Raras
Buscar doenças, sintomas, genes...
Craniossinostose sindrômica
ORPHA:139393DOENÇA RARA

O fechamento precoce dos ossos da cabeça, que faz parte de uma síndrome maior.

Mantido por Agente Raras·Colaborar como especialista →

Introdução

O que você precisa saber de cara

📋

O fechamento precoce dos ossos da cabeça, que faz parte de uma síndrome maior.

Publicações científicas
767 artigos
Último publicado: 2026 Apr 6
Medicamentos
4 registrados
EPOPROSTENOL, PRAVASTATIN, IRBESARTAN

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4 medicamentos registrados
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EPOPROSTENOLPRAVASTATINIRBESARTANRITUXIMAB
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SUS: Sem cobertura SUSScore: 0%
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Entender a doença

Do básico ao detalhe, leia no seu ritmo

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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
161 sintomas
😀
Face
67 sintomas
👁️
Olhos
42 sintomas
🫃
Digestivo
40 sintomas
🧠
Neurológico
36 sintomas
🫘
Rins
35 sintomas

+ 352 sintomas em outras categorias

Características mais comuns

Acne
Hemangioma capilar de terço médio da face
Microcefalia congênita
Palato alto e estreito
Pigmentação mosqueada do epitélio pigmentar da retina
Clinodactilia
877sintomas
Sem dados (877)

Os sintomas variam de pessoa para pessoa. Abaixo estão as 877 características clínicas mais associadas, ordenadas por frequência.

Acne
Hemangioma capilar de terço médio da faceMidface capillary hemangioma
Microcefalia congênitaCongenital microcephaly
Palato alto e estreitoHigh, narrow palate
Pigmentação mosqueada do epitélio pigmentar da retinaRetinal pigment epithelial mottling

Linha do tempo da pesquisa

Publicações por ano — veja quando o interesse científico cresceu
Anos de pesquisa1desde 2026
Total histórico767PubMed
Últimos 10 anos200publicações
Pico202571 papers
Linha do tempo
2026Hoje · 2026🧪 2020Primeiro 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

27 genes identificados com associação a esta condição.

WDR35WD repeat-containing protein 35Disease-causing germline mutation(s) inTolerante
FUNÇÃO

As a component of the IFT complex A (IFT-A), a complex required for retrograde ciliary transport and entry into cilia of G protein-coupled receptors (GPCRs), it is involved in ciliogenesis and ciliary protein trafficking (PubMed:21473986, PubMed:28400947, PubMed:29220510). May promote CASP3 activation and TNF-stimulated apoptosis

LOCALIZAÇÃO

Cytoplasm, cytoskeleton, microtubule organizing center, centrosomeCytoplasm, cytoskeleton, cilium axonemeCytoplasm, cytoskeleton, cilium basal body

VIAS BIOLÓGICAS (2)
Hedgehog 'off' stateIntraflagellar transport
MECANISMO DE DOENÇA

Cranioectodermal dysplasia 2

A disorder characterized by craniofacial, skeletal and ectodermal abnormalities. Clinical features include short stature, dolichocephaly, craniosynostosis, narrow thorax with pectus excavatum, short limbs, brachydactyly, joint laxity, narrow palpebral fissures, telecanthus with hypertelorism, low-set simple ears, everted lower lip, and short neck. Teeth abnormalities include widely spaced, hypoplastic and fused teeth.

EXPRESSÃO TECIDUAL(Ubíquo)
Testículo
20.6 TPM
Pituitária
17.9 TPM
Ovário
17.3 TPM
Útero
17.2 TPM
Nervo tibial
16.9 TPM
OUTRAS DOENÇAS (4)
cranioectodermal dysplasia 2short-rib thoracic dysplasia 7 with or without polydactylyasphyxiating thoracic dystrophy 3cranioectodermal dysplasia
HGNC:29250UniProt:Q9P2L0
PORNADPH--cytochrome P450 reductaseDisease-causing germline mutation(s) inTolerante
FUNÇÃO

This enzyme is required for electron transfer from NADP to cytochrome P450 in microsomes. It can also provide electron transfer to heme oxygenase and cytochrome B5

LOCALIZAÇÃO

Endoplasmic reticulum membrane

VIAS BIOLÓGICAS (1)
Cytochrome P450 - arranged by substrate type
MECANISMO DE DOENÇA

Antley-Bixler syndrome, with genital anomalies and disordered steroidogenesis

A disease characterized by the association of Antley-Bixler syndrome with steroidogenesis defects and abnormal genitalia. Antley-Bixler syndrome is characterized by craniosynostosis, radiohumeral synostosis present from the perinatal period, midface hypoplasia, choanal stenosis or atresia, femoral bowing and multiple joint contractures.

EXPRESSÃO TECIDUAL(Ubíquo)
Glândula adrenal
164.2 TPM
Fígado
149.3 TPM
Tireoide
116.3 TPM
Pituitária
84.0 TPM
Pulmão
67.2 TPM
OUTRAS DOENÇAS (2)
congenital adrenal hyperplasia due to cytochrome P450 oxidoreductase deficiencyAntley-Bixler syndrome with genital anomalies and disordered steroidogenesis
HGNC:9208UniProt:P16435
FGFR1Fibroblast growth factor receptor 1Disease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Tyrosine-protein kinase that acts as a cell-surface receptor for fibroblast growth factors and plays an essential role in the regulation of embryonic development, cell proliferation, differentiation and migration. Required for normal mesoderm patterning and correct axial organization during embryonic development, normal skeletogenesis and normal development of the gonadotropin-releasing hormone (GnRH) neuronal system. Phosphorylates PLCG1, FRS2, GAB1 and SHB. Ligand binding leads to the activati

LOCALIZAÇÃO

Cell membraneNucleusCytoplasm, cytosolCytoplasmic vesicle

VIAS BIOLÓGICAS (2)
Epithelial-Mesenchymal Transition (EMT) during gastrulationFormation of paraxial mesoderm
MECANISMO DE DOENÇA

Pfeiffer syndrome

A syndrome characterized by the association of craniosynostosis, broad and deviated thumbs and big toes, and partial syndactyly of the fingers and toes. Three subtypes are known: mild autosomal dominant form (type 1); cloverleaf skull, elbow ankylosis, early death, sporadic (type 2); craniosynostosis, early demise, sporadic (type 3).

EXPRESSÃO TECIDUAL(Ubíquo)
Aorta
144.8 TPM
Ovário
142.9 TPM
Artéria tibial
134.1 TPM
Fallopian Tube
122.3 TPM
Cérebro - Hemisfério cerebelar
122.0 TPM
OUTRAS DOENÇAS (20)
Hartsfield-Bixler-Demyer syndromeencephalocraniocutaneous lipomatosisosteoglophonic dysplasiaPfeiffer syndrome
HGNC:3688UniProt:P11362
MEGF8Multiple epidermal growth factor-like domains protein 8Disease-causing germline mutation(s) inRestrito
FUNÇÃO

Acts as a negative regulator of hedgehog signaling

LOCALIZAÇÃO

Membrane

MECANISMO DE DOENÇA

Carpenter syndrome 2

An autosomal recessive multiple congenital malformation disorder characterized by multisuture craniosynostosis and polysyndactyly of the hands and feet, in association with abnormal left-right patterning and other features, most commonly obesity, umbilical hernia, cryptorchidism, and congenital heart disease.

EXPRESSÃO TECIDUAL(Ubíquo)
Cerebelo
51.8 TPM
Cérebro - Hemisfério cerebelar
46.8 TPM
Córtex cerebral
41.0 TPM
Brain Frontal Cortex BA9
38.5 TPM
Fibroblastos
28.9 TPM
OUTRAS DOENÇAS (2)
MEGF8-related Carpenter syndromeCarpenter syndrome
HGNC:3233UniProt:Q7Z7M0
IFT52Intraflagellar transport protein 52 homologDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Involved in ciliogenesis as part of a complex involved in intraflagellar transport (IFT), the bi-directional movement of particles required for the assembly, maintenance and functioning of primary cilia (PubMed:27466190). Required for the anterograde transport of IFT88 (PubMed:27466190)

LOCALIZAÇÃO

Cell projection, cilium

VIAS BIOLÓGICAS (2)
Hedgehog 'off' stateIntraflagellar transport
MECANISMO DE DOENÇA

Short-rib thoracic dysplasia 16 with or without polydactyly

A form of short-rib thoracic dysplasia, a group of autosomal recessive ciliopathies that are characterized by a constricted thoracic cage, short ribs, shortened tubular bones, and a 'trident' appearance of the acetabular roof. Polydactyly is variably present. Non-skeletal involvement can include cleft lip/palate as well as anomalies of major organs such as the brain, eye, heart, kidneys, liver, pancreas, intestines, and genitalia. Some forms of the disease are lethal in the neonatal period due to respiratory insufficiency secondary to a severely restricted thoracic cage, whereas others are compatible with life. Disease spectrum encompasses Ellis-van Creveld syndrome, asphyxiating thoracic dystrophy (Jeune syndrome), Mainzer-Saldino syndrome, and short rib-polydactyly syndrome.

EXPRESSÃO TECIDUAL(Ubíquo)
Ovário
44.8 TPM
Cervix Ectocervix
43.8 TPM
Cervix Endocervix
43.3 TPM
Testículo
41.5 TPM
Útero
37.8 TPM
OUTRAS DOENÇAS (2)
short-rib thoracic dysplasia 16 with or without polydactylycranioectodermal dysplasia
HGNC:15901UniProt:Q9Y366
IL11RAInterleukin-11 receptor subunit alphaDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Receptor for interleukin-11 (IL11). The receptor systems for IL6, LIF, OSM, CNTF, IL11 and CT1 can utilize IL6ST for initiating signal transmission. The IL11/IL11RA/IL6ST complex may be involved in the control of proliferation and/or differentiation of skeletogenic progenitor or other mesenchymal cells (Probable). Essential for the normal development of craniofacial bones and teeth. Restricts suture fusion and tooth number Soluble form of IL11 receptor (sIL11RA) that acts as an agonist of IL11 a

LOCALIZAÇÃO

MembraneSecreted

VIAS BIOLÓGICAS (1)
IL-6-type cytokine receptor ligand interactions
MECANISMO DE DOENÇA

Craniosynostosis and dental anomalies

A disorder characterized by craniosynostosis, maxillary hypoplasia, and dental anomalies, including malocclusion, delayed and ectopic tooth eruption, and/or supernumerary teeth. Some patients also display minor digit anomalies, such as syndactyly and/or clinodactyly.

EXPRESSÃO TECIDUAL(Ubíquo)
Aorta
80.6 TPM
Cervix Ectocervix
74.6 TPM
Tireoide
67.1 TPM
Cólon sigmoide
64.2 TPM
Útero
62.3 TPM
OUTRAS DOENÇAS (1)
craniosynostosis and dental anomalies
HGNC:5967UniProt:Q14626
MSX2Homeobox protein MSX-2Disease-causing germline mutation(s) inTolerante
FUNÇÃO

Acts as a transcriptional regulator in bone development. Represses the ALPL promoter activity and antagonizes the stimulatory effect of DLX5 on ALPL expression during osteoblast differentiation. Probable morphogenetic role. May play a role in limb-pattern formation. In osteoblasts, suppresses transcription driven by the osteocalcin FGF response element (OCFRE). Binds to the homeodomain-response element of the ALPL promoter

LOCALIZAÇÃO

Nucleus

VIAS BIOLÓGICAS (1)
Regulation of RUNX2 expression and activity
MECANISMO DE DOENÇA

Parietal foramina 1

Autosomal dominant disease characterized by oval defects of the parietal bones caused by deficient ossification around the parietal notch, which is normally obliterated during the fifth fetal month.

EXPRESSÃO TECIDUAL(Tecido-específico)
Bladder
17.6 TPM
Artéria tibial
11.3 TPM
Testículo
11.2 TPM
Cerebelo
7.3 TPM
Skin Sun Exposed Lower leg
6.0 TPM
OUTRAS DOENÇAS (4)
parietal foramina 1parietal foramina with cleidocranial dysplasiacraniosynostosis 2parietal foramina
HGNC:7392UniProt:P35548
FBN1Fibrillin-1Candidate gene tested inAltamente restrito
FUNÇÃO

Structural component of the 10-12 nm diameter microfibrils of the extracellular matrix, which conveys both structural and regulatory properties to load-bearing connective tissues (PubMed:15062093, PubMed:1860873). Fibrillin-1-containing microfibrils provide long-term force bearing structural support (PubMed:27026396). In tissues such as the lung, blood vessels and skin, microfibrils form the periphery of the elastic fiber, acting as a scaffold for the deposition of elastin (PubMed:27026396). In

LOCALIZAÇÃO

SecretedSecreted, extracellular space, extracellular matrix

VIAS BIOLÓGICAS (2)
Post-translational protein phosphorylationRegulation of Insulin-like Growth Factor (IGF) transport and uptake by Insulin-like Growth Factor Binding Proteins (IGFBPs)
MECANISMO DE DOENÇA

Marfan syndrome

A hereditary disorder of connective tissue that affects the skeletal, ocular, and cardiovascular systems. A wide variety of skeletal abnormalities occurs with Marfan syndrome, including scoliosis, chest wall deformity, tall stature, abnormal joint mobility. Ectopia lentis occurs in most of the patients and is almost always bilateral. The leading cause of premature death is progressive dilation of the aortic root and ascending aorta, causing aortic incompetence and dissection. Neonatal Marfan syndrome is the most severe form resulting in death from cardiorespiratory failure in the first few years of life.

EXPRESSÃO TECIDUAL(Ubíquo)
Fibroblastos
295.9 TPM
Artéria coronária
63.8 TPM
Aorta
63.1 TPM
Tecido adiposo
54.3 TPM
Esôfago - Junção
48.0 TPM
OUTRAS DOENÇAS (14)
geleophysic dysplasia 2Weill-Marchesani syndrome 2, dominantMASS syndromeectopia lentis 1, isolated, autosomal dominant
HGNC:3603UniProt:P35555
ERFETS domain-containing transcription factor ERFDisease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Potent transcriptional repressor that binds to the H1 element of the Ets2 promoter. May regulate other genes involved in cellular proliferation. Required for extraembryonic ectoderm differentiation, ectoplacental cone cavity closure, and chorioallantoic attachment (By similarity). May be important for regulating trophoblast stem cell differentiation (By similarity)

LOCALIZAÇÃO

Nucleus

VIAS BIOLÓGICAS (1)
Oncogene Induced Senescence
MECANISMO DE DOENÇA

Craniosynostosis 4

A primary abnormality of skull growth involving premature fusion of one or more cranial sutures. The growth velocity of the skull often cannot match that of the developing brain resulting in an abnormal head shape and, in some cases, increased intracranial pressure, which must be treated promptly to avoid permanent neurodevelopmental disability.

EXPRESSÃO TECIDUAL(Ubíquo)
Fallopian Tube
95.0 TPM
Nervo tibial
86.2 TPM
Útero
85.8 TPM
Ovário
72.8 TPM
Cervix Endocervix
71.0 TPM
INTERAÇÕES PROTEICAS (5)
OUTRAS DOENÇAS (4)
Chitayat syndromecraniosynostosis 4Crouzon syndromecraniosynostosis-facial dysmorphism-chiari-1 malformation-developmental and language delay syndrome
HGNC:3444UniProt:P50548
CYP26B1Cytochrome P450 26B1Disease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

A cytochrome P450 monooxygenase involved in the metabolism of retinoates (RAs), the active metabolites of vitamin A, and critical signaling molecules in animals (PubMed:10823918, PubMed:22020119). RAs exist as at least four different isomers: all-trans-RA (atRA), 9-cis-RA, 13-cis-RA, and 9,13-dicis-RA, where atRA is considered to be the biologically active isomer, although 9-cis-RA and 13-cis-RA also have activity (Probable). Catalyzes the hydroxylation of atRA primarily at C-4 and C-18, thereby

LOCALIZAÇÃO

Endoplasmic reticulum membraneMicrosome membrane

VIAS BIOLÓGICAS (2)
VitaminsRA biosynthesis pathway
MECANISMO DE DOENÇA

Radiohumeral fusions with other skeletal and craniofacial anomalies

A disease characterized by craniofacial malformations, occipital encephalocele, radiohumeral fusions, oligodactyly, advanced osseous maturation, and calvarial mineralization defects.

EXPRESSÃO TECIDUAL(Ubíquo)
Cérebro - Hemisfério cerebelar
65.3 TPM
Cerebelo
56.0 TPM
Skin Sun Exposed Lower leg
46.0 TPM
Skin Not Sun Exposed Suprapubic
39.2 TPM
Tecido adiposo
35.6 TPM
OUTRAS DOENÇAS (1)
lethal occipital encephalocele-skeletal dysplasia syndrome
HGNC:20581UniProt:Q9NR63
LRP5Low-density lipoprotein receptor-related protein 5Candidate gene tested inRestrito
FUNÇÃO

Acts as a coreceptor with members of the frizzled family of seven-transmembrane spanning receptors to transduce signal by Wnt proteins (PubMed:11336703, PubMed:11448771, PubMed:11719191, PubMed:15778503, PubMed:15908424, PubMed:16252235). Activates the canonical Wnt signaling pathway that controls cell fate determination and self-renewal during embryonic development and adult tissue regeneration (PubMed:11336703, PubMed:11719191). In particular, may play an important role in the development of t

LOCALIZAÇÃO

MembraneEndoplasmic reticulum

VIAS BIOLÓGICAS (1)
Negative regulation of TCF-dependent signaling by WNT ligand antagonists
MECANISMO DE DOENÇA

Vitreoretinopathy, exudative 1

An autosomal dominant disorder of the retinal vasculature characterized by an abrupt cessation of growth of peripheral capillaries, leading to an avascular peripheral retina. This may lead to compensatory retinal neovascularization, which is thought to be induced by hypoxia from the initial avascular insult. New vessels are prone to leakage and rupture causing exudates and bleeding, followed by scarring, retinal detachment and blindness. Clinical features can be highly variable, even within the same family. Patients with mild forms of the disease are asymptomatic, and their only disease related abnormality is an arc of avascular retina in the extreme temporal periphery. In many ways the disease resembles retinopathy of prematurity but there is no evidence of prematurity or small birth weight in the patient history.

EXPRESSÃO TECIDUAL(Ubíquo)
Aorta
83.2 TPM
Artéria tibial
67.8 TPM
Glândula salivar
64.2 TPM
Útero
56.7 TPM
Tireoide
51.4 TPM
OUTRAS DOENÇAS (12)
polycystic liver disease 4 with or without kidney cystsobsolete bone mineral density quantitative trait locus 1autosomal dominant osteosclerosis, Worth typeosteoporosis-pseudoglioma syndrome
HGNC:6697UniProt:O75197
IFT140Intraflagellar transport protein 140 homologDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Component of the IFT complex A (IFT-A), a complex required for retrograde ciliary transport and entry into cilia of G protein-coupled receptors (GPCRs) (PubMed:20889716, PubMed:22503633). Plays a pivotal role in proper development and function of ciliated cells through its role in ciliogenesis and/or cilium maintenance (PubMed:22503633). Required for the development and maintenance of the outer segments of rod and cone photoreceptor cells. Plays a role in maintenance and the delivery of opsin to

LOCALIZAÇÃO

Cytoplasm, cytoskeleton, cilium basal bodyCytoplasm, cytoskeleton, microtubule organizing center, centrosomeCell projection, cilium

VIAS BIOLÓGICAS (2)
Hedgehog 'off' stateIntraflagellar transport
MECANISMO DE DOENÇA

Short-rib thoracic dysplasia 9 with or without polydactyly

A form of short-rib thoracic dysplasia, a group of autosomal recessive ciliopathies that are characterized by a constricted thoracic cage, short ribs, shortened tubular bones, and a 'trident' appearance of the acetabular roof. Polydactyly is variably present. Non-skeletal involvement can include cleft lip/palate as well as anomalies of major organs such as the brain, eye, heart, kidneys, liver, pancreas, intestines, and genitalia. Some forms of the disease are lethal in the neonatal period due to respiratory insufficiency secondary to a severely restricted thoracic cage, whereas others are compatible with life. Disease spectrum encompasses Ellis-van Creveld syndrome, asphyxiating thoracic dystrophy (Jeune syndrome), Mainzer-Saldino syndrome, and short rib-polydactyly syndrome. SRTD9 is characterized by phalangeal cone-shaped epiphyses, chronic renal disease, nearly constant retinal dystrophy, and mild radiographic abnormality of the proximal femur. Occasional features include short stature, cerebellar ataxia, and hepatic fibrosis.

EXPRESSÃO TECIDUAL(Ubíquo)
Tireoide
59.8 TPM
Testículo
47.9 TPM
Pituitária
34.4 TPM
Ovário
31.1 TPM
Cervix Endocervix
29.6 TPM
OUTRAS DOENÇAS (8)
retinitis pigmentosa 80cranioectodermal dysplasia 5short-rib thoracic dysplasia 9 with or without polydactylyJeune syndrome
HGNC:29077UniProt:Q96RY7
RAB23Ras-related protein Rab-23Disease-causing germline mutation(s) inTolerante
FUNÇÃO

The small GTPases Rab are key regulators of intracellular membrane trafficking, from the formation of transport vesicles to their fusion with membranes. Rabs cycle between an inactive GDP-bound form and an active GTP-bound form that is able to recruit to membranes different set of downstream effectors directly responsible for vesicle formation, movement, tethering and fusion. In conjunction with IFT57 and KIF17, it drives the localization of specific G protein-coupled receptors, such as the dopa

LOCALIZAÇÃO

Cell membraneCytoplasmCytoplasmic vesicle, autophagosomeEndosome membraneCytoplasmic vesicle, phagosomeCytoplasmic vesicle, phagosome membrane

VIAS BIOLÓGICAS (1)
RAB geranylgeranylation
MECANISMO DE DOENÇA

Carpenter syndrome 1

A rare autosomal recessive disorder characterized by acrocephaly with variable synostosis of the sagittal, lambdoid, and coronal sutures; peculiar facies; brachydactyly of the hands with syndactyly; preaxial polydactyly and syndactyly of the feet; congenital heart defects; growth retardation; intellectual disability; hypogenitalism; and obesity. In addition, cerebral malformations, oral and dental abnormalities, coxa valga, genu valgum, hydronephrosis, precocious puberty, and hearing loss may be observed.

VIAS REACTOME (1)
EXPRESSÃO TECIDUAL(Ubíquo)
Cólon sigmoide
83.1 TPM
Útero
74.4 TPM
Artéria tibial
68.7 TPM
Aorta
53.9 TPM
Cervix Endocervix
49.7 TPM
OUTRAS DOENÇAS (2)
RAB23-related Carpenter syndromeCarpenter syndrome
HGNC:14263UniProt:Q9ULC3
CD96T-cell surface protein tactileDisease-causing germline mutation(s) inTolerante
FUNÇÃO

May be involved in adhesive interactions of activated T and NK cells during the late phase of the immune response. Promotes NK cell-target adhesion by interacting with PVR present on target cells. May function at a time after T and NK cells have penetrated the endothelium using integrins and selectins, when they are actively engaging diseased cells and moving within areas of inflammation

LOCALIZAÇÃO

Membrane

VIAS BIOLÓGICAS (1)
Immunoregulatory interactions between a Lymphoid and a non-Lymphoid cell
MECANISMO DE DOENÇA

C syndrome

A syndrome characterized by trigonocephaly, severe intellectual disability, hypotonia, variable cardiac defects, redundant skin, and dysmorphic facial features, including upslanted palpebral fissures, epicanthal folds, depressed nasal bridge, and low-set, posteriorly rotated ears.

OUTRAS DOENÇAS (1)
C syndrome
HGNC:16892UniProt:P40200
RNU12Disease-causing germline mutation(s) inDesconhecido
LOCALIZAÇÃO

VIAS BIOLÓGICAS (1)
RNA polymerase II transcribes snRNA genes
OUTRAS DOENÇAS (3)
craniosynostosis-anal anomalies-porokeratosis syndromespinocerebellar ataxia, autosomal recessive 33congenital cerebellar ataxia due to RNU12 mutation
HGNC:19380
KAT6AHistone acetyltransferase KAT6ADisease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Histone acetyltransferase that acetylates lysine residues in histone H3 and histone H4 (in vitro) (PubMed:11742995, PubMed:11965546). Component of the MOZ/MORF complex which has a histone H3 acetyltransferase activity (PubMed:11965546). May act as a transcriptional coactivator for RUNX1 and RUNX2 (PubMed:12771199). Acetylates p53/TP53 at 'Lys-120' and 'Lys-382' and controls its transcriptional activity via association with PML (PubMed:23431171). May play a role in leukemogenic gene transcription

LOCALIZAÇÃO

NucleusNucleus, nucleolusNucleus, nucleoplasmNucleus, PML body

VIAS BIOLÓGICAS (2)
Regulation of TP53 Activity through AcetylationHATs acetylate histones
EXPRESSÃO TECIDUAL(Ubíquo)
Pulmão
28.6 TPM
Útero
26.8 TPM
Cervix Ectocervix
25.7 TPM
Skin Sun Exposed Lower leg
25.1 TPM
Nervo tibial
24.6 TPM
OUTRAS DOENÇAS (2)
autosomal dominant intellectual disability-craniofacial anomalies-cardiac defects syndromeacute myeloid leukemia with t(8;16)(p11;p13) translocation
HGNC:13013UniProt:Q92794
WDR19WD repeat-containing protein 19Disease-causing germline mutation(s) inTolerante
FUNÇÃO

As component of the IFT complex A (IFT-A), a complex required for retrograde ciliary transport and entry into cilia of G protein-coupled receptors (GPCRs), it is involved in cilia function and/or assembly (PubMed:20889716). Essential for functional IFT-A assembly and ciliary entry of GPCRs (PubMed:20889716). Associates with the BBSome complex to mediate ciliary transport (By similarity)

LOCALIZAÇÃO

Cell projection, ciliumCytoplasm, cytoskeleton, cilium basal bodyCell projection, cilium, photoreceptor outer segmentCell projection, cilium, flagellum

VIAS BIOLÓGICAS (2)
Hedgehog 'off' stateIntraflagellar transport
MECANISMO DE DOENÇA

Cranioectodermal dysplasia 4

A disorder primarily characterized by craniofacial, skeletal and ectodermal abnormalities. Clinical features include craniosynostosis, narrow rib cage, short limbs, brachydactyly, hypoplastic and widely spaced teeth, sparse hair, skin laxity and abnormal nails. Nephronophthisis leading to progressive renal failure, hepatic fibrosis, heart defects, and retinitis pigmentosa have also been described.

EXPRESSÃO TECIDUAL(Ubíquo)
Ovário
43.9 TPM
Tireoide
41.9 TPM
Fallopian Tube
40.6 TPM
Pituitária
39.0 TPM
Útero
37.9 TPM
OUTRAS DOENÇAS (9)
Senior-Loken syndrome 8asphyxiating thoracic dystrophy 5nephronophthisis 13cranioectodermal dysplasia 4
HGNC:18340UniProt:Q8NEZ3
TCF12Transcription factor 12Disease-causing germline mutation(s) inTolerante
FUNÇÃO

Transcriptional regulator. Involved in the initiation of neuronal differentiation. Activates transcription by binding to the E box (5'-CANNTG-3') (By similarity). May be involved in the functional network that regulates the development of the GnRH axis (PubMed:32620954)

LOCALIZAÇÃO

Nucleus

VIAS BIOLÓGICAS (5)
Negative Regulation of CDH1 Gene TranscriptionRUNX1 regulates transcription of genes involved in differentiation of HSCsNGF-stimulated transcriptionMyogenesisTGFBR3 expression
MECANISMO DE DOENÇA

Craniosynostosis 3

A primary abnormality of skull growth involving premature fusion of one or more cranial sutures. The growth velocity of the skull often cannot match that of the developing brain resulting in an abnormal head shape and, in some cases, increased intracranial pressure, which must be treated promptly to avoid permanent neurodevelopmental disability.

EXPRESSÃO TECIDUAL(Ubíquo)
Cervix Endocervix
39.7 TPM
Cervix Ectocervix
38.1 TPM
Fibroblastos
37.1 TPM
Útero
32.3 TPM
Nervo tibial
28.4 TPM
OUTRAS DOENÇAS (4)
hypogonadotropic hypogonadism 26 with or without anosmiaTCF12-related craniosynostosisextraskeletal myxoid chondrosarcomaobsolete isolated brachycephaly
HGNC:11623UniProt:Q99081
IFT122Intraflagellar transport protein 122 homologDisease-causing germline mutation(s) inTolerante
FUNÇÃO

As a component of the IFT complex A (IFT-A), a complex required for retrograde ciliary transport and entry into cilia of G protein-coupled receptors (GPCRs), it is required in ciliogenesis and ciliary protein trafficking (PubMed:27932497, PubMed:29220510). Involved in cilia formation during neuronal patterning. Acts as a negative regulator of Shh signaling. Required to recruit TULP3 to primary cilia (By similarity)

LOCALIZAÇÃO

Cell projection, ciliumCytoplasm, cytoskeleton, cilium basal body

VIAS BIOLÓGICAS (2)
Hedgehog 'off' stateIntraflagellar transport
MECANISMO DE DOENÇA

Cranioectodermal dysplasia 1

A disorder characterized by craniofacial, skeletal and ectodermal abnormalities. Clinical features include dolichocephaly (with or without sagittal suture synostosis), scaphocephaly, short stature, limb shortening, short ribs, narrow chest, brachydactyly, renal failure and hepatic fibrosis, small and abnormally shaped teeth, sparse hair, skin laxity and abnormal nails.

EXPRESSÃO TECIDUAL(Ubíquo)
Testículo
157.0 TPM
Ovário
43.0 TPM
Pituitária
38.0 TPM
Cervix Endocervix
35.0 TPM
Nervo tibial
33.9 TPM
OUTRAS DOENÇAS (3)
cranioectodermal dysplasia 1Beemer-Langer syndromecranioectodermal dysplasia
HGNC:13556UniProt:Q9HBG6
SKISki oncogeneDisease-causing germline mutation(s) inRestrito
FUNÇÃO

May play a role in terminal differentiation of skeletal muscle cells but not in the determination of cells to the myogenic lineage. Functions as a repressor of TGF-beta signaling

LOCALIZAÇÃO

Nucleus

VIAS BIOLÓGICAS (2)
Downregulation of SMAD2/3:SMAD4 transcriptional activitySignaling by BMP
MECANISMO DE DOENÇA

Shprintzen-Goldberg craniosynostosis syndrome

A very rare syndrome characterized by a marfanoid habitus, craniosynostosis, characteristic dysmorphic facial features, skeletal and cardiovascular abnormalities, intellectual disability, developmental delay and learning disabilities.

EXPRESSÃO TECIDUAL(Ubíquo)
Cérebro - Hemisfério cerebelar
235.4 TPM
Cerebelo
233.1 TPM
Artéria tibial
154.4 TPM
Aorta
150.9 TPM
Útero
107.2 TPM
OUTRAS DOENÇAS (2)
Shprintzen-Goldberg syndromechromosome 1p36 deletion syndrome
HGNC:10896UniProt:P12755
IFT43Intraflagellar transport protein 43 homologDisease-causing germline mutation(s) inTolerante
FUNÇÃO

As a component of IFT complex A (IFT-A), a complex required for retrograde ciliary transport and entry into cilia of G protein-coupled receptors (GPCRs), it is involved in ciliogenesis (PubMed:28400947, PubMed:28973684). Involved in retrograde ciliary transport along microtubules from the ciliary tip to the base (PubMed:21378380)

LOCALIZAÇÃO

Cytoplasm, cytoskeletonCell projection, cilium

VIAS BIOLÓGICAS (1)
Intraflagellar transport
MECANISMO DE DOENÇA

Cranioectodermal dysplasia 3

A disorder primarily characterized by craniofacial, skeletal and ectodermal abnormalities. Clinical features include craniosynostosis, narrow rib cage, short limbs, brachydactyly, hypoplastic and widely spaced teeth, sparse hair, skin laxity and abnormal nails. Nephronophthisis leading to progressive renal failure, hepatic fibrosis, heart defects, and retinitis pigmentosa have also been described.

VIAS REACTOME (1)
EXPRESSÃO TECIDUAL(Ubíquo)
Testículo
38.9 TPM
Pituitária
32.1 TPM
Tireoide
31.6 TPM
Fallopian Tube
30.3 TPM
Cervix Endocervix
29.3 TPM
OUTRAS DOENÇAS (5)
cranioectodermal dysplasia 3retinitis pigmentosa 81short-rib thoracic dysplasia 18 with polydactylycranioectodermal dysplasia
HGNC:29669UniProt:Q96FT9
TWIST1Twist-related protein 1Disease-causing germline mutation(s) inTolerante
FUNÇÃO

Acts as a transcriptional regulator. Inhibits myogenesis by sequestrating E proteins, inhibiting trans-activation by MEF2, and inhibiting DNA-binding by MYOD1 through physical interaction. This interaction probably involves the basic domains of both proteins. Also represses expression of pro-inflammatory cytokines such as TNFA and IL1B. Regulates cranial suture patterning and fusion. Activates transcription as a heterodimer with E proteins. Regulates gene expression differentially, depending on

LOCALIZAÇÃO

Nucleus

VIAS BIOLÓGICAS (5)
Negative Regulation of CDH1 Gene TranscriptionRegulation of RUNX2 expression and activityTranscriptional regulation by RUNX2Nuclear events stimulated by ALK signaling in cancerInterleukin-4 and Interleukin-13 signaling
MECANISMO DE DOENÇA

Saethre-Chotzen syndrome

A craniosynostosis syndrome characterized by coronal synostosis, brachycephaly, low frontal hairline, facial asymmetry, hypertelorism, broad halluces, and clinodactyly.

EXPRESSÃO TECIDUAL(Ubíquo)
Fibroblastos
128.4 TPM
Mama
42.0 TPM
Tecido adiposo
36.0 TPM
Útero
28.8 TPM
Aorta
26.2 TPM
OUTRAS DOENÇAS (6)
Sweeney-Cox syndromeTWIST1-related craniosynostosisRobinow-Sorauf syndromeSaethre-Chotzen syndrome
HGNC:12428UniProt:Q15672
SMOSpermine oxidaseDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Flavoenzyme which catalyzes the oxidation of spermine to spermidine. Can also use N(1)-acetylspermine and spermidine as substrates, with different affinity depending on the isoform (isozyme) and on the experimental conditions. Plays an important role in the regulation of polyamine intracellular concentration and has the potential to act as a determinant of cellular sensitivity to the antitumor polyamine analogs. May contribute to beta-alanine production via aldehyde dehydrogenase conversion of 3

LOCALIZAÇÃO

CytoplasmNucleus

VIAS BIOLÓGICAS (1)
Hedgehog 'off' state
EXPRESSÃO TECIDUAL(Ubíquo)
Cervix Endocervix
62.8 TPM
Ovário
60.7 TPM
Cervix Ectocervix
56.8 TPM
Útero
50.1 TPM
Pituitária
38.7 TPM
OUTRAS DOENÇAS (5)
congenital hypothalamic hamartoma syndromebasal cell carcinoma, susceptibility to, 1Curry-Jones syndromemeningioma
HGNC:11119UniProt:Q9NWM0
RECQL4ATP-dependent DNA helicase Q4Disease-causing germline mutation(s) inTolerante
FUNÇÃO

An ATP-dependent DNA helicase which unwinds dsDNA with a 3'-overhang in a 3'-5' direction (PubMed:28653661). Does not unwind more than 18 bp of dsDNA (PubMed:28653661). May modulate chromosome segregation. The N-terminal domain (residues 1-54) binds DNA Y-shaped DNA better than ss- or dsDNA (PubMed:22730300). The core helicase domain binds ssDNA (PubMed:22730300, PubMed:28653661)

LOCALIZAÇÃO

CytoplasmNucleus

MECANISMO DE DOENÇA

RAPADILINO syndrome

Disease characterized by radial and patellar aplasia or hypoplasia.

EXPRESSÃO TECIDUAL(Ubíquo)
Testículo
56.5 TPM
Linfócitos
32.2 TPM
Cerebelo
26.9 TPM
Cérebro - Hemisfério cerebelar
23.8 TPM
Esôfago - Mucosa
13.9 TPM
OUTRAS DOENÇAS (3)
Baller-Gerold syndromerapadilino syndromeRothmund-Thomson syndrome type 2
HGNC:9949UniProt:O94761
FGFR2Fibroblast growth factor receptor 2Disease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Tyrosine-protein kinase that acts as a cell-surface receptor for fibroblast growth factors and plays an essential role in the regulation of cell proliferation, differentiation, migration and apoptosis, and in the regulation of embryonic development. Required for normal embryonic patterning, trophoblast function, limb bud development, lung morphogenesis, osteogenesis and skin development. Plays an essential role in the regulation of osteoblast differentiation, proliferation and apoptosis, and is

LOCALIZAÇÃO

Cell membraneGolgi apparatusCytoplasmic vesicleSecreted

VIAS BIOLÓGICAS (1)
Signaling by FGFR2 amplification mutants
MECANISMO DE DOENÇA

Crouzon syndrome

An autosomal dominant syndrome characterized by craniosynostosis, hypertelorism, exophthalmos and external strabismus, parrot-beaked nose, short upper lip, hypoplastic maxilla, and a relative mandibular prognathism.

EXPRESSÃO TECIDUAL(Ubíquo)
Brain Spinal cord cervical c-1
130.1 TPM
Útero
43.5 TPM
Skin Sun Exposed Lower leg
41.0 TPM
Cólon sigmoide
39.0 TPM
Skin Not Sun Exposed Suprapubic
37.3 TPM
OUTRAS DOENÇAS (15)
Saethre-Chotzen syndromegastric cancerJackson-Weiss syndromePfeiffer syndrome
HGNC:3689UniProt:P21802
FGFR3Fibroblast growth factor receptor 3Disease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Tyrosine-protein kinase that acts as a cell-surface receptor for fibroblast growth factors and plays an essential role in the regulation of cell proliferation, differentiation and apoptosis. Plays an essential role in the regulation of chondrocyte differentiation, proliferation and apoptosis, and is required for normal skeleton development. Regulates both osteogenesis and postnatal bone mineralization by osteoblasts. Promotes apoptosis in chondrocytes, but can also promote cancer cell proliferat

LOCALIZAÇÃO

Cell membraneCytoplasmic vesicleEndoplasmic reticulumSecreted

VIAS BIOLÓGICAS (2)
Signaling by FGFR3 in diseaset(4;14) translocations of FGFR3
MECANISMO DE DOENÇA

Achondroplasia

A frequent form of short-limb dwarfism. It is characterized by a long, narrow trunk, short extremities, particularly in the proximal (rhizomelic) segments, a large head with frontal bossing, hypoplasia of the midface and a trident configuration of the hands. ACH is an autosomal dominant disease.

EXPRESSÃO TECIDUAL(Ubíquo)
Skin Not Sun Exposed Suprapubic
364.6 TPM
Skin Sun Exposed Lower leg
356.5 TPM
Esôfago - Mucosa
199.7 TPM
Brain Caudate basal ganglia
148.4 TPM
Brain Nucleus accumbens basal ganglia
135.4 TPM
OUTRAS DOENÇAS (19)
nevus, epidermalsevere achondroplasia-developmental delay-acanthosis nigricans syndromelacrimoauriculodentodigital syndrome 2testicular germ cell tumor
HGNC:3690UniProt:P22607
ZNF462Zinc finger protein 462Disease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Zinc finger nuclear factor involved in transcription by regulating chromatin structure and organization (PubMed:20219459, PubMed:21570965). Involved in the pluripotency and differentiation of embryonic stem cells by regulating SOX2, POU5F1/OCT4, and NANOG (PubMed:21570965). By binding PBX1, prevents the heterodimerization of PBX1 and HOXA9 and their binding to DNA (By similarity). Regulates neuronal development and neural cell differentiation (PubMed:21570965)

LOCALIZAÇÃO

Nucleus

MECANISMO DE DOENÇA

Weiss-Kruszka syndrome

An autosomal dominant, multiple congenital anomaly syndrome with variable expressivity and complete penetrance. Patients manifest developmental delay, hypotonia, feeding difficulties, craniofacial abnormalities including ptosis, abnormal head shape, downslanting palpebral fissures, metopic ridging, and craniosynostosis. Variable congenital heart defects can be observed in some patients. A few patients show agenesis of the corpus callosum on brain imaging.

EXPRESSÃO TECIDUAL(Ubíquo)
Nervo tibial
10.7 TPM
Ovário
9.7 TPM
Cervix Ectocervix
9.2 TPM
Testículo
9.0 TPM
Tireoide
8.8 TPM
INTERAÇÕES PROTEICAS (2)
OUTRAS DOENÇAS (1)
Weiss-Kruszka syndrome
HGNC:21684UniProt:Q96JM2

Medicamentos e terapias

EPOPROSTENOLPhase 3

Mecanismo: Prostanoid IP receptor agonist

PRAVASTATINPhase 3

Mecanismo: HMG-CoA reductase inhibitor

IRBESARTANPhase 3

Mecanismo: Type-1 angiotensin II receptor antagonist

RITUXIMABPhase 2

Mecanismo: B-lymphocyte antigen CD20 binding agent

Ver mais no OpenTargets

Variantes genéticas (ClinVar)

828 variantes patogênicas registradas no ClinVar.

🧬 WDR35: NM_020779.4(WDR35):c.1990C>T (p.Arg664Ter) ()
🧬 WDR35: NM_020779.4(WDR35):c.1525-2_1527dup ()
🧬 WDR35: NM_020779.4(WDR35):c.2956del (p.Ser986fs) ()
🧬 WDR35: NM_020779.4(WDR35):c.2306G>A (p.Trp769Ter) ()
🧬 WDR35: NM_020779.4(WDR35):c.721_727del (p.His241fs) ()
Ver todas no ClinVar

Classificação de variantes (ClinVar)

Distribuição de 6 variantes classificadas pelo ClinVar.

6
Patogênica (100.0%)
VARIANTES MAIS SIGNIFICATIVAS
HDAC9: GRCh37/hg19 7p21.1(chr7:17930686-19059254)x1 [Likely pathogenic]
HNMT: GRCh37/hg19 2q22.1(chr2:138458639-141918297)x1 [Likely pathogenic]
CMPK2: GRCh37/hg19 2p25.2-25.1(chr2:5689487-7379378)x3 [Likely pathogenic]
PHF5A: GRCh37/hg19 22q13.1-13.2(chr22:40545592-42096995)x3 [Likely pathogenic]
CIC: NC_000019.10:g.42032860_42297536del [Pathogenic]

Vias biológicas (Reactome)

86 vias biológicas associadas aos genes desta condição.

Hedgehog 'off' state Intraflagellar transport Cytochrome P450 - arranged by substrate type PI3K Cascade PIP3 activates AKT signaling Signaling by FGFR1 amplification mutants Signaling by activated point mutants of FGFR1 FGFR1b ligand binding and activation FGFR1c ligand binding and activation FGFR1c and Klotho ligand binding and activation Constitutive Signaling by Aberrant PI3K in Cancer NCAM signaling for neurite out-growth Signal transduction by L1 Phospholipase C-mediated cascade: FGFR1 Downstream signaling of activated FGFR1 SHC-mediated cascade:FGFR1 PI-3K cascade:FGFR1 FRS-mediated FGFR1 signaling Negative regulation of FGFR1 signaling Signaling by FGFR1 in disease RAF/MAP kinase cascade PI5P, PP2A and IER3 Regulate PI3K/AKT Signaling Signaling by plasma membrane FGFR1 fusions Epithelial-Mesenchymal Transition (EMT) during gastrulation Formation of paraxial mesoderm IL-6-type cytokine receptor ligand interactions Regulation of RUNX2 expression and activity Degradation of the extracellular matrix Elastic fibre formation Molecules associated with elastic fibres Integrin cell surface interactions TGF-beta receptor signaling activates SMADs Regulation of Insulin-like Growth Factor (IGF) transport and uptake by Insulin-like Growth Factor Binding Proteins (IGFBPs) Post-translational protein phosphorylation Oncogene Induced Senescence Vitamins RA biosynthesis pathway Defective CYP26B1 causes RHFCA TCF dependent signaling in response to WNT Negative regulation of TCF-dependent signaling by WNT ligand antagonists Disassembly of the destruction complex and recruitment of AXIN to the membrane Regulation of FZD by ubiquitination Signaling by LRP5 mutants Signaling by RNF43 mutants RAB geranylgeranylation Immunoregulatory interactions between a Lymphoid and a non-Lymphoid cell RNU12 gene U12 snRNA U1,U2,U4,U4atac,U5,U11,U12 gene HATs acetylate histones Regulation of TP53 Activity through Acetylation Myogenesis RUNX1 regulates transcription of genes involved in differentiation of HSCs NGF-stimulated transcription Negative Regulation of CDH1 Gene Transcription TGFBR3 expression Signaling by BMP Downregulation of SMAD2/3:SMAD4 transcriptional activity Interleukin-4 and Interleukin-13 signaling Transcriptional regulation by RUNX2 Nuclear events stimulated by ALK signaling in cancer PAOs oxidise polyamines to amines Interconversion of polyamines FGFR2c ligand binding and activation FGFR2b ligand binding and activation Signaling by FGFR2 amplification mutants Activated point mutants of FGFR2 Phospholipase C-mediated cascade; FGFR2 PI-3K cascade:FGFR2 SHC-mediated cascade:FGFR2 FRS-mediated FGFR2 signaling Negative regulation of FGFR2 signaling Signaling by FGFR2 in disease Signaling by FGFR2 IIIa TM Signaling by FGFR2 fusions Signaling by activated point mutants of FGFR3 FGFR3b ligand binding and activation FGFR3c ligand binding and activation t(4;14) translocations of FGFR3 Phospholipase C-mediated cascade; FGFR3 SHC-mediated cascade:FGFR3 FRS-mediated FGFR3 signaling PI-3K cascade:FGFR3 Negative regulation of FGFR3 signaling Signaling by FGFR3 in disease Signaling by FGFR3 fusions in cancer

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Pipeline de tratamentos
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·Pré-clínico1
Medicamentos catalogadosEnsaios clínicos· 4 medicamentos · 1 ensaio
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Onde tratar no SUS

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

🇧🇷 Atendimento SUS — Craniossinostose sindrômica

🗺️

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Ensaios clínicos abertos e novidades científicas recentes

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Publicações mais relevantes

Timeline de publicações
516 papers (10 anos)
#1

Daily executive function in children and adolescents with non-syndromic craniosynostosis: association with timing of surgical intervention.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery2026 Mar 13

This study aimed to assess executive functioning in everyday activities of participants with non-syndromic craniosynostosis, to compare their performance with a control group, to assess the relationship between the timing of surgery and executive functioning, and to determine the proportion of participants scoring in the clinical range for executive functions. Twenty-nine participants with craniosynostosis and 30 controls were assessed with the parent version of the Behavior Rating Inventory of Executive Function 2 (BRIEF-2). Both groups scored similarly. However, the older the participant with craniosynostosis at the time of the surgery, the greater their difficulties in Task Initiation, Working Memory, Planning, and Monitoring. Only a small percentage (0-17%) of participants with craniosynostosis scored in the clinically significant range; this proportion was similar to that of the control group. This study did not find substantial difficulties in executive functions in daily activities in participants with non-syndromic craniosynostosis, but it did find an association between late surgery and lower scores in some cold executive functions. The study suggests that early surgical intervention could have a potential neurodevelopmental benefit for children and adolescents with craniosynostosis.

#2

En-bloc posterior vault distraction including the foramen magnum for Chiari I malformation in severe syndromic craniosynostosis: First cases and literature review.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery2026 Mar 10

This case series describes two patients with severe Crouzon syndrome and persistent Chiari I malformation. Initial treatment with standard posterior vault distraction osteogenesis and frontoorbital advancement failed to resolve the patient's symptoms. One patient developed severe syringomyelia and myelopathy refractory even to standard suboccipital decompression. Given the persistent posterior fossa constriction, an en-bloc posterior vault distraction incorporating the foramen magnum was performed over 21 days, achieving an expansion of 20 mm. This approach resulted in complete symptom resolution in both patients, disappearance of syringomyelia, and normalization of posterior fossa anatomy. One procedure was guided by intraoperative cone-beam CT (Brainlab Loop-X) to optimize the distraction vector and ensure optimal ventricular catheter placement during the concomitant shunt revision. These are the first reported cases of en-bloc posterior vault distractions incorporating the foramen magnum to simultaneously address both posterior cranial restriction and secondary Chiari I malformation. They proof the feasibility of a mono-bloc technique to simultaneously distract the posterior cranial vault and the posterior fossa down to the foramen magnum.

#3

Ophthalmic Pathologies in Craniosynostosis: Risk Factors and Disparities in the United States.

The Journal of craniofacial surgery2026

Known ophthalmic manifestations of craniosynostosis include strabismus, papilledema, refractive errors, and amblyopia. This study uses a national database to identify risk factors and disparities in the presentation of these ocular manifestations of craniosynostosis in the last decade. Hospital discharges with diagnoses of craniosynostosis were identified in the 2016 to 2022 National Inpatient Sample. Admissions were additionally characterized by concurrent ophthalmic pathologies, as well as sociodemographic and clinical variables. A multivariable logistic regression model was used to evaluate independent predictors of comorbid ophthalmic pathology (P<0.05). The final cohort included 39,385 discharges with craniosynostosis diagnoses from 2016 to 2022. The overall rate of ocular pathologies was 7.7%. The most common pathologies were disorders of the orbit (2.1%) and strabismus (2.0%). Syndromic craniosynostosis (OR: 2.06, 95% CI: 1.90-2.24, P<0.001) predicted higher odds of concurrent ophthalmic pathologies. Black (OR: 1.30, 95% CI: 1.15-1.47, P<0.001) and Hispanic (OR: 1.23, 95% CI: 1.11-1.35, P<0.001) race/ethnicity predicted higher odds of ophthalmic pathologies, whereas private insurance (OR: 0.85, 95% CI: 0.78-0.92, P<0.001) predicted lower odds. Comorbid ophthalmic pathologies in patients with craniosynostosis may be common. Historically underserved populations, such as Black and Hispanic patients and publicly insured patients, may experience a disproportionate epidemiological and clinical burden from these comorbid conditions. Such findings emphasize the need for multidisciplinary partnership and early ophthalmology referral for both syndromic and nonsyndromic craniosynostosis patients.

#4

Morphological analysis of posterior fossa in Apert and Crouzon syndromes before and after posterior cranial vault expansion.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery2026 Feb 25

Syndromic craniosynostosis, such as Apert and Crouzon syndromes, involve premature fusion of cranial sutures, leading to cranial deformities and potential neurological complications. Posterior cranial vault expansion (PCVE) is commonly used to address intracranial volume (ICV) anomalies in these cases, especially in the posterior fossa region. However, the morphological variation of this region between normal, Apert and Crouzon individuals remains understudied. This study aimed to carry out a detailed morphological analysis of the posterior fossa between the aforementioned groups before and after PCVE. Retrospective computed tomography (CT) data from 28 children with Apert (n = 13) and Crouzon (n = 15) syndromes, alongside 51 age-matched controls, were analyzed. Intracranial and posterior fossa volumes (ICV and PFV) were computed pre- and post-operatively. Morphometric shape analyses were conducted for posterior fossa and intracranial cavity using principal component analysis (PCA). Pre-operatively, Apert syndrome patients exhibited larger PFV and ICV compared to Crouzon syndrome patients and controls (e.g., p = 0.009 for pre-operative Apert vs. controls), while Crouzon syndrome patients showed more variability in PFV and ICV. Post-operatively, Apert patients demonstrated significant increases in both PFV and PFV/ICV ratios (e.g., post-operative vs. pre-operative PFV: p < 0.001). In contrast, Crouzon patients exhibited limited improvements in PFV, with PFV/ICV ratios often declining post-surgery (p = 0.890; median post-operative-pre-operative difference = -0.095). PCA revealed distinct intracranial and posterior fossa shape differences between Apert and Crouzon patients. While PCVE increased posterior fossa volume, it did not appear to improve posterior fossa shape in either Apert or Crouzon patients. Future studies are required to address the limitations of small sample sizes, lack of post-operative data on Chiari anomaly, and surgical variability to further explore possible correlations between the aforementioned parameters.

#5

Feasibility and reproducibility of handheld and table-mounted optical coherence tomography in children with craniosynostosis.

Eye (London, England)2026 Feb 24

Optical coherence tomography (OCT) can be a valuable tool for non-invasively monitoring the optic nerve status in children with craniosynostosis. However, it is currently unknown whether optic nerve parameters derived from handheld OCT are comparable to those derived from table-mounted OCT, which is more widely used. This study aims to assess the feasibility and reproducibility of handheld and table-mounted OCT in craniosynostosis. This was a cross-sectional study conducted at Great Ormond Street Hospital (GOSH), London. Twenty children aged 4-18 years with a clinical/genetic diagnosis of craniosynostosis were included. Bilateral optic nerve head OCT imaging was performed using the Spectralis (Heidelberg Engineering), followed by the handheld Envisu C2300 (Leica Microsystems). Primary outcome measures were quantitative cup, disc, rim and peripapillary parameters. Intraclass correlation coefficients (ICC) and coefficient of variation (CoV) were calculated for each quantitative OCT parameter. 20 children (100%) were successfully recruited. Median age at the time of OCT examination was 6 years (range: 4-16; IQR: 5-8). Ten participants (50%) were female. Seven participants (35%) had syndromic craniosynostosis and 13 participants (65%) had non-syndromic craniosynostosis. Bilateral imaging success was 100% for both machines. ICCs were good-to-excellent for all parameters, ranging from 0.81 to 1.00. The coefficient of variation was low for all parameters. OCT imaging of the optic nerve is feasible in school-aged children with craniosynostosis and comparable between the Spectralis and handheld Envisu OCT. This could allow comparison and pooling of data between the two machines, greatly enhancing patient care and future research.

Publicações recentes

Ver todas no PubMed

📚 EuropePMC377 artigos no totalmostrando 198

2026

Daily executive function in children and adolescents with non-syndromic craniosynostosis: association with timing of surgical intervention.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2026

En-bloc posterior vault distraction including the foramen magnum for Chiari I malformation in severe syndromic craniosynostosis: First cases and literature review.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2026

Ophthalmic Pathologies in Craniosynostosis: Risk Factors and Disparities in the United States.

The Journal of craniofacial surgery
2026

Morphological analysis of posterior fossa in Apert and Crouzon syndromes before and after posterior cranial vault expansion.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2026

Feasibility and reproducibility of handheld and table-mounted optical coherence tomography in children with craniosynostosis.

Eye (London, England)
2026

Tracheal cartilaginous sleeve prevalence in syndromic craniosynostosis: A single institution study.

International journal of pediatric otorhinolaryngology
2026

Current Management of Craniosynostosis: A Nordic Pediatric Neurosurgery Network Study.

The Journal of craniofacial surgery
2026

Sagittal Craniosynostosis Associated With Chromosome 16p13.3 Duplication.

The Journal of craniofacial surgery
2026

Concurrent posterior vault expansion and extradural Chiari decompression in syndromic and non-syndromic craniosynostosis: a case series.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2026

Long-term morphometric and functional outcomes of frontofacial advancement in syndromic craniosynostosis.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2026

Posterior Cranial Decompression in ERF-Mutated Multisuture Craniosynostosis.

The Journal of craniofacial surgery
2026

Examining Sociodemographic Disparities in Diagnostic Delays and Surgical Management of Non-Syndromic Craniosynostosis: A 10-Year Review.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2025

Blended Phenotypes in Siblings: Dual Diagnoses of Nicolaides-Baraitser and Craniosynostosis Syndromes.

Molecular syndromology
2025

P38α MAPK-induced senescence in cranial suture progenitor cells promotes craniosynostosis.

Communications biology
2025

Midface Hypoplasia and Cranial Base Morphology in Syndromic Craniosynostosis: A Comparative Analysis Study Using a Predictive Regression Model.

Journal of visualized experiments : JoVE
2026

Secondary Coronal Synostosis After Posterior Vault Distraction Osteogenesis.

The Journal of craniofacial surgery
2025

Difference in anterior fontanelle closure between non-syndromic craniosynostosis and normal controls: a retrospective cross-sectional study.

Italian journal of pediatrics
2026

Early posterior cranial vault distraction in syndromic craniosynostosis: orbital and ocular changes.

International journal of oral and maxillofacial surgery
2025

[Dynamics of caudal dystopia of cerebellar tonsils in patients with craniosynostosis following posterior cranial distraction].

Zhurnal voprosy neirokhirurgii imeni N. N. Burdenko
2025

Management of upper airway obstruction in syndromic craniosynostosis: A lifespan approach from childhood to adulthood.

Archives de pediatrie : organe officiel de la Societe francaise de pediatrie
2025

Posterior Vault Distraction Osteogenesis: A Systematic Review and Single-Arm Metanalysis.

The Journal of craniofacial surgery
2026

Are There Morphophysiological Airway Alterations in Syndromic Craniosynostosis? A 3D Computed Tomography and CFD Analysis.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2025

Practical Use of Mixed Reality for Transfacial Pinning in Monobloc Advancement for Syndromic Craniosynostosis.

The Journal of craniofacial surgery
2025

Generation of human induced pluripotent stem cell lines from patients with FGFR2-linked syndromic craniosynostosis.

Disease models &amp; mechanisms
2025

LeFort III distraction in patients with syndromic craniosynostosis: Is overcorrection beneficial?

Journal of plastic, reconstructive &amp; aesthetic surgery : JPRAS
2025

Peripapillary Hyperreflective Ovoid Mass-like Structures before and after Intracranial Decompression: A Longitudinal Cohort Study.

Ophthalmology
2025

Syndromic vs Nonsyndromic Management of Multisuture Craniosynostosis: A Single-Center Experience.

Operative neurosurgery (Hagerstown, Md.)
2025

Highlighting posterior cranial vault expansion remodeling for treating various types of craniosynostosis in children.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2025

Long-term neurocognitive and behavioral outcomes in patients with non-syndromic craniosynostosis.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2025

Enhancing Predictive Tools for Skeletal Growth and Craniofacial Morphology in Syndromic Craniosynostosis: A Focus on Cranial Base Variables.

Diagnostics (Basel, Switzerland)
2025

Differential impact of Crouzon and Apert syndromes on upper airways morphology: implications for Obstructive Sleep Apnoea.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2026

The influence of closed sutures on cranial morphology in Apert and Crouzon syndromes: A quantitative analysis.

Journal of anatomy
2025

Eye Tracking Technology as an Adjunct in Medical Education: Our Experience at the National Paediatric Craniofacial Centre in Ireland.

The Journal of craniofacial surgery
2025

Association of Preoperative Nutritional Status With Outcomes in Fronto-Orbital Advancement for Syndromic Craniosynostosis.

Journal of oral and maxillofacial surgery : official journal of the American Association of Oral and Maxillofacial Surgeons
2025

Facial asymmetry in syndromic craniosynostosis patients undergoing midface surgery compared to a large general population.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2025

Syndromic Craniosynostosis: The Hidden Burden of Comorbidities on Surgical Outcomes.

Annals of plastic surgery
2025

A Rare Case of Prenatally Diagnosed Pfeiffer Syndrome with Multicystic Kidney.

Journal of obstetrics and gynaecology of India
2025

Craniosynostosis: Experience From a Single Tertiary Center in India.

Cureus
2025

Further Evidence of Early-Onset Osteoporosis and Bone Fractures as a New FGFR2-Related Phenotype.

International journal of molecular sciences
2025

Fathoming the scientific paradox of intangibles: protocol reappraisal for optimizing cognitive outcomes in faciocraniosynostosis-an institutional experience.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2025

Psychosocial Support for Australian Families Impacted by Craniosynostosis: A Qualitative Study.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2025

Three-dimensional assessment of outcomes of surgical midface advancement in syndromic craniosynostosis: A systematic review.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2025

Chaos to Flow: Categorization, Nomenclature to Problem Mapping: Unified Interdisciplinary Language in Craniosynostoses.

The Journal of craniofacial surgery
2025

Reversible abducens nerve palsy following cranial vault expansion in the setting of multisutural craniosynostosis: illustrative case.

Journal of neurosurgery. Case lessons
2025

Current Understanding of Crouzon Syndrome Pathophysiology and New Therapeutic Approaches.

The Journal of craniofacial surgery
2024

Targeting of C-ROS-1 Activity Using a Controlled Release Carrier to Treat Craniosynostosis in a Preclinical Model of Saethre-Chotzen Syndrome.

Journal of tissue engineering and regenerative medicine
2025

Skull Base and Craniocervical Anomalies in Syndromic Craniosynostosis.

Neurosurgery
2025

Non-syndromic craniosynostosis.

Nature reviews. Disease primers
2025

Upper Airway Obstruction Trends in Craniofacial Syndromes: A Comparative Study.

The Journal of craniofacial surgery
2025

Determining the cause of optic nerve atrophy in syndromic craniosynostosis using logistic regression.

International ophthalmology
2025

Overlap of genetic factors among craniosynostosis and autism spectrum disorder: the presence of autistic cases without craniosynostosis in carriers of shared variants.

Journal of neurosurgery. Pediatrics
2025

Clinical Nasal Deviation Following Midface Advancement in Patients With Syndromic Craniosynostosis.

The Journal of craniofacial surgery
2025

Craniosynostosis and Chiari I Malformation Managed With Middle 1/3 Calvarial Vault Expansion.

The Journal of craniofacial surgery
2025

The Efficacy of Postoperative Helmet Therapy After Open Cranial Vault Remodeling for Nonsyndromic Craniosynostosis.

The Journal of craniofacial surgery
2025

Novel digital measurement system for predicting surgical outcomes in patients with primary non-syndromic craniosynostosis.

Journal of oral biology and craniofacial research
2025

The cranial base and midface characteristics in apert and Crouzon syndrome: A 3-dimensional analysis of morphological variations.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2025

Discussion: Long-Term Orbito-Ocular Outcomes following Le Fort III and Monobloc Distraction Osteogenesis in Patients with Syndromic Craniosynostosis.

Plastic and reconstructive surgery
2025

Moving beyond surgical excellence: a qualitative systematic review into the perspectives and experiences of children, adolescents, and adults living with a rare congenital craniofacial condition and their parents.

Journal of plastic surgery and hand surgery
2025

Perioperative Airway Management for Midface Surgery in Children With Syndromic Craniosynostosis; a Single Center Experience With Immediate Extubation.

Paediatric anaesthesia
2025

Prevalence and treatment outcomes of hydrocephalus among children with craniofacial syndromes.

Journal of plastic surgery and hand surgery
2025

Orthognathic surgery in syndromic craniosynostosis: Incorporating midface morphometric analysis and dental parameters in surgical planning.

Current problems in surgery
2025

Impact of Lefort III/ monobloc advancement on midface growth in children with syndromic craniosynostosis: A systematic review.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2025

Factors influencing blood loss in fronto-orbital advancement and remodeling: A retrospective review of 231 cases.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2025

Prevalence and Clinical Associations of Peripapillary Hyperreflective Ovoid Mass-like Structures in Craniosynostosis.

Journal of neuro-ophthalmology : the official journal of the North American Neuro-Ophthalmology Society
2025

A Preliminary Study of Hearing Loss in Children With Craniosynostosis.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2025

Utility of Papilledema for Detection of Elevated Intracranial Pressure in Craniosynostosis.

The Journal of craniofacial surgery
2025

Success of Bone Ossification After Posterior Vault Distraction Osteogenesis in Patients With Craniosynostosis.

The Journal of craniofacial surgery
2025

Simultaneous Lefort 2 Distraction and Fronto-Orbito-Malar Advancement: Correcting Severe Upper and Midface Retrusion in a Patient With Crouzon Syndrome.

The Journal of craniofacial surgery
2026

Prenatal Diagnosis of Nonsyndromic Craniosynostosis: A Scoping Review.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2025

Intraoperative Skull Fracture During Halo Application in Subcranial Le Fort III: Strategies for Managing a Rare Complication.

The Journal of craniofacial surgery
2024

Cerebral Cortical Changes in Craniosynostosis: A Systematic Review.

Cureus
2025

Temporal bone thickness analysis in craniofacial anomalies: key considerations for bone conduction hearing implants.

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
2025

Discussion: Thirty-Year Experience Treating Syndromic Craniosynostosis: Long-Term Outcomes following Cranial Expansions.

Plastic and reconstructive surgery
2024

Open versus endoscopic surgery with helmet molding therapy in non-syndromic patients with craniosynostosis: an updated systematic review and meta-analysis of clinical outcomes and treatment-related costs.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2025

Factors Influencing Fronto-Orbital Relapse in Patients With Syndromic Craniosynostosis: A 2 Decade Review.

The Journal of craniofacial surgery
2025

Risk of Reading Difficulties in School-age Children Treated for Non-syndromic Craniosynostosis.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2025

Craniosynostosis in Africa: Insights from 8 Countries-A Systematic Review and Meta-Analysis.

World neurosurgery
2025

Social Perceptions of Preoperative and Postoperative Photographs of Patients With Syndromic Craniosynostosis Undergoing Le Fort III Advancement.

The Journal of craniofacial surgery
2024

Management of Midface Deficiency in Syndromic Craniosynostosis with Lefort III Distraction Osteogenesis, Outcomes, and Pitfalls.

Journal of maxillofacial and oral surgery
2024

Bilevel positive airway pressure treatment of sleep apnea syndrome in a patient with Crouzon syndrome.

Sleep &amp; breathing = Schlaf &amp; Atmung
2024

Comparison of syndromic and non-syndromic craniosynostosis cases followed in a tertiary pediatric intensive care unit: a case control study.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2025

Free-floating bone flap posterior cranial vault release in syndromic craniosynostosis.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2024

Discussion: A European Multicenter Outcome Study of Perioperative Airway Management Policies following Midface Surgery in Syndromic Craniosynostosis.

Plastic and reconstructive surgery
2026

Information Needs of Australian Families Living with Craniosynostosis: A Qualitative Study.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2025

Influence of Bone Defects After Endoscope-Assisted Suturectomy on Monobloc Advancement in Syndromic Bilateral Coronal Craniosynostosis Patients.

The Journal of craniofacial surgery
2025

A study of the cerebral venous drainage patterns in craniosynostosis: nonsyndromic cases and the induction effect of Virchow's law on venous sinuses.

Journal of neurosurgery. Pediatrics
2025

Software-assisted bone thickness evaluation in patients with syndromic craniosynostosis undergoing Le Fort III osteotomy: a technical note.

International journal of oral and maxillofacial surgery
2025

Confronting Craniosynostosis: Maternal Concerns, Challenges, and Coping for their Children.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2024

A cephalometric study on Le Fort Ⅲ osteotomy related anatomical features of anterior cranial base in syndromic craniosynostosis.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2024

Skeletal and Soft Tissue Surgeries in the Long-term Management of Patients With Syndromic Craniosynostosis: A 20-Year Review.

Annals of plastic surgery
2024

Reducing the risk of unfavourable fractures in Le Fort III osteotomy via a navigation-guided technique.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2025

A 3-Dimensional Morphometric Analysis of 4 Midsagittal Planes for CT Scan Reference Determination in Children with Syndromic Craniosynostosis.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2024

Ocular manifestations of craniosynostosis in Saudi Arabia.

Journal francais d'ophtalmologie
2024

Clinical analysis of Le Fort III distraction for obstructive sleep apnea in pediatric patients with syndromic craniosynostosis.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2025

Tracheal Ultrasound for Diagnosis of Tracheal Cartilaginous Sleeve in Patients with Syndromic Craniosynostosis.

Otolaryngology--head and neck surgery : official journal of American Academy of Otolaryngology-Head and Neck Surgery
2025

Using a Disentangled Neural Network to Objectively Assess the Outcomes of Midfacial Surgery in Syndromic Craniosynostosis.

Plastic and reconstructive surgery
2024

Discussion: Comparative Study of Internal Device versus External Device in Le Fort III Distraction for Syndromic Craniosynostosis.

Plastic and reconstructive surgery
2024

AI-based diagnosis and phenotype - Genotype correlations in syndromic craniosynostoses.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2024

New CRISPR/Cas9-based Fgfr2C361Y/+ mouse model of Crouzon syndrome exhibits skull and behavioral abnormalities.

Journal of molecular medicine (Berlin, Germany)
2024

Cranial bone microarchitecture in a mouse model for syndromic craniosynostosis.

Journal of anatomy
2024

The role of pathogenic TCF12 variants in children with coronal craniosynostosis-a systematic review with addition of two novel cases.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2024

Genetic Heterogeneity, Craniofacial Surgical Burden, and Surgical Techniques in Patients With Saethre-Chotzen Syndrome.

The Journal of craniofacial surgery
2024

Exploring Different Management Modalities of Nonsyndromic Craniosynostosis.

Cureus
2024

Early posterior vault distraction osteogenesis changes the syndromic craniosynostosis treatment paradigm: long-term outcomes of a 23-year cohort study.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2024

A rare case report on imaging on severe form of syndromic craniosynostosis-oxycephaly.

QJM : monthly journal of the Association of Physicians
2024

Loss-of-function variants in ERF are associated with a Noonan syndrome-like phenotype with or without craniosynostosis.

European journal of human genetics : EJHG
2024

Nonsyndromic Craniosynostosis Correlation Between Ethnicity, Race, and Pattern of Affected Suture Type: Meta-Analysis.

The Journal of craniofacial surgery
2024

Attention Deficit/Hyperactivity Disorder in Individuals with Non-Syndromic Craniosynostosis: A Systematic Review and Meta-Analysis.

Developmental neuropsychology
2024

Parental satisfaction with hospital care for children with non-syndromic craniosynostosis: A mixed-method study.

Journal of pediatric nursing
2024

Noonan syndrome-like phenotype associated with an ERF frameshift variant.

American journal of medical genetics. Part A
2024

Three-dimensional quantification of soft tissue changes and its relationship to skeletal changes after Le Fort III, monobloc, and facial bipartition in syndromic craniosynostosis.

International journal of oral and maxillofacial surgery
2024

Apert syndrome: neurosurgical outcomes and complications following posterior vault distraction osteogenesis.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2024

Syndromic craniosynostosis caused by a novel missense variant in MAP4K4: Expanding the genotype-phenotype relationship in RASopathies.

Clinical genetics
2024

Skeletal changes after midface surgery in patients with craniofacial deformities: a three-dimensional quantification method.

International journal of oral and maxillofacial surgery
2025

Thirty-Year Experience Treating Syndromic Craniosynostosis: Long-Term Outcomes following Cranial Expansions.

Plastic and reconstructive surgery
2024

The effect of Le Fort III procedure in the treatment of obstructive sleep apnea in children with syndromic craniosynostosis.

Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine
2025

Health-Related Quality of Life in Mexican Children and Adolescents with Non-Syndromic Craniosynostosis.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2024

In Vitro Assessment of the Neuro-Compatibility of Fe-20Mn as a Potential Bioresorbable Material for Craniofacial Surgery.

Medicina (Kaunas, Lithuania)
2024

Skeletal expansion via craniofacial distraction osteogenesis technique in syndromic craniosynostosis: impact on ophthalmic parameters.

International ophthalmology
2024

Neurocognitive outcomes and associated clinical factors 5 years after surgery in children with craniosynostosis.

Journal of neurosurgery. Pediatrics
2024

Severe obstructive sleep apnea in children with syndromic craniosynostosis: analysis of pulse transit time.

Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine
2024

Development of Calvarial Bone Osteomyelitis in a Noonan Syndrome Patient Following Fronto-Orbital Advancement of Craniosynostosis: A Novel Technique of Treatment Using Povidone and Hydrogen Peroxide Solutions.

Cureus
2024

Prevalence of craniosynostosis in Finland, 1987-2010: A population-based study.

Birth defects research
2025

Long-Term Orbito-Ocular Outcomes following Le Fort III and Monobloc Distraction Osteogenesis in Patients with Syndromic Craniosynostosis.

Plastic and reconstructive surgery
2024

Changes in mandibular position during midface distraction in patients with syndromic craniosynostosis.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2024

Evaluation of neurocognitive and social developments after craniosynostosis surgery.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2024

A European Multicenter Outcome Study of Perioperative Airway Management Policies following Midface Surgery in Syndromic Craniosynostosis.

Plastic and reconstructive surgery
2024

Endoscopy-Assisted Craniosynostosis Surgery Versus Cranial Vault Remodeling for Non-Syndromic Craniosynostosis: Experience of a Single Center.

Turkish neurosurgery
2024

Delayed Postnatal Synostosis without Spheno-occipital Synchondrosis Fusion: A Curious Case of Apert Syndrome.

Plastic and reconstructive surgery. Global open
2023

Syndromic Craniosynostosis: A Comprehensive Review.

Cureus
2024

The Role of Virtual Surgical Planning in Surgery for Complex Craniosynostosis.

Plastic and reconstructive surgery. Global open
2024

The effect of continuous positive airway pressure on obstructive sleep apnea in children with syndromic craniosynostosis.

Sleep &amp; breathing = Schlaf &amp; Atmung
2024

Strabismus and refraction in non-syndromic craniosynostosis - A longitudinal study up to 5 years of age.

Acta ophthalmologica
2023

Intracranial Pressure, Autoregulation, and Cerebral Perfusion in Infants With Nonsyndromic Craniosynostosis at the Time of Surgical Correction.

Neurosurgery
2023

Spring-assisted posterior vault expansion: a parametric study to improve the intracranial volume increase prediction.

Scientific reports
2023

Perioperative Management of Obstructive Sleep Apnea in Patients With Syndromic Craniosynostosis Undergoing LeFort III Osteotomy With Distraction: A Case Series.

Craniomaxillofacial trauma &amp; reconstruction
2024

Social Media and Website Use: The Experiences of Parents and Carers Accessing Care at the Oxford Craniofacial Unit.

The Journal of craniofacial surgery
2024

Syndromic Craniofacial Disorders.

Facial plastic surgery clinics of North America
2024

The Outcomes of Endoscopic Suturectomy in Syndromic Craniosynostosis.

The Journal of craniofacial surgery
2024

Posterior cranial vault distraction in children with syndromic craniosynostosis: the era of biodegradable materials-a comprehensive review of the literature and proposed novel global application.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2024

Incidence of Ventriculomegaly in Patients With Craniosynostosis.

The Journal of craniofacial surgery
2023

Morphometric analysis and outcomes following posterior cranial vault distraction in syndromic and multisuture craniosynostosis.

Journal of plastic, reconstructive &amp; aesthetic surgery : JPRAS
2024

LeFort III Versus Monobloc Frontofacial Advancement: A Comparative Analysis of Soft Tissue Changes.

The Journal of craniofacial surgery
2023

Plea for systematic prenatal genes panel testing when facing isolated craniosynostosis on fetal imaging.

European journal of obstetrics, gynecology, and reproductive biology
2024

Incidence of Airway Abnormalities in Children With Craniosynostosis.

The Journal of craniofacial surgery
2023

Artificial Intelligence Applications in Diagnosing and Managing Non-syndromic Craniosynostosis: A Comprehensive Review.

Cureus
2024

Sensitivity, Specificity, and Cutoff Identifying Optic Atrophy by Macular Ganglion Cell Layer Volume in Syndromic Craniosynostosis.

Ophthalmology
2024

Midface hypoplasia in syndromic craniosynostosis: predicting craniofacial growth via a novel regression model from anatomical morphometric analysis.

International journal of oral and maxillofacial surgery
2023

Genetic diagnostic yield in an 11-year cohort of craniosynostosis patients.

European journal of medical genetics
2023

Can Craniosynostosis be Diagnosed on Physical Examination? A Retrospective Review.

The Journal of craniofacial surgery
2024

Comparative Study of Internal Device versus External Device in Le Fort III Distraction for Syndromic Craniosynostosis.

Plastic and reconstructive surgery
2023

What We Know About Intracranial Hypertension in Children With Syndromic Craniosynostosis.

The Journal of craniofacial surgery
2023

Minor Suture Fusion is Associated With Chiari Malformation in Nonsyndromic Craniosynostosis.

The Journal of craniofacial surgery
2024

Frequency and predictors of concurrent complications in multi-suture release for syndromic craniosynostosis.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2023

[Application and development of orthognathic surgery in treatment of syndromic craniosynostosis].

Zhongguo xiu fu chong jian wai ke za zhi = Zhongguo xiufu chongjian waike zazhi = Chinese journal of reparative and reconstructive surgery
2024

A Rare Case of Sagittal Sinus Obstruction Following Posterior Cranial Vault Distraction Osteogenesis.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2023

CLINICOROENTGENOLOGICAL PECULIARITIES OF THE CONGENITAL AND ACQUIRED CRANIOFACIAL ANOMALIES.

Georgian medical news
2024

Are Patients with Syndromic Craniosynostosis at Greater Risk for Epilepsy than Patients with Nonsyndromic Craniosynostosis?

World neurosurgery
2023

FGF signaling in cranial suture development and related diseases.

Frontiers in cell and developmental biology
2023

Apert Syndrome: Selection Rationale for Midface Advancement Technique.

Advances and technical standards in neurosurgery
2023

Patient Tailored Surgery in Saethre-Chotzen Syndrome: Analysis of Reoperation for Intracranial Hypertension.

The Journal of craniofacial surgery
2023

Facial skeleton dysmorphology in syndromic craniosynostosis: differences between FGFR2 and no-FGFR2-related syndromes and relationship with skull base and facial sutural patterns.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2023

Familial monoallelic CYP26B1 truncating variant causes a syndromic craniosynostosis due to haploinsufficiency ?

European journal of medical genetics
2023

De novo variants implicate chromatin modification, transcriptional regulation, and retinoic acid signaling in syndromic craniosynostosis.

American journal of human genetics
2024

Systematic Review of Nonsyndromic Craniosynostosis: Genomic Alterations and Impacted Signaling Pathways.

Plastic and reconstructive surgery
2024

Posterior Cranial Distraction in Craniosynostosis: A Systematic Review of the Literature.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2023

Utilization of carbonated calcium phosphate cement for contouring cranioplasty in patients with syndromic craniosynostosis.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2023

The Need for Additional Surgery after Passive versus Active Approaches to Syndromic Craniosynostosis: A Meta-analysis.

Plastic and reconstructive surgery. Global open
2023

Useful Genioplasty for Repeated Recurrent Sleep Apnea of Congenital Anomalies and Its Evaluation.

Plastic and reconstructive surgery. Global open
2022

Comprehensive Genetic Evaluation of Bulgarian Children with Syndromic Craniosynostosis.

Balkan journal of medical genetics : BJMG
2023

Long-term neurocognitive outcomes in 204 single-suture craniosynostosis patients.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2023

Comparison of Internal and External Distraction in Frontofacial Monobloc Advancement: A Three-Dimensional Quantification.

Plastic and reconstructive surgery
2024

Cognitive performance in preschoolers with non-syndromic craniosynostosis undergoing surgery: A comparison with typically developing children.

Applied neuropsychology. Child
2023

Prior fronto-orbital advancement associated with complications from transcranial midface surgery in patients with syndromic craniosynostosis.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2023

Outcomes after Endoscope-Assisted Strip Craniectomy and Orthotic Therapy for Syndromic Craniosynostosis.

Plastic and reconstructive surgery
2023

Practical Algorithm for the Management of Multisutural Craniosynostosis with Associated Chiari Malformation and/or Hydrocephalus.

Pediatric neurosurgery
2023

Three-Dimensional Evaluation of Dental Arches in Individuals with Syndromic Craniosynostosis.

International journal of dentistry
2023

Molecular Mechanisms Involved in Craniosynostosis.

In vivo (Athens, Greece)
2023

Sex Differences in Comorbidities of Pediatric Craniosynostosis at Presentation.

Pediatric neurosurgery
2022

Retroperitoneal liposarcoma and craniosynostosis: possible genomic relationship, case report, and literature review.

Functional &amp; integrative genomics
2023

Health-related quality of life of children treated for non-syndromic craniosynostosis.

Journal of plastic surgery and hand surgery
2023

Synchondrosis fusion contributes to the progression of postnatal craniofacial dysmorphology in syndromic craniosynostosis.

Journal of anatomy
2023

Spring forces and calvarial thickness predict cephalic index changes following spring-mediated cranioplasty for sagittal craniosynostosis.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2022

Evaluation and Management of Nonsyndromic Craniosynostosis.

Journal of pediatric neurosciences
2022

Management of Chiari 1 Malformation and Hydrocephalus in Syndromic Craniosynostosis: A Review.

Journal of pediatric neurosciences
2022

Craniosynostosis: A Pediatric Neurologist's Perspective.

Journal of pediatric neurosciences
2022

FGFR2 Mutation p.Cys342Arg Enhances Mitochondrial Metabolism-Mediated Osteogenesis via FGF/FGFR-AMPK-Erk1/2 Axis in Crouzon Syndrome.

Cells
2023

ERF-related craniosynostosis and surgical management in the paediatric cohort.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2022

An Elderly Patient With Crouzon Syndrome Treated With Monobloc Distraction.

The Journal of craniofacial surgery
2022

Comparing the Increased Intracranial Volume From Different Surgical Methods for Syndromic Craniosynostosis.

The Journal of craniofacial surgery
2022

ERN CRANIO patient coverage of craniosynostosis in Europe.

Orphanet journal of rare diseases
2023

De novo mutations in the BMP signaling pathway in lambdoid craniosynostosis.

Human genetics
2022

TWIST1, a gene associated with Saethre-Chotzen syndrome, regulates extraocular muscle organization in mouse.

Developmental biology
2022

Factors Affecting Optic Nerve Damage in Le Fort III Osteotomy: A Retrospective Study.

The Journal of craniofacial surgery
2022

Changes in venous drainage after posterior cranial vault distraction and foramen magnum decompression in syndromic craniosynostosis.

Journal of neurosurgery. Pediatrics
2022

Accuracy of Detecting Obstructive Sleep Apnea Using Ambulatory Sleep Studies in Patients With Syndromic Craniosynostosis.

The Journal of craniofacial surgery
2022

Orthognathic Surgery in Patients with Syndromic Craniosynostosis.

Oral and maxillofacial surgery clinics of North America
2022

Syndromic Craniosynostosis: Cranial Vault Expansion in Infancy.

Oral and maxillofacial surgery clinics of North America
Ver todos os 377 no EuropePMC

Associações

Organizações que acompanham esta doença — pra ter apoio e orientação

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Comunidades

Grupos ativos de quem convive com esta doença aqui no Raras

Ainda não existe comunidade no Raras para Craniossinostose sindrômica

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Doenças relacionadas

Doenças com sintomas parecidos — ajudam quem ainda está buscando diagnóstico

Ordenadas pelo número de sintomas em comum.

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. Daily executive function in children and adolescents with non-syndromic craniosynostosis: association with timing of surgical intervention.
    Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery· 2026· PMID 41820702mais citado
  2. En-bloc posterior vault distraction including the foramen magnum for Chiari I malformation in severe syndromic craniosynostosis: First cases and literature review.
    Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery· 2026· PMID 41812278mais citado
  3. Ophthalmic Pathologies in Craniosynostosis: Risk Factors and Disparities in the United States.
    The Journal of craniofacial surgery· 2026· PMID 41773839mais citado
  4. Morphological analysis of posterior fossa in Apert and Crouzon syndromes before and after posterior cranial vault expansion.
    Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery· 2026· PMID 41747663mais citado
  5. Feasibility and reproducibility of handheld and table-mounted optical coherence tomography in children with craniosynostosis.
    Eye (London, England)· 2026· PMID 41735680mais citado
  6. Septorhinoplasty in Congenital Craniofacial Anomalies: A Narrative Review of Contemporary Surgical Approaches.
    Facial Plast Surg· 2026· PMID 41941884recente
  7. Surgical Outcomes of Cranial Vault Expansion Following Treatment of Craniosynostosis in Complex Syndromes: A Retrospective-Prospective Patient Series.
    J Craniofac Surg· 2026· PMID 41931363recente
  8. Predictors of Epilepsy in Syndromic Craniosynostosis.
    J Craniofac Surg· 2026· PMID 41911563recente
  9. Digital Planning and Patient-Specific Implants Facilitate Accurate Conventional Sub-Cranial Le Fort III Advancement.
    J Oral Maxillofac Surg· 2026· PMID 41887612recente

Bases de dados e fontes oficiais

Identificadores e referências canônicas usadas para montar este verbete.

  1. ORPHA:139393(Orphanet)
  2. MONDO:0015338(MONDO)
  3. GARD:19911(GARD (NIH))
  4. Variantes catalogadas(ClinVar)
  5. Busca completa no PubMed(PubMed)
  6. Q55785407(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

Craniossinostose sindrômica
Compêndio · Raras BR

Craniossinostose sindrômica

ORPHA:139393 · MONDO:0015338
Medicamentos
4 registrados
MedGen
UMLS
C5680624
EuropePMC
Wikidata
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