missense Mutation in the ECEL1 Underlies Distal Arthrogryposis Type 5 (DA5D).

Frontiers in pediatrics
2020

When fingers point to the diagnosis.

Archives of disease in childhood
2019

One-stage release by double surgical approach for neglected congenital vertical talus: results in a series of walking children in Tanzania.

Journal of pediatric orthopedics. Part B
2019

Mutations in PIEZO2 contribute to Gordon syndrome, Marden-Walker syndrome and distal arthrogryposis: A bioinformatics analysis of mechanisms.

Experimental and therapeutic medicine
2019

Distal arthrogryposis type 5 and PIEZO2 novel variant in a Canadian family.

American journal of medical genetics. Part A
2019

Distal Arthrogryposis with Impaired Proprioception and Touch: Description of an Early Phenotype in a Boy with Compound Heterozygosity of PIEZO2 Mutations and Review of the Literature.

Molecular syndromology
2019

A novel nonsense PIEZO2 mutation in a family with scoliosis and proprioceptive defect.

Neuromuscular disorders : NMD
2018

The Primary Causes of Muscle Dysfunction Associated with the Point Mutations in Tpm3.12; Conformational Analysis of Mutant Proteins as a Tool for Classification of Myopathies.

International journal of molecular sciences
2018

Revisiting the Many Names of Freeman-Sheldon Syndrome.

The Journal of craniofacial surgery
2019

Reductions in ATPase activity, actin sliding velocity, and myofibril stability yield muscle dysfunction in Drosophila models of myosin-based Freeman-Sheldon syndrome.

Molecular biology of the cell
2019

New Insights of a Neuronal Peptidase DINE/ECEL1: Nerve Development, Nerve Regeneration and Neurogenic Pathogenesis.

Neurochemical research
2018

Skeletal myosin binding protein-C: An increasingly important regulator of striated muscle physiology.

Archives of biochemistry and biophysics
2018

Novel mutations in TPM2 and PIEZO2 are responsible for distal arthrogryposis (DA) 2B and mild DA in two Chinese families.

BMC medical genetics
2019

Intrafamilial variability of clinical features in distal arthrogryposis type 2B.

Clinical dysmorphology
2018

ECEL1 gene related contractural syndrome: Long-term follow-up and update on clinical and pathological aspects.

Neuromuscular disorders : NMD
2018

Difficult airway management in children and young adults with arthrogryposis.

World journal of otorhinolaryngology - head and neck surgery
2018

Extending the phenotype and an ECEL1 gene mutation in distal arthrogryposis type 5D.

Clinical dysmorphology
2018

Novel Type of Complicated Autosomal Dominant Hereditary Spastic Paraplegia Associated with Congenital Distal Arthrogryposis Type I.

Brain sciences
2018

Arthrogryposis in children: Etiological assessments and preparation of a protocol for etiological investigations.

Archives de pediatrie : organe officiel de la Societe francaise de pediatrie
2018

Recessive Spondylocarpotarsal Synostosis Syndrome Due to Compound Heterozygosity for Variants in MYH3.

American journal of human genetics
2018

A novel ECEL1 mutation expands the phenotype of distal arthrogryposis multiplex congenita type 5D to include pretibial vertical skin creases.

American journal of medical genetics. Part A
2018

A MYH3 mutation identified for the first time in a Chinese family with Sheldon-Hall syndrome (DA2B).

Neuromuscular disorders : NMD
2018

Chitayat-Hall and Schaaf-Yang syndromes:a common aetiology: expanding the phenotype of MAGEL2-related disorders.

Journal of medical genetics
2018

A novel pathogenic MYH3 mutation in a child with Sheldon-Hall syndrome and vertebral fusions.

American journal of medical genetics. Part A
2018

Nemaline myopathy and distal arthrogryposis associated with an autosomal recessive TNNT3 splice variant.

Human mutation
2017

[A pedigree with distal arthrogryposis].

Zhonghua yi xue yi chuan xue za zhi = Zhonghua yixue yichuanxue zazhi = Chinese journal of medical genetics
2017

Distinct functional consequences of ECEL1/DINE missense mutations in the pathogenesis of congenital contracture disorders.

Acta neuropathologica communications
2017

The reason for a high Ca2+-sensitivity associated with Arg91Gly substitution in TPM2 gene is the abnormal behavior and high flexibility of tropomyosin during the ATPase cycle.

Biochemical and biophysical research communications
2017

A mutation of beta-tropomyosin gene in a Chinese family with distal arthrogryposis type I.

International journal of clinical and experimental pathology
2017

Research progress of myosin heavy chain genes in human genetic diseases.

Yi chuan = Hereditas
2017

Genetic Diseases of PIEZO1 and PIEZO2 Dysfunction.

Current topics in membranes
2017

Interbrachial Pinch by Trapezius Transfer in Amyoplasia Congenita: A Case Report.

Plastic and reconstructive surgery. Global open
2017

Treatment of congenital clasped thumb in arthrogryposis.

The Journal of hand surgery, European volume
2017

Genetics and Classifications.

Journal of pediatric orthopedics
2017

Freeman-Sheldon Syndrome: First Molecularly Confirmed Case from Sub-Saharan Africa.

Case reports in genetics
2017

Fetal costello syndrome with neuromuscular spindles excess and p.Gly12Val HRAS mutation.

European journal of medical genetics
2017

Recessive mutations in SLC35A3 cause early onset epileptic encephalopathy with skeletal defects.

American journal of medical genetics. Part A
2017

Findings, phenotypes, and outcomes in Freeman-Sheldon and Sheldon-Hall syndromes and distal arthrogryposis types 1 and 3: protocol for systematic review and patient-level data meta-analysis.

Systematic reviews
2017

Arthrogryposis multiplex congenita: classification, diagnosis, perioperative care, and anesthesia.

Frontiers of medicine
2017

A Novel Variant in the Endothelin-Converting Enzyme-Like 1 (ECEL1) Gene in an Emirati Child.

Medical principles and practice : international journal of the Kuwait University, Health Science Centre
2017

Anesthetic Outcomes of Children With Arthrogryposis Syndromes: No Evidence of Hyperthermia.

Anesthesia and analgesia
2017

Recessive PIEZO2 stop mutation causes distal arthrogryposis with distal muscle weakness, scoliosis and proprioception defects.

Journal of human genetics
2016

Amino Acid Changes at Arginine 204 of Troponin I Result in Increased Calcium Sensitivity of Force Development.

Frontiers in physiology
2016

Novel NALCN variant: altered respiratory and circadian rhythm, anesthetic sensitivity.

Annals of clinical and translational neurology
2016

Biallelic Loss of Proprioception-Related PIEZO2 Causes Muscular Atrophy with Perinatal Respiratory Distress, Arthrogryposis, and Scoliosis.

American journal of human genetics
2016

A Highly Conserved Yet Flexible Linker Is Part of a Polymorphic Protein-Binding Domain in Myosin-Binding Protein C.

Structure (London, England : 1993)
2016

Distal arthrogryposis with variable clinical expression caused by TNNI2 mutation.

Human genome variation
2017

Homozygous SYNE1 mutation causes congenital onset of muscular weakness with distal arthrogryposis: a genotype-phenotype correlation.

European journal of human genetics : EJHG
2017

A novel TPM2 gene splice-site mutation causes severe congenital myopathy with arthrogryposis and dysmorphic features.

Journal of applied genetics
2017

Familial Gordon syndrome associated with a PIEZO2 mutation.

American journal of medical genetics. Part A
2016

MYBPC1, an Emerging Myopathic Gene: What We Know and What We Need to Learn.

Frontiers in physiology
2017

Loss of the proprioception and touch sensation channel PIEZO2 in siblings with a progressive form of contractures.

Clinical genetics
2016

Spinal muscular atrophy with respiratory distress syndrome (SMARD1): Case report and review of literature.

Annals of Indian Academy of Neurology
2016

Review of the recurrent 8q13.2q13.3 branchio-oto-renal related microdeletion, and report of an additional case with associated distal arthrogryposis.

American journal of medical genetics. Part A
2016

Muscle biopsy findings in a child with NALCN gene mutation.

Journal of clinical neuroscience : official journal of the Neurosurgical Society of Australasia
2016

Protein-altering MYH3 variants are associated with a spectrum of phenotypes extending to spondylocarpotarsal synostosis syndrome.

European journal of human genetics : EJHG
2016

Novel Mutations in the Nonselective Sodium Leak Channel (NALCN) Lead to Distal Arthrogryposis with Increased Muscle Tone.

Neuropediatrics
2015

Recessive REEP1 mutation is associated with congenital axonal neuropathy and diaphragmatic palsy.

Neurology. Genetics
2016

ECEL1 mutation implicates impaired axonal arborization of motor nerves in the pathogenesis of distal arthrogryposis.

Acta neuropathologica
2016

The Most Prevalent Freeman-Sheldon Syndrome Mutations in the Embryonic Myosin Motor Share Functional Defects.

The Journal of biological chemistry
2016

Expanding the MYBPC1 phenotypic spectrum: a novel homozygous mutation causes arthrogryposis multiplex congenita.

Clinical genetics
2015

Developmental MYH3 Myopathy Associated with Expression of Mutant Protein and Reduced Expression Levels of Embryonic MyHC.

PloS one
2015

AMC: amyoplasia and distal arthrogryposis.

Journal of children's orthopaedics
2015

Management of the knees in arthrogryposis.

Journal of children's orthopaedics
2015

Freeman-Sheldon syndrome in a 29-year-old woman presenting with rare and previously undescribed features.

BMJ case reports
2016

The clubfoot painted by Jusepe de Ribera: a controversial diagnosis.

The journal of maternal-fetal &amp; neonatal medicine : the official journal of the European Association of Perinatal Medicine, the Federation of Asia and Oceania Perinatal Societies, the International Society of Perinatal Obstetricians
2016

A novel missense mutation of TNNI2 in a Chinese family cause distal arthrogryposis type 1.

American journal of medical genetics. Part A
2015

Bilateral Coronoidectomy by Craniofacial Approach for Hecht Syndrome-Related Trismus.

The Journal of craniofacial surgery
2015

Myosin Binding Protein-C Slow Phosphorylation is Altered in Duchenne Dystrophy and Arthrogryposis Myopathy in Fast-Twitch Skeletal Muscles.

Scientific reports
2015

Developmental myosins: expression patterns and functional significance.

Skeletal muscle

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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. Distal arthrogryposis with impaired proprioception and touch: description of 9 additional cases harbouring novel PIEZO2 variants and literature review.
    European journal of paediatric neurology : EJPN : official journal of the European Paediatric Neurology Society· 2026· PMID 41780226mais citado
  2. Biallelic MYH3 Variants Cause Distal Arthrogryposis in Compound Heterozygosity and a Subclinical Phenotype in Simple Heterozygosity. Codominance or Recessive Inheritance?
    Clinical genetics· 2026· PMID 41562327mais citado
  3. Expanding the Genotype-Phenotype Correlation of Marden-Walker Syndrome due to PIEZO2 Gene Variants: A Case Report From Brazil.
    American journal of medical genetics. Part A· 2026· PMID 41572728mais citado
  4. Impact of growth hormone treatment on a 12-year-old female with newly diagnosed panhypopituitarism and distal arthrogryposis.
    Endocrinology, diabetes &amp; metabolism case reports· 2026· PMID 41499205mais citado
  5. Psychometric properties of functional mobility outcome measures in children with arthrogryposis multiplex congenita.
    Developmental medicine and child neurology· 2025· PMID 41124586mais citado
  6. Freeman-Sheldon Syndrome: A Rare Case Report with Dental Perspective.
    Prague Med Rep· 2025· PMID 41480702recente

Bases de dados e fontes oficiais

Identificadores e referências canônicas usadas para montar este verbete.

  1. ORPHA:97120(Orphanet)
  2. MONDO:0019942(MONDO)
  3. GARD:786(GARD (NIH))
  4. Variantes catalogadas(ClinVar)
  5. Busca completa no PubMed(PubMed)
  6. Q18553375(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.

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Artrogripose distal

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