A síndrome de Nakajo, também chamada de eritema nodular com alterações digitais, é um distúrbio congênito autossômico recessivo raro relatado pela primeira vez em 1939 por A. Nakajo em descendentes de pais consanguíneos. A síndrome pode ser caracterizada por eritema, perda de gordura corporal na parte superior do corpo e olhos, orelhas, nariz, lábios e dedos desproporcionalmente grandes.
Introdução
O que você precisa saber de cara
Visão geral
Sinais e sintomas
O quadro clínico da condição envolve manifestações cutâneas e craniofaciais, incluindo aparência facial progeroide, nariz proeminente, pele rígida, hiperpigmentação da pele, envelhecimento prematuro dos cabelos e proptose.[1][2]
Também são observadas alterações osteoarticulares e de crescimento, como anormalidade de crescimento, atraso de crescimento, dedos longos, contratura em flexão do cotovelo, hálux valgo e dor óssea.[1][2]
Entre os sinais oculares, inflamatórios e sistêmicos, relatam-se epiesclerite, opacificação puntiforme da córnea, parotidite, sinusite recorrente, epididimite, constipação crônica, abdome protuberante e arritmia.[1][2]
Alterações laboratoriais e neurológicas incluem concentração elevada de transaminase hepática circulante, aumento da concentração circulante de interleucina 8, concentração elevada de hormônio tireoestimulante circulante, concentração diminuída de colesterol HDL e deficiência intelectual.[1][2]
Causas genéticas
A condição está associada a variantes em genes ligados a subunidades e montagem do proteassoma, destacando-se PSMB8 (Proteasome subunit beta type-8), PSMG2 (Proteasome assembly chaperone 2), PSMB4 (Proteasome subunit beta type-4), PSMB10 (Proteasome subunit beta type-10), PSMB9 (Proteasome subunit beta type-9) e POMP (Proteasome maturation protein).[1][3]
Diagnóstico
Conteúdo informativo gerado e mantido automaticamente a partir de fontes oficiais (Orphanet, HPO, OMIM, SUS). Não substitui avaliação médica.
A **síndrome Nakajo-Nishimura** é uma condição genética muito rara que provoca alterações inflamatórias no corpo e no tecido adiposo. A pessoa acometida pode apresentar endurecimento e manchas na pele, dores ósseas, perda de flexibilidade nas articulações e traços faciais característicos. A causa está ligada a alterações em genes responsáveis pelo funcionamento dos proteassomos das células, principalmente o gene PSMB8. Esta doença não consta na lista oficial de PCDTs de doenças raras do Ministério da Saúde e não possui remédios dispensados diretamente pelo SUS para ela. O acompanhamento clínico busca aliviar os sintomas e monitorar as manifestações inflamatórias na rede de saúde.
A síndrome de Nakajo, também chamada de eritema nodular com alterações digitais, é um distúrbio congênito autossômico recessivo raro relatado pela primeira vez em 1939 por A. Nakajo em descendentes de pais consanguíneos. A síndrome pode ser caracterizada por eritema, perda de gordura corporal na parte superior do corpo e olhos, orelhas, nariz, lábios e dedos desproporcionalmente grandes.
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Sinais e sintomas
O que aparece no corpo e com que frequência cada sintoma acontece
Partes do corpo afetadas
+ 66 sintomas em outras categorias
Características mais comuns
Os sintomas variam de pessoa para pessoa. Abaixo estão as 137 características clínicas mais associadas, ordenadas por frequência.
Linha do tempo da pesquisa
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Genética e causas
O que está alterado no DNA e como passa nas famílias
Genes associados
6 genes identificados com associação a esta condição.
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.
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.
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.
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.
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.
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.
Variantes genéticas (ClinVar)
170 variantes patogênicas registradas no ClinVar.
Vias biológicas (Reactome)
33 vias biológicas associadas aos genes desta condição.
Diagnóstico
Os sinais que médicos procuram e os exames que confirmam
Tratamento e manejo
Remédios, cuidados de apoio e o que precisa acompanhar
Onde tratar no SUS
Hospitais de referência no Brasil e o protocolo oficial do SUS (PCDT)
🇧🇷 Atendimento SUS — Síndrome Nakajo-Nishimura
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Pesquisa ativa
Ensaios clínicos abertos e novidades científicas recentes
Ensaios em destaque
Nenhum país com ensaio aberto no momento.
Pesquisa e ensaios clínicos
1 ensaios clínicos encontrados.
Publicações mais relevantes
Molecular dynamics simulation of wild and mutant proteasome subunit beta type 8 (PSMB8) protein: Implications for restoration of inflammation in experimental autoimmune encephalomyelitis pathogenesis.
Multiple Sclerosis (MS) is an autoimmune and chronic disease in the brain and spinal cord. MS has inflammatory progression characterized by its hallmark inflammatory plaques. The histological and clinical characteristics of MS are shared by Experimental Autoimmune Encephalomyelitis (EAE). Genetic and environmental factors contribute to the development of MS. In EAE-MS disease, the level of proteasome subunit beta type-8 (PSMB8), encoded by the PSMB8 gene, is increased and regulates the inflammatory response in this disease. In humans, the Nakajo-Nishimura Syndrome is caused by a mutation in the gene PSMB8, a part of the immunoproteasome subunit. Therefore, special attention to wild and mutant (G210V) PSMB8 protein is imperative. In this study, we performed a 100 ns molecular dynamics (MD) simulation for wild-type PSMB8 and the mutant G210V. Then, we analyzed the fundamental and essential simulation results using another Google Colab system. The energy analysis ensures the structural deviation due to point mutation. The trajectory of the fundamental simulation (RMSD, RMSF, and Rg) describes that the G210V mutated protein is more flexible and less stable than the wild type. We observed the conformational changes due to mutation by analyzing the RMSD average linkage hierarchical clustering, total SASA, and SASA autocorrelation. The differences in the protein's overall motion and the atoms' precise location are identified by the principal component analysis, showing that the overall motion and location of the atoms are different. Our study provides valuable insights into the dynamics and structure of this protein, which can aid in further understanding its biological functions and potential implications for disease.
Proteasome inhibitor-associated histiocytoid Sweet's syndrome: Clinical and histological similarities to Nakajo-Nishimura syndrome suggest a potential mechanism.
Histiocytoid Sweet's syndrome (HSS) is a variant of Sweet's syndrome (SS) that clinically resembles SS but differs histologically by infiltrates, predominantly composed of immature cells of the myeloid lineage. Medications such as proteasome inhibitors have been reported to cause HSS but there has been little discussion on the underlying mechanism. Here we report two cases of HSS associated with a proteasome inhibitor. Both patients were on ixazomib for the treatment of multiple myeloma and presented with acute erythematous plaques on the upper half of the body. Pathological findings were consistent with HSS. Similarities between proteasome inhibitor-induced HSS and Nakajo-Nishimura syndrome, an inherited inflammatory disease, can be identified both clinically and histologically, suggesting a potential explanation of the mechanism behind proteasome inhibitor-associated HSS.
Panniculitis: A Cardinal Sign of Autoinflammation.
Panniculitis was first described in the nineteenth century and is characterized by inflammation of the subcutaneous fat. It may be categorized in septal or lobular subtypes, but other histopathological features (e.g., presence of vasculitis, nature of inflammatory infiltrates, characteristics of fat necrosis) are also important for diagnostic purposes. Clinically, panniculitis is characterized by the presence of subcutaneous nodules, and both ulcerative and nonulcerative clinical subtypes have been proposed. In this review, we aimed to describe the occurrence of panniculitis in autoinflammatory disorders (AIDs) and related diseases. Among monogenic AIDs, panniculitis is common in IFN-mediated disorders. Panniculitis is a distinctive feature in proteasome-associated autoinflammatory syndromes (PRAAS), including chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature (CANDLE) syndrome and Nakajo-Nishimura syndrome. On the other hand, erythema nodosum corresponds to the most common clinical form of panniculitis and is common in polygenic AIDs, such as Behçet's syndrome, inflammatory bowel disease, and sarcoidosis. Cytophagic histiocytic panniculitis, lipoatrophic panniculitis of children, and otulipenia are rare disorders that may also present with inflammation of the subcutaneous fat. Therefore, panniculitis can identify a specific subgroup of patients with AIDs and may potentially be regarded as a cardinal sign of autoinflammation.
Correction to: Pluripotent Stem Cell-Based Screening Identifies CUDC-907 as an Effective Compound for Restoring the In Vitro Phenotype of Nakajo-Nishimura Syndrome.
Nakajo-Nishimura Syndrome: The First African Case.
Nakajo-Nishimura syndrome is a hereditary autoinflammatory disorder caused by an autosomal recessive homozygous mutation of the PSMB8 gene, which encodes the immunoproteasome subunit beta 5i. The clinical manifestations of NNS are mainly pernio-like skin rashes, nodular erythema, lipodystrophy, clubbed fingers, remittent fever, hepatosplenomegaly, and basal ganglia calcifications. Here we are reporting a case of NNS in an 11-year-old girl, who lives in eastern Algeria, born from a first-degree consanguineous marriage, she presented with erythematous patches on her face and her back, nodular erythema on her neck, swollen and painful fingers with acrocyanosis and recurrent fever that mainly occurred in cold weather. The patient received long-term treatment with low-dose glucocorticoids, along with immunomodulatory drugs (hydroxychloroquine with methotrexate), partial improvement clinically and biologically was observed. Colchicine was added to her treatment, with increased prednisone doses when she recently developed an AA amyloidosis. Our patient was diagnosed clinically with a probable NNS because she exhibited six of the eight characteristics. To the best of our knowledge, this is the first case of NNS in Africa.
Publicações recentes
Molecular dynamics simulation of wild and mutant proteasome subunit beta type 8 (PSMB8) protein: Implications for restoration of inflammation in experimental autoimmune encephalomyelitis pathogenesis.
Proteasome inhibitor-associated histiocytoid Sweet's syndrome: Clinical and histological similarities to Nakajo-Nishimura syndrome suggest a potential mechanism.
Panniculitis: A Cardinal Sign of Autoinflammation.
Nakajo-Nishimura Syndrome: The First African Case.
Efficacy and safety of baricitinib in Japanese patients with autoinflammatory type I interferonopathies (NNS/CANDLE, SAVI, And AGS).
📚 EuropePMC20 artigos no totalmostrando 16
Molecular dynamics simulation of wild and mutant proteasome subunit beta type 8 (PSMB8) protein: Implications for restoration of inflammation in experimental autoimmune encephalomyelitis pathogenesis.
HeliyonProteasome inhibitor-associated histiocytoid Sweet's syndrome: Clinical and histological similarities to Nakajo-Nishimura syndrome suggest a potential mechanism.
The Journal of dermatologyPanniculitis: A Cardinal Sign of Autoinflammation.
Current rheumatology reviewsNakajo-Nishimura Syndrome: The First African Case.
Mediterranean journal of rheumatologyEfficacy and safety of baricitinib in Japanese patients with autoinflammatory type I interferonopathies (NNS/CANDLE, SAVI, And AGS).
Pediatric rheumatology online journalPluripotent stem cell-based screening identifies CUDC-907 as an effective compound for restoring the in vitro phenotype of Nakajo-Nishimura syndrome.
Stem cells translational medicineA Chinese case of Nakajo-Nishimura syndrome with novel compound heterozygous mutations of the PSMB8 gene.
BMC medical geneticsDesignation of Autoinflammatory Skin Manifestations With Specific Genetic Backgrounds.
Frontiers in immunologyMyositis with sarcoplasmic inclusions in Nakajo-Nishimura syndrome: a genetic inflammatory myopathy.
Neuropathology and applied neurobiologyNakajo-Nishimura syndrome and related proteasome-associated autoinflammatory syndromes.
Journal of inflammation researchInduced pluripotent stem cells representing Nakajo-Nishimura syndrome.
Inflammation and regenerationBeneficial effect of methotrexate on a child case of Nakajo-Nishimura syndrome.
The Journal of dermatologyChinese case of Nakajo-Nishimura syndrome with a novel mutation of the PSMB8 gene.
The Journal of dermatologyPluripotent Stem Cell Model of Nakajo-Nishimura Syndrome Untangles Proinflammatory Pathways Mediated by Oxidative Stress.
Stem cell reportsDysfunctional immunoproteasomes in autoinflammatory diseases.
Inflammation and regeneration[Type I interferonopathies].
Annales de dermatologie et de venereologieAssociações
Organizações que acompanham esta doença — pra ter apoio e orientação
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Doença com base genética
Um médico geneticista pode ajudar no diagnóstico de Síndrome Nakajo-Nishimura e no aconselhamento genético da família.
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
A síndrome é causada por alterações genéticas que comprometem os proteassomos celulares, em especial mutações no gene PSMB8. Outros genes do proteassomo, como PSMB9, PSMB10, PSMB4, PSMG2 e POMP, também estão associados a esse grupo de condições.
Referências
Fontes citadas no texto, publicações do grafo e bases de dados usadas neste verbete
4 fontes citadas no texto · 6 publicações do grafo RarasNet (PubMed) · 5 bases de dados. Títulos, periódicos e PMIDs vêm direto da fonte, sem intermediação de IA.
Dados compilados pelo RarasNet a partir de fontes abertas (Orphanet, OMIM, MONDO, PubMed/EuropePMC, ClinicalTrials.gov, DATASUS, PCDT/MS). Este conteúdo é informativo e não substitui avaliação médica.
Citar este verbete
Raras. (2026). Síndrome Nakajo-Nishimura. Em Raras — Enciclopédia de Doenças Raras do Brasil. https://raras.org/doenca/sindrome-nakajo-nishimura
Formato APA. Conteúdo sob CC BY 4.0 — reuso livre com atribuição.
Conteúdo mantido por Agente Raras · Médicos e pesquisadores podem colaborar
Síndrome Nakajo-Nishimura
📋 Origem dos dados
Esta página agrega dados de fontes públicas e oficiais. Dados sobre cobertura no SUS (PCDT, CEAF) são verificados ativamente por agente proativo (ver badge no infobox). Demais dados têm atribuição de fonte + data da última sincronização — clique para abrir o original.
- Doença rara (ontologia)
- fonte: Orphanet
- Identificador unificado
- fonte: MONDO
- NIH/GARD
- fonte: GARD (NIH)
- Indexação biomédica
- fonte: MeSH (NLM)
- Dado público estruturado
- fonte: Wikidata