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Síndrome autoinflamatório
ORPHA:93665CID-10 · A68.9DOENÇA RARA
Esta doença tem causa genética. Recomendamos procurar um médico geneticista para diagnóstico, exames genéticos e aconselhamento familiar.
neuro

É um grupo de doenças que afeta o sistema imunológico inato (a primeira defesa do corpo), caracterizadas por crises de inflamação que surgem sem um motivo aparente. Diferente das doenças autoimunes, não há quantidades significativas de autoanticorpos (anticorpos que atacam o próprio corpo) nem de células T (células de defesa) que reagem contra o próprio organismo, que são achados mais típicos de condições autoimunes.

Em construçãohistóricoSugerir correção
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Introdução

O que você precisa saber de cara

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EM LINGUAGEM SIMPLES

O síndrome autoinflamatório é um conjunto de doenças raras em que o sistema de defesa do próprio corpo causa episódios repetidos de inflamação sem uma causa externa evidente. Ao contrário de doenças autoimunes tradicionais, o quadro não envolve a produção de autoanticorpos específicos que atacam as células. As pessoas podem manifestar alterações em vasos sanguíneos, problemas oculares, inchaço pelo corpo e inflamações em órgãos internos. No Brasil, a condição não conta com um Protocolo Clínico e Diretrizes Terapêuticas (PCDT) no Ministério da Saúde, de modo que o acompanhamento é individualizado nos serviços de saúde.

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Informacoes curadas por IA — podem conter imprecisoes

É um grupo de doenças que afeta o sistema imunológico inato (a primeira defesa do corpo), caracterizadas por crises de inflamação que surgem sem um motivo aparente. Diferente das doenças autoimunes, não há quantidades significativas de autoanticorpos (anticorpos que atacam o próprio corpo) nem de células T (células de defesa) que reagem contra o próprio organismo, que são achados mais típicos de condições autoimunes.

Pesquisas ativas
6 ensaios
17 total registrados no ClinicalTrials.gov
Publicações científicas
633 artigos
Último publicado: 2026 Apr 7
Medicamentos
6 com registro
COLCHICINA, GOLTRITE, COLCHIS

Tem tratamento?

✓ 6 medicamentos específicos desta doença (registro ANVISA, FDA ou SUS/CEAF)
Ver detalhes, fases e interações →
COLCHICINAGOLTRITECOLCHISCOXYMCOCICHIMILCIXIN
Mais 6 moléculas em estudo para esta doença.
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SUS: Cobertura mínimaScore: 0%
CID-10: A68.9
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Entender a doença

Do básico ao detalhe, leia no seu ritmo

Preparando trilha educativa...

Sinais e sintomas

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

Partes do corpo afetadas

🧠
Neurológico
73 sintomas
🦴
Ossos e articulações
71 sintomas
🧬
Pele e cabelo
58 sintomas
🫃
Digestivo
53 sintomas
🩸
Sangue
52 sintomas
👁️
Olhos
49 sintomas

+ 419 sintomas em outras categorias

Características mais comuns

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Edema
—
Estenose arterial
—
Anormalidade vascular da pele
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Neuropatia sensorimotora
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Acidente vascular cerebral tromboembólico
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Oftalmoplegia internuclear
997sintomas
Sem dados (997)

Os sintomas variam de pessoa para pessoa. Abaixo estão as 997 características clínicas mais associadas, ordenadas por frequência.

Estenose arterialArterial stenosis
Anormalidade vascular da peleVascular skin abnormality
Neuropatia sensorimotoraSensorimotor neuropathy

Linha do tempo da pesquisa

Publicações por ano — veja quando o interesse científico cresceu
Anos de pesquisa1desde 2026
Total histórico633PubMed
Últimos 10 anos200publicações
Pico202550 papers
Linha do tempo
2026Hoje · 2026🧪 2005Primeiro 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

55 genes identificados com associação a esta condição.

Curadoria gene-doença

fontes oficiais
UNC93B1IL36RNDPP9GIMAP6
LPIN2Phosphatidate phosphatase LPIN2Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (3)
Synthesis of PESynthesis of PCTriglyceride biosynthesis
MECANISMO DE DOENÇA

Majeed syndrome

An autosomal recessive syndrome characterized by chronic recurrent multifocal osteomyelitis that is of early onset with a lifelong course, congenital dyserythropoietic anemia that presents as hypochromic, microcytic anemia during the first year of life and ranges from mild to transfusion-dependent, and transient inflammatory dermatosis, often manifesting as Sweet syndrome (neutrophilic skin infiltration).

EXPRESSÃO TECIDUAL(Ubíquo)
Fígado
47.5 TPM
Testículo
37.8 TPM
Pulmão
31.4 TPM
Cérebro - Hemisfério cerebelar
29.9 TPM
Cerebelo
29.9 TPM
OUTRAS DOENÇAS (1)
Majeed syndrome
HGNC:14450UniProt:Q92539
F12Coagulation factor XIICandidate gene tested inTolerante
VIAS BIOLÓGICAS (1)
Defective factor XII causes hereditary angioedema
MECANISMO DE DOENÇA

Factor XII deficiency

An asymptomatic anomaly of in vitro blood coagulation. Its diagnosis is based on finding a low plasma activity of the factor in coagulating assays. It is usually only accidentally discovered through pre-operative blood tests. Factor XII deficiency is divided into two categories, a cross-reacting material (CRM)-negative group (negative F12 antigen detection) and a CRM-positive group (positive F12 antigen detection).

EXPRESSÃO TECIDUAL(Tecido-específico)
Fígado
376.1 TPM
Esôfago - Mucosa
10.0 TPM
Brain Nucleus accumbens basal ganglia
6.6 TPM
Skin Sun Exposed Lower leg
5.4 TPM
Vagina
5.3 TPM
OUTRAS DOENÇAS (3)
congenital factor XII deficiencyhereditary angioedema type 3F12-associated cold autoinflammatory syndrome
HGNC:3530UniProt:P00748
HLA-DRB1HLA class II histocompatibility antigen, DRB1 beta chainCandidate gene tested inRestrito
VIAS BIOLÓGICAS (7)
Generation of second messenger moleculesTranslocation of ZAP-70 to Immunological synapsePhosphorylation of CD3 and TCR zeta chainsCo-inhibition by PD-1Downstream TCR signaling
EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
1832.6 TPM
Pulmão
1234.8 TPM
Baço
990.6 TPM
Intestino delgado
607.2 TPM
Nervo tibial
598.3 TPM
OUTRAS DOENÇAS (16)
narcolepsy-cataplexy syndromefollicular lymphomaVogt-Koyanagi-Harada diseasepediatric multiple sclerosis
HGNC:4948UniProt:P01911
LACC1Purine nucleoside phosphorylase LACC1Candidate gene tested inTolerante
MECANISMO DE DOENÇA

Juvenile arthritis

A rare, familial form of juvenile arthritis characterized by autosomal recessive inheritance and onset in early childhood of symmetric, chronic joint inflammation. It causes joint swelling, pain, stiffness and restricted joint movement. JUVAR has high clinical variability. Some patients exhibit systemic symptoms, including quotidian fever, erythematous rash, generalized lymphadenopathy, hepatomegaly, and/or splenomegaly. Others display polyarthritis without systemic inflammation.

EXPRESSÃO TECIDUAL(Ubíquo)
Brain Spinal cord cervical c-1
14.4 TPM
Substância negra
12.1 TPM
Testículo
11.5 TPM
Fibroblastos
10.3 TPM
Hipocampo
9.7 TPM
INTERAÇÕES PROTEICAS (4)
OUTRAS DOENÇAS (2)
juvenile arthritis due to defect in LACC1systemic-onset juvenile idiopathic arthritis
HGNC:26789UniProt:Q8IV20
DOCK11Dedicator of cytokinesis protein 11Candidate gene tested inRestrito
VIAS BIOLÓGICAS (3)
Factors involved in megakaryocyte development and platelet productionRAC1 GTPase cycleCDC42 GTPase cycle
MECANISMO DE DOENÇA

Autoinflammatory disease, multisystem, with immune dysregulation, X-linked

An X-linked recessive disorder apparent in infancy or early childhood, and characterized by immune dysregulation, variable cytopenias, and systemic or organ-specific autoinflammatory manifestations. Clinical features include systemic lupus erythematosus, panniculitis, inflammatory bowel disease, pulmonary disease, or arthritis associated with recurrent fever, leukocytosis, lymphoproliferation, and hepatosplenomegaly in the absence of an infectious agent. Death in childhood has been reported.

EXPRESSÃO TECIDUAL(Ubíquo)
Tecido adiposo
45.4 TPM
Artéria tibial
31.9 TPM
Adipose Visceral Omentum
29.7 TPM
Esôfago - Muscular
29.2 TPM
Esôfago - Junção
25.1 TPM
INTERAÇÕES PROTEICAS (5)
OUTRAS DOENÇAS (3)
autoinflammatory disease, multisystem, with immune dysregulation, X-linkedearly-onset autoimmune disorder due to DOCK11 partial deficiencyearly-onset immune dysregulation due to DOCK11 complete deficiency
HGNC:23483UniProt:Q5JSL3
CDC42Cell division control protein 42 homologCandidate gene tested inAltamente restrito
VIAS BIOLÓGICAS (10)
VEGFA-VEGFR2 PathwayRHO GTPases Activate ForminsRHO GTPases activate IQGAPsRHO GTPases activate PAKsMAPK6/MAPK4 signaling
MECANISMO DE DOENÇA

Takenouchi-Kosaki syndrome

An autosomal dominant syndrome characterized by macrothrombocytopenia, lymphedema, intellectual disability, developmental delay, and distinctive facial features.

OUTRAS DOENÇAS (2)
macrothrombocytopenia-lymphedema-developmental delay-facial dysmorphism-camptodactyly syndromeneonatal-onset severe multisystemic autoinflammatory disease with increased IL18
HGNC:1736UniProt:P60953
SAMD9LSterile alpha motif domain-containing protein 9-likeCandidate gene tested inTolerante
MECANISMO DE DOENÇA

Ataxia-pancytopenia syndrome

An autosomal dominant disorder characterized by cerebellar ataxia, variable hematologic cytopenias, and predisposition to bone marrow failure and myeloid leukemia.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
55.2 TPM
Baço
34.1 TPM
Pulmão
22.9 TPM
Nervo tibial
17.2 TPM
Cervix Endocervix
14.3 TPM
OUTRAS DOENÇAS (4)
monosomy 7 myelodysplasia and leukemia syndrome 1ataxia-pancytopenia syndromespinocerebellar ataxia 49SAMD9L-associated autoinflammatory syndrome
HGNC:1349UniProt:Q8IVG5
RBCK1RanBP-type and C3HC4-type zinc finger-containing protein 1Candidate gene tested inTolerante
VIAS BIOLÓGICAS (4)
Antigen processing: Ubiquitination & Proteasome degradationTNFR1-induced proapoptotic signalingRegulation of TNFR1 signalingTNFR1-induced NF-kappa-B signaling pathway
MECANISMO DE DOENÇA

Polyglucosan body myopathy 1 with or without immunodeficiency

A disease characterized by polyglucosan storage myopathy associated with early-onset progressive muscle weakness and progressive dilated cardiomyopathy, which may necessitate cardiac transplant in severe cases. Some patients present with severe immunodeficiency, invasive bacterial infections and chronic autoinflammation.

EXPRESSÃO TECIDUAL(Ubíquo)
Cerebelo
135.7 TPM
Cérebro - Hemisfério cerebelar
126.4 TPM
Pituitária
116.3 TPM
Baço
102.0 TPM
Tireoide
101.6 TPM
OUTRAS DOENÇAS (2)
polyglucosan body myopathy 1 with or without immunodeficiencyautoinflammatory syndrome with pyogenic bacterial infection and amylopectinosis
HGNC:15864UniProt:Q9BYM8
NCSTNNicastrinCandidate gene tested inAltamente restrito
VIAS BIOLÓGICAS (10)
Activated NOTCH1 Transmits Signal to the NucleusNOTCH3 Activation and Transmission of Signal to the NucleusNOTCH4 Activation and Transmission of Signal to the NucleusNoncanonical activation of NOTCH3NOTCH2 Activation and Transmission of Signal to the Nucleus
MECANISMO DE DOENÇA

Acne inversa, familial, 1

A chronic relapsing inflammatory disease of the hair follicles characterized by recurrent draining sinuses, painful skin abscesses, and disfiguring scars. Manifestations typically appear after puberty.

EXPRESSÃO TECIDUAL(Ubíquo)
Fibroblastos
123.9 TPM
Ovário
75.8 TPM
Tireoide
75.3 TPM
Pulmão
70.3 TPM
Nervo tibial
69.0 TPM
OUTRAS DOENÇAS (2)
acne inversa, familial, 1pyoderma gangrenosum-acne-suppurative hidradenitis syndrome
HGNC:17091UniProt:Q92542
RNASEH2BRibonuclease H2 subunit BCandidate gene tested inTolerante
MECANISMO DE DOENÇA

Aicardi-Goutieres syndrome 2

A form of Aicardi-Goutieres syndrome, a genetically heterogeneous disease characterized by cerebral atrophy, leukoencephalopathy, intracranial calcifications, chronic cerebrospinal fluid (CSF) lymphocytosis, increased CSF alpha-interferon, and negative serologic investigations for common prenatal infection. Clinical features as thrombocytopenia, hepatosplenomegaly and elevated hepatic transaminases along with intermittent fever may erroneously suggest an infective process. Severe neurological dysfunctions manifest in infancy as progressive microcephaly, spasticity, dystonic posturing and profound psychomotor retardation. Death often occurs in early childhood.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
17.9 TPM
Útero
14.7 TPM
Cervix Ectocervix
12.1 TPM
Fallopian Tube
12.0 TPM
Cólon sigmoide
11.6 TPM
OUTRAS DOENÇAS (2)
Aicardi-Goutieres syndrome 2Aicardi-Goutieres syndrome
HGNC:25671UniProt:Q5TBB1
BTNL2Butyrophilin-like protein 2Candidate gene tested inTolerante
VIAS BIOLÓGICAS (1)
Butyrophilin (BTN) family interactions
MECANISMO DE DOENÇA

Sarcoidosis 2

An idiopathic, systemic, inflammatory disease characterized by the formation of immune granulomas in involved organs. Granulomas predominantly invade the lungs and the lymphatic system, but also skin, liver, spleen, eyes and other organs may be involved.

OUTRAS DOENÇAS (2)
sarcoidosissarcoidosis, susceptibility to, 2
HGNC:1142UniProt:Q9UIR0
PTPN6Tyrosine-protein phosphatase non-receptor type 6Candidate gene tested inAltamente restrito
VIAS BIOLÓGICAS (10)
GPVI-mediated activation cascadeInterferon alpha/beta signalingSARS-CoV-2 activates/modulates innate and adaptive immune responsesCo-inhibition by PD-1Co-inhibition by BTLA
EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
442.6 TPM
Sangue
278.5 TPM
Baço
267.0 TPM
Pulmão
74.1 TPM
Intestino delgado
57.8 TPM
OUTRAS DOENÇAS (5)
bullous pyoderma gangrenosumvegetative pyoderma gangrenosumpustular pyoderma gangrenosumclassic pyoderma gangrenosum
HGNC:9658UniProt:P29350
AP1S3AP-1 complex subunit sigma-3Candidate gene tested inTolerante
VIAS BIOLÓGICAS (2)
MHC class II antigen presentationLysosome Vesicle Biogenesis
MECANISMO DE DOENÇA

Psoriasis 15, pustular

A form of pustular psoriasis, a life-threatening disease defined by repeated flares of sudden onset consisting of diffuse erythematous skin eruption characterized by rapid coverage with pustules, high-grade fever, asthenia, marked leukocytosis, and elevated serum levels of C-reactive protein.

OUTRAS DOENÇAS (4)
psoriasis 14, pustularpustulosis palmaris et plantarisgeneralized pustular psoriasispsoriasis 15, pustular, susceptibility to
HGNC:18971UniProt:Q96PC3
MIFMacrophage migration inhibitory factorCandidate gene tested inTolerante
VIAS BIOLÓGICAS (2)
Cell surface interactions at the vascular wallNeutrophil degranulation
MECANISMO DE DOENÇA

Persistent Muellerian duct syndrome 1

A form of male pseudohermaphroditism characterized by a failure of Muellerian duct regression in otherwise normal males.

EXPRESSÃO TECIDUAL(Ubíquo)
Testículo
29.1 TPM
Cerebelo
21.8 TPM
Cérebro - Hemisfério cerebelar
20.5 TPM
Córtex cerebral
5.6 TPM
Próstata
5.6 TPM
OUTRAS DOENÇAS (3)
systemic-onset juvenile idiopathic arthritiscystic fibrosisjuvenile idiopathic arthritis
HGNC:7097UniProt:P14174
RNF31E3 ubiquitin-protein ligase RNF31Candidate gene tested inAltamente restrito
VIAS BIOLÓGICAS (3)
TNFR1-induced proapoptotic signalingRegulation of TNFR1 signalingTNFR1-induced NF-kappa-B signaling pathway
MECANISMO DE DOENÇA

Immunodeficiency 115 with autoinflammation

An autosomal recessive immunologic disorder manifesting in early infancy and characterized by combined immunodeficiency, recurrent bacterial, viral, and fungal infections, as well as autoinflammatory features, including arthritis and dermatitis.

EXPRESSÃO TECIDUAL(Ubíquo)
Baço
58.6 TPM
Útero
50.4 TPM
Linfócitos
49.4 TPM
Cervix Ectocervix
49.0 TPM
Cervix Endocervix
47.7 TPM
OUTRAS DOENÇAS (2)
immunodeficiency 115 with autoinflammationautoinflammatory syndrome with pyogenic bacterial infection and amylopectinosis
HGNC:16031UniProt:Q96EP0
RNASEH2CRibonuclease H2 subunit CDisease-causing germline mutation(s) inTolerante
MECANISMO DE DOENÇA

Aicardi-Goutieres syndrome 3

A form of Aicardi-Goutieres syndrome, a genetically heterogeneous disease characterized by cerebral atrophy, leukoencephalopathy, intracranial calcifications, chronic cerebrospinal fluid (CSF) lymphocytosis, increased CSF alpha-interferon, and negative serologic investigations for common prenatal infection. Clinical features as thrombocytopenia, hepatosplenomegaly and elevated hepatic transaminases along with intermittent fever may erroneously suggest an infective process. Severe neurological dysfunctions manifest in infancy as progressive microcephaly, spasticity, dystonic posturing and profound psychomotor retardation. Death often occurs in early childhood.

EXPRESSÃO TECIDUAL(Ubíquo)
Cerebelo
70.5 TPM
Cérebro - Hemisfério cerebelar
68.3 TPM
Útero
67.2 TPM
Ovário
60.3 TPM
Cervix Endocervix
59.7 TPM
OUTRAS DOENÇAS (2)
Aicardi-Goutieres syndrome 3Aicardi-Goutieres syndrome
HGNC:24116UniProt:Q8TDP1
CEBPECCAAT/enhancer-binding protein epsilonDisease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (1)
Transcriptional regulation of granulopoiesis
MECANISMO DE DOENÇA

Specific granule deficiency 1

An immunologic disorder characterized by recurrent pyogenic infections, defective neutrophil chemotaxis and bactericidal activity, and lack of neutrophil secondary granule proteins. Neutrophils of affected individuals lack lactoferrin and show abnormal nuclear segmentation, bilobed nuclei, low alkaline phosphatase, and increased number of neutrophil mitochondria and ribosomes. SGD1 inheritance can be autosomal dominant or recessive.

OUTRAS DOENÇAS (4)
Pelger-Huet-like anomaly and episodic fever with abdominal painspecific granule deficiency 1CEBPE-associated autoinflammation-immunodeficiency-neutrophil dysfunction syndromespecific granule deficiency
HGNC:1836UniProt:Q15744
SAMHD1Deoxynucleoside triphosphate triphosphohydrolase SAMHD1Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (2)
Interferon alpha/beta signalingNucleotide catabolism
MECANISMO DE DOENÇA

Aicardi-Goutieres syndrome 5

A form of Aicardi-Goutieres syndrome, a genetically heterogeneous disease characterized by cerebral atrophy, leukoencephalopathy, intracranial calcifications, chronic cerebrospinal fluid (CSF) lymphocytosis, increased CSF alpha-interferon, and negative serologic investigations for common prenatal infection. Clinical features as thrombocytopenia, hepatosplenomegaly and elevated hepatic transaminases along with intermittent fever may erroneously suggest an infective process. Severe neurological dysfunctions manifest in infancy as progressive microcephaly, spasticity, dystonic posturing and profound psychomotor retardation. Death often occurs in early childhood.

EXPRESSÃO TECIDUAL(Ubíquo)
Nervo tibial
453.4 TPM
Esôfago - Muscular
225.4 TPM
Esôfago - Junção
209.5 TPM
Artéria coronária
194.4 TPM
Fallopian Tube
174.3 TPM
OUTRAS DOENÇAS (4)
chilblain lupus 2Aicardi-Goutieres syndrome 5familial chilblain lupusAicardi-Goutieres syndrome
HGNC:15925UniProt:Q9Y3Z3
NLRC4NLR family CARD domain-containing protein 4Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (2)
The IPAF inflammasomeTP53 Regulates Transcription of Caspase Activators and Caspases
MECANISMO DE DOENÇA

Autoinflammation with infantile enterocolitis

An autosomal dominant disorder characterized by neonatal-onset enterocolitis, periodic fever, and fatal or near-fatal episodes of autoinflammation. Affected individuals tend to have poor overall growth and gastrointestinal symptoms in infancy, recurrent febrile episodes with splenomegaly, and sometimes hematologic disturbances, arthralgias, or myalgias.

EXPRESSÃO TECIDUAL(Tecido-específico)
Sangue
25.8 TPM
Baço
13.8 TPM
Pulmão
5.5 TPM
Adipose Visceral Omentum
2.1 TPM
Glândula adrenal
1.7 TPM
OUTRAS DOENÇAS (2)
periodic fever-infantile enterocolitis-autoinflammatory syndromefamilial cold autoinflammatory syndrome 4
HGNC:16412UniProt:Q9NPP4
RNU7-1Disease-causing germline mutation(s) inDesconhecido
OUTRAS DOENÇAS (2)
Aicardi-Goutieres syndrome 9Aicardi-Goutieres syndrome
HGNC:34033
OTULINUbiquitin thioesterase otulinDisease-causing germline mutation(s) inRestrito
VIAS BIOLÓGICAS (2)
Regulation of TNFR1 signalingSynthesis of active ubiquitin: roles of E1 and E2 enzymes
MECANISMO DE DOENÇA

Autoinflammation, panniculitis, and dermatosis syndrome, autosomal recessive

An autosomal recessive autoinflammatory disorder characterized by neonatal-onset of fever, neutrophilic dermatitis, panniculitis, painful joints, failure to thrive. Patients do not exhibit overt primary immunodeficiency.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
21.5 TPM
Skin Not Sun Exposed Suprapubic
20.0 TPM
Skin Sun Exposed Lower leg
18.8 TPM
Aorta
13.0 TPM
Vagina
12.4 TPM
OUTRAS DOENÇAS (3)
autoinflammation, panniculitis, and dermatosis syndrome, autosomal dominantautoinflammation, panniculitis, and dermatosis syndrome, autosomal recessiveimmunodeficiency 107, susceptibility to invasive staphylococcus aureus infection
HGNC:25118UniProt:Q96BN8
PSMG2Proteasome assembly chaperone 2Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (1)
Proteasome assembly
MECANISMO DE DOENÇA

Proteasome-associated autoinflammatory syndrome 4

An autosomal recessive, autoinflammatory disorder characterized by panniculitis and erythematous skin lesions apparent in early infancy. Additional features include hepatosplenomegaly, lymphadenopathy, autoimmune hemolytic anemia, fever, generalized lipodystrophy, myositis, joint contractures, and mild motor and speech delay.

VIAS REACTOME (1)
EXPRESSÃO TECIDUAL(Ubíquo)
Testículo
68.7 TPM
Linfócitos
63.5 TPM
Artéria tibial
57.5 TPM
Cervix Ectocervix
54.2 TPM
Aorta
54.0 TPM
OUTRAS DOENÇAS (1)
proteasome-associated autoinflammatory syndrome 4
HGNC:HGNC:24929UniProt:Q969U7
NOD2Nucleotide-binding oligomerization domain-containing protein 2Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (7)
SARS-CoV-2 activates/modulates innate and adaptive immune responsesNOD1/2 Signaling PathwayTAK1-dependent IKK and NF-kappa-B activation Interleukin-1 signalingJNK (c-Jun kinases) phosphorylation and activation mediated by activated human TAK1
MECANISMO DE DOENÇA

Blau syndrome

An autosomal dominant inflammatory disorder characterized by the formation of immune granulomas invading the skin, joints and eye. Other organs may be involved. Clinical manifestations are variable and include early-onset granulomatous arthritis, uveitis and skin rash. Blindness, joint destruction and visceral involvement have been reported in severe cases.

EXPRESSÃO TECIDUAL(Tecido-específico)
Skin Sun Exposed Lower leg
18.6 TPM
Vagina
16.5 TPM
Linfócitos
16.2 TPM
Skin Not Sun Exposed Suprapubic
15.8 TPM
Sangue
14.2 TPM
OUTRAS DOENÇAS (3)
Blau syndromeinflammatory bowel disease 1Yao syndrome
HGNC:5331UniProt:Q9HC29
TNFAIP3Tumor necrosis factor alpha-induced protein 3Disease-causing germline mutation(s) inAltamente restrito
VIAS BIOLÓGICAS (6)
Ovarian tumor domain proteasesNOD1/2 Signaling PathwayNegative regulators of DDX58/IFIH1 signalingRegulation of TNFR1 signalingTNFR1-induced proapoptotic signaling
MECANISMO DE DOENÇA

Autoinflammatory syndrome, familial, Behcet-like 1

An autosomal dominant, autoinflammatory disorder with early onset, characterized by ulceration of mucosal surfaces, particularly in the oral and genital areas. Additional variable features include skin rash, uveitis, and polyarthritis.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
245.4 TPM
Ovário
109.7 TPM
Pulmão
74.4 TPM
Adipose Visceral Omentum
63.8 TPM
Skin Not Sun Exposed Suprapubic
58.3 TPM
OUTRAS DOENÇAS (2)
autoinflammatory syndrome, familial, Behcet-like 1systemic lupus erythematosus
HGNC:11896UniProt:P21580
LYNTyrosine-protein kinase LynDisease-causing germline mutation(s) inAltamente restrito
VIAS BIOLÓGICAS (10)
GPVI-mediated activation cascadePlatelet Adhesion to exposed collagenCell surface interactions at the vascular wallFCGR3A-mediated phagocytosisFCGR3A-mediated IL10 synthesis
MECANISMO DE DOENÇA

Autoinflammatory disease, systemic, with vasculitis

An autosomal dominant disorder characterized by systemic autoinflammation manifesting in the first hours of life with diffuse purpuric skin lesions, fever, hepatosplenomegaly, and increased C-reactive protein. Additional clinical features include periorbital edema, conjunctivitis, urticaria, atopic dermatitis, abdominal pain, and arthralgia. Laboratory studies may show leukocytosis, thrombocytopenia, and autoantibodies.

EXPRESSÃO TECIDUAL(Ubíquo)
Sangue
128.4 TPM
Linfócitos
119.3 TPM
Baço
74.3 TPM
Pulmão
34.0 TPM
Intestino delgado
16.6 TPM
OUTRAS DOENÇAS (1)
autoinflammatory disease, systemic, with vasculitis
HGNC:HGNC:6735UniProt:P07948
NLRP12NACHT, LRR and PYD domains-containing protein 12Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (1)
SARS-CoV-2 activates/modulates innate and adaptive immune responses
MECANISMO DE DOENÇA

Familial cold autoinflammatory syndrome 2

A rare autosomal dominant systemic inflammatory disease characterized by recurrent episodes of maculopapular rash associated with arthralgias, myalgias, fever and chills, swelling of the extremities, and conjunctivitis after generalized exposure to cold.

EXPRESSÃO TECIDUAL(Tecido-específico)
Sangue
34.0 TPM
Baço
7.7 TPM
Pulmão
2.6 TPM
Adipose Visceral Omentum
0.5 TPM
Tecido adiposo
0.5 TPM
OUTRAS DOENÇAS (1)
familial cold autoinflammatory syndrome 2
HGNC:22938UniProt:P59046
SH3BP2SH3 domain-binding protein 2Disease-causing germline mutation(s) inTolerante
MECANISMO DE DOENÇA

Cherubism

An autosomal dominant syndrome characterized by excessive bone degradation of the upper and lower jaws, which often begins around three years of age. It is followed by development of fibrous tissue masses, which causes a characteristic facial swelling.

EXPRESSÃO TECIDUAL(Ubíquo)
Baço
67.6 TPM
Skin Not Sun Exposed Suprapubic
58.0 TPM
Skin Sun Exposed Lower leg
55.9 TPM
Sangue
53.0 TPM
Pulmão
39.0 TPM
OUTRAS DOENÇAS (1)
cherubism
HGNC:10825UniProt:P78314
RNASEH2ARibonuclease H2 subunit ADisease-causing germline mutation(s) inTolerante
MECANISMO DE DOENÇA

Aicardi-Goutieres syndrome 4

A form of Aicardi-Goutieres syndrome, a genetically heterogeneous disease characterized by cerebral atrophy, leukoencephalopathy, intracranial calcifications, chronic cerebrospinal fluid (CSF) lymphocytosis, increased CSF alpha-interferon, and negative serologic investigations for common prenatal infection. Clinical features as thrombocytopenia, hepatosplenomegaly and elevated hepatic transaminases along with intermittent fever may erroneously suggest an infective process. Severe neurological dysfunctions manifest in infancy as progressive microcephaly, spasticity, dystonic posturing and profound psychomotor retardation. Death often occurs in early childhood.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
81.3 TPM
Testículo
49.8 TPM
Fibroblastos
44.5 TPM
Esôfago - Mucosa
24.3 TPM
Cervix Ectocervix
23.4 TPM
OUTRAS DOENÇAS (2)
Aicardi-Goutieres syndrome 4Aicardi-Goutieres syndrome
HGNC:18518UniProt:O75792
PSMB4Proteasome subunit beta type-4Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (10)
Vif-mediated degradation of APOBEC3GVpu mediated degradation of CD4Degradation of GLI2 by the proteasomeDegradation of GLI1 by the proteasomeHedgehog 'on' state
MECANISMO DE DOENÇA

Proteasome-associated autoinflammatory syndrome 3

An autoinflammatory disorder characterized by onset in early infancy and recurrent fever, nodular dermatitis, myositis, panniculitis-induced lipodystrophy, lymphadenopathy, and immune dysregulation. Variable accompanying features may include joint contractures, hepatosplenomegaly, anemia, thrombocytopenia, recurrent infections, autoantibodies, and hypergammaglobulinemia. Some patients may have intracranial calcifications. PRAAS3 inheritance is autosomal recessive or digenic.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
509.3 TPM
Fibroblastos
468.9 TPM
Útero
432.8 TPM
Aorta
427.2 TPM
Cervix Ectocervix
417.9 TPM
OUTRAS DOENÇAS (1)
proteasome-associated autoinflammatory syndrome 3
HGNC:HGNC:9541UniProt:P28070
LSM11U7 snRNA-associated Sm-like protein LSm11Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (3)
SLBP independent Processing of Histone Pre-mRNAsRNA Polymerase II Transcription TerminationSLBP Dependent Processing of Replication-Dependent Histone Pre-mRNAs
MECANISMO DE DOENÇA

Aicardi-Goutieres syndrome 8

A form of Aicardi-Goutieres syndrome, a genetically heterogeneous disease characterized by cerebral atrophy, leukoencephalopathy, intracranial calcifications, chronic cerebrospinal fluid (CSF) lymphocytosis, increased CSF alpha-interferon, and negative serologic investigations for common prenatal infection. Clinical features as thrombocytopenia, hepatosplenomegaly and elevated hepatic transaminases along with intermittent fever may erroneously suggest an infective process. Severe neurological dysfunctions manifest in infancy as progressive microcephaly, spasticity, dystonic posturing and profound psychomotor retardation. Death often occurs in early childhood. AGS8 inheritance is autosomal recessive.

EXPRESSÃO TECIDUAL(Ubíquo)
Esôfago - Muscular
19.5 TPM
Bladder
17.4 TPM
Cérebro - Hemisfério cerebelar
16.2 TPM
Cerebelo
12.9 TPM
Útero
12.7 TPM
OUTRAS DOENÇAS (2)
Aicardi-Goutieres syndrome 8Aicardi-Goutieres syndrome
HGNC:30860UniProt:P83369
TREX1Three-prime repair exonuclease 1Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (2)
IRF3-mediated induction of type I IFNRegulation by TREX1
MECANISMO DE DOENÇA

Aicardi-Goutieres syndrome 1

A form of Aicardi-Goutieres syndrome, a genetically heterogeneous disease characterized by cerebral atrophy, leukoencephalopathy, intracranial calcifications, chronic cerebrospinal fluid (CSF) lymphocytosis, increased CSF alpha-interferon, and negative serologic investigations for common prenatal infection. Clinical features as thrombocytopenia, hepatosplenomegaly and elevated hepatic transaminases along with intermittent fever may erroneously suggest an infective process. Severe neurological dysfunctions manifest in infancy as progressive microcephaly, spasticity, dystonic posturing and profound psychomotor retardation. Death often occurs in early childhood.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
80.6 TPM
Baço
45.2 TPM
Skin Sun Exposed Lower leg
42.6 TPM
Skin Not Sun Exposed Suprapubic
42.3 TPM
Pituitária
42.0 TPM
OUTRAS DOENÇAS (6)
retinal vasculopathy with cerebral leukoencephalopathy and systemic manifestationschilblain lupus 1Aicardi-Goutieres syndrome 1Aicardi-Goutieres syndrome
HGNC:12269UniProt:Q9NSU2
POLA1DNA polymerase alpha catalytic subunitDisease-causing germline mutation(s) inAltamente restrito
VIAS BIOLÓGICAS (10)
Polymerase switching on the C-strand of the telomereTelomere C-strand synthesis initiationPolymerase switchingRemoval of the Flap IntermediateProcessive synthesis on the lagging strand
MECANISMO DE DOENÇA

Pigmentary disorder, reticulate, with systemic manifestations, X-linked

An X-linked recessive disorder characterized by recurrent infections and sterile inflammation in various organs. Diffuse skin hyperpigmentation with a distinctive reticulate pattern is universally evident by early childhood. This is later followed in many patients by hypohidrosis, corneal inflammation and scarring, enterocolitis that resembles inflammatory bowel disease, and recurrent urethral strictures. Melanin and amyloid deposition is present in the dermis. Affected males also have a characteristic facies with frontally upswept hair and flared eyebrows. Female carriers have only restricted pigmentary changes along Blaschko's lines.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
21.1 TPM
Nervo tibial
11.8 TPM
Ovário
9.4 TPM
Cervix Endocervix
9.2 TPM
Útero
8.9 TPM
OUTRAS DOENÇAS (2)
X-linked intellectual disability, van Esch typeX-linked reticulate pigmentary disorder
HGNC:9173UniProt:P09884
SKIC3Superkiller complex protein 3Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (1)
mRNA decay by 3' to 5' exoribonuclease
MECANISMO DE DOENÇA

Trichohepatoenteric syndrome 1

A syndrome characterized by intrauterine growth retardation, severe diarrhea in infancy requiring total parenteral nutrition, facial dysmorphism, immunodeficiency, and hair abnormalities, mostly trichorrhexis nodosa. Hepatic involvement contributes to the poor prognosis of affected patients.

OUTRAS DOENÇAS (2)
trichohepatoenteric syndrome 1trichohepatoenteric syndrome
HGNC:23639UniProt:Q6PGP7
ACP5Tartrate-resistant acid phosphatase type 5Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (1)
Vitamin B2 (riboflavin) metabolism
MECANISMO DE DOENÇA

Spondyloenchondrodysplasia with immune dysregulation

A disease characterized by vertebral and metaphyseal dysplasia, spasticity with cerebral calcifications, and strong predisposition to autoimmune diseases. The skeletal dysplasia is characterized by radiolucent and irregular spondylar and metaphyseal lesions that represent islands of chondroid tissue within bone.

OUTRAS DOENÇAS (1)
Spondyloenchondrodysplasia with immune dysregulation
HGNC:124UniProt:P13686
SKIC2Superkiller complex protein 2Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (2)
mRNA decay by 3' to 5' exoribonucleaseAssociation of TriC/CCT with target proteins during biosynthesis
MECANISMO DE DOENÇA

Trichohepatoenteric syndrome 2

A syndrome characterized by intrauterine growth retardation, severe diarrhea in infancy requiring total parenteral nutrition, facial dysmorphism, immunodeficiency, and hair abnormalities, mostly trichorrhexis nodosa. Hepatic involvement contributes to the poor prognosis of affected patients.

OUTRAS DOENÇAS (2)
trichohepatoenteric syndrome 2trichohepatoenteric syndrome
HGNC:10898UniProt:Q15477
USP18Ubl carboxyl-terminal hydrolase 18Disease-causing germline mutation(s) inAltamente restrito
VIAS BIOLÓGICAS (5)
Regulation of NF-kappa B signalingTAK1-dependent IKK and NF-kappa-B activation ISG15 antiviral mechanismRegulation of IFNA/IFNB signalingUb-specific processing proteases
MECANISMO DE DOENÇA

Pseudo-TORCH syndrome 2

An autosomal recessive multisystem disorder characterized by antenatal onset of intracranial hemorrhage, calcification, brain malformations, liver dysfunction, and often thrombocytopenia. Affected individuals tend to have respiratory insufficiency and seizures, and die in infancy. The phenotype resembles the sequelae of intrauterine infection, but there is no evidence of an infectious agent.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
45.3 TPM
Tecido adiposo
8.7 TPM
Nervo tibial
8.2 TPM
Tireoide
6.8 TPM
Adipose Visceral Omentum
6.7 TPM
OUTRAS DOENÇAS (1)
pseudo-TORCH syndrome 2
HGNC:12616UniProt:Q9UMW8
PSMB10Proteasome subunit beta type-10Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (4)
Antigen processing: Ub, ATP-independent proteasomal degradationCross-presentation of soluble exogenous antigens (endosomes)ER-Phagosome pathwayProteasome assembly
MECANISMO DE DOENÇA

Proteasome-associated autoinflammatory syndrome 5

An autosomal recessive, autoinflammatory disorder characterized by recurrent, polymorphic disseminated cutaneous rash with annular lesions, non-specific lymphocytic infiltration in the skin, fever, failure to thrive, and persistent hepatosplenomegaly. Disease onset is in early infancy.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
302.5 TPM
Baço
186.4 TPM
Sangue
148.5 TPM
Intestino delgado
126.3 TPM
Pulmão
94.2 TPM
OUTRAS DOENÇAS (3)
immunodeficiency 121 with autoinflammationproteasome-associated autoinflammatory syndrome 5Omenn syndrome
HGNC:9538UniProt:P40306
UBA1Ubiquitin-like modifier-activating enzyme 1Disease-causing germline mutation(s) inAltamente restrito
VIAS BIOLÓGICAS (3)
Antigen processing: Ubiquitination & Proteasome degradationDengue Virus Attachment and EntrySynthesis of active ubiquitin: roles of E1 and E2 enzymes
MECANISMO DE DOENÇA

Spinal muscular atrophy X-linked 2

A lethal infantile form of spinal muscular atrophy, a neuromuscular disorder characterized by degeneration of the anterior horn cells of the spinal cord, leading to symmetrical muscle weakness and atrophy. Clinical features include hypotonia, areflexia, and multiple congenital contractures.

EXPRESSÃO TECIDUAL(Ubíquo)
Tireoide
191.7 TPM
Útero
173.1 TPM
Fibroblastos
172.9 TPM
Cervix Endocervix
161.0 TPM
Linfócitos
156.0 TPM
OUTRAS DOENÇAS (2)
VEXAS syndromeinfantile-onset X-linked spinal muscular atrophy
HGNC:12469UniProt:P22314
IFIH1Interferon-induced helicase C domain-containing protein 1Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (10)
DDX58/IFIH1-mediated induction of interferon-alpha/betaNegative regulators of DDX58/IFIH1 signalingTRAF3-dependent IRF activation pathwaySARS-CoV-2 activates/modulates innate and adaptive immune responsesOvarian tumor domain proteases
MECANISMO DE DOENÇA

Type 1 diabetes mellitus 19

A multifactorial disorder of glucose homeostasis that is characterized by susceptibility to ketoacidosis in the absence of insulin therapy. Clinical features are polydipsia, polyphagia and polyuria which result from hyperglycemia-induced osmotic diuresis and secondary thirst. These derangements result in long-term complications that affect the eyes, kidneys, nerves, and blood vessels.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
87.0 TPM
Baço
15.9 TPM
Pulmão
13.4 TPM
Nervo tibial
11.7 TPM
Útero
11.1 TPM
OUTRAS DOENÇAS (5)
immunodeficiency 95Singleton-Merten syndrome 1Aicardi-Goutieres syndrome 7Singleton-Merten dysplasia
HGNC:18873UniProt:Q9BYX4
STING1Stimulator of interferon genes proteinDisease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (5)
SARS-CoV-2 activates/modulates innate and adaptive immune responsesSTING mediated induction of host immune responsesRegulation of innate immune responses to cytosolic DNASARS-CoV-1 activates/modulates innate immune responsesDengue virus activates/modulates innate and adaptive immune responses
MECANISMO DE DOENÇA

STING-associated vasculopathy, infantile-onset

An autoinflammatory disease characterized by early-onset systemic inflammation and cutaneous vasculopathy, resulting in severe skin lesions. Violaceous, scaling lesions of fingers, toes, nose, cheeks and ears progress to acral necrosis in most of the patients. Some patients have severe interstitial lung disease.

OUTRAS DOENÇAS (2)
STING-associated vasculopathy with onset in infancyfamilial chilblain lupus
HGNC:27962UniProt:Q86WV6
ADARDouble-stranded RNA-specific adenosine deaminaseDisease-causing germline mutation(s) inAltamente restrito
VIAS BIOLÓGICAS (3)
Interferon alpha/beta signalingFormation of editosomes by ADAR proteinsC6 deamination of adenosine
MECANISMO DE DOENÇA

Dyschromatosis symmetrica hereditaria

An autosomal dominant pigmentary genodermatosis characterized by a mixture of hyperpigmented and hypopigmented macules distributed on the face and the dorsal parts of the hands and feet, that appear in infancy or early childhood.

OUTRAS DOENÇAS (4)
dyschromatosis symmetrica hereditariaAicardi-Goutieres syndrome 6Aicardi-Goutieres syndromefamilial infantile bilateral striatal necrosis
HGNC:225UniProt:P55265
IL1RNInterleukin-1 receptor antagonist proteinDisease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (2)
Interleukin-1 signalingInterleukin-10 signaling
MECANISMO DE DOENÇA

Microvascular complications of diabetes 4

Pathological conditions that develop in numerous tissues and organs as a consequence of diabetes mellitus. They include diabetic retinopathy, diabetic nephropathy leading to end-stage renal disease, and diabetic neuropathy. Diabetic retinopathy remains the major cause of new-onset blindness among diabetic adults. It is characterized by vascular permeability and increased tissue ischemia and angiogenesis.

EXPRESSÃO TECIDUAL(Tecido-específico)
Esôfago - Mucosa
1638.9 TPM
Vagina
322.5 TPM
Skin Not Sun Exposed Suprapubic
130.8 TPM
Skin Sun Exposed Lower leg
124.0 TPM
Sangue
85.4 TPM
OUTRAS DOENÇAS (3)
sterile multifocal osteomyelitis with periostitis and pustulosismicrovascular complications of diabetes, susceptibility to, 4gastric cancer
HGNC:6000UniProt:P18510
MEFVPyrinDisease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (2)
Purinergic signaling in leishmaniasis infectionThe NLRP3 inflammasome
MECANISMO DE DOENÇA

Familial Mediterranean fever, autosomal recessive

A hereditary periodic fever syndrome characterized by recurrent episodic fever, serosal inflammation and pain in the abdomen, chest or joints. It is frequently complicated by reactive amyloidosis, which leads to renal failure and can be prophylactically treated with colchicine.

EXPRESSÃO TECIDUAL(Tecido-específico)
Sangue
81.4 TPM
Baço
19.9 TPM
Pulmão
10.6 TPM
Testículo
4.1 TPM
Adipose Visceral Omentum
1.9 TPM
OUTRAS DOENÇAS (6)
autosomal recessive familial Mediterranean feversweet syndromefamilial Mediterranean fever, autosomal dominantBehcet disease
HGNC:6998UniProt:O15553
PSTPIP1Proline-serine-threonine phosphatase-interacting protein 1Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (2)
Purinergic signaling in leishmaniasis infectionThe NLRP3 inflammasome
MECANISMO DE DOENÇA

Pyogenic sterile arthritis, pyoderma gangrenosum, and acne

A rare autosomal dominant autoinflammatory disease that typically presents with recurrent sterile, erosive arthritis in childhood, occurring spontaneously or after minor trauma, occasionally resulting in significant joint destruction. By puberty, joint symptoms tend to subside and cutaneous symptoms increase. Cutaneous manifestations include pathergy, frequently with abscesses at the sites of injections, severe cystic acne, and recurrent nonhealing sterile ulcers, often diagnosed as pyoderma gangrenosum.

EXPRESSÃO TECIDUAL(Ubíquo)
Sangue
99.1 TPM
Baço
47.7 TPM
Pulmão
14.8 TPM
Aorta
11.8 TPM
Linfócitos
7.5 TPM
OUTRAS DOENÇAS (2)
pyogenic arthritis-pyoderma gangrenosum-acne syndromehyperzincemia with functional zinc depletion
HGNC:9580UniProt:O43586
PSMB9Proteasome subunit beta type-9Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (4)
Antigen processing: Ub, ATP-independent proteasomal degradationCross-presentation of soluble exogenous antigens (endosomes)ER-Phagosome pathwayProteasome assembly
MECANISMO DE DOENÇA

Proteasome-associated autoinflammatory syndrome 3

An autoinflammatory disorder characterized by onset in early infancy and recurrent fever, nodular dermatitis, myositis, panniculitis-induced lipodystrophy, lymphadenopathy, and immune dysregulation. Variable accompanying features may include joint contractures, hepatosplenomegaly, anemia, thrombocytopenia, recurrent infections, autoantibodies, and hypergammaglobulinemia. Some patients may have intracranial calcifications. PRAAS3 inheritance is autosomal recessive or digenic.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
537.0 TPM
Baço
236.5 TPM
Sangue
182.6 TPM
Intestino delgado
116.3 TPM
Pulmão
105.2 TPM
OUTRAS DOENÇAS (1)
proteasome-associated autoinflammatory syndrome 6
HGNC:HGNC:9546UniProt:P28065
NLRP3NACHT, LRR and PYD domains-containing protein 3Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (6)
SARS-CoV-2 activates/modulates innate and adaptive immune responsesPurinergic signaling in leishmaniasis infectionThe NLRP3 inflammasomeCytoprotection by HMOX1SARS-CoV-1 activates/modulates innate immune responses
MECANISMO DE DOENÇA

Familial cold autoinflammatory syndrome 1

A rare autosomal dominant systemic inflammatory disease characterized by recurrent episodes of maculopapular rash associated with arthralgias, myalgias, fever and chills, swelling of the extremities, and conjunctivitis after generalized exposure to cold. Rarely, some patients may also develop late-onset renal amyloidosis.

EXPRESSÃO TECIDUAL(Tecido-específico)
Sangue
23.3 TPM
Pulmão
6.7 TPM
Baço
6.5 TPM
Adipose Visceral Omentum
3.4 TPM
Nervo tibial
2.9 TPM
OUTRAS DOENÇAS (6)
Muckle-Wells syndromekeratitis fugax hereditariafamilial cold autoinflammatory syndrome 1CINCA syndrome
HGNC:16400UniProt:Q96P20
TNFRSF1ATumor necrosis factor receptor superfamily member 1ADisease-causing germline mutation(s) inAltamente restrito
VIAS BIOLÓGICAS (6)
TNFR1-induced proapoptotic signalingTNF signalingTNFR1-induced NF-kappa-B signaling pathwayRegulation of TNFR1 signalingTNFR1-mediated ceramide production
MECANISMO DE DOENÇA

Periodic fever, familial, autosomal dominant

A hereditary periodic fever syndrome characterized by recurrent fever, abdominal pain, localized tender skin lesions and myalgia. Reactive amyloidosis is the main complication and occurs in 25% of cases.

EXPRESSÃO TECIDUAL(Ubíquo)
Adipose Visceral Omentum
162.0 TPM
Fallopian Tube
161.6 TPM
Pulmão
157.1 TPM
Útero
154.1 TPM
Nervo tibial
151.6 TPM
OUTRAS DOENÇAS (3)
TNF receptor 1-associated periodic fever syndromeintermittent hydrarthrosismultiple sclerosis, susceptibility to, 5
HGNC:11916UniProt:P19438
PLCG21-phosphatidylinositol 4,5-bisphosphate phosphodiesterase gamma-2Disease-causing germline mutation(s) inAltamente restrito
VIAS BIOLÓGICAS (2)
GPVI-mediated activation cascadeSynthesis of IP3 and IP4 in the cytosol
MECANISMO DE DOENÇA

Familial cold autoinflammatory syndrome 3

An autosomal dominant immune disorder characterized by the development of cutaneous urticaria, erythema, and pruritis in response to cold exposure. Affected individuals have variable additional immunologic defects, including antibody deficiency, decreased numbers of B-cells, defective B-cells, increased susceptibility to infection, and increased risk of autoimmune disorders.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
95.9 TPM
Baço
37.4 TPM
Sangue
25.6 TPM
Rim - Córtex
14.7 TPM
Intestino delgado
13.6 TPM
OUTRAS DOENÇAS (2)
familial cold autoinflammatory syndrome 3autoinflammation-PLCG2-associated antibody deficiency-immune dysregulation
HGNC:9066UniProt:P16885
ADA2Adenosine deaminase 2Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (2)
Neutrophil degranulationSurfactant metabolism
MECANISMO DE DOENÇA

Vasculitis, autoinflammation, immunodeficiency, and hematologic defects syndrome

An autosomal recessive, systemic necrotizing vasculitis that affects medium and small arteries. The ensuing tissue ischemia can affect any organ, including the skin, musculoskeletal system, kidneys, gastrointestinal tract, and the cardiovascular and nervous systems. Organ involvement and disease severity are highly variable. Clinical features include recurrent ischemic stroke affecting the small vessels of the brain and resulting in neurologic dysfunction, recurrent fever, myalgias, livedoid rash, gastrointestinal pain and hepatosplenomegaly.

OUTRAS DOENÇAS (3)
vasculitis due to ADA2 deficiencySneddon syndromeDiamond-Blackfan anemia
HGNC:1839UniProt:Q9NZK5
RIGIAntiviral innate immune response receptor RIG-IDisease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (10)
SARS-CoV-2 activates/modulates innate and adaptive immune responsesDDX58/IFIH1-mediated induction of interferon-alpha/betaModulation of host responses by IFN-stimulated genesNegative regulators of DDX58/IFIH1 signalingUb-specific processing proteases
MECANISMO DE DOENÇA

Singleton-Merten syndrome 2

A form of Singleton-Merten syndrome, an autosomal dominant disorder characterized by marked aortic calcification, dental anomalies, osteopenia, acro-osteolysis, and to a lesser extent glaucoma, psoriasis, muscle weakness, and joint laxity. Additional clinical manifestations include particular facial characteristics and abnormal joint and muscle ligaments. SGMRT2 is an atypical form characterized by variable expression of glaucoma, aortic calcification, and skeletal abnormalities, without dental anomalies.

OUTRAS DOENÇAS (2)
Singleton-Merten syndrome 2Singleton-Merten dysplasia
HGNC:19102UniProt:O95786
IL36RNInterleukin-36 receptor antagonist proteinDisease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (1)
Interleukin-36 pathway
MECANISMO DE DOENÇA

Psoriasis 14, pustular

A life-threatening disease defined by repeated flares of sudden onset consisting of diffuse erythematous skin eruption characterized by rapid coverage with pustules, high-grade fever, asthenia, marked leukocytosis, and elevated serum levels of C-reactive protein.

VIAS REACTOME (1)
EXPRESSÃO TECIDUAL(Tecido-específico)
Skin Sun Exposed Lower leg
82.0 TPM
Skin Not Sun Exposed Suprapubic
49.1 TPM
Esôfago - Mucosa
21.0 TPM
Vagina
3.8 TPM
Testículo
0.2 TPM
OUTRAS DOENÇAS (3)
psoriasis 14, pustulargeneralized pustular psoriasispustulosis palmaris et plantaris
HGNC:15561UniProt:Q9UBH0
POMPProteasome maturation proteinDisease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (1)
Proteasome assembly
MECANISMO DE DOENÇA

Keratosis linearis with ichthyosis congenita and sclerosing keratoderma

A keratinizing disorder characterized by ichthyosis, palmoplantar keratoderma with constricting bands around fingers, flexural deformities of fingers and keratotic papules in a linear distribution on the flexural side of large joints. Histological examination of the skin of affected individuals shows hypertrophy and hyperplasia of the spinous, granular and horny epidermal layer.

VIAS REACTOME (1)
EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
254.5 TPM
Fibroblastos
199.6 TPM
Testículo
152.9 TPM
Artéria tibial
132.7 TPM
Artéria coronária
128.2 TPM
OUTRAS DOENÇAS (2)
proteasome-associated autoinflammatory syndrome 2keratosis linearis-ichthyosis congenita-sclerosing keratoderma syndrome
HGNC:20330UniProt:Q9Y244
PSMB8Proteasome subunit beta type-8Disease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (4)
Antigen processing: Ub, ATP-independent proteasomal degradationCross-presentation of soluble exogenous antigens (endosomes)ER-Phagosome pathwayProteasome assembly
MECANISMO DE DOENÇA

Proteasome-associated autoinflammatory syndrome 1

An autosomal recessive autoinflammatory disorder characterized by early childhood onset of recurrent fever, joint stiffness and severe contractures of the hands and feet, and erythematous skin lesions with subsequent development of lipodystrophy and laboratory evidence of immune dysregulation. Accompanying features may include muscle weakness and atrophy, hepatosplenomegaly, and microcytic anemia.

EXPRESSÃO TECIDUAL(Ubíquo)
Linfócitos
416.8 TPM
Baço
277.5 TPM
Pulmão
166.8 TPM
Intestino delgado
160.6 TPM
Sangue
155.0 TPM
OUTRAS DOENÇAS (2)
proteasome-associated autoinflammatory syndrome 1proteosome-associated autoinflammatory syndrome
HGNC:9545UniProt:P28062
EGFREpidermal growth factor receptorDisease-causing germline mutation(s) inRestrito
VIAS BIOLÓGICAS (10)
Signaling by EGFRInhibition of Signaling by Overexpressed EGFRSignaling by ERBB2Signaling by ERBB4Signaling by ERBB2 TMD/JMD mutants
MECANISMO DE DOENÇA

Lung cancer

A common malignancy affecting tissues of the lung. The most common form of lung cancer is non-small cell lung cancer (NSCLC) that can be divided into 3 major histologic subtypes: squamous cell carcinoma, adenocarcinoma, and large cell lung cancer. NSCLC is often diagnosed at an advanced stage and has a poor prognosis.

EXPRESSÃO TECIDUAL(Ubíquo)
Skin Sun Exposed Lower leg
78.3 TPM
Skin Not Sun Exposed Suprapubic
75.9 TPM
Fibroblastos
60.6 TPM
Nervo tibial
43.1 TPM
Vagina
40.6 TPM
OUTRAS DOENÇAS (4)
inflammatory skin and bowel disease, neonatal, 2lung cancergliosarcomagiant cell glioblastoma
HGNC:3236UniProt:P00533
MVKMevalonate kinaseDisease-causing germline mutation(s) inTolerante
VIAS BIOLÓGICAS (2)
Lanosterol biosynthesisActivation of gene expression by SREBF (SREBP)
MECANISMO DE DOENÇA

Mevalonic aciduria

Accumulation of mevalonic acid which causes a variety of symptoms such as psychomotor retardation, dysmorphic features, cataracts, hepatosplenomegaly, lymphadenopathy, anemia, hypotonia, myopathy, and ataxia.

EXPRESSÃO TECIDUAL(Ubíquo)
Testículo
41.6 TPM
Esôfago - Mucosa
40.7 TPM
Skin Sun Exposed Lower leg
34.1 TPM
Rim - Medula
33.4 TPM
Skin Not Sun Exposed Suprapubic
32.7 TPM
OUTRAS DOENÇAS (5)
mevalonic aciduriaporokeratosis 3, disseminated superficial actinic typehyperimmunoglobulinemia D with periodic feverporokeratosis of Mibelli
HGNC:7530UniProt:Q03426

Medicamentos e terapias

RILONACEPTPhase 4

Mecanismo: Interleukin-1 beta inhibitor

CANAKINUMABPhase 4

Mecanismo: Interleukin-1 beta inhibitor

COLCHICINEPhase 4

Mecanismo: Tubulin inhibitor

PREDNISOLONEPhase 4

Mecanismo: Glucocorticoid receptor agonist

DEXAMETHASONEPhase 4

Mecanismo: Glucocorticoid receptor agonist

ANAKINRAPhase 4

Mecanismo: Interleukin-1 receptor antagonist

Ver mais no OpenTargets

Variantes genéticas (ClinVar)

343 variantes patogênicas registradas no ClinVar.

🧬 LPIN2: GRCh38/hg38 18p11.32-11.21(chr18:136227-15181209)x4 ()
🧬 LPIN2: NM_001375808.2(LPIN2):c.441del (p.Phe147fs) ()
🧬 LPIN2: NM_001375808.2(LPIN2):c.973_976del (p.Val325fs) ()
🧬 LPIN2: NM_001375808.2(LPIN2):c.1168+1G>C ()
🧬 LPIN2: NM_001375808.2(LPIN2):c.1794-2_1821del ()
Ver todas no ClinVar

Classificação de variantes (ClinVar)

Distribuição de 5.624 variantes classificadas pelo ClinVar.

2531
3093
VUS (45.0%)
Benigna (55.0%)
VARIANTES MAIS SIGNIFICATIVAS
PLCG2: NM_002661.5(PLCG2):c.914A>G (p.Lys305Arg) [Uncertain significance]
PLCG2: NM_002661.5(PLCG2):c.2458C>G (p.Pro820Ala) [Uncertain significance]
NLRP12: NM_144687.4(NLRP12):c.689T>C (p.Val230Ala) [Uncertain significance]
NLRP12: NM_144687.4(NLRP12):c.615G>C (p.Glu205Asp) [Uncertain significance]
PLCG2: NM_002661.5(PLCG2):c.164G>A (p.Ser55Asn) [Uncertain significance]

Vias biológicas (Reactome)

234 vias biológicas associadas aos genes desta condição.

Synthesis of PC Synthesis of PE Depolymerization of the Nuclear Lamina Triglyceride biosynthesis Intrinsic Pathway of Fibrin Clot Formation Defective factor XII causes hereditary angioedema Defective SERPING1 causes hereditary angioedema Downstream TCR signaling Phosphorylation of CD3 and TCR zeta chains Translocation of ZAP-70 to Immunological synapse Generation of second messenger molecules MHC class II antigen presentation Co-inhibition by PD-1 Interferon gamma signaling CDC42 GTPase cycle RAC1 GTPase cycle Factors involved in megakaryocyte development and platelet production GPVI-mediated activation cascade EGFR downregulation Regulation of actin dynamics for phagocytic cup formation CD28 dependent Vav1 pathway EPHB-mediated forward signaling DCC mediated attractive signaling Inactivation of CDC42 and RAC1 VEGFA-VEGFR2 Pathway Myogenesis RHO GTPases activate KTN1 RHO GTPases activate IQGAPs RHO GTPases activate PAKs RHO GTPases Activate WASPs and WAVEs RHO GTPases Activate Formins MAPK6/MAPK4 signaling Gene and protein expression by JAK-STAT signaling after Interleukin-12 stimulation G beta:gamma signalling through CDC42 RAC2 GTPase cycle RHOQ GTPase cycle RHOG GTPase cycle RHOJ GTPase cycle RHOU GTPase cycle RAC3 GTPase cycle RHOV GTPase cycle FCGR3A-mediated phagocytosis TNFR1-induced proapoptotic signaling Regulation of TNFR1 signaling TNFR1-induced NF-kappa-B signaling pathway Antigen processing: Ubiquitination & Proteasome degradation Nuclear signaling by ERBB4 Degradation of the extracellular matrix Regulated proteolysis of p75NTR NRIF signals cell death from the nucleus Activated NOTCH1 Transmits Signal to the Nucleus Constitutive Signaling by NOTCH1 PEST Domain Mutants Constitutive Signaling by NOTCH1 HD+PEST Domain Mutants NOTCH2 Activation and Transmission of Signal to the Nucleus EPH-ephrin mediated repulsion of cells Neutrophil degranulation NOTCH3 Activation and Transmission of Signal to the Nucleus NOTCH4 Activation and Transmission of Signal to the Nucleus Noncanonical activation of NOTCH3 Amyloid fiber formation TGFBR3 PTM regulation Butyrophilin (BTN) family interactions Regulation of KIT signaling Signaling by ALK PECAM1 interactions Signal regulatory protein family interactions Platelet sensitization by LDL Interleukin-3, Interleukin-5 and GM-CSF signaling CD22 mediated BCR regulation Regulation of IFNG signaling Interleukin-37 signaling Interferon alpha/beta signaling Interleukin receptor SHC signaling Regulation of IFNA/IFNB signaling SARS-CoV-2 activates/modulates innate and adaptive immune responses Nuclear events stimulated by ALK signaling in cancer Growth hormone receptor signaling Antigen activates B Cell Receptor (BCR) leading to generation of second messengers Co-inhibition by BTLA Nef mediated downregulation of MHC class I complex cell surface expression Lysosome Vesicle Biogenesis Golgi Associated Vesicle Biogenesis Signaling by BMP Transcriptional regulation of testis differentiation Transcriptional regulation of granulopoiesis Nucleotide catabolism TP53 Regulates Transcription of Caspase Activators and Caspases The IPAF inflammasome Synthesis of active ubiquitin: roles of E1 and E2 enzymes Proteasome assembly NOD1/2 Signaling Pathway TAK1-dependent IKK and NF-kappa-B activation activated TAK1 mediates p38 MAPK activation JNK (c-Jun kinases) phosphorylation and activation mediated by activated human TAK1 Ovarian tumor domain proteases Interleukin-1 signaling Negative regulators of DDX58/IFIH1 signaling Signaling by SCF-KIT Cell surface interactions at the vascular wall FCGR activation Fc epsilon receptor (FCERI) signaling EPH-Ephrin signaling Role of LAT2/NTAL/LAB on calcium mobilization FCERI mediated MAPK activation FCERI mediated Ca+2 mobilization FCERI mediated NF-kB activation Co-stimulation by CD28 Co-inhibition by CTLA4 EPHA-mediated growth cone collapse Dectin-2 family CD209 (DC-SIGN) signaling Cyclin D associated events in G1 Platelet Adhesion to exposed collagen Signaling by Erythropoietin Erythropoietin activates Phosphoinositide-3-kinase (PI3K) Erythropoietin activates Phospholipase C gamma (PLCG) Erythropoietin activates STAT5 Erythropoietin activates RAS Regulation of signaling by CBL FCGR3A-mediated IL10 synthesis Activation of NF-kappaB in B cells Oxygen-dependent proline hydroxylation of Hypoxia-inducible Factor Alpha ER-Phagosome pathway Cross-presentation of soluble exogenous antigens (endosomes) Autodegradation of Cdh1 by Cdh1:APC/C SCF-beta-TrCP mediated degradation of Emi1 APC/C:Cdc20 mediated degradation of Securin APC/C:Cdh1 mediated degradation of Cdc20 and other APC/C:Cdh1 targeted proteins in late mitosis/early G1 Cdc20:Phospho-APC/C mediated degradation of Cyclin A Vpu mediated degradation of CD4 Vif-mediated degradation of APOBEC3G SCF(Skp2)-mediated degradation of p27/p21 Degradation of beta-catenin by the destruction complex Regulation of activated PAK-2p34 by proteasome mediated degradation Separation of Sister Chromatids Autodegradation of the E3 ubiquitin ligase COP1 Regulation of ornithine decarboxylase (ODC) ABC-family proteins mediated transport AUF1 (hnRNP D0) binds and destabilizes mRNA Asymmetric localization of PCP proteins Degradation of AXIN Degradation of DVL Hedgehog ligand biogenesis Hh mutants are degraded by ERAD Dectin-1 mediated noncanonical NF-kB signaling CLEC7A (Dectin-1) signaling Degradation of GLI1 by the proteasome Degradation of GLI2 by the proteasome SLBP independent Processing of Histone Pre-mRNAs RNA Polymerase II Transcription Termination SLBP Dependent Processing of Replication-Dependent Histone Pre-mRNAs Regulation by TREX1 IRF3-mediated induction of type I IFN Inhibition of replication initiation of damaged DNA by RB1/E2F1 Polymerase switching on the C-strand of the telomere Telomere C-strand synthesis initiation DNA replication initiation Activation of the pre-replicative complex Polymerase switching Removal of the Flap Intermediate Processive synthesis on the lagging strand G1/S-Specific Transcription Defective pyroptosis mRNA decay by 3' to 5' exoribonuclease Vitamin B2 (riboflavin) metabolism Association of TriC/CCT with target proteins during biosynthesis ISG15 antiviral mechanism Ub-specific processing proteases Regulation of NF-kappa B signaling Antigen processing: Ub, ATP-independent proteasomal degradation Dengue Virus Attachment and Entry DDX58/IFIH1-mediated induction of interferon-alpha/beta TRAF3-dependent IRF activation pathway TRAF6 mediated IRF7 activation TRAF6 mediated NF-kB activation NF-kB activation through FADD/RIP-1 pathway mediated by caspase-8 and -10 SARS-CoV-1 activates/modulates innate immune responses Evasion by RSV of host interferon responses Modulation of host responses by IFN-stimulated genes Dengue virus activates/modulates innate and adaptive immune responses STING mediated induction of host immune responses Regulation of innate immune responses to cytosolic DNA STAT6-mediated induction of chemokines C6 deamination of adenosine Formation of editosomes by ADAR proteins PKR-mediated signaling Interleukin-10 signaling The NLRP3 inflammasome Purinergic signaling in leishmaniasis infection Metalloprotease DUBs Cytoprotection by HMOX1 TNFR1-mediated ceramide production TNFs bind their physiological receptors TNF signaling Toll Like Receptor 4 (TLR4) Cascade Synthesis of IP3 and IP4 in the cytosol Role of phospholipids in phagocytosis DAP12 signaling Potential therapeutics for SARS Signaling by CSF1 (M-CSF) in myeloid cells Surfactant metabolism OAS antiviral response RSV-host interactions Interleukin-36 pathway Signaling by ERBB2 Constitutive Signaling by Ligand-Responsive EGFR Cancer Variants Signaling by ERBB4 SHC1 events in ERBB2 signaling PLCG1 events in ERBB2 signaling PIP3 activates AKT signaling Signaling by EGFR GRB2 events in EGFR signaling GAB1 signalosome SHC1 events in EGFR signaling GRB2 events in ERBB2 signaling PI3K events in ERBB2 signaling EGFR interacts with phospholipase C-gamma EGFR Transactivation by Gastrin Constitutive Signaling by Aberrant PI3K in Cancer Signal transduction by L1 Constitutive Signaling by EGFRvIII Inhibition of Signaling by Overexpressed EGFR RAF/MAP kinase cascade ERBB2 Regulates Cell Motility PI5P, PP2A and IER3 Regulate PI3K/AKT Signaling ERBB2 Activates PTK6 Signaling Cargo recognition for clathrin-mediated endocytosis Clathrin-mediated endocytosis PTK6 promotes HIF1A stabilization Downregulation of ERBB2 signaling TFAP2 (AP-2) family regulates transcription of growth factors and their receptors Extra-nuclear estrogen signaling Activation of gene expression by SREBF (SREBP) Lanosterol biosynthesis

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.
✓Aprovado6
3Fase 35
2Fase 23
·Pré-clínico9
Medicamentos catalogadosEnsaios clínicos· 6 medicamentos · 17 ensaios
✓ Aprovados — podem ser usados hoje
RILONACEPTCANAKINUMABCOLCHICINEPREDNISOLONEDEXAMETHASONEANAKINRA
Carregando informações de tratamento...

Onde tratar no SUS

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

🇧🇷 Atendimento SUS — Síndrome autoinflamatório

🗺️

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

Onde estão os ensaios

Com ensaio aberto em 25 países. O ponto verde marca onde há vaga agora.

🟢 Recrutando agora

5 pesquisas recrutando participantes. Converse com seu médico sobre a possibilidade de participar.

Outros ensaios clínicos

17 ensaios clínicos encontrados, 6 ativos.

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

Timeline de publicações
434 papers (10 anos)
#1

Independent mechanisms of inflammation and myeloid bias in VEXAS syndrome.

Nature2026 Jan

Somatically acquired mutations in the E1 ubiquitin-activating enzyme UBA1 within haematopoietic stem and progenitor cells (HSPCs) were recently identified as the cause of the adult-onset autoinflammatory syndrome VEXAS (vacuoles, E1 enzyme, X linked, autoinflammatory, somatic)1. UBA1 mutations in VEXAS lead to clonal expansion within the HSPC and myeloid, but not lymphoid, compartments. Despite its severity and prevalence, the mechanisms by which UBA1 mutations cause multiorgan autoinflammation and haematological disease are unclear. Here we use somatic gene-editing approaches to model VEXAS-associated UBA1 mutations in primary macrophages and HSPCs. Uba1-mutant mouse macrophages exposed to inflammatory stimuli underwent aberrant apoptotic and necroptotic cell death mediated by caspase-8 (CASP8) and RIPK3-MLKL, respectively. Accordingly, in mice challenged with TNF or LPS, the UBA1 inhibitor TAK-243 exacerbated inflammation in a RIPK3-CASP8-dependent manner. By contrast, Uba1 mutation in HSPCs induced an unfolded protein response and myeloid bias independently of RIPK3-CASP8. Mechanistically, aberrant cell death of Uba1-mutant macrophages coincided with a kinetic defect in Lys63/Met1 (that is, linear) polyubiquitylation of inflammatory signalling complexes. Collectively, our results link VEXAS pathogenesis to the pathogenesis of rarer monogenic autoinflammatory syndromes, highlight specific ubiquitin-associated defects stemming from an apical mutation in the ubiquitylation cascade and support therapeutic targeting of the inflammatory cell death axis in VEXAS.

#2

A Rare RIPK3 Variant Enhances Necroptosis and Promotes Inflammation in a Still's Disease-like Autoinflammatory Syndrome.

Arthritis & rheumatology (Hoboken, N.J.)2026 Mar 19

Still's Disease represents a prototypical polygenic systemic autoinflammatory diseases (SAIDs), characterized by recurrent systemic inflammation and dysregulation of innate immunity. Despite extensive clinical characterization, familial clustering Still's disease remains unreported. This study investigates a novel variant underlying a distinct Still's disease-like autoinflammatory syndrome. Whole-exome sequencing was performed in a three-generation family with three affected individuals. Functional analyses were conducted to assess the impact of the RIPK3 p.Q134K variant on kinase activity, necroptosis and inflammatory signaling. Transcriptomic profiling of peripheral blood mononuclear cells (PBMCs) and in vitro cytokine assays were used to characterize inflammatory responses. A heterozygous RIPK3 p.Q134K variant was identified in all affected individuals. Functional analyses demonstrated that this variant exhibits gain-of-function activity, characterized by enhanced RIPK3 kinase activity and increased RIPK3 self-association, which in turn promote MLKL phosphorylation and necroptosis. RNA sequencing of PBMCs from one patient revealed marked upregulation of NF-κB and MAPK signaling pathways and elevated expression of IL-6, and TNF-α, which was effectively suppressed by pharmacological inhibition of RIPK3. This study describes a familial Still's disease-like autoinflammatory syndrome caused by a gain-of-function RIPK3 variant. Our findings establish aberrant RIPK3 activation as a driver of autoinflammation and expand the spectrum of human RIPK3-associated diseases.

#3

NEMO-NDAS: Case Report and Review of the Literature.

Pediatric dermatology2026 Mar 11

NEMO-deleted exon 5 autoinflammatory syndrome (NEMO-NDAS) is the result of a gain-of-function IKBKG pathogenic variant leading to dysregulated NF-κB signaling and systemic inflammation. We present a case of NEMO-NDAS in a 2-year-old female presenting with recurrent fevers, subcutaneous nodules, lymphadenopathy, and splenomegaly. This article highlights the symptoms that aid in diagnosis and how to differentiate it from other autoinflammatory diseases and reviews therapies for NEMO-NDAS.

#4

Marked regression of calcinosis with canakinumab in hyperphosphatemic familial tumoral calcinosis.

JBMR plus2026 Apr

Hyperphosphatemic familial tumoral calcinosis (HFTC) is a rare autosomal recessive disease caused by pathogenic variants in genes involved in phosphate homeostasis, and characterized by elevated serum phosphate levels and the development of ectopic calcifications. Management typically includes phosphate-lowering strategies and, when clinically necessary, surgical debulking of calcifications. Inflammatory flares occur variably and remain poorly understood. We report the case of a 27-yr-old woman with HFTC due to a homozygous GALNT3 mutation, who experienced recurrent painful flares, elevated inflammatory markers, and progressive calcinosis. Based on the hypothesis that HFTC may behave like an autoinflammatory syndrome, the patient was treated with canakinumab, an anti-interleukin-1 antibody, with 7 yr of follow-up to date. The treatment led to rapid and sustained clinical and biological remission, significant regression of calcified lesions without surgical intervention, and demonstrated good safety.

#5

Mice humanized by syntenic replacement with full-length NLRP3 disease-associated variants model the clinical cryopyrinopathy continuum.

JCI insight2026 Mar 09

Next-generation sequencing technologies are increasingly used to diagnose genetic disorders, particularly immunological diseases with broad and overlapping immune dysregulation. Cryopyrin-associated periodic syndromes (CAPS) are caused by gain-of-function mutations in NLRP3 and include 3 autoinflammatory diseases spanning a continuum of severity: familial cold autoinflammatory syndrome (FCAS), Muckle-Wells syndrome (MWS), and neonatal-onset multisystem inflammatory disease (NOMID). Linking NLRP3 variants to protein dysfunction and clinical phenotype remains challenging because of genetic modifiers and environmental factors. We report the generation and phenotyping of 5 mouse lines expressing either the common human NLRP3 allele or 1 of 4 CAPS mutations spanning the disease spectrum from FCAS to NOMID. In these lines, the murine Nlrp3 locus is replaced by syntenic integration of the human NLRP3 locus, yielding 1 line with the common allele and 4 lines each carrying a distinct CAPS mutation. Unlike models in which a human mutation is introduced into the mouse protein, these lines recapitulate the spectrum of disease severity observed in humans. These findings support a model in which evaluation of nonsynonymous mutations in mice is optimized when introduced in the context of the human gene. This suggests that species-specific regulation and/or intramolecular epistasis may impact modeling of disease-associated variants.

Publicações recentes

Ver todas no PubMed

📚 EuropePMC156 artigos no totalmostrando 192

2026

A Rare RIPK3 Variant Enhances Necroptosis and Promotes Inflammation in a Still's Disease-like Autoinflammatory Syndrome.

Arthritis & rheumatology (Hoboken, N.J.)
2026

NEMO-NDAS: Case Report and Review of the Literature.

Pediatric dermatology
2026

Marked regression of calcinosis with canakinumab in hyperphosphatemic familial tumoral calcinosis.

JBMR plus
2026

Mice humanized by syntenic replacement with full-length NLRP3 disease-associated variants model the clinical cryopyrinopathy continuum.

JCI insight
2026

An unusual case of RIPK1-related immunodeficiency: The importance of a timely diagnosis for a novel clinical and therapeutic pattern.

Pediatric allergy and immunology : official publication of the European Society of Pediatric Allergy and Immunology
2026

Clinical features of patients with familial Mediterranean fever over 50 years of age: a single-center experience.

Internal and emergency medicine
2026

Bilateral lung transplantation for severe lung disease caused by a STING1 mutation: A case report.

Transplant immunology
2026

Canakinumab as effective therapy in anakinra-refractory Schnitzler syndrome: a case-based review.

Internal medicine journal
2025

Commentary: Autoimmune/Autoinflammatory Syndrome Induced by Adjuvants (ASIA Syndrome) After Polypropylene Mesh Implantation - Protocol of a Pilot Study for Diagnostics and Treatment.

Journal of abdominal wall surgery : JAWS
2026

Autoinflammatory syndrome associated with a variant of the NLRP1 gene (NAIAD): description of a pediatric case treated with canakinumab.

Medicina clinica
2025

Adult-Onset Still's Disease Presenting with Atypical Kikuchi-Fujimoto Disease-Like Histiocytic Necrotizing Lymphadenitis: A Diagnostic Pitfall.

European journal of case reports in internal medicine
2025

Cold-Induced Urticarias with Familial Background: Clinical Spectrum, Pathogenesis, and Diagnostic Challenges.

Diagnostics (Basel, Switzerland)
2026

Neonatal familial cold autoinflammatory syndrome: A newborn with recurrent cold-induced urticarial eruptions.

JAAD case reports
2025

RM1mAb avoids Intracellular degradation to Synergistically inhibit NLRP3 Inflammasome Activation in Familial Cold Autoinflammatory Syndrome.

bioRxiv : the preprint server for biology
2025

Clinical Images: Periumbilical wound dehiscence and pathergy in a neonate with OTULIN-related autoinflammatory syndrome.

ACR open rheumatology
2025

Symptoms of ASIA Syndrome in a Female Patient with Tattoos - A Case Report.

Clinical, cosmetic and investigational dermatology
2025

Safety and effectiveness of canakinumab in Japanese patients with cryopyrin-associated periodic syndrome: final results from a post-marketing surveillance in Japan.

Modern rheumatology
2026

Novel Variant in the NLRP12 Gene: Insights From a Case Report and Systematic Review.

International journal of immunogenetics
2026

A new digenic inheritance of proteasome-associated autoinflammatory syndrome involving the PSMA6 gene.

The journal of allergy and clinical immunology. In practice
2025

VEXAS syndrome: cutaneous manifestations and UBA1 gene variants in the diagnosis of a rare autoinflammatory syndrome - Case report.

Anais brasileiros de dermatologia
2026

Independent mechanisms of inflammation and myeloid bias in VEXAS syndrome.

Nature
2025

Mevalonate kinase deficiency (hyperimmunoglobulin D syndrome) in a Tanzanian girl: a case report.

Journal of medical case reports
2025

Understanding PFAPA Syndrome in Palestine: A Retrospective Cohort Analysis of Epidemiological and Clinical Data.

Mediterranean journal of rheumatology
2025

Case Report: Mevalonate kinase deficiency: an underdiagnosed cause of ischemic stroke-characterization of a novel genetic variant.

Frontiers in immunology
2025

Early-onset neutropenia and mixed phenotype in ADA2 deficiency: diagnostic and therapeutic challenges.

Frontiers in immunology
2025

[Genetic analysis of a child with X-linked familial Behcet-like autoinflammatory syndrome-2 due to variant of ELF4 gene].

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

A Patient with Vexas Syndrome Presenting with Complete Ophthalmoplegia.

European journal of case reports in internal medicine
2025

Adjuvant-induced autoimmune syndrome (ASIA syndrome) and the role of sonography in its diagnosis: a case-based review.

Rheumatology international
2025

Serum Galectin-3 and Presepsin Levels in Pediatric Familial Mediterranean Fever Patients During Remission: A Prospective Study.

Diagnostics (Basel, Switzerland)
2025

Severe acneiform eruption associated with TNFRSF13B mutation: Clinical response to adalimumab.

JAAD case reports
2025

Severe Complications Due to Biopolymers in a Patient With Autoimmune Disease: A Case Report and Review of the Literature.

Aesthetic surgery journal. Open forum
2025

A rare autoinflammatory syndrome associated with a C2orf69 frameshift mutation: a case report.

Immunologic research
2025

UBA1-depleted neutrophils disrupt immune homeostasis and induce VEXAS-like autoinflammatory disease in mice.

The Journal of clinical investigation
2025

Case Report: A heterozygous mutation of NLRP3 in a Chinese child with NLRP3-AID.

Frontiers in pediatrics
2025

Case Report: Exploring the spectrum of A20 haploinsufficiency in children.

Frontiers in pediatrics
2025

Long-term clinical course and treatment response in two patients with OTULIN-related autoinflammatory syndrome.

Rheumatology (Oxford, England)
2025

Chronic urticaria and autoinflammatory syndromes.

Current opinion in allergy and clinical immunology
2025

TH5487 specifically targets NLRP3 in FCAS patients resistant to MCC950.

bioRxiv : the preprint server for biology
2025

Autoimmune/Autoinflammatory Syndrome Induced by Adjuvants (ASIA Syndrome) After Polypropylene Mesh Implantation - Protocol of a Pilot Study for Diagnostics and Treatment.

Journal of abdominal wall surgery : JAWS
2025

Paracentral acute middle maculopathy as a major clinical manifestation of adenosine deaminase-2 deficiency.

BMC ophthalmology
2025

Clinical relevance of loss-of-function mutations of NEMO/IKBKG.

Genes & diseases
2025

[Genetic diagnostics in fever syndromes].

Zeitschrift fur Rheumatologie
2025

Association Between Pathogenic Variants in NLRP12 and Autoinflammatory Disease: A Comprehensive Systematic Review.

International journal of immunogenetics
2025

Genetic Basis of Autoinflammatory Skin Diseases. Part I. Genetic Pathways of Complex Autoinflammatory Skin Diseases.

Journal of the American Academy of Dermatology
2025

Use of anakinra in cryopyrin-associated periodic syndromes: case report and review of literature.

Frontiers in immunology
2025

Rapid clinical deployment of UBA1 testing in patients with VEXAS syndrome.

American journal of clinical pathology
2025

OTULIN-Related Autoinflammatory Syndrome With a Novel Mutation Treated With Tumor Necrosis Factor Inhibitor Therapy: A Rare Case Report.

The Journal of rheumatology
2025

Pyoderma Gangrenosum-Associated Autoinflammatory Syndrome With Skin Abscesses: Successful Treatment With Anti-Interleukin 1 Therapy.

The Journal of rheumatology
2025

Toll-like receptor 8 deficiency syndrome result in diffuse pigmented skin rash and autoinflammatory syndrome.

Journal der Deutschen Dermatologischen Gesellschaft = Journal of the German Society of Dermatology : JDDG
2025

Is it VEXAS or is it vasculitis?

Clinical and experimental rheumatology
2025

Digital clubbing, an unfamiliar feature of cryopyrin-associated periodic syndrome: A cohort study.

Journal of the American Academy of Dermatology
2025

Vacuoles, E1 Enzyme, X-linked, Autoinflammatory, and Somatic (VEXAS) Syndrome: A Diagnostic and Therapeutic Conundrum.

Cureus
2025

Basiliximab for IL-2-Associated Inflammatory Disorder With Atopic Dermatitis.

Pediatric dermatology
2025

Fasciitis-panniculitis syndrome with autoantibodies reacting to adipocyte pericellular fibers: a case report.

Pediatric rheumatology online journal
2025

Nucleotide-binding domain, leucine-rich repeat, and pyrin domain-containing protein 3 inflammasome: From action mechanism to therapeutic target in clinical trials.

World journal of gastrointestinal oncology
2025

IFN-γ licenses normal and pathogenic ALPK1/TIFA pathway in human monocytes.

iScience
2025

Linear ubiquitination at damaged lysosomes induces local NFKB activation and controls cell survival.

Autophagy
2024

[New Advances in the Study of VEXAS Syndrome --Review].

Zhongguo shi yan xue ye xue za zhi
2024

Enforced CARD11/MALT1 signaling in dendritic cells triggers hemophagocytic lymphohistiocytosis.

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

The role of PET/CT scan in addressing autoinflammatory diseases; A case report of a young man presenting with fever, splenomegaly and exanthem.

Hellenic journal of nuclear medicine
2024

Misdiagnosed for 14 Years: Adenosine Deaminase 2 (ADA2) Deficiency in a Teen Mimicking Polyarteritis Nodosa.

Clinical case reports
2025

Sirolimus as a possible treatment in cleavage-resistant RIPK1-induced autoinflammatory syndrome.

The journal of allergy and clinical immunology. In practice
2026

Pyoderma Gangrenosum Associated With Iatrogenic Interleukin 17A Blockade: A Report of Two Cases and a Review of the Literature.

Journal of cutaneous pathology
2024

Familial Mediterranean Fever: An Autoinflammatory Genetic Disorder.

Cureus
2024

Three cases of autoinflammatory disease with novel NLRC4 mutations, and the first mutation reported in the CARD domain of NLRC4 associated with autoinflammatory infantile enterocolitis (AIFEC).

Pediatric rheumatology online journal
2024

Rapid identification of primary atopic disorders (PAD) by a clinical landmark-guided, upfront use of genomic sequencing.

Allergologie select
2024

A Challenging Case of Recurrent Fever.

Cureus
2024

Non-Skewed X-inactivation Results in NF-κB Essential Modulator (NEMO) Δ-exon 5-autoinflammatory Syndrome (NEMO-NDAS) in a Female with Incontinentia Pigmenti.

Journal of clinical immunology
2025

Clinical spectrum of and outcomes for Indian children with deficiency of adenosine deaminase 2 (DADA2): a multicentric study.

Rheumatology (Oxford, England)
2024

Approaches for the diagnosis and treatment of VEXAS syndrome: the importance of clinical suspicion and the use of methotrexate.

Annals of hematology
2024

Activation of autoreactive lymphocytes in the lung by radioresistant cells expressing a STING gain-of-function mutation.

JCI insight
2024

TNFRSF11A variants contribute to systemic autoinflammatory diseases: A case series of 12 patients.

Seminars in arthritis and rheumatism
2024

Unraveling the diagnostic odyssey: stimulator of interferon gene-associated vasculopathy with onset in infancy in a 30-year-old female.

Journal of rheumatic diseases
2024

VEXAS syndrome as a cause for multifocal, relapsing head and neck inflammation.

Clinical case reports
2024

OTULIN-related conditions: Report of a new case and review of the literature using GenIA.

Clinical immunology (Orlando, Fla.)
2024

Adult-Onset Still's Disease Following COVID-19 Infection in a Patient Receiving Nirmatrelvir/Ritonavir: A Case Report.

Cureus
2024

A case of mother and child with CANDLE syndrome: Diagnosis and subsequent treatment with baricitinib.

Pediatric dermatology
2024

Case report: VEXAS syndrome: an atypical indolent presentation as sacroiliitis with molecular response to azacitidine.

Frontiers in immunology
2024

OTULIN haploinsufficiency predisposes to environmentally directed inflammation.

Frontiers in immunology
2024

A rare case of uncharacterized autoinflammatory disease: Patient carrying variations in NLRP3 and TNFRSF1A genes.

American journal of medical genetics. Part A
2024

Foreign substance injection for esthetic purposes associated with delayed unilateral sciatic nerve impairment: A surgical case report.

Clinical neurology and neurosurgery
2024

Disease flares with baricitinib dose reductions and development of flare criteria in patients with CANDLE/PRAAS.

Annals of the rheumatic diseases
2024

A de novo dominant-negative variant is associated with OTULIN-related autoinflammatory syndrome.

The Journal of experimental medicine
2024

Dominant negative OTULIN-related autoinflammatory syndrome.

The Journal of experimental medicine
2024

Unveiling the clinical spectrum of relapsing polychondritis: insights into its pathogenesis, novel monogenic causes, and therapeutic strategies.

Advances in rheumatology (London, England)
2024

Proteasome inhibitor-associated histiocytoid Sweet's syndrome: Clinical and histological similarities to Nakajo-Nishimura syndrome suggest a potential mechanism.

The Journal of dermatology
2024

Clinical Manifestations in Vacuoles, E1 Enzyme, X-Linked, Autoinflammatory, Somatic (VEXAS) Syndrome: A Narrative Review.

Cureus
2024

Identification of a Novel NLRP12 Frameshift Mutation (Val730Glyfs∗41) by Whole-Exome Sequencing in Patients with Crohn's Disease.

Human mutation
2023

Post-COVID-19 Vaccination and Thyrotoxicosis (ASIA Syndrome): Single-Centre Experience from India with Review of Literature.

Indian journal of endocrinology and metabolism
2024

Case report: VEXAS as an example of autoinflammatory syndrome in pulmonology clinical practice.

Frontiers in medicine
2025

Susceptibility to mycobacterial infection in VEXAS syndrome.

Rheumatology (Oxford, England)
2024

VEXAS syndrome: An update.

Joint bone spine
2024

Clinical Characteristics of Cryopyrin-Associated Periodic Syndrome and Long-Term Real-World Efficacy and Tolerability of Canakinumab in Japan: Results of a Nationwide Survey.

Arthritis & rheumatology (Hoboken, N.J.)
2024

Trappc1 intrinsically prevents ferroptosis of naive T cells to avoid spontaneous autoinflammatory disease in mice.

European journal of immunology
2024

Heterozygous mutations in the C-terminal domain of COPA underlie a complex autoinflammatory syndrome.

The Journal of clinical investigation
2024

VEXAS syndrome: Clinical manifestations, diagnosis, and treatment.

Reumatologia clinica
2024

The discovery of NLRP3 and its function in cryopyrin-associated periodic syndromes and innate immunity.

Immunological reviews
2023

Atypical cutaneous presentation of AOSD with persistent itchy urticaria: A case report.

Medicine
2024

Proteasome-associated autoinflammatory syndrome 2 in a neonate.

Pediatric dermatology
2023

Phagocytic cell death leads to enhanced release of pro-inflammatory S100A12 in familial Mediterranean fever.

Molecular and cellular pediatrics
2024

Features of immune mediated diseases in JAK2 (V617F)-positive myeloproliferative neoplasms and the potential therapeutic role of JAK inhibitors.

European journal of internal medicine
2024

A case of cryopyrin-associated periodic syndrome due to somatic mosaic mutation complicated with recurrent circinate erythematous psoriasis.

Modern rheumatology case reports
2023

Autoinflammatory Recurrent Pericarditis Associated with a New NLRP12 Mutation in a Male Adolescent.

Life (Basel, Switzerland)
2023

Myeloid OTULIN deficiency couples RIPK3-dependent cell death to Nlrp3 inflammasome activation and IL-1β secretion.

Science immunology
2023

[Síndrome de fiebre periódica, adenitis, faringitis y aftosis en un preescolar con síndrome de Asperger: reporte de caso].

Revista alergia Mexico (Tecamachalco, Puebla, Mexico : 1993)
2023

Lipin-2 regulates the antiviral and anti-inflammatory responses to interferon.

EMBO reports
2023

NLRP12-associated autoinflammatory disease: much more than the FCAS phenotype.

Clinical and experimental rheumatology
2023

Anakinra and hepatotoxicity in pediatric rheumatology: a case series.

Pediatric rheumatology online journal
2024

Autoimmune Autoinflammatory Syndrome Induced by Adjuvants (ASIA) After Injection of Foreign Materials for Cosmetic Purposes: Retrospective Analysis of 1027 Cases.

Aesthetic plastic surgery
2023

Neuroinflammation, autoinflammation, splenomegaly and anemia caused by bi-allelic mutations in IRAK4.

Frontiers in immunology
2023

Variant STAT4 and Treatment of an Autoinflammatory Syndrome. Reply.

The New England journal of medicine
2023

Variant STAT4 and Treatment of an Autoinflammatory Syndrome.

The New England journal of medicine
2023

Hepatic safety and efficacy of immunomodulatory drugs used in patients with autoimmune hepatitis.

Journal of autoimmunity
2024

Breast implant illness: Is it causally related to breast implants?

Autoimmunity reviews
2023

Autoinflammatory gene mutations associated with eosinophilia and asthma.

Allergy, asthma, and clinical immunology : official journal of the Canadian Society of Allergy and Clinical Immunology
2023

Pediatric-onset limited ANCA-associated vasculitis arising during pre-existing chronic recurrent multifocal osteomyelitis.

Pediatric rheumatology online journal
2024

Cleavage Resistant RIP Kinase1 Induced Autoinflammatory Syndrome (CRIA) - A Novel Autoinflammatory Syndrome.

Indian journal of pediatrics
2023

Identification of eight novel proteasome variants in five unrelated cases of proteasome-associated autoinflammatory syndromes (PRAAS).

Frontiers in immunology
2023

OTULIN protects the intestinal epithelium from apoptosis during inflammation and infection.

Cell death & disease
2023

NLRC4 gain-of-function variant is identified in a patient with systemic lupus erythematosus.

Clinical immunology (Orlando, Fla.)
2023

Activation of Autoreactive Lymphocytes in the Lung by STING Gain-of-function Mutation Radioresistant Cells.

bioRxiv : the preprint server for biology
2024

Cleavage-resistant RIPK1-induced autoinflammatory syndrome-A report of three generations with periodic fever and clinical response to colchicine.

International journal of rheumatic diseases
2023

Update on autoinflammatory diseases.

Current opinion in rheumatology
2023

Clinical and histopathological correlations in symptomatic patients with salpingectomy for Essure® implant removal: a cross-sectional study.

The European journal of contraception & reproductive health care : the official journal of the European Society of Contraception
2023

A case series of ten plus one deficiency of adenosine deaminase 2 (DADA2) patients in Iran.

Pediatric rheumatology online journal
2023

Cold Urticaria Syndromes: Diagnosis and Management.

The journal of allergy and clinical immunology. In practice
2023

Variant STAT4 and Response to Ruxolitinib in an Autoinflammatory Syndrome.

The New England journal of medicine
2023

JAK inhibitors for the treatment of VEXAS syndrome.

Experimental biology and medicine (Maywood, N.J.)
2023

Rare Cases of Polymyalgia Rheumatica After Receiving COVID-19 Vaccinations.

Cureus
2023

Feasibility study of busulfan, fludarabine, and thiotepa conditioning regimen for allogeneic hematopoietic stemcell transplantationfor children and young adults with nonmalignant disorders.

Pediatric blood & cancer
2023

Case report: De novo SAMD9L truncation causes neonatal-onset autoinflammatory syndrome which was successfully treated with hematopoietic stem cell transplantation.

Frontiers in pediatrics
2023

Socio-professional impact and quality of life of cryopyrin-associated periodic syndromes in 54 patients in adulthood.

Clinical and experimental rheumatology
2023

Similarities and differences in autoinflammatory diseases with urticarial rash, cryopyrin-associated periodic syndrome and Schnitzler syndrome.

Allergology international : official journal of the Japanese Society of Allergology
2023

Early-onset recurrent panniculitis as a phenotype of NLRC4-associated autoinflammatory syndrome: Characterization of pathogenicity of the p.Ser445Pro NLRC4 variant.

The Australasian journal of dermatology
2023

Two Siblings With Recurrent Fevers: The Path to Mevalonate Kinase Deficiency Diagnosis.

Cureus
2023

Identification of a variant in NLRP3 gene in a patient with Muckle-Wells syndrome: a case report and review of literature.

Pediatric rheumatology online journal
2023

Autoimmune/inflammatory syndrome induced by adjuvants (ASIA) in 2023.

Autoimmunity reviews
2024

[Macrocytic anemia and polychondritis: VEXAS syndrome].

Zeitschrift fur Rheumatologie
2023

Mimickers of Classical Urticaria: Cryopyrin-Associated Autoinflammatory Syndrome Presenting as Isolated Urticaria in an Infant.

The journal of allergy and clinical immunology. In practice
2023

Pulmonary manifestations in VEXAS (vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic) syndrome: a systematic review.

Rheumatology international
2022

A novel frameshift variant in the ADA2 gene of a patient with a neurological phenotype: a case report.

Pediatric rheumatology online journal
2022

Hemophagocytic Lymphohistiocytosis in a Patient With Post-acute COVID-19 Infection.

Cureus
2023

Cold-induced loss of interaction with HSC70 triggers inflammasome activity of familial cold autoinflammatory syndrome-causing mutants of NLRP3.

Biochemical and biophysical research communications
2023

Histiocytoid Sweet Syndrome Presenting in Two Sisters With Deficiency of Deaminase Type 2.

The American Journal of dermatopathology
2022

Anesthesia Management of a Patient With Familial Cold Autoinflammatory Syndrome: A Case Report.

Anesthesia progress
2022

Assessment of type I interferon signatures in undifferentiated inflammatory diseases: A Japanese multicenter experience.

Frontiers in immunology
2022

Equally potent: Nlrp3 mutation in macrophages or neutrophils is sufficient to drive autoinflammation.

EMBO reports
2022

Autoimmune and autoinflammatory manifestations in inborn errors of immunity.

Current opinion in allergy and clinical immunology
2022

Successful Treatment of a Patient with Pyoderma Gangrenosum, Plaque Psoriasis and Palmoplantar Pustulosis with Adalimumab.

Clinical, cosmetic and investigational dermatology
2022

Periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis syndrome: Clinical characteristics and treatment outcomes - a single center study in Japan.

Pediatrics international : official journal of the Japan Pediatric Society
2022

Vasculitis associated with VEXAS syndrome: A literature review.

Frontiers in medicine
2022

SOCS1 Haploinsufficiency Presenting as Severe Enthesitis, Bone Marrow Hypocellularity, and Refractory Thrombocytopenia in a Pediatric Patient with Subsequent Response to JAK Inhibition.

Journal of clinical immunology
2022

A comprehensive summary of disease variants implicated in metal allergy.

Journal of toxicology and environmental health. Part B, Critical reviews
2022

NLRP12 and IL36RN mutations in a Portuguese woman with autoinflammatory syndrome.

JAAD case reports
2022

Frightening Fever: Familial Cold Autoinflammatory Syndrome 2 (FCAS-2) with Macrophage Activation Syndrome (MAS).

Indian journal of pediatrics
2022

The efficacy and safety of allogeneic stem cell transplantation in Mevalonate Kinase Deficiency.

Pediatric rheumatology online journal
2022

VEXAS Syndrome: A Novelty in MDS Landscape.

Diagnostics (Basel, Switzerland)
2022

Comparison between idiopathic and VEXAS-relapsing polychondritis: analysis of a French case series of 95 patients.

RMD open
2022

Rare mutations in NLRP3 and NLRP12 associated with familial cold autoinflammatory syndrome: two Chinese pedigrees.

Clinical rheumatology
2022

Autoimmune/autoinflammatory syndrome induced by adjuvants: a focus on silicone.

Clinical rheumatology
2022

[Autoinflammatory diseases associated with RIPK1 mutations: A review of the literature].

La Revue de medecine interne
2022

Case Report: Comorbid Hyper-IgD Syndrome and Hidradenitis Suppurativa - A New Syndromic Form of HS? A Report of Two Cases.

Frontiers in immunology
2022

Characterization of Novel Pathogenic Variants Leading to Caspase-8 Cleavage-Resistant RIPK1-Induced Autoinflammatory Syndrome.

Journal of clinical immunology
2022

Radioresistant cells initiate lymphocyte-dependent lung inflammation and IFNγ-dependent mortality in STING gain-of-function mice.

Proceedings of the National Academy of Sciences of the United States of America
2022

SARS-CoV-2 the ASIA virus (Autoimmune/autoinflammatory Syndrome Induced by Adjuvants), the risk of infertility and vaccine hesitancy.

Expert review of vaccines
2023

Clinical heterogeneity of NLRP12-associated autoinflammatory diseases.

Genes & diseases
2022

Cryo-sensitive aggregation triggers NLRP3 inflammasome assembly in cryopyrin-associated periodic syndrome.

eLife
2022

Autoimmune/autoinflammatory syndrome induced by adjuvants after multi-component meningococcal serogroup B vaccination in a 7-year-old girl: a case report.

The Turkish journal of pediatrics
2022

An autoinflammatory syndrome with compound heterozygous MEFV and NOD2/CARD15 gene mutations successfully treated with tocilizumab.

Rheumatology advances in practice
2022

Low-ratio somatic NLRC4 mutation causes late-onset autoinflammatory disease.

Annals of the rheumatic diseases
2022

VEXAS syndrome: lessons learnt from an early Australian case series.

Internal medicine journal
2022

Pyoderma Gangrenosum, Acne, and Hidradenitis Suppurativa Syndrome: A Case Report and Literature Review.

Frontiers in medicine
2022

Variants in autoinflammatory diseases-related genes in a family with cold-induced autoinflammatory syndrome.

Clinical and experimental rheumatology
2022

NEMO splice variant causes distinct autoinflammatory syndrome.

Nature reviews. Rheumatology
2022

A patient with urticarial lesions, recurrent fever, and IgM-type monoclonal gammopathy.

Acta dermatovenerologica Alpina, Pannonica, et Adriatica
2022

Outcomes of SARS-CoV-2 infection among children and young people with pre-existing rheumatic and musculoskeletal diseases.

Annals of the rheumatic diseases
2022

The 2021 European Alliance of Associations for Rheumatology/American College of Rheumatology Points to Consider for Diagnosis and Management of Autoinflammatory Type I Interferonopathies: CANDLE/PRAAS, SAVI, and AGS.

Arthritis & rheumatology (Hoboken, N.J.)
2022

Screening of OTULIN gene mutation with targeted next generation sequencing in Turkish populations and in silico analysis of these mutations.

Molecular biology reports
2022

Genetically programmed alternative splicing of NEMO mediates an autoinflammatory disease phenotype.

The Journal of clinical investigation
2022

AA amyloidosis complicating cryopyrin-associated periodic syndrome: a study of 86 cases including 23 French patients and systematic review.

Rheumatology (Oxford, England)
2022

Rubicon promotes rather than restricts murine lupus and is not required for LC3-associated phagocytosis.

JCI insight
2022

Compound heterozygous variants in OTULIN are associated with fulminant atypical late-onset ORAS.

EMBO molecular medicine
2022

RNA sensing via the RIG-I-like receptor LGP2 is essential for the induction of a type I IFN response in ADAR1 deficiency.

The EMBO journal
2023

Kynurenine pathway of tryptophan metabolism in patients with familial Mediterranean fever.

Modern rheumatology
2021

Case Report: Genetic Double Strike: VEXAS and TET2-Positive Myelodysplastic Syndrome in a Patient With Long-Standing Refractory Autoinflammatory Disease.

Frontiers in immunology
2022

NLRP12-associated systemic autoinflammatory diseases in children.

Pediatric rheumatology online journal
2023

VEXAS syndrome (vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic) for the dermatologist.

Journal of the American Academy of Dermatology
2022

NEMO-NDAS: A Panniculitis in the Young Representing an Autoinflammatory Disorder in Disguise.

The American Journal of dermatopathology
2022

[Analysis of clinical characteristics of 35 inflammasomopathies cases].

Zhonghua er ke za zhi = Chinese journal of pediatrics
2022

The 2021 European Alliance of Associations for Rheumatology/American College of Rheumatology points to consider for diagnosis and management of autoinflammatory type I interferonopathies: CANDLE/PRAAS, SAVI and AGS.

Annals of the rheumatic diseases
2022

Haploinsufficiency of PSMD12 Causes Proteasome Dysfunction and Subclinical Autoinflammation.

Arthritis & rheumatology (Hoboken, N.J.)
2022

Thyrotoxicosis following SARS-COV-2 vaccination: a case series and discussion.

Journal of endocrinological investigation

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Doença com base genética

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Conselhos e sociedades

Doenças relacionadas

Doenças com sintomas parecidos — ajudam quem ainda está buscando diagnóstico

Perguntas frequentes

O que as famílias mais perguntam sobre esta doença — cada resposta com a fonte de onde saiu

Respostas geradas por IA a partir das fontes citadas

O quadro é desencadeado por falhas no sistema imunológico inato, frequentemente associadas a alterações genéticas. Diversos genes foram catalogados em conexão com essas respostas inflamatórias, como LPIN2, F12 e LACC1.

Referências

Fontes citadas no texto, publicações do grafo e bases de dados usadas neste verbete

Citar este verbete

Raras. (s.d.). Síndrome autoinflamatório. Em Raras — Enciclopédia de Doenças Raras do Brasil. https://raras.org/doenca/sindrome-autoinflamatorio

Formato APA. Conteúdo sob CC BY 4.0 — reuso livre com atribuição.

Conteúdo mantido por Agente Raras · Médicos e pesquisadores podem colaborar

Compêndio · Raras BR

Síndrome autoinflamatório

ORPHA:93665 · MONDO:0019751
CID-10
A68.9 · Febre recorrente não especificada
Ensaios
6 ativos
Medicamentos
6 registrados
MedGen
UMLS
C0015974
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Doença rara (ontologia)
fonte: Orphanet
Identificador unificado
fonte: MONDO
Indexação biomédica
fonte: MeSH (NLM)
Dado público estruturado
fonte: Wikidata
Moléculas estudadas na doença
fonte: OpenTargets
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