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Síndrome Saethre-Chotzen
ORPHA:794CID-10 · Q87.0CID-11 · LD24.GYOMIM 101400DOENÇA RARA

A síndrome de Saethre-Chotzen (SCS) é uma síndrome de craniossinostose hereditária caracterizada por sinostose coronal unilateral ou bilateral, assimetria facial, ptose, estrabismo e orelhas pequenas com crus proeminentes, entre outras manifestações menos comuns.

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Introdução

O que você precisa saber de cara

📋

A síndrome de Saethre-Chotzen (SCS) é uma síndrome de craniossinostose hereditária caracterizada por sinostose coronal unilateral ou bilateral, assimetria facial, ptose, estrabismo e orelhas pequenas com crus proeminentes, entre outras manifestações menos comuns.

Pesquisas ativas
2 ensaios
39 total registrados no ClinicalTrials.gov
Publicações científicas
276 artigos
Último publicado: 2026 Mar 25

Escala de raridade

CLASSIFICAÇÃO ORPHANET · BRASIL 2024
1-9 / 100 000
Ultra-rara
<1/50k
Muito rara
1/20k
Rara
1/10k
Pouco freq.
1/5k
Incomum
1/2k
Prevalência
0.0
Europe
Início
Antenatal
+ neonatal
🏥
SUS: Cobertura mínimaScore: 15%
CID-10: Q87.0
🇧🇷Dados SUS / DATASUS
PROCEDIMENTOS SIGTAP (5)
0202010503
Cariótipo — bandas G, Q ou Rgenetic_test
0202010600
Pesquisa de microdeleções/microduplicações por FISHlab_test
0202010694
Sequenciamento completo do exoma (WES)rehabilitation
0202010260
Dosagem de alfa-fetoproteína
0301070040
Atendimento em reabilitação — doenças raras
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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
19 sintomas
😀
Face
16 sintomas
👂
Ouvidos
5 sintomas
👁️
Olhos
4 sintomas
🧠
Neurológico
3 sintomas
🫘
Rins
2 sintomas

+ 29 sintomas em outras categorias

Características mais comuns

90%prev.
Craniossinostose coronal
Muito frequente (99-80%)
90%prev.
Craniossinostose
Muito frequente (99-80%)
90%prev.
Assimetria facial
Muito frequente (99-80%)
90%prev.
Morfologia anormal do crânio
Muito frequente (99-80%)
90%prev.
Testa alta
Muito frequente (99-80%)
90%prev.
Sindactilia dos dedos
Muito frequente (99-80%)
83sintomas
Muito frequente (7)
Frequente (22)
Ocasional (33)
Sem dados (21)

Os sintomas variam de pessoa para pessoa. Abaixo estão as 83 características clínicas mais associadas, ordenadas por frequência.

Craniossinostose coronalCoronal craniosynostosis
Muito frequente (99-80%)90%
CraniossinostoseCraniosynostosis
Muito frequente (99-80%)90%
Assimetria facialFacial asymmetry
Muito frequente (99-80%)90%
Morfologia anormal do crânioAbnormal skull morphology
Muito frequente (99-80%)90%
Testa altaHigh forehead
Muito frequente (99-80%)90%

Linha do tempo da pesquisa

Publicações por ano — veja quando o interesse científico cresceu
Anos de pesquisa1desde 2026
Total histórico276PubMed
Últimos 10 anos83publicações
Pico201815 papers
Linha do tempo
2026Hoje · 2026🧪 2001Primeiro ensaio clínico📈 2018Ano 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

3 genes identificados com associação a esta condição. Padrão de herança: Autosomal dominant.

TWIST1Twist-related protein 1Disease-causing germline mutation(s) (loss of function) 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
FGFR2Fibroblast growth factor receptor 2Candidate gene tested 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 3Candidate gene tested 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

Variantes genéticas (ClinVar)

914 variantes patogênicas registradas no ClinVar.

🧬 TWIST1: NM_000474.4(TWIST1):c.430A>G (p.Ser144Gly) ()
🧬 TWIST1: NM_000474.4(TWIST1):c.454G>A (p.Ala152Thr) ()
🧬 TWIST1: NM_000474.4(TWIST1):c.364C>T (p.Gln122Ter) ()
🧬 TWIST1: NM_000474.4(TWIST1):c.480C>G (p.Tyr160Ter) ()
🧬 TWIST1: GRCh37/hg19 7p22.3-14.3(chr7:158725-29918785)x3 ()
Ver todas no ClinVar

Classificação de variantes (ClinVar)

Distribuição de 367 variantes classificadas pelo ClinVar.

147
202
18
Patogênica (40.1%)
VUS (55.0%)
Benigna (4.9%)
VARIANTES MAIS SIGNIFICATIVAS
TWIST1: NM_000474.4(TWIST1):c.364C>T (p.Gln122Ter) [Pathogenic]
LOC129998021: NM_000474.4(TWIST1):c.480C>G (p.Tyr160Ter) [Pathogenic]
TWIST1: NM_000474.4(TWIST1):c.364del (p.Gln122fs) [Likely pathogenic]
LOC129998021: NM_000474.4(TWIST1):c.480C>A (p.Tyr160Ter) [Pathogenic]
TWIST1: NM_000474.4(TWIST1):c.124del (p.Ser42fs) [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-clínico2
Medicamentos catalogadosEnsaios clínicos· 0 medicamentos · 2 ensaios
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Onde tratar no SUS

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

🇧🇷 Atendimento SUS — Síndrome Saethre-Chotzen

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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

🟢 Recrutando agora

1 pesquisa recrutando participantes. Converse com seu médico sobre a possibilidade de participar.

Outros ensaios clínicos

39 ensaios clínicos encontrados, 2 ativos.

Distribuição por fase
Ver todos no ClinicalTrials.gov
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Publicações mais relevantes

Timeline de publicações
86 papers (10 anos)
#1

Semi-automated three-dimensional analysis of maxillary anomalies in patients with Muenke syndrome, Saethre-Chotzen syndrome or TCF12-related craniosynostosis: A retrospective study.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery2025 Aug

In craniosynostosis distinctive craniofacial and oral growth patterns are to be expected. This study aims to determine maxillary anomalies in craniosynostosis patients with Muenke syndrome, Saethre-Chotzen syndrome or TCF12-related craniosynostosis, using a three-dimensional semi-automated setup measurement of digital dental casts. Symmetry analysis of the maxilla of craniosynostosis patients was performed with creation of a reference frame, a palatal mesh and a distance map. The outline of the palate was determined by landmarks with the use of a semi-automated technique and software algorithm and compared to healthy controls. All transverse dimensions were smaller in the craniosynostosis group compared to the. control group (p < 0.001; canine premolar right TCF12 p = 0.005). In Muenke syndrome, the palate was higher compared to the control group (left and right; p < 0.001). In Saethre-Chotzen syndrome, the palate was shallower (left p < 0.001; right p = 0.003) and the left palatal surface was smaller compared to the control group (p < 0.001). This retrospective case-control study indicates that Muenke syndrome, Saethre-Chotzen syndrome and TCF12-related craniosynostosis have distinctive maxillary characteristics compared to healthy controls. Muenke syndrome had a higher arched palate. and Saethre-Chotzen had a shallower palate compared to healthy controls. The intersurface distance (ISD) in both syndromes indicated a palatal anomaly.

#2

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

Journal of plastic surgery and hand surgery2025 Feb 20

Hydrocephalus is more common in patients with craniofacial syndromes (CS) relative to non-syndromic craniosynostosis, and the optimal management is challenging. This study examined the prevalence and treatment outcomes of hydrocephalus among children with CS. We performed a retrospective review of medical records for all children with established CS and registered in the Gothenburg Craniofacial Registry between 1975 and 2022. This review included analyses of data regarding patient demographics, radiological imaging, hydrocephalus treatment modalities, and shunt revisions. Eligible patients (n = 193) included those with CS, including Pfeiffer (n = 13), Crouzon (n = 57), Apert (n = 49), Muenke (n = 25), and Saethre-Chotzen (n = 49) syndromes. A total of 22 patients (11.4%) presented hydrocephalus requiring treatment [Pfeiffer, n = 8 (61.5%); Crouzon, n = 13 (22.8%); and Apert, n = 1 (2.0%)]. Nineteen (9.8%) patients underwent ventricular shunt insertion, and three (1.6%) underwent endoscopic third ventriculostomy as a first procedure. None of the Muenke or Saethre-Chotzen patients required hydrocephalus treatment. Seventeen (85%) patients with shunts required revision mainly due to shunt obstruction. Pfeiffer patients had the highest risk of both developing hydrocephalus requiring treatment and needing shunt revision (p < 0.001 and p = 0.004, respectively). Approximately 40% of patients with Pfeiffer, Crouzon, or Apert presented ventriculomegaly not requiring treatment. Hydrocephalus requiring treatment is common in Pfeiffer and Crouzon patients but rare in Apert, Muenke, or Saethre-Chotzen syndrome. Shunt treatment is often associated with complications that require revisions, emphasizing the importance of distinguishing non-progressive ventriculomegaly from hydrocephalus requiring treatment.

#3

Prenatal diagnosis of chromothripsis causing complex chromosomal rearrangement involving chromosomes 5, 7 and 11 leading to TWIST1 deletion and Saethre-Chotzen syndrome.

Taiwanese journal of obstetrics &amp; gynecology2025 Jan

Prenatal detection of complex chromosomal rearrangements (CCR) is extremely rare, but is of great clinical importance, since CCR can be causative of different congenital disorders. We present an exceptionally rare case of prenatally diagnosed Saethre-Chotzen syndrome (SCS) rising as a consequence of chromothripsis involving chromosomes 5, 7 and 11 and deletion of TWIST1 gene. Brachycephaly, hypertelorism, flat face, micrognathia, relative macroglossia and small posterior fossa were noted on ultrasound examination at 28th gestational week. Fetal karyotyping revealed de novo translocation 46,XY,t(7;11)(p15.5;q21)dn. Chromosomal microarray showed presence of three microdeletions on chromosome 7 (7p21.1p15.3 including TWIST1, 7p12.1p11.2 and 7q21.11), and one on chromosome 5p12p11. Use of advanced molecular diagnostic techniques in combination with cytogenetic methods allows for precise characterization of CCRs and detection of molecular mechanisms of their origin. Phenomenon of chromothripsis can be causative of rare genetic syndromes such as SCS.

#4

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 medicine2024

Saethre-Chotzen syndrome (SCS) is one of the most prevalent craniosynostosis, caused by a loss-of-function mutation in the TWIST-1 gene, with current treatment options relying on major invasive transcranial surgery. TWIST-1 haploinsufficient osteogenic progenitor cells exhibit increased osteogenic differentiation potential due to an upregulation of the transmembrane tyrosine kinase receptor, C-ROS-1, a TWIST-1 target gene known to promote bone formation. The present study assessed the efficacy of suppressing C-ROS-1 activity using a known chemical inhibitor to C-ROS-1, crizotinib, to halt premature coronal suture fusion in a preclinical mouse model of SCS. Crizotinib (1 μM, 2 μM, or 4 μM) was administered locally over the calvaria of Twist-1del/+ heterozygous mice prior to coronal suture fusion using either a nonresorbable collagen sponge (quick drug release) or a resorbable sodium carboxymethylcellulose microdisk (slow sustained release). Coronal suture fusion rates and bone parameters were determined by μCT imaging and histomorphometric analysis of calvaria postcoronal suture fusion. Results demonstrated a dose-dependent increase in the efficacy of crizotinib to maintain coronal suture patency, with no adverse effects to brain, kidney, liver, and spleen tissue, or blood cell parameters. Moreover, crizotinib delivered on microdisks resulted in a greater efficacy at a lower concentration to reduce bone formation at the coronal suture sites compared to sponges. However, the bone inhibitory effects were found to be diminished by over time following cessation of treatment. Our findings lay the foundation for the development of a pharmacological nonsurgical, targeted approach to temporarily maintain open coronal sutures in SCS patients. This study could potentially be used to develop similar therapeutic strategies to treat different syndromic craniosynostosis conditions caused by known genetic mutations.

#5

Prediction and design of transcriptional repressor domains with large-scale mutational scans and deep learning.

bioRxiv : the preprint server for biology2024 Sep 24

Regulatory proteins have evolved diverse repressor domains (RDs) to enable precise context-specific repression of transcription. However, our understanding of how sequence variation impacts the functional activity of RDs is limited. To address this gap, we generated a high-throughput mutational scanning dataset measuring the repressor activity of 115,000 variant sequences spanning more than 50 RDs in human cells. We identified thousands of clinical variants with loss or gain of repressor function, including TWIST1 HLH variants associated with Saethre-Chotzen syndrome and MECP2 domain variants associated with Rett syndrome. We also leveraged these data to annotate short linear interacting motifs (SLiMs) that are critical for repression in disordered RDs. Then, we designed a deep learning model called TENet ( T ranscriptional E ffector Net work) that integrates sequence, structure and biochemical representations of sequence variants to accurately predict repressor activity. We systematically tested generalization within and across domains with varying homology using the mutational scanning dataset. Finally, we employed TENet within a directed evolution sequence editing framework to tune the activity of both structured and disordered RDs and experimentally test thousands of designs. Our work highlights critical considerations for future dataset design and model training strategies to improve functional variant prioritization and precision design of synthetic regulatory proteins.

Publicações recentes

Ver todas no PubMed

📚 EuropePMC135 artigos no totalmostrando 82

2025

Semi-automated three-dimensional analysis of maxillary anomalies in patients with Muenke syndrome, Saethre-Chotzen syndrome or TCF12-related craniosynostosis: A retrospective study.

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

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

Journal of plastic surgery and hand surgery
2025

Prenatal diagnosis of chromothripsis causing complex chromosomal rearrangement involving chromosomes 5, 7 and 11 leading to TWIST1 deletion and Saethre-Chotzen syndrome.

Taiwanese journal of obstetrics &amp; gynecology
2024

Prediction and design of transcriptional repressor domains with large-scale mutational scans and deep learning.

bioRxiv : the preprint server for biology
2024

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

The Journal of craniofacial surgery
2023

Syndromic Craniosynostosis: A Comprehensive Review.

Cureus
2024

Automated three-dimensional analysis of facial asymmetry in patients with syndromic coronal synostosis: A retrospective study.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2023

Skull progenitor cell-driven meningeal lymphatic restoration improves neurocognitive functions in craniosynostosis.

Cell stem cell
2024

TWIST1 Gene Variants Cause Craniosynostosis with Limb Abnormalities in Asian Patients.

Journal of pediatric genetics
2023

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

The Journal of craniofacial surgery
2023

A Twist-Box domain of the C. elegans Twist homolog, HLH-8, plays a complex role in transcriptional regulation.

Genetics
2022

Case report: A third variant in the 5' UTR of TWIST1 creates a novel upstream translation initiation site in a child with Saethre-Chotzen syndrome.

Frontiers in genetics
2023

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

Journal of anatomy
2022

The impact of obstructive sleep apnea on growth in patients with syndromic and complex craniosynostosis: a retrospective study.

European journal of pediatrics
2023

Auricles Anomalies in Patients With a TCF12 Gene Mutation.

The Journal of craniofacial surgery
2022

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

Developmental biology
2022

Case Report: Posterolateral Epidural Supra-C2-Root Approach (PESCA) for Biopsy of a Retro-Odontoid Lesions in Same Sitting After Occipitocervical Fixation and Decompression in a Case of Crowned Dens Syndrome With Brainstem Compression and Displacement.

Frontiers in surgery
2023

Postnatal Progressive Craniosynostosis in Syndromic Conditions: Two Patients With Saethre-Chotzen Due to TWIST1 Gene Deletions and Review of the Literature.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2022

Quantitative Morphologic Analysis of Cranial Vault in Twist1+/- Mice: Implications in Craniosynostosis.

Plastic and reconstructive surgery
2022

Craniofacial morphology and growth in Muenke syndrome, Saethre-Chotzen syndrome, and TCF12-related craniosynostosis.

Clinical oral investigations
2022

Embryonic requirements for Tcf12 in the development of the mouse coronal suture.

Development (Cambridge, England)
2021

Facial Dysmorphology in Saethre-Chotzen Syndrome.

The Journal of craniofacial surgery
2022

Differential diagnosis of syndromic craniosynostosis: a case series.

Archives of gynecology and obstetrics
2022

Evaluation of dental maturity in Muenke syndrome, Saethre-Chotzen syndrome, and TCF12-related craniosynostosis.

European journal of orthodontics
2021

The developing mouse coronal suture at single-cell resolution.

Nature communications
2021

Syndrome-related outcomes following posterior vault distraction osteogenesis.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2021

[Saethre-Chotzen syndrome: a case report].

Archivos argentinos de pediatria
2021

Intracranial Volume Measured and Correlated to Cephalic Index in Syndromic and Nonsyndromic Anterior Brachycephaly.

Annals of plastic surgery
2022

Invited Commentary from the authors of: Abulezz TA, et al. Saethre-Chotzen Syndrome: A Report of 7 Patients and Review of the Literature.

Annals of plastic surgery
2021

Cranial Suture Regeneration Mitigates Skull and Neurocognitive Defects in Craniosynostosis.

Cell
2021

Evolution of Cranioorbital Shape in Nonsyndromic, Muenke, and Saethre-Chotzen Bilateral Coronal Synostosis: A Case-Control Study of 2-Year Outcomes.

Plastic and reconstructive surgery
2021

Vertical transmission of a large calvarial ossification defect due to heterozygous variants of ALX4 and TWIST1.

American journal of medical genetics. Part A
2021

Adult Saethre-Chotzen Syndrome: A Unique Abnormal Breathing Pattern.

The Journal of craniofacial surgery
2020

Pharmacological targeting of KDM6A and KDM6B, as a novel therapeutic strategy for treating craniosynostosis in Saethre-Chotzen syndrome.

Stem cell research &amp; therapy
2021

Lateral and Frontal Cephalometric Measurements in a Cohort With Saethre-Chotzen Syndrome.

The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association
2020

Caput membranaceum: A novel clinical presentation of ZIC1 related skull malformation and craniosynostosis.

American journal of medical genetics. Part A
2021

Saethre-Chotzen syndrome: long-term outcome of a syndrome-specific management protocol.

Developmental medicine and child neurology
2020

50 Years Ago in TheJournalofPediatrics: Clinical and Genetic Delineation of Saethre-Chotzen Syndrome.

The Journal of pediatrics
2020

Saethre-Chotzen Syndrome: A Report of 7 Patients and Review of the Literature.

Annals of plastic surgery
2020

An ENU-induced mutation in Twist1 transactivation domain causes hindlimb polydactyly with complete penetrance and dominant-negatively impairs E2A-dependent transcription.

Scientific reports
2019

Language Development, Hearing Loss, and Intracranial Hypertension in Children With TWIST1-Confirmed Saethre-Chotzen Syndrome.

The Journal of craniofacial surgery
2019

Craniofacial abnormalities in a murine model of Saethre-Chotzen Syndrome.

Annals of anatomy = Anatomischer Anzeiger : official organ of the Anatomische Gesellschaft
2019

Nonsyndromic craniosynostosis: novel coding variants.

Pediatric research
2018

From 3D imaging to 3D printing in dentistry - a practical guide.

International journal of computerized dentistry
2018

Ablepharon and craniosynostosis in a patient with a localized TWIST1 basic domain substitution.

American journal of medical genetics. Part A
2019

Deviating dental arch morphology in mild coronal craniosynostosis syndromes.

Clinical oral investigations
2018

Altered bone growth dynamics prefigure craniosynostosis in a zebrafish model of Saethre-Chotzen syndrome.

eLife
2018

Unraveling the transcriptional regulation of TWIST1 in limb development.

PLoS genetics
2018

Saethre-Chotzen syndrome: Case report and literature review.

Dental and medical problems
2019

Prenatal sonographic findings and prognosis of craniosynostosis diagnosed during the fetal and neonatal periods.

Congenital anomalies
2018

Intracranial Volume and Head Circumference in Children with Unoperated Syndromic Craniosynostosis.

Plastic and reconstructive surgery
2018

Staged Raising of a Coronal Flap for Fronto-Orbital Advancement and Remodeling in Saethre-Chotzen Syndrome Complicated by Sinus Pericranii.

The Journal of craniofacial surgery
2018

Disruption of TWIST1 translation by 5' UTR variants in Saethre-Chotzen syndrome.

Human mutation
2018

Inhaled nitric oxide mitigates need for extracorporeal membrane oxygenation in a patient with refractory acute hypoxemic respiratory failure due to cardiac and pulmonary shunts.

Respiratory medicine case reports
2018

Recombinant mouse periostin ameliorates coronal sutures fusion in Twist1+/- mice.

Journal of translational medicine
2018

Tyrosine kinase receptor c-ros-oncogene 1 inhibition alleviates aberrant bone formation of TWIST-1 haploinsufficient calvarial cells from Saethre-Chotzen syndrome patients.

Journal of cellular physiology
2018

Craniosynostosis as a clinical and diagnostic problem: molecular pathology and genetic counseling.

Journal of applied genetics
2018

Reduced Intercarotid Artery Distance in Syndromic and Isolated Brachycephaly.

Pediatric neurology
2018

A craniosynostosis massively parallel sequencing panel study in 309 Australian and New Zealand patients: findings and recommendations.

Genetics in medicine : official journal of the American College of Medical Genetics
2017

Severe persistent pulmonary hypertension of the newborn and dysmorphic features in neonate with a deletion involving TWIST1 and PHF14: a case report.

Journal of medical case reports
2017

Destabilization of the TWIST1/E12 complex dimerization following the R154P point-mutation of TWIST1: an in silico approach.

BMC structural biology
2017

A Surgical Technique for Management of the Metopic Suture in Syndromic Craniosynostosis.

The Journal of craniofacial surgery
2017

Localized TWIST1 and TWIST2 basic domain substitutions cause four distinct human diseases that can be modeled in Caenorhabditis elegans.

Human molecular genetics
2017

Ocular Morbidity in the Correction of Orbital Hypertelorism and Dystopia: A 15-Year Experience.

Plastic and reconstructive surgery
2018

Contiguous gene deletion neighboring TWIST1 identified in a patient with Saethre-Chotzen syndrome associated with neurodevelopmental delay: Possible contribution of HDAC9.

Congenital anomalies
2017

Multiple biological functions of Twist1 in various cancers.

Oncotarget
2017

A novel TWIST1 gene mutation in a patient with Saethre-Chotzen syndrome.

Clinical dysmorphology
2016

Syndromic Craniosynostosis.

Facial plastic surgery clinics of North America
2016

[Repeated General Anesthesia for a Child with Saethre-Chotzen Syndrome].

Masui. The Japanese journal of anesthesiology
2016

Prevention and management of hearing loss in syndromic craniosynostosis: A case series.

International journal of pediatric otorhinolaryngology
2016

Genetic Syndromes Associated with Craniosynostosis.

Journal of Korean Neurosurgical Society
2015

Clinical and genetic characteristics of craniosynostosis in Hungary.

American journal of medical genetics. Part A
2015

New Pattern of Sutural Synostosis Associated With TWIST Gene Mutation and Saethre-Chotzen Syndrome: Peace Sign Synostosis.

The Journal of craniofacial surgery
2015

A twist tale of cancer metastasis and tumor angiogenesis.

Histology and histopathology
2015

Progressive postnatal pansynostosis: an insidious and pernicious form of craniosynostosis.

Journal of neurosurgery. Pediatrics
2015

The occipitofrontal circumference: reliable prediction of the intracranial volume in children with syndromic and complex craniosynostosis.

Neurosurgical focus
2015

Y-craniosynostosis by premature fusion of the metopic and coronal sutures: a new nosological entity or a variety of Saethre-Chotzen syndrome?

Birth defects research. Part A, Clinical and molecular teratology
2015

Morphology of the foramen magnum in syndromic and non-syndromic brachycephaly.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2014

Child with Saethre-Chotzen syndrome: anesthetic management and literature review.

Acta anaesthesiologica Belgica
2015

Venous hypertension in syndromic and complex craniosynostosis: the abnormal anatomy of the jugular foramen and collaterals.

Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery
2014

A case of Robinow-Sorauf syndrome (Craniosynostosis-Bifid Hallux Syndrome): The allelic variant of the Saethre-Chotzen syndrome.

Indian journal of dentistry
Ver todos os 135 no EuropePMC

Associações

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Comunidades

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Doenças relacionadas

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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. Semi-automated three-dimensional analysis of maxillary anomalies in patients with Muenke syndrome, Saethre-Chotzen syndrome or TCF12-related craniosynostosis: A retrospective study.
    Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery· 2025· PMID 40393840mais citado
  2. Prevalence and treatment outcomes of hydrocephalus among children with craniofacial syndromes.
    Journal of plastic surgery and hand surgery· 2025· PMID 39976487mais citado
  3. Prenatal diagnosis of chromothripsis causing complex chromosomal rearrangement involving chromosomes 5, 7 and 11 leading to TWIST1 deletion and Saethre-Chotzen syndrome.
    Taiwanese journal of obstetrics &amp; gynecology· 2025· PMID 39794020mais citado
  4. 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· 2024· PMID 40225751mais citado
  5. Prediction and design of transcriptional repressor domains with large-scale mutational scans and deep learning.
    bioRxiv : the preprint server for biology· 2024· PMID 39386603mais citado
  6. Prenatal Tympanic Ring Anomaly Without Microtia: A Subtle Clue Toward Severe Early-Onset Monogenic Disorders.
    Prenat Diagn· 2026· PMID 41882499recente
  7. Pfeiffer Syndrome.
    · 2026· PMID 30422477recente

Bases de dados e fontes oficiais

Identificadores e referências canônicas usadas para montar este verbete.

  1. ORPHA:794(Orphanet)
  2. OMIM OMIM:101400(OMIM)
  3. MONDO:0007042(MONDO)
  4. GARD:7598(GARD (NIH))
  5. Variantes catalogadas(ClinVar)
  6. Busca completa no PubMed(PubMed)
  7. Artigo Wikipedia(Wikipedia)
  8. Q3508686(Wikidata)

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

Conteúdo mantido por Agente Raras · Médicos e pesquisadores podem colaborar

Síndrome Saethre-Chotzen
Compêndio · Raras BR

Síndrome Saethre-Chotzen

ORPHA:794 · MONDO:0007042
Prevalência
1-9 / 100 000
Herança
Autosomal dominant
CID-10
Q87.0 · Síndromes com malformações congênitas afetando predominantemente o aspecto da face
CID-11
Ensaios
2 ativos
Início
Antenatal, Neonatal
Prevalência
0.0 (Europe)
MedGen
UMLS
C0175699
EuropePMC
Wikidata
Wikipedia
Papers 10a
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