Raras
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Síndrome Adams-Oliver
ORPHA:974CID-10 · Q87.2CID-11 · LD2F.1YDOENÇA RARA

A Síndrome de Adams-Oliver (SAO) é uma doença rara que se caracteriza pela combinação de malformações nos braços e pernas (presentes desde o nascimento) e falhas no couro cabeludo, sendo muitas vezes acompanhada por problemas na formação dos ossos da cabeça (o crânio).

Mantido por Agente Raras·Colaborar como especialista →

Introdução

O que você precisa saber de cara

📋

A Síndrome de Adams-Oliver (SAO) é uma doença rara que se caracteriza pela combinação de malformações nos braços e pernas (presentes desde o nascimento) e falhas no couro cabeludo, sendo muitas vezes acompanhada por problemas na formação dos ossos da cabeça (o crânio).

Publicações científicas
245 artigos
Último publicado: 2026 Apr 13

Escala de raridade

CLASSIFICAÇÃO ORPHANET · BRASIL 2024
1-9 / 1 000 000
Ultra-rara
<1/50k
Muito rara
1/20k
Rara
1/10k
Pouco freq.
1/5k
Incomum
1/2k
Prevalência
0.44
Worldwide
Casos conhecidos
398
pacientes catalogados
Início
Antenatal
+ neonatal
🏥
SUS: Cobertura mínimaScore: 15%
CID-10: Q87.2
🇧🇷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
26 sintomas
🧠
Neurológico
18 sintomas
❤️
Coração
12 sintomas
🫃
Digestivo
8 sintomas
😀
Face
8 sintomas
🧬
Pele e cabelo
5 sintomas

+ 43 sintomas em outras categorias

Características mais comuns

90%prev.
Cutis marmorata
Muito frequente (99-80%)
90%prev.
Defeito craniano
Muito frequente (99-80%)
90%prev.
Cabelo esparso
Muito frequente (99-80%)
90%prev.
Déficit de crescimento
Muito frequente (99-80%)
90%prev.
Anormalidade do membro superior
Muito frequente (99-80%)
90%prev.
Aplasia/Hipoplasia da pele
Muito frequente (99-80%)
133sintomas
Muito frequente (10)
Frequente (13)
Ocasional (23)
Sem dados (87)

Os sintomas variam de pessoa para pessoa. Abaixo estão as 133 características clínicas mais associadas, ordenadas por frequência.

Cutis marmorata
Muito frequente (99-80%)90%
Defeito cranianoSkull defect
Muito frequente (99-80%)90%
Cabelo esparsoSparse hair
Muito frequente (99-80%)90%
Déficit de crescimentoFailure to thrive
Muito frequente (99-80%)90%
Anormalidade do membro superiorAbnormality of the upper limb
Muito frequente (99-80%)90%

Linha do tempo da pesquisa

Publicações por ano — veja quando o interesse científico cresceu
Anos de pesquisa1desde 2026
Total histórico245PubMed
Últimos 10 anos121publicações
Pico202518 papers
Linha do tempo
2026Hoje · 2026📈 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

6 genes identificados com associação a esta condição. Padrão de herança: Autosomal dominant, Autosomal recessive.

ARHGAP31Rho GTPase-activating protein 31Disease-causing germline mutation(s) (gain of function) inAltamente restrito
FUNÇÃO

Functions as a GTPase-activating protein (GAP) for RAC1 and CDC42. Required for cell spreading, polarized lamellipodia formation and cell migration

LOCALIZAÇÃO

Cell projection, lamellipodiumCell junction, focal adhesion

VIAS BIOLÓGICAS (4)
RHOU GTPase cycleRAC1 GTPase cycleCDC42 GTPase cycleRHOA GTPase cycle
MECANISMO DE DOENÇA

Adams-Oliver syndrome 1

A disorder characterized by the congenital absence of skin (aplasia cutis congenita) in combination with transverse limb defects. Aplasia cutis congenita can be located anywhere on the body, but in the vast majority of the cases, it is present on the posterior parietal region where it is often associated with an underlying defect of the parietal bones. Limb abnormalities are typically limb truncation defects affecting the distal phalanges or entire digits (true ectrodactyly). Only rarely, metatarsals/metacarpals or more proximal limb structures are also affected. Apart from transverse limb defects, syndactyly, most commonly of second and third toes, can also be observed. The clinical features are highly variable and can also include cardiovascular malformations, brain abnormalities and vascular defects such as cutis marmorata and dilated scalp veins.

OUTRAS DOENÇAS (2)
Adams-Oliver syndrome 1Adams-Oliver syndrome
HGNC:29216UniProt:Q2M1Z3
NOTCH1Neurogenic locus notch homolog protein 1Disease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Functions as a receptor for membrane-bound ligands Jagged-1 (JAG1), Jagged-2 (JAG2) and Delta-1 (DLL1) to regulate cell-fate determination. Upon ligand activation through the released notch intracellular domain (NICD) it forms a transcriptional activator complex with RBPJ/RBPSUH and activates genes of the enhancer of split locus. Affects the implementation of differentiation, proliferation and apoptotic programs. Involved in angiogenesis; negatively regulates endothelial cell proliferation and m

LOCALIZAÇÃO

Cell membraneLate endosome membraneNucleus

VIAS BIOLÓGICAS (2)
NFE2L2 regulating tumorigenic genesFormation of paraxial mesoderm
MECANISMO DE DOENÇA

Aortic valve disease 1

A common defect in the aortic valve in which two rather than three leaflets are present. It is often associated with aortic valve calcification, stenosis and insufficiency. In extreme cases, the blood flow may be so restricted that the left ventricle fails to grow, resulting in hypoplastic left heart syndrome.

EXPRESSÃO TECIDUAL(Ubíquo)
Pulmão
47.5 TPM
Baço
45.2 TPM
Skin Not Sun Exposed Suprapubic
42.4 TPM
Skin Sun Exposed Lower leg
41.7 TPM
Nervo tibial
38.9 TPM
OUTRAS DOENÇAS (4)
Adams-Oliver syndrome 5aortic valve disease 1Adams-Oliver syndromefamilial bicuspid aortic valve
HGNC:7881UniProt:P46531
DOCK6Dedicator of cytokinesis protein 6Disease-causing germline mutation(s) (loss of function) inTolerante
FUNÇÃO

Acts as a guanine nucleotide exchange factor (GEF) for CDC42 and RAC1 small GTPases. Through its activation of CDC42 and RAC1, may regulate neurite outgrowth (By similarity)

LOCALIZAÇÃO

CytoplasmCytoplasm, perinuclear region

VIAS BIOLÓGICAS (3)
Factors involved in megakaryocyte development and platelet productionRAC1 GTPase cycleCDC42 GTPase cycle
MECANISMO DE DOENÇA

Adams-Oliver syndrome 2

A disorder characterized by the congenital absence of skin (aplasia cutis congenita) in combination with transverse limb defects. Aplasia cutis congenita can be located anywhere on the body, but in the vast majority of the cases, it is present on the posterior parietal region where it is often associated with an underlying defect of the parietal bones. Limb abnormalities are typically limb truncation defects affecting the distal phalanges or entire digits (true ectrodactyly). Only rarely, metatarsals/metacarpals or more proximal limb structures are also affected. Apart from transverse limb defects, syndactyly, most commonly of second and third toes, can also be observed. The clinical features are highly variable and can also include cardiovascular malformations, brain abnormalities and vascular defects such as cutis marmorata and dilated scalp veins.

EXPRESSÃO TECIDUAL(Ubíquo)
Pulmão
85.2 TPM
Tireoide
66.5 TPM
Tecido adiposo
56.9 TPM
Mama
48.0 TPM
Adipose Visceral Omentum
44.0 TPM
OUTRAS DOENÇAS (2)
Adams-Oliver syndrome 2Adams-Oliver syndrome
HGNC:19189UniProt:Q96HP0
EOGTEGF domain-specific O-linked N-acetylglucosamine transferaseDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Catalyzes the transfer of a single N-acetylglucosamine from UDP-GlcNAc to a serine or threonine residue in extracellular proteins resulting in their modification with a beta-linked N-acetylglucosamine (O-GlcNAc). Specifically glycosylates the Thr residue located between the fifth and sixth conserved cysteines of folded EGF-like domains

LOCALIZAÇÃO

Endoplasmic reticulum lumen

MECANISMO DE DOENÇA

Adams-Oliver syndrome 4

A form of Adams-Oliver syndrome, a disorder characterized by the congenital absence of skin (aplasia cutis congenita) in combination with transverse limb defects. Aplasia cutis congenita can be located anywhere on the body, but in the vast majority of the cases, it is present on the posterior parietal region where it is often associated with an underlying defect of the parietal bones. Limb abnormalities are typically limb truncation defects affecting the distal phalanges or entire digits (true ectrodactyly). Only rarely, metatarsals/metacarpals or more proximal limb structures are also affected. Apart from transverse limb defects, syndactyly, most commonly of second and third toes, can also be observed. The clinical features are highly variable and can also include cardiovascular malformations, brain abnormalities and vascular defects such as cutis marmorata and dilated scalp veins.

EXPRESSÃO TECIDUAL(Ubíquo)
Aorta
73.6 TPM
Artéria tibial
56.9 TPM
Artéria coronária
52.8 TPM
Esôfago - Muscular
43.4 TPM
Pulmão
39.1 TPM
INTERAÇÕES PROTEICAS (4)
OUTRAS DOENÇAS (2)
Adams-Oliver syndrome 4Adams-Oliver syndrome
HGNC:28526UniProt:Q5NDL2
DLL4Delta-like protein 4Disease-causing germline mutation(s) (loss of function) inAltamente restrito
FUNÇÃO

Involved in the Notch signaling pathway as Notch ligand (PubMed:11134954). Activates NOTCH1 and NOTCH4. Involved in angiogenesis; negatively regulates endothelial cell proliferation and migration and angiogenic sprouting (PubMed:20616313). Essential for retinal progenitor proliferation. Required for suppressing rod fates in late retinal progenitors as well as for proper generation of other retinal cell types (By similarity). During spinal cord neurogenesis, inhibits V2a interneuron fate (PubMed:

LOCALIZAÇÃO

Cell membrane

VIAS BIOLÓGICAS (8)
NOTCH2 Activation and Transmission of Signal to the NucleusActivated NOTCH1 Transmits Signal to the NucleusConstitutive Signaling by NOTCH1 PEST Domain MutantsConstitutive Signaling by NOTCH1 t(7;9)(NOTCH1:M1580_K2555) Translocation MutantNOTCH4 Activation and Transmission of Signal to the Nucleus
MECANISMO DE DOENÇA

Adams-Oliver syndrome 6

A form of Adams-Oliver syndrome, a disorder characterized by the congenital absence of skin (aplasia cutis congenita) in combination with transverse limb defects. Aplasia cutis congenita can be located anywhere on the body, but in the vast majority of the cases, it is present on the posterior parietal region where it is often associated with an underlying defect of the parietal bones. Limb abnormalities are typically limb truncation defects affecting the distal phalanges or entire digits (true ectrodactyly). Only rarely, metatarsals/metacarpals or more proximal limb structures are also affected. Apart from transverse limb defects, syndactyly, most commonly of second and third toes, can also be observed. The clinical features are highly variable and can also include cardiovascular malformations, brain abnormalities and vascular defects such as cutis marmorata and dilated scalp veins.

EXPRESSÃO TECIDUAL(Ubíquo)
Tecido adiposo
46.1 TPM
Mama
39.1 TPM
Pulmão
38.5 TPM
Adipose Visceral Omentum
35.9 TPM
Tireoide
34.3 TPM
OUTRAS DOENÇAS (3)
Adams-Oliver syndrome 6Adams-Oliver syndromeaplasia cutis congenita
HGNC:2910UniProt:Q9NR61
RBPJRecombining binding protein suppressor of hairlessDisease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Transcriptional regulator that plays a central role in Notch signaling, a signaling pathway involved in cell-cell communication that regulates a broad spectrum of cell-fate determinations. Acts as a transcriptional repressor when it is not associated with Notch proteins. When associated with some NICD product of Notch proteins (Notch intracellular domain), it acts as a transcriptional activator that activates transcription of Notch target genes. Probably represses or activates transcription via

LOCALIZAÇÃO

NucleusCytoplasm

VIAS BIOLÓGICAS (10)
NOTCH1 Intracellular Domain Regulates TranscriptionPre-NOTCH Transcription and TranslationNOTCH4 Intracellular Domain Regulates TranscriptionNOTCH3 Intracellular Domain Regulates TranscriptionRegulation of gene expression in late stage (branching morphogenesis) pancreatic bud precursor cells
MECANISMO DE DOENÇA

Adams-Oliver syndrome 3

An autosomal dominant form of Adams-Oliver syndrome, a disorder characterized by the congenital absence of skin (aplasia cutis congenita) in combination with transverse limb defects. Aplasia cutis congenita can be located anywhere on the body, but in the vast majority of the cases, it is present on the posterior parietal region where it is often associated with an underlying defect of the parietal bones. Limb abnormalities are typically limb truncation defects affecting the distal phalanges or entire digits (true ectrodactyly). Only rarely, metatarsals/metacarpals or more proximal limb structures are also affected. Apart from transverse limb defects, syndactyly, most commonly of second and third toes, can also be observed. The clinical features are highly variable and can also include cardiovascular malformations, brain abnormalities and vascular defects such as cutis marmorata and dilated scalp veins. AOS3 patients manifest characteristic vertex scalp defects and terminal limb defects, but without congenital heart defects, other associated defects, or immune defects.

EXPRESSÃO TECIDUAL(Ubíquo)
Útero
58.7 TPM
Cervix Ectocervix
56.0 TPM
Tecido adiposo
52.7 TPM
Nervo tibial
51.7 TPM
Linfócitos
47.3 TPM
OUTRAS DOENÇAS (2)
Adams-Oliver syndrome 3Adams-Oliver syndrome
HGNC:5724UniProt:Q06330

Variantes genéticas (ClinVar)

1,083 variantes patogênicas registradas no ClinVar.

🧬 ARHGAP31: NM_020754.4(ARHGAP31):c.2647C>T (p.Gln883Ter) ()
🧬 ARHGAP31: NM_020754.4(ARHGAP31):c.3166_3167del (p.Gln1056fs) ()
🧬 ARHGAP31: NM_020754.4(ARHGAP31):c.2101A>C (p.Ser701Arg) ()
🧬 ARHGAP31: NM_020754.4(ARHGAP31):c.1593G>T (p.Glu531Asp) ()
🧬 ARHGAP31: NM_020754.4(ARHGAP31):c.1315G>C (p.Glu439Gln) ()
Ver todas no ClinVar

Classificação de variantes (ClinVar)

Distribuição de 3,807 variantes classificadas pelo ClinVar.

571
1332
1904
Patogênica (15.0%)
VUS (35.0%)
Benigna (50.0%)
VARIANTES MAIS SIGNIFICATIVAS
DOCK6-AS1: NM_020812.4(DOCK6):c.4708G>T (p.Glu1570Ter) [Likely pathogenic]
NOTCH1: NM_017617.5(NOTCH1):c.1342del (p.Arg448fs) [Pathogenic]
NOTCH1: NM_017617.5(NOTCH1):c.1650C>A (p.Tyr550Ter) [Pathogenic]
NOTCH1: NM_017617.5(NOTCH1):c.6475C>G (p.Arg2159Gly) [Uncertain significance]
NOTCH1: NM_017617.5(NOTCH1):c.6218G>A (p.Ser2073Asn) [Uncertain significance]

Diagnóstico

Os sinais que médicos procuram e os exames que confirmam

Carregando...

Tratamento e manejo

Remédios, cuidados de apoio e o que precisa acompanhar

Carregando informações de tratamento...

Onde tratar no SUS

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

🇧🇷 Atendimento SUS — Síndrome Adams-Oliver

🗺️

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.

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Ensaios clínicos abertos e novidades científicas recentes

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Publicações mais relevantes

Timeline de publicações
119 papers (10 anos)
#1

Noncirrhotic Portopulmonary Hypertension Due to Hepatoportal Sclerosis in Adams-Oliver Syndrome.

Pediatrics2026 Jan 01

A 13-year-old girl presented for evaluation of pulmonary hypertension after symptoms of dyspnea and exercise intolerance. Full evaluation was negative except for abdominal ultrasonography with splenomegaly and esophageal varices suggestive of portal hypertension. Cardiac and hepatic vein catheterization confirmed portopulmonary hypertension. Liver biopsy demonstrated sinusoidal dilatation, nodularity, and minimal fibrosis, which was interpreted as possible nodular regenerative hyperplasia but not cirrhosis. Radiographic imaging, including computed tomography venography, demonstrated an elongated and severely stenotic extrahepatic portal vein, and portal hypertension was presumed to be secondary to congenital portal vein hypoplasia. She was treated with ambrisentan with initial improvement in symptoms and estimated pulmonary pressure. Whole-exome sequencing revealed a likely pathogenic missense mutation in Delta-like canonical Notch ligand 4 associated with Adams-Oliver syndrome. After 2 years, pulmonary hypertension and right heart failure symptoms worsened, along with liver failure. She then had fulminant liver failure and cardiorespiratory arrest. Resuscitative efforts included extracorporeal membranous oxygenation (ECMO), but because of hypoxic brain injury, care was compassionately withdrawn. Autopsy limited to the thorax and abdomen revealed high-grade pulmonary plexiform arteriopathy, splenomegaly, esophageal varices, and large splenorenal shunt. The liver was small with a nodular surface but not fibrotic. The entire length of the extrahepatic portal vein was severely stenotic, and intrahepatic portal veins were missing or diminutive-findings diagnostic of hepatoportal sclerosis. Noncirrhotic portopulmonary hypertension is rare and should include evaluation of immunologic, infectious, toxic, thrombotic, and genetic etiologies. Unfortunately, there is no known treatment of hepatoportal sclerosis.

#2

Adams-Oliver Syndrome Type 3: A Case Report of Concurrent RBPJ, CACNA1A, and Double-Heterozygous MTHFR Variants.

Diagnostics (Basel, Switzerland)2026 Jan 15

Background and Clinical Significance: Adams-Oliver syndrome type 3 (AOS3) is a rare congenital disorder typically characterised by terminal transverse limb defects and variable involvement of other organ systems. Although pathogenic variants in RBPJ are well established in AOS3, associated neurodevelopmental or psychiatric features have been only sporadically documented. Case Presentation: We describe a male patient first evaluated at the age of 10 years and subsequently re-evaluated at 14 years, with AOS3 presenting terminal limb defects together with autistic-like behaviour, cognitive difficulties, dyslexia, and recurrent depressive symptoms. Whole-exome sequencing (WES) identified a heterozygous pathogenic variant in RBPJ (c.505A>G; p.Lys169Glu), confirming the molecular diagnosis of autosomal dominant AOS3. Additional findings included a heterozygous missense variant in CACNA1A (p.Arg1678Cys), a gene linked to neurological disorders with broad phenotypic variability. Because of elevated homocysteine levels, the patient was also tested for MTHFR variants and was found to be heterozygous for C677T and A1298C. Conclusions: This case illustrates a rare combination of a validated AOS3-associated RBPJ variant, along with additional CACNA1A and MTHFR variants that may influence the patient's neurocognitive and psychiatric characteristics. The results underscore the importance of comprehensive genetic testing in atypical AOS presentations and highlight the complexity of interpreting overlapping genetic factors.

#3

Aplasia Cutis Congenita and Congenital Heart Disease: A Case Report, Highlighting the Limitation of Antenatal Screening.

The American journal of case reports2026 Jan 01

BACKGROUND Aplasia cutis congenita (ACC) is a rare congenital defect involving localized absence of skin, occasionally associated with deeper tissue anomalies and congenital malformations. CASE REPORT We report the case of a full-term newborn presenting with extensive ACC involving 40% of the scalp, alongside multiple congenital heart defects, ultimately consistent with tetralogy of Fallot (ToF). Initial management included conservative wound care, cardiologic and neurologic evaluations, and genetic testing. Despite clinical stability at discharge, the infant was readmitted 3 weeks later with hemorrhagic shock secondary to sagittal sinus bleeding, requiring surgical intervention and intensive care. Subsequent follow-up revealed tonic seizures and radiological findings suggestive of prior mild hypoxic injury. While the Rapid Aneuploidy Detection test confirmed a normal female karyotype (46,XX), whole-exome sequencing was pending; however, Adams-Oliver syndrome was strongly suspected. This case illustrates the diagnostic and management challenges posed by extensive ACC with syndromic associations. It also highlights the limitations of antenatal screening, as no abnormalities were detected during pregnancy despite fetal growth restriction. CONCLUSIONS The case underscores the need for a multidisciplinary approach, parental education, and long-term follow-up. Early recognition and coordinated care are essential for managing potential complications and guiding prognosis in complex cases of ACC with associated congenital anomalies.

#4

Adams-Oliver Syndrome: A Comprehensive Literature Review of Clinical, Nutritional, Genetic, and Molecular Aspects with Nursing Care Considerations.

International journal of molecular sciences2025 Dec 23

The present review aims to provide a comprehensive overview of the current literature on Adams-Oliver syndrome (AOS), synthesizing information on its clinical features, genetic and molecular underpinnings, nutritional aspects, and key nursing care considerations. AOS is a rare congenital disorder. Its genetic basis is heterogeneous, involving mutations in at least six key genes (ARHGAP31, RBPJ, NOTCH1, DLL4, DOCK6, and EOGT), which primarily affect vascular development through pathways like Notch signaling and Rho GTPase regulation. The management of AOS is complex and requires a multidisciplinary approach. The clinical presentation of AOS is highly variable, ranging from mild to severe and includes a wide spectrum of clinical manifestations, most notably aplasia cutis congenita and terminal transverse limb defects. The underlying molecular mechanisms predominantly point towards vasculopathy, disrupting embryonic development. Emerging evidence also highlights the presence of nutritional issues, such as poor feeding and growth failure, which are often overlooked. Management demands an integrated, multidisciplinary management approach, requiring coordinated effort from specialists in pediatrics, genetics, molecular biology, cardiology, surgery, and nutrition. Specialized nursing care is crucial for managing complex symptoms, particularly wound care for aplasia cutis, and for providing family support.

#5

Case Report: A novel DLL4 variant in a neonate with Adams-Oliver syndrome.

Frontiers in pediatrics2025

Adams-Oliver syndrome is a rare congenital disorder with six subtypes that have been identified. Subtypes 1, 3, 5, and 6 have an autosomal dominant inheritance pattern, whereas subtypes 2 and 4 have an autosomal recessive inheritance pattern. The clinical phenotype of Adams-Oliver syndrome is heterogeneous and can be accompanied by abnormalities in other organs, especially the cardiovascular system, such as cutis marmorata telangiectatica congenita, pulmonary hypertension, vascular abnormalities in other organs, and congenital heart defects. Herein, we report a case of Adams-Oliver syndrome caused by a de novo variant in DLL4. The patient was a neonate with clinical manifestations of skin defects who was diagnosed with Adams-Oliver syndrome on the basis of genetic testing.

Publicações recentes

Ver todas no PubMed

📚 EuropePMC178 artigos no totalmostrando 119

2026

Adams-Oliver Syndrome Type 3: A Case Report of Concurrent RBPJ, CACNA1A, and Double-Heterozygous MTHFR Variants.

Diagnostics (Basel, Switzerland)
2025

Adams-Oliver Syndrome: A Clinical Diagnosis in the Genomic Era.

Cureus
2025

Adams-Oliver Syndrome: A Comprehensive Literature Review of Clinical, Nutritional, Genetic, and Molecular Aspects with Nursing Care Considerations.

International journal of molecular sciences
2026

Aplasia Cutis Congenita and Congenital Heart Disease: A Case Report, Highlighting the Limitation of Antenatal Screening.

The American journal of case reports
2025

ROP mimicker in a big premature baby: Adams-Oliver syndrome with DOCK6 mutation: a case report and review of the literature.

Ophthalmic genetics
2026

Noncirrhotic Portopulmonary Hypertension Due to Hepatoportal Sclerosis in Adams-Oliver Syndrome.

Pediatrics
2025

Mouse scalp development requires Rac1 and SRF for the maintenance of mechanoresponsive mesenchyme.

Development (Cambridge, England)
2025

Defective Notch1 signaling in endothelial cells drives pathogenesis in a mouse model of Adams-Oliver syndrome.

The Journal of clinical investigation
2025

Novel De Novo DLL4 Missense and Highly Accurate Protein Structure Prediction in Adams-Oliver Type 6 Syndrome.

Clinical case reports
2025

Adams-Oliver syndrome: an unusual congenital disorder.

Oxford medical case reports
2025

Cutaneous Features of Adams-Oliver Syndrome: Diagnosis, Differentiation, and Management.

Pediatric dermatology
2025

Familial Exudative Vitreoretinopathy-Like Retinal Findings in Adams-Oliver Syndrome Type 2.

Clinical &amp; experimental ophthalmology
2025

Adams-Oliver Syndrome in a Newborn: A Case Report and Comprehensive Literature Review.

Cureus
2025

Novel compound heterozygous DOCK6 variants expand the mutational spectrum in prenatal diagnosis of Adams-Oliver syndrome 2.

BMC medical genomics
2025

A case of congenital heart defects and familial exudative vitreoretinopathy caused by activation of a cryptic splice donor in NOTCH1.

BMC medical genomics
2025

Perinatal outcomes in cases of umbilical-portal-systemic venous shunts: experience of a tertiary center.

BMC pregnancy and childbirth
2025

Aplasia Cutis: From Diagnosis to Management-2 Decades of Clinical Insights.

Plastic and reconstructive surgery
2025

Case Report: A novel DLL4 variant in a neonate with Adams-Oliver syndrome.

Frontiers in pediatrics
2025

Mouse scalp development requires Rac1 and SRF for the maintenance of mechanosensing mesenchyme.

bioRxiv : the preprint server for biology
2025

Effective in vivo binding energy landscape illustrates kinetic stability of RBPJ-DNA binding.

Nature communications
2025

Outpatient management of large scalp aplasia cutis congenita without skull defect in a case of Adams-Oliver syndrome.

The Kaohsiung journal of medical sciences
2024

Human Genetics of Ventricular Septal Defect.

Advances in experimental medicine and biology
2024

Adams-Oliver syndrome associated with refractory glaucoma.

Journal of AAPOS : the official publication of the American Association for Pediatric Ophthalmology and Strabismus
2024

Characterization of a New Variant in ARHGAP31 Probably Involved in Adams-Oliver Syndrome in a Family with a Variable Phenotypic Spectrum.

Genes
2024

Expanding the phenotypic spectrum of NOTCH1 variants: clinical manifestations in families with congenital heart disease.

European journal of human genetics : EJHG
2024

Adams-Oliver syndrome: About a case.

Clinical case reports
2024

Encephalocele as a rare complication of conservatively managed cranial aplasia cutis in a boy with Adams-Oliver syndrome.

Pediatrics and neonatology
2023

Cutis marmorata telangiectatica congenita: Incidence of extracutaneous manifestations and a proposed clinical definition.

Pediatric dermatology
2023

Adams-Oliver Syndrome - A Case Report.

Indian dermatology online journal
2023

A novel pathogenic variation of DOCK6 gene: the genotype-phenotype correlation in Adams-Oliver syndrome.

Molecular biology reports
2023

NOTCH1 loss of the TAD and PEST domain: An antimorph?

American journal of medical genetics. Part A
2022

Case report: Recombinant human epidermal growth factor gel plus kangfuxin solution in the treatment of aplasia cutis congenita in a case with Adams-Oliver syndrome.

Frontiers in surgery
2023

The role of Notch signaling pathway in metabolic bone diseases.

Biochemical pharmacology
2022

Adams-Oliver syndrome and associated complications: Report of a family in Colombia and review of the literature.

Biomedica : revista del Instituto Nacional de Salud
2022

Intrafamilial phenotypic variability in autosomal recessive DOCK6-related Adams-Oliver syndrome.

European journal of medical genetics
2022

FOSL2 truncating variants in the last exon cause a neurodevelopmental disorder with scalp and enamel defects.

Genetics in medicine : official journal of the American College of Medical Genetics
2022

Synergistic effects of rare variants of ARHGAP31 and FBLN1 in vitro in terminal transverse limb defects.

Frontiers in genetics
2022

Cutaneous squamous cell carcinoma in an autosomal-recessive Adams-Oliver syndrome patient with a novel frameshift pathogenic variant in the EOGT gene.

American journal of medical genetics. Part A
2022

Atypical Adams-Oliver syndrome with typical ocular signs of familial exudative vitreoretinopathy.

International journal of ophthalmology
2022

A Drosophila Su(H) model of Adams-Oliver Syndrome reveals cofactor titration as a mechanism underlying developmental defects.

PLoS genetics
2022

Heterozygous NOTCH1 Variants Cause CNS Immune Activation and Microangiopathy.

Annals of neurology
2022

Adams-Oliver Syndrome: Vestigial Tail and Genetics Update.

Archives of plastic surgery
2022

Adams-Oliver Syndrome: A Rare Congenital Disorder.

Cureus
2023

Severe Adams-Oliver Syndrome after Maternal COVID-19 Infection Could Be Another Effect of the SARS-CoV-2 Inflammatory Storm? Case Report.

Fetal and pediatric pathology
2022

Trichorhinophalangeal syndrome type II associated with aplasia cutis congenita in a neonate.

Pediatric dermatology
2022

Adams-Oliver syndrome, intestinal obstruction and heart defects: a case series of aplasia cutis congenita.

Oxford medical case reports
2022

A novel DLL4 mutation in Adams-Oliver syndrome with absence of the right pulmonary artery in newborn.

American journal of medical genetics. Part A
2021

Murine Model of Cardiac Defects Observed in Adams-Oliver Syndrome Driven by Delta-Like Ligand-4 Haploinsufficiency.

Stem cells and development
2021

Case report and review of literature of a rare congenital disorder: Adams-Oliver syndrome.

BMC anesthesiology
2020

Born in the Purple: An Exceptional Case of Cutis Marmorata Telangiectatica Congenita.

Acta dermatovenerologica Croatica : ADC
2021

Two AOS genes attributed to familial exudative vitreoretinopathy with microcephaly: Two case reports.

Medicine
2021

"Health status of children with chronic liver disease during the SARS-CoV-2 outbreak: results from a multicentre study".

Clinics and research in hepatology and gastroenterology
2021

Multiomics Integration in Skin Diseases with Alterations in Notch Signaling Pathway: PlatOMICs Phase 1 Deployment.

International journal of molecular sciences
2021

The prognosis of common arterial trunk from a fetal perspective: A prenatal cohort study and systematic literature review.

Prenatal diagnosis
2021

Ex Vivo Models to Decipher the Molecular Mechanisms of Genetic Notch Cardiovascular Disorders.

Tissue engineering. Part C, Methods
2021

Diseases related to Notch glycosylation.

Molecular aspects of medicine
2022

PROLIFERATIVE RETINOPATHY IN A 13-YEAR-OLD WITH ADAMS-OLIVER SYNDROME.

Retinal cases &amp; brief reports
2021

Aplasia cutis congenita in a CDC42-related developmental phenotype.

American journal of medical genetics. Part A
2020

From Skin to Kidneys: Cutaneous Clues of Renal Disease in Children.

Dermatology practical &amp; conceptual
2020

Aplasia cutis congenita: a report of two cases from National Hospital Abuja, Nigeria and review of the literature.

The Pan African medical journal
2020

Pleiotropic Role of Notch Signaling in Human Skin Diseases.

International journal of molecular sciences
2020

Adams-Oliver syndrome: a case of bilateral progressive ischemic maculopathy.

Journal of AAPOS : the official publication of the American Association for Pediatric Ophthalmology and Strabismus
2020

A novel variant in DOCK6 gene associated with Adams-Oliver syndrome type 2.

Ophthalmic genetics
2020

Adams Oliver syndrome: A mimicker of familial exudative vitreoretinopathy.

American journal of ophthalmology case reports
2020

Multiple Aplasia Cutis Congenita Lesions of the Scalp: A Case Study.

Neonatal network : NN
2020

Treatment of a Large Skull Defect and Brain Herniation in a Newborn With Adams-Oliver Syndrome.

Cureus
2020

Novel In-Frame Deletion Mutation in NOTCH1 in a Chinese Sporadic Case of Adams-Oliver Syndrome.

DNA and cell biology
2020

Functional genomics and gene-environment interaction highlight the complexity of congenital heart disease caused by Notch pathway variants.

Human molecular genetics
2020

Expanding the phenotype in Adams-Oliver syndrome correlating with the genotype.

American journal of medical genetics. Part A
2019

Adams-Oliver syndrome caused by mutations of the EOGT gene.

American journal of medical genetics. Part A
2019

A novel DLL4 missense mutation in a Chinese patient with Adams-Oliver syndrome.

Chinese medical journal
2019

Ocular Manifestations of Cutis Marmorata Telangiectatica Congenita.

Ophthalmology. Retina
2019

Familial aggregation of "apple peel" intestinal atresia and cardiac left-sided obstructive lesions: A possible causal relationship with NOTCH1 gene mutations.

American journal of medical genetics. Part A
2019

[Analysis of DOCK6 gene mutation in a child affected with Adams-Oliver syndrome type 2].

Zhonghua yi xue yi chuan xue za zhi = Zhonghua yixue yichuanxue zazhi = Chinese journal of medical genetics
2019

Novel compound heterozygous mutations of the DOCK6 gene in a familial case of Adams-Oliver syndrome 2.

Gene
2019

Structure and function of extracellular O-GlcNAc.

Current opinion in structural biology
2019

[Adams-Oliver syndrome and cutis marmorata telangiectatica congenita].

Annales de dermatologie et de venereologie
2018

Congenital diseases caused by defective O-glycosylation of Notch receptors.

Nagoya journal of medical science
2018

Dermatoscopy of Common Lesions in Pediatric Dermatology.

Dermatologic clinics
2018

Intracranial Calcifications in Young Children.

Seminars in pediatric neurology
2019

Adams-Oliver Syndrome: Limited Expression.

Indian journal of pediatrics
2018

Elucidating the genetic architecture of Adams-Oliver syndrome in a large European cohort.

Human mutation
2019

Overlapping but distinct roles for NOTCH receptors in human cardiovascular disease.

Clinical genetics
2018

Adams Oliver syndrome with cerebellar cortical dysplasia.

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
2018

Epileptic Encephalopathy in Adams-Oliver Syndrome Associated to a New DOCK6 Mutation: A Peculiar Behavioral Phenotype.

Neuropediatrics
2018

CdGAP/ARHGAP31 is regulated by RSK phosphorylation and binding to 14-3-3β adaptor protein.

Oncotarget
2018

Importance of complete phenotyping in prenatal whole exome sequencing.

Human genetics
2017

A Case of Adams-Oliver Syndrome.

Advanced biomedical research
2019

Adams-Oliver Syndrome With Moyamoya Disease for Cerebral Revascularisation Surgery.

Journal of neurosurgical anesthesiology
2017

Adams-Oliver Syndrome Type 2 in Association with Compound Heterozygous DOCK6 Mutations.

Pediatric dermatology
2017

Corrigendum: Novel missense mutation in DLL4 in a Japanese sporadic case of Adams-Oliver syndrome.

Journal of human genetics
2017

The scaffold protein Ajuba suppresses CdGAP activity in epithelia to maintain stable cell-cell contacts.

Scientific reports
2017

Adams-Oliver Syndrome with Unusual Central Nervous System Findings and an Extrahepatic Portosystemic Shunt.

Pediatric reports
2017

Use of an epidermal growth factor-infused foam dressing in a complicated case of Adams-Oliver syndrome.

Journal of wound care
2017

Visual Diagnosis: A Baby with a Scalp Lesion, Rash, and Left-Foot Deformity.

Pediatrics in review
2017

The developmental biology of genetic Notch disorders.

Development (Cambridge, England)
2017

Novel missense mutation in DLL4 in a Japanese sporadic case of Adams-Oliver syndrome.

Journal of human genetics
2017

EOGT and O-GlcNAc on secreted and membrane proteins.

Biochemical Society transactions
2019

Mechanism of cell-intrinsic adaptation to Adams-Oliver Syndrome gene DOCK6 disruption highlights ubiquitin-like modifier ISG15 as a regulator of RHO GTPases.

Small GTPases
2017

Adams-Oliver syndrome review of the literature: Refining the diagnostic phenotype.

American journal of medical genetics. Part A
2016

Adams-Oliver syndrome associated with gastrointestinal malformations.

BMJ case reports
2016

[Aplasia cutis congenita: Update and management].

Annales de chirurgie plastique et esthetique
2016

Cell-Intrinsic Adaptation Arising from Chronic Ablation of a Key Rho GTPase Regulator.

Developmental cell
2016

Adams-Oliver Syndrome: A Case with Full Expression.

Pediatric reports
2016

Letter regarding "Distal Limb Defects and Aplasia Cutis: Adams-Oliver Syndrome".

The Journal of hand surgery
2016

Distal Limb Defects and Aplasia Cutis: Adams-Oliver Syndrome.

The Journal of hand surgery
2016

Novel copy number variants and major limb reduction malformation: Report of three cases.

American journal of medical genetics. Part A
2015

DOCK6 Mutations Are Responsible for a Distinct Autosomal-Recessive Variant of Adams-Oliver Syndrome Associated with Brain and Eye Anomalies.

Human mutation
2015

DLL4 loss-of-function heterozygous mutations cause Adams-Oliver syndrome.

Clinical genetics
2015

Network-Informed Gene Ranking Tackles Genetic Heterogeneity in Exome-Sequencing Studies of Monogenic Disease.

Human mutation
2015

Heterozygous Loss-of-Function Mutations in DLL4 Cause Adams-Oliver Syndrome.

American journal of human genetics
2015

Intracellular and extracellular O-linked N-acetylglucosamine in the nervous system.

Experimental neurology
2015

Idiopathic non-cirrhotic portal hypertension: a review.

Orphanet journal of rare diseases
2015

Aplasia cutis congenita: report of 22 cases.

International journal of dermatology
2015

Haploinsufficiency of the NOTCH1 Receptor as a Cause of Adams-Oliver Syndrome With Variable Cardiac Anomalies.

Circulation. Cardiovascular genetics
2015

Cardiovascular malformations in Adams-Oliver syndrome.

American journal of medical genetics. Part A
2015

N-acetylglucosamine modification in the lumen of the endoplasmic reticulum.

Biochimica et biophysica acta
2015

Multiple tics in a patient with Adams-Oliver syndrome.

The Journal of neuropsychiatry and clinical neurosciences
2015

Adams-Oliver syndrome: a case report.

Pediatric dermatology
Ver todos os 178 no EuropePMC

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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. Noncirrhotic Portopulmonary Hypertension Due to Hepatoportal Sclerosis in Adams-Oliver Syndrome.
    Pediatrics· 2026· PMID 41406992mais citado
  2. Adams-Oliver Syndrome Type 3: A Case Report of Concurrent RBPJ, CACNA1A, and Double-Heterozygous MTHFR Variants.
    Diagnostics (Basel, Switzerland)· 2026· PMID 41594250mais citado
  3. Aplasia Cutis Congenita and Congenital Heart Disease: A Case Report, Highlighting the Limitation of Antenatal Screening.
    The American journal of case reports· 2026· PMID 41477812mais citado
  4. Adams-Oliver Syndrome: A Comprehensive Literature Review of Clinical, Nutritional, Genetic, and Molecular Aspects with Nursing Care Considerations.
    International journal of molecular sciences· 2025· PMID 41516051mais citado
  5. Case Report: A novel DLL4 variant in a neonate with Adams-Oliver syndrome.
    Frontiers in pediatrics· 2025· PMID 40098638mais citado
  6. Mutations in VCP cause Adams-Oliver syndrome with or without pulmonary hypertension.
    Genet Med· 2026· PMID 41979051recente
  7. Molecular mechanism study of novel compound heterozygous EOGT mutations leading to Adams-Oliver syndrome type 4.
    Glob Med Genet· 2026· PMID 41959640recente
  8. Adams-Oliver Syndrome: A Clinical Diagnosis in the Genomic Era.
    Cureus· 2025· PMID 41552262recente

Bases de dados e fontes oficiais

Identificadores e referências canônicas usadas para montar este verbete.

  1. ORPHA:974(Orphanet)
  2. MONDO:0007034(MONDO)
  3. GARD:5739(GARD (NIH))
  4. Variantes catalogadas(ClinVar)
  5. Busca completa no PubMed(PubMed)
  6. Q351708(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 Adams-Oliver
Compêndio · Raras BR

Síndrome Adams-Oliver

ORPHA:974 · MONDO:0007034
Prevalência
1-9 / 1 000 000
Casos
398 casos conhecidos
Herança
Autosomal dominant, Autosomal recessive
CID-10
Q87.2 · Síndromes com malformações congênitas afetando predominantemente os membros
CID-11
Início
Antenatal, Neonatal
Prevalência
0.44 (Worldwide)
MedGen
UMLS
C0265268
EuropePMC
Wikidata
Papers 10a
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