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Síndrome Prader-Willi
ORPHA:739CID-10 · Q87.1CID-11 · LD90.3OMIM 176270DOENÇA RARA

A síndrome de Prader-Willi é uma doença genética rara caracterizada por anomalias hipotálamo-hipófise com hipotonia grave durante o período neonatal e primeiros dois anos de vida e início de hiperfagia com risco de obesidade mórbida durante a infância e a idade adulta, dificuldades de aprendizagem e problemas comportamentais ou problemas psiquiátricos graves.

Mantido por Agente Raras·Colaborar como especialista →

Introdução

O que você precisa saber de cara

📋

A síndrome de Prader-Willi é uma doença genética rara caracterizada por anomalias hipotálamo-hipófise com hipotonia grave durante o período neonatal e primeiros dois anos de vida e início de hiperfagia com risco de obesidade mórbida durante a infância e a idade adulta, dificuldades de aprendizagem e problemas comportamentais ou problemas psiquiátricos graves.

Pesquisas ativas
26 ensaios
149 total registrados no ClinicalTrials.gov
Publicações científicas
4.023 artigos
Último publicado: 2026 Apr 14
Medicamentos
5 registrados
SOMATROPIN, RIMONABANT, OCTREOTIDE ACETATE

Tem tratamento?

5 medicamentos registrados
Ver detalhes, fases e interações →
SOMATROPINRIMONABANTOCTREOTIDE ACETATEOCTREOTIDETOPIRAMATE

Escala de raridade

CLASSIFICAÇÃO ORPHANET · BRASIL 2024
1-9 / 100 000
Ultra-rara
<1/50k
Muito rara
1/20k
Rara
1/10k
Pouco freq.
1/5k
Incomum
1/2k
Prevalência
0.0
Worldwide
Início
Antenatal
+ neonatal
🏥
SUS: Cobertura mínimaScore: 35%
Centros em: PA, PR, SC, RS, ES +10CID-10: Q87.1
🇧🇷Dados SUS / DATASUS
PROCEDIMENTOS SIGTAP (5)
0202010503
Cariótipo — bandas G, Q ou Rgenetic_test
0202010600
Pesquisa de microdeleções/microduplicações por FISHlab_test
0202010694
Sequenciamento completo do exoma (WES)rehabilitation
0202010260
Dosagem de alfa-fetoproteína
0301070040
Atendimento em reabilitação — doenças raras
Você se identifica com essa condição?
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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

📏
Crescimento
31 sintomas
🧠
Neurológico
27 sintomas
🦴
Ossos e articulações
22 sintomas
😀
Face
17 sintomas
👁️
Olhos
10 sintomas
🧬
Pele e cabelo
8 sintomas

+ 98 sintomas em outras categorias

Características mais comuns

100%prev.
Hipotonia neonatal
Frequência: 244/244
100%prev.
Deficiência intelectual
Frequência: 12/12
91%prev.
Criptorquidia
Muito frequente (99-80%)
90%prev.
Hipogonadismo hipogonadotrófico
Muito frequente (99-80%)
90%prev.
Hipotonia generalizada
Muito frequente (99-80%)
90%prev.
Palma curta
Muito frequente (99-80%)
241sintomas
Muito frequente (28)
Frequente (65)
Ocasional (39)
Muito raro (3)
Sem dados (106)

Os sintomas variam de pessoa para pessoa. Abaixo estão as 241 características clínicas mais associadas, ordenadas por frequência.

Hipotonia neonatalNeonatal hypotonia
Frequência: 244/244100%
Deficiência intelectualIntellectual disability
Frequência: 12/12100%
CriptorquidiaCryptorchidism
Muito frequente (99-80%)91%
Hipogonadismo hipogonadotróficoHypogonadotropic hypogonadism
Muito frequente (99-80%)90%
Hipotonia generalizadaGeneralized hypotonia
Muito frequente (99-80%)90%

Linha do tempo da pesquisa

Publicações por ano — veja quando o interesse científico cresceu
Anos de pesquisa1desde 2026
Total histórico4.023PubMed
Últimos 10 anos200publicações
Pico2025150 papers
Linha do tempo
2026Hoje · 2026🧪 1999Primeiro 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

13 genes identificados com associação a esta condição. Padrão de herança: Autosomal dominant, Not applicable.

MAGEL2MAGE-like protein 2Disease-causing germline mutation(s) inDesconhecido
FUNÇÃO

Probably enhances ubiquitin ligase activity of RING-type zinc finger-containing E3 ubiquitin-protein ligases, possibly through recruitment and/or stabilization of the Ubl-conjugating enzyme (E2) at the E3:substrate complex. Acts as a regulator of retrograde transport via its interaction with VPS35. Recruited to retromer-containing endosomes and promotes the formation of 'Lys-63'-linked polyubiquitin chains at 'Lys-220' of WASHC1 together with TRIM27, leading to promote endosomal F-actin assembly

LOCALIZAÇÃO

Early endosomeCytoplasmNucleus

MECANISMO DE DOENÇA

Schaaf-Yang syndrome

A disease characterized by clinical features of Prader-Willi syndrome, including neonatal hypotonia with poor suck, feeding problems in infancy, obesity, developmental delay, short stature, and hypogonadism. Additionally, patients manifest autism spectrum disorder. Some patients have dysmorphic facial features.

EXPRESSÃO TECIDUAL(Tecido-específico)
Hipotálamo
16.6 TPM
Pituitária
15.9 TPM
Brain Nucleus accumbens basal ganglia
8.1 TPM
Cervix Endocervix
6.5 TPM
Cervix Ectocervix
4.4 TPM
OUTRAS DOENÇAS (6)
Schaaf-Yang syndromePrader-Willi syndrome due to paternal deletion of 15q11q13 type 2fetal akinesia deformation sequence 1Prader-Willi syndrome due to maternal uniparental disomy of chromosome 15
HGNC:6814UniProt:Q9UJ55
IPWMENDELIANDesconhecido
LOCALIZAÇÃO

MKRN3E3 ubiquitin-protein ligase makorin-3MENDELIANDesconhecido
FUNÇÃO

E3 ubiquitin ligase catalyzing the covalent attachment of ubiquitin moieties onto substrate proteins. Acts as a key developmental timer that helps ensure puberty begins at the appropriate age, by inhibiting premature activation of the reproductive hormone cascade. Epigenetically regulates GNRH1 transcription by disrupting the binding of methyl-DNA binding protein 3/MBD3 to the promoter of GNRH1. Mechanistically, mediates the non-proteolytic ubiquitination of MBD3 at multiple sites with 'Lys27' u

LOCALIZAÇÃO

Nucleus

MECANISMO DE DOENÇA

Precocious puberty, central 2

A condition defined as the development of secondary sexual characteristics in boys and girls at a chronological age that is 2.5 standard deviations below the mean age at onset of puberty in the population. Central precocious puberty results from premature activation of the hypothalamic-pituitary-gonadal axis.

EXPRESSÃO TECIDUAL(Baixa expressão)
Brain Spinal cord cervical c-1
3.4 TPM
Testículo
1.9 TPM
Glândula salivar
1.7 TPM
Hipocampo
1.6 TPM
Esôfago - Mucosa
1.4 TPM
OUTRAS DOENÇAS (3)
precocious puberty, central, 2genetic central precocious puberty in malegenetic central precocious puberty in female
HGNC:7114UniProt:Q13064
PWRN1MENDELIANDesconhecido
LOCALIZAÇÃO

HERC2E3 ubiquitin-protein ligase HERC2MENDELIANAltamente restrito
FUNÇÃO

E3 ubiquitin-protein ligase that regulates ubiquitin-dependent retention of repair proteins on damaged chromosomes. Recruited to sites of DNA damage in response to ionizing radiation (IR) and facilitates the assembly of UBE2N and RNF8 promoting DNA damage-induced formation of 'Lys-63'-linked ubiquitin chains. Acts as a mediator of binding specificity between UBE2N and RNF8. Involved in the maintenance of RNF168 levels. E3 ubiquitin-protein ligase that promotes the ubiquitination and proteasomal

LOCALIZAÇÃO

CytoplasmCytoplasm, cytoskeleton, microtubule organizing center, centrosome, centrioleNucleus

VIAS BIOLÓGICAS (2)
Recruitment and ATM-mediated phosphorylation of repair and signaling proteins at DNA double strand breaksSUMOylation of DNA damage response and repair proteins
MECANISMO DE DOENÇA

Intellectual developmental disorder, autosomal recessive 38

A disorder characterized by significantly below average general intellectual functioning associated with impairments in adaptive behavior and manifested during the developmental period. MRT38 is characterized by global developmental delay affecting motor, speech, adaptive, and social development. Patients manifest autistic features, aggression, self-injury, impulsivity, and distractibility.

EXPRESSÃO TECIDUAL(Ubíquo)
Cerebelo
31.1 TPM
Cérebro - Hemisfério cerebelar
28.8 TPM
Ovário
25.9 TPM
Útero
25.0 TPM
Cólon sigmoide
24.5 TPM
OUTRAS DOENÇAS (2)
obsolete skin/hair/eye pigmentation, variation in, 1developmental delay with autism spectrum disorder and gait instability
HGNC:4868UniProt:O95714
PWAR1MENDELIANDesconhecido
LOCALIZAÇÃO

SNORD116-1MENDELIANDesconhecido
LOCALIZAÇÃO

NPAP1MENDELIANDesconhecido
LOCALIZAÇÃO

FUNÇÃO (UNIPROT)

May be involved in spermatogenesis

EXPRESSÃO TECIDUAL(Tecido-específico)
Testículo
7.5 TPM
Rim - Medula
1.2 TPM
Tireoide
1.1 TPM
Ovário
1.1 TPM
Pituitária
0.9 TPM
INTERAÇÕES PROTEICAS (4)
UniProt:Q9NZP6
SNORD115-1MENDELIANDesconhecido
LOCALIZAÇÃO

OCA2P proteinCandidate gene tested inTolerante
FUNÇÃO

Contributes to a melanosome-specific anion (chloride) current that modulates melanosomal pH for optimal tyrosinase activity required for melanogenesis and the melanosome maturation (PubMed:11310796, PubMed:15262401, PubMed:22234890, PubMed:25513726). One of the components of the mammalian pigmentary system (PubMed:15262401, PubMed:18252222, PubMed:7601462). May serve as a key control point at which ethnic skin color variation is determined. Major determinant of brown and/or blue eye color (PubMe

LOCALIZAÇÃO

Melanosome membrane

VIAS BIOLÓGICAS (1)
Melanin biosynthesis
MECANISMO DE DOENÇA

Albinism, oculocutaneous, 2

An autosomal recessive disorder in which the biosynthesis of melanin pigment is reduced in skin, hair, and eyes. Although affected infants may appear at birth to have complete absence of melanin pigment, most patients acquire small amounts of pigment with age. Visual anomalies include decreased acuity and nystagmus. The phenotype is highly variable. The hair of affected individuals may turn darker with age, and pigmented nevi or freckles may be seen. African and African American individuals may have yellow hair and blue-gray or hazel irides. One phenotypic variant, 'brown OCA,' has been described in African and African American populations and is characterized by light brown hair and skin color and gray to tan irides.

VIAS REACTOME (1)
EXPRESSÃO TECIDUAL(Tecido-específico)
Artéria tibial
10.5 TPM
Tireoide
9.2 TPM
Aorta
8.2 TPM
Skin Sun Exposed Lower leg
7.1 TPM
Testículo
5.3 TPM
OUTRAS DOENÇAS (6)
obsolete skin/hair/eye pigmentation, variation in, 1oculocutaneous albinism type 2Angelman syndrome due to maternal 15q11q13 deletionPrader-Willi syndrome due to maternal uniparental disomy of chromosome 15
HGNC:8101UniProt:Q04671
NDNNecdinCandidate gene tested inDesconhecido
FUNÇÃO

Growth suppressor that facilitates the entry of the cell into cell cycle arrest. Functionally similar to the retinoblastoma protein it binds to and represses the activity of cell-cycle-promoting proteins such as SV40 large T antigen, adenovirus E1A, and the transcription factor E2F. Necdin also interacts with p53 and works in an additive manner to inhibit cell growth. Also functions as a transcription factor and directly binds to specific guanosine-rich DNA sequences (By similarity)

LOCALIZAÇÃO

PerikaryonNucleus

VIAS BIOLÓGICAS (1)
Interleukin-4 and Interleukin-13 signaling
EXPRESSÃO TECIDUAL(Ubíquo)
Cervix Endocervix
169.7 TPM
Cervix Ectocervix
166.8 TPM
Ovário
140.4 TPM
Útero
125.6 TPM
Pituitária
108.7 TPM
OUTRAS DOENÇAS (4)
Prader-Willi syndrome due to paternal deletion of 15q11q13 type 1Prader-Willi syndrome due to imprinting mutationPrader-Willi syndrome due to paternal deletion of 15q11q13 type 2Prader-Willi syndrome due to maternal uniparental disomy of chromosome 15
HGNC:7675UniProt:Q99608
SNRPNSmall nuclear ribonucleoprotein-associated protein NCandidate gene tested inAltamente restrito
FUNÇÃO

May be involved in tissue-specific alternative RNA processing events

LOCALIZAÇÃO

Nucleus

VIAS BIOLÓGICAS (3)
mRNA Splicing - Major PathwayDengue Virus-Host InteractionsmRNA Polyadenylation
EXPRESSÃO TECIDUAL(Ubíquo)
Cérebro - Hemisfério cerebelar
258.4 TPM
Brain Frontal Cortex BA9
241.0 TPM
Cerebelo
226.7 TPM
Pituitária
189.2 TPM
Córtex cerebral
187.8 TPM
OUTRAS DOENÇAS (6)
Prader-Willi syndrome due to paternal deletion of 15q11q13 type 2Angelman syndrome due to imprinting defect in 15q11-q13Prader-Willi syndrome due to maternal uniparental disomy of chromosome 15Prader-Willi syndrome due to translocation
HGNC:11164UniProt:P63162

Medicamentos e terapias

SOMATROPINPhase 4

Mecanismo: Growth hormone receptor agonist

RIMONABANTPhase 3

Mecanismo: Cannabinoid CB1 receptor antagonist

OCTREOTIDE ACETATEPhase 3

Mecanismo: Somatostatin receptor agonist

OCTREOTIDEPhase 3

Mecanismo: Somatostatin receptor agonist

TOPIRAMATEPhase 3

Mecanismo: Sodium channel alpha subunit blocker

Ver mais no OpenTargets

Variantes genéticas (ClinVar)

1,696 variantes patogênicas registradas no ClinVar.

🧬 MAGEL2: GRCh38/hg38 15q11.2-13.1(chr15:23387531-28281759)x3 ()
🧬 MAGEL2: NM_019066.5(MAGEL2):c.2515C>G (p.Gln839Glu) ()
🧬 MAGEL2: NM_019066.5(MAGEL2):c.1757T>C (p.Ile586Thr) ()
🧬 MAGEL2: NM_019066.5(MAGEL2):c.74G>A (p.Arg25His) ()
🧬 MAGEL2: NM_019066.5(MAGEL2):c.3106C>T (p.Gln1036Ter) ()
Ver todas no ClinVar

Classificação de variantes (ClinVar)

Distribuição de 53 variantes classificadas pelo ClinVar.

19
29
5
Patogênica (35.8%)
VUS (54.7%)
Benigna (9.4%)
VARIANTES MAIS SIGNIFICATIVAS
MKRN3: NM_005664.4(MKRN3):c.137C>A (p.Ser46Ter) [Likely pathogenic]
CYFIP1: GRCh37/hg19 15q11.2(chr15:22698522-25199682)x1 [Pathogenic]
ATP10A: GRCh37/hg19 15q11.2-13.1(chr15:22770421-28635058) [Pathogenic]
SNHG14: NM_022807.5(SNRPN):c.-390-9497_-390-228del [Pathogenic]
MAGEL2: NM_019066.5(MAGEL2):c.2099dup (p.Val701fs) [Pathogenic]

Diagnóstico

Os sinais que médicos procuram e os exames que confirmam

Carregando...

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.
Aprovado2
3Fase 39
2Fase 23
·Pré-clínico11
Medicamentos catalogadosEnsaios clínicos· 5 medicamentos · 20 ensaios
✓ Aprovados — podem ser usados hoje
SOMATROPIN
Carregando informações de tratamento...

Onde tratar no SUS

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

🇧🇷 Atendimento SUS — Síndrome Prader-Willi

Centros de Referência SUS

24 centros habilitados pelo SUS para Síndrome Prader-Willi

Centros para Síndrome Prader-Willi

Detalhes dos centros

Hospital Universitário Prof. Edgard Santos (HUPES)

R. Dr. Augusto Viana, s/n - Canela, Salvador - BA, 40110-060 · CNES 0003808

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do Metabolismo

Hospital Infantil Albert Sabin

R. Tertuliano Sales, 544 - Vila União, Fortaleza - CE, 60410-794 · CNES 2407876

Serviço de Referência

Rota
Anomalias CongênitasDeficiência Intelectual

Hospital de Apoio de Brasília (HAB)

AENW 3 Lote A Setor Noroeste - Plano Piloto, Brasília - DF, 70684-831 · CNES 0010456

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do MetabolismoDeficiência Intelectual

Hospital Estadual Infantil e Maternidade Alzir Bernardino Alves (HIABA)

Av. Min. Salgado Filho, 918 - Soteco, Vila Velha - ES, 29106-010 · CNES 6631207

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do MetabolismoDeficiência Intelectual

Hospital das Clínicas da UFG

Rua 235 QD. 68 Lote Área, Nº 285, s/nº - Setor Leste Universitário, Goiânia - GO, 74605-050 · CNES 2338424

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do Metabolismo

Hospital Universitário da UFJF

R. Catulo Breviglieri, Bairro - s/n - Santa Catarina, Juiz de Fora - MG, 36036-110 · CNES 2297442

Atenção Especializada

Rota
Anomalias Congênitas

Hospital das Clínicas da UFMG

Av. Prof. Alfredo Balena, 110 - Santa Efigênia, Belo Horizonte - MG, 30130-100 · CNES 2280167

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do MetabolismoDeficiência Intelectual

Hospital Universitário Julio Müller (HUJM)

R. Luis Philippe Pereira Leite, s/n - Alvorada, Cuiabá - MT, 78048-902 · CNES 2726092

Atenção Especializada

Rota
Anomalias Congênitas

Hospital Universitário João de Barros Barreto

R. dos Mundurucus, 4487 - Guamá, Belém - PA, 66073-000 · CNES 2337878

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do MetabolismoDeficiência Intelectual

Hospital Universitário Lauro Wanderley (HULW)

R. Tabeliao Estanislau Eloy, 585 - Castelo Branco, João Pessoa - PB, 58050-585 · CNES 0002470

Atenção Especializada

Rota
Anomalias Congênitas

Instituto de Medicina Integral Prof. Fernando Figueira (IMIP)

R. dos Coelhos, 300 - Boa Vista, Recife - PE, 50070-902 · CNES 0000647

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do MetabolismoDeficiência Intelectual

Hospital Pequeno Príncipe

R. Des. Motta, 1070 - Água Verde, Curitiba - PR, 80250-060 · CNES 3143805

Serviço de Referência

Rota
Anomalias CongênitasDeficiência Intelectual

Hospital Universitário Regional de Maringá (HUM)

Av. Mandacaru, 1590 - Parque das Laranjeiras, Maringá - PR, 87083-240 · CNES 2216108

Atenção Especializada

Rota
Anomalias Congênitas

Hospital de Clínicas da UFPR

R. Gen. Carneiro, 181 - Alto da Glória, Curitiba - PR, 80060-900 · CNES 2364980

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do MetabolismoDeficiência Intelectual

Hospital Universitário Pedro Ernesto (HUPE-UERJ)

Blvd. 28 de Setembro, 77 - Vila Isabel, Rio de Janeiro - RJ, 20551-030 · CNES 2280221

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do Metabolismo

Instituto Nacional de Saúde da Mulher, da Criança e do Adolescente Fernandes Figueira (IFF/Fiocruz)

Av. Rui Barbosa, 716 - Flamengo, Rio de Janeiro - RJ, 22250-020 · CNES 2269988

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do MetabolismoDeficiência Intelectual

Hospital São Lucas da PUCRS

Av. Ipiranga, 6690 - Jardim Botânico, Porto Alegre - RS, 90610-000 · CNES 2232928

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do Metabolismo

Hospital de Clínicas de Porto Alegre (HCPA)

Rua Ramiro Barcelos, 2350 Bloco A - Av. Protásio Alves, 211 - Bloco B e C - Santa Cecília, Porto Alegre - RS, 90035-903 · CNES 2237601

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do MetabolismoDeficiência Intelectual

Hospital Universitário da UFSC (HU-UFSC)

R. Profa. Maria Flora Pausewang - Trindade, Florianópolis - SC, 88036-800 · CNES 2560356

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do Metabolismo

Hospital das Clínicas da FMUSP

R. Dr. Ovídio Pires de Campos, 225 - Cerqueira César, São Paulo - SP, 05403-010 · CNES 2077485

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do MetabolismoDeficiência Intelectual

Hospital de Base de São José do Rio Preto

Av. Brg. Faria Lima, 5544 - Vila Sao Jose, São José do Rio Preto - SP, 15090-000 · CNES 2079798

Atenção Especializada

Rota
Anomalias Congênitas

Hospital de Clínicas da UNICAMP

R. Vital Brasil, 251 - Cidade Universitária, Campinas - SP, 13083-888 · CNES 2748223

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do MetabolismoDeficiência Intelectual

Hospital de Clínicas de Ribeirão Preto (HCRP-USP)

R. Ten. Catão Roxo, 3900 - Vila Monte Alegre, Ribeirão Preto - SP, 14015-010 · CNES 2082187

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do MetabolismoDeficiência Intelectual

UNIFESP / Hospital São Paulo

R. Napoleão de Barros, 715 - Vila Clementino, São Paulo - SP, 04024-002 · CNES 2688689

Serviço de Referência

Rota
Anomalias CongênitasErros Inatos do Metabolismo
Sobre os centros SUS: Estes centros são habilitados pelo Ministério da Saúde como Serviços de Referência em Doenças Raras ou Serviços de Atenção Especializada. O atendimento é pelo SUS, com encaminhamento da rede de atenção básica.

Dados de DATASUS/CNES, SBGM, ABNeuro e Ministério da Saúde. Sempre confirme a disponibilidade diretamente com o estabelecimento.

Pesquisa ativa

Ensaios clínicos abertos e novidades científicas recentes

🟢 Recrutando agora

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

Outros ensaios clínicos

149 ensaios clínicos encontrados, 26 ativos.

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

🥈Melhor nível de evidência: Ensaio clínico
Timeline de publicações
1.691 papers (10 anos)

Mostrando amostra de 200 publicações de um total de 1.691

#1

Understanding the impact of growth hormone on ventilatory control stability in children with Prader-Willi syndrome.

European journal of pediatrics2026 Mar 20

A hallmark of Prader-Willi syndrome (PWS) is hypothalamic-pituitary axis dysfunction, which can result in reduced growth hormone (GH) production. While GH replacement therapy is common in children with PWS, it has also been implicated in the development of obstructive sleep apnoea (OSA) in some children. The mechanisms underlying this development are poorly understood but may be related to alterations in ventilatory control. Our study investigated the impact of GH treatment on ventilatory control stability during sleep in children with PWS. Polysomnographic data pre- and post-GH therapy in 25 children (aged 2mo-18y) were used to assess ventilatory control using a validated method that estimates loop gain (dimensionless ratio) from ventilation changes following spontaneous sighs during sleep. Data were analysed using linear mixed-effects modelling with GH as a fixed effect and participant as a random intercept. Covariates that could impact loop gain including age, obstructive-apnoea hypopnoea index (OAHI) and central apnoea-hypopnoea index (CAHI) were each added separately to the base model in a stepwise, manual forward selection approach. Loop gain was not altered by GH treatment (β = 0.003, 95% CI: [-0.042, 0.049], p = 0.878, Cohen's d = 0.031). Age, OAHI and CAHI did not alter the impact of GH on loop gain. No difference in sleep or respiratory characteristics were found, however 20% of children developed OSA post-GH. Initiation of GH therapy was not associated with a change in loop gain, suggesting that changes in ventilatory control are unlikely to contribute to the development of OSA in children with PWS. • Prader-Willi syndrome is associated with abnormal ventilatory control and increased risk of sleep-disordered breathing. • Growth hormone therapy may influence respiratory physiology but its effect on the stability of ventilatory control (loop gain) remains unclear. • In this cohort of children with Prader-Willi syndrome, growth hormone therapy did not alter loop gain despite inter-individual variability. • Our findings suggest that any sleep-disordered breathing that emerges following growth hormone therapy is likely driven by mechanisms other than altered loop gain.

#2

Hypercoagulability in Prader-Willi Syndrome: A case-control study exploring coagulation profiles and thrombotic risk.

Genetics in medicine : official journal of the American College of Medical Genetics2026 Feb 10

Prader-Willi syndrome (PWS) is a complex imprinting disorder associated with severe obesity and endocrine dysfunction, both contributing to increased cardiovascular morbidity. Emerging data suggest a disproportionately high incidence of thromboembolic events in PWS, potentially implicating an intrinsic hypercoagulable state. We conducted a cross-sectional, case-control study including 49 genetically confirmed PWS patients (22 pediatric and 27 adult) and 85 age-, sex-, and body-mass-index-matched controls. Participants underwent comprehensive hemostatic assessment including standard coagulation tests, thrombophilia screening, and factor VIII and von Willebrand factor (vWF: Ag) and platelet function analysis. Thrombin generation test and thromboelastography in PWS were also explored. Routine coagulation and thrombophilia parameters were largely normal across groups. Thrombin generation test and platelet function analysis were unremarkable. However, D-dimer and vWF: Ag levels were significantly elevated in both pediatric and adult PWS groups with no association to obesity or inflammatory markers. Thromboelastography showed a hypercoagulable pattern in 89.76% of PWS participants, independent of body mass index or metabolic status. This study identifies a distinct hypercoagulable profile in individuals with PWS not attributable solely to obesity and likely linked to endothelial dysfunction rather than conventional thrombophilic mechanisms. This may justify personalized thrombotic risk assessment in PWS and further investigation into preventive strategies.

#3

Clinical Presentation, Genetics, and Laboratory Testing with Integrated Genetic Analysis of Molecular Mechanisms in Prader-Willi and Angelman Syndromes: A Review.

International journal of molecular sciences2026 Jan 27

Prader-Willi (PWS) and Angelman (AS) syndromes were the first examples in humans with errors in genomic imprinting, usually from de novo 15q11-q13 deletions of different parent origin (paternal in PWS and maternal in AS). Dozens of genes and transcripts are found in the 15q11-q13 region, and may play a role in PWS, specifically paternally expressed SNURF-SNRPN and MAGEL2 genes, while AS is due to the maternally expressed UBE3A gene. These three causative genes, including their encoding proteins, were targeted. This review article summarizes and illustrates the current understanding and cause of both PWS and AS using strategies to include the literature sources of key words and searchable web-based programs with databases for integrated gene and protein interactions, biological processes, and molecular mechanisms available for the two imprinting disorders. The SNURF-SNRPN gene is key in developing complex spliceosomal snRNP assemblies required for mRNA processing, cellular events, splicing, and binding required for detailed protein production and variation, neurodevelopment, immunodeficiency, and cell migration. The MAGEL2 gene is involved with the regulation of retrograde transport and promotion of endosomal assembly, oxytocin and reproduction, as well as circadian rhythm, transcriptional activity control, and appetite. The UBE3A gene encodes a key enzyme for the ubiquitin protein degradation system, apoptosis, tumor suppression, cell adhesion, and targeting proteins for degradation, autophagy, signaling pathways, and circadian rhythm. PWS is characterized early with infantile hypotonia, a poor suck, and failure to thrive with hypogenitalism/hypogonadism. Later, growth and other hormone deficiencies, developmental delays, and behavioral problems are noted with hyperphagia and morbid obesity, if not externally controlled. AS is characterized by seizures, lack of speech, severe learning disabilities, inappropriate laughter, and ataxia. This review captures the clinical presentation, natural history, causes with genetics, mechanisms, and description of established laboratory testing for genetic confirmation of each disorder. Three separate searchable web-based programs and databases that included information from the updated literature and other sources were used to identify and examine integrated genetic findings with predicted gene and protein interactions, molecular mechanisms and functions, biological processes, pathways, and gene-disease associations for candidate or causative genes per disorder. The natural history, review of pathophysiology, clinical presentation, genetics, and genetic-phenotypic findings were described along with computational biology, molecular mechanisms, genetic testing approaches, and status for each disorder, management and treatment options, clinical trial experiences, and future strategies. Conclusions and limitations were discussed to improve understanding, clinical care, genetics, diagnostic protocols, therapeutic agents, and genetic counseling for those with these genomic imprinting disorders.

#4

Genomic Imprinting, Epigenetic Dysregulation, and Neuropsychiatric Mechanisms in Prader-Willi Syndrome: A Multi-Level Integrative Review.

Cells2026 Jan 31

Prader-Willi syndrome (PWS) is a rare imprinting-related neurodevelopmental disorder caused by loss of paternally expressed genes within the chromosome 15q11-q13 region, including SNORD116, MAGEL2, and NDN. It provides a natural model for examining how genomic imprinting disruptions shape neural development and psychiatric vulnerability. This review synthesizes current evidence to clarify the mechanistic pathways linking imprinting defects and epigenetic dysregulation to neuropsychiatric outcomes in PWS. Published studies-including patient-derived induced pluripotent stem cell (iPSC) models, animal knockout systems (e.g., Magel2-null models), transcriptomic and DNA methylation datasets, and human neuroimaging research-were identified through targeted searches of PubMed and Web of Science and integrated narratively rather than through systematic procedures. Across these data sources, deletion-type PWS is primarily associated with impaired neuronal maturation, altered serotonergic signaling, and locus-specific transcriptional dysregulation. Maternal uniparental disomy (mUPD) is characterized by broader epigenetic alterations within the imprinted domain, genome-wide transcriptional effects, dopaminergic pathway alterations, and disrupted prefrontal-limbic connectivity linked to increased psychosis risk. Importantly, available evidence supports substantial phenotypic and mechanistic overlap between PWS subtypes, with genotype-phenotype associations reflecting probabilistic tendencies rather than categorical distinctions. Collectively, convergent findings across molecular, neurochemical, and systems-level studies support a mechanistic continuum extending from imprinting defects to behavioral phenotypes. These insights position PWS as a translational model for understanding how epigenetic dysregulation contributes to psychiatric risk and highlight the need for genotype-informed, mechanistically grounded research to advance biomarker development and targeted therapeutic strategies.

#5

Unravelling Narcolepsy: A Series of Complex Pediatric Cases.

Neurology. Clinical practice2026 Apr

Narcolepsy type 1 (NT1) and narcolepsy type 2 (NT2) are rare, chronic neurologic disorders of hypersomnolence. Narcolepsy type 1 results from the selective loss of orexin-producing neurons, leading to markedly reduced levels of orexin neuropeptides in the brain and CSF. NT2 shares some symptoms with the former but has no orexin deficiency. Both disorders manifest as a spectrum of debilitating symptoms, including excessive daytime sleepiness (EDS), cataplexy (NT1 only), fragmented nocturnal sleep, sleep paralysis, and hallucinations. Diagnosis is particularly challenging, especially in pediatric patients. We describe 7 pediatric patients presenting with complex narcolepsy phenotype of EDS or cataplexy with a diverse array of comorbid genetic, neurologic, and neuropsychiatric conditions. These cases illustrate the diagnostic challenges in differentiating "primary narcolepsy" from "narcolepsy because of a medical disorder" (e.g., Prader-Willi Syndrome) or "narcolepsy associated with autism spectrum disorder or very early-onset schizophrenia." The patients underwent a comprehensive diagnostic workup, including polysomnography, multiple sleep latency testing (performed after wash-out of concomitant medications), brain magnetic resonance imaging, CSF hypocretin-1 assay, and, in case of consistent clues, autoimmune, and genetic testing. Ensuring accurate and prompt narcolepsy diagnosis allows effective and patient-centered management.

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2026

Respiratory Syncytial Virus Infection Triggering a Pulmonary Hypertensive Crisis in a Boy With Prader-Willi Syndrome-Associated Sleep-Disordered Breathing.

Cureus
2026

Epidemiology, Comorbidities, and Healthcare Costs of Prader-Willi Syndrome in South Korea Using the Korean National Health Insurance Service Database.

Journal of obesity &amp; metabolic syndrome
2026

Understanding the impact of growth hormone on ventilatory control stability in children with Prader-Willi syndrome.

European journal of pediatrics
2026

The disproportionate burden of severe obesity in youth with special needs and the role of metabolic and bariatric surgery.

Seminars in pediatric surgery
2026

Spectrum of Hypogonadism and Its Management in Adolescents With Prader-Willi Syndrome: A Retrospective Cohort Study Over 35 Years.

Clinical endocrinology
2026

ARD-101, a gut-restricted TAS2R agonist, reduces hunger in adults and promotes weight loss in DIO mice with DPP-4 inhibition.

Molecular metabolism
2026

Neonatal Bradypnea as an Under-Recognized Manifestation of Prader-Willi Syndrome: A Case Report.

Cureus
2026

Circulating levels of ghrelin and hyperphagia in patients with rare genetic neurodevelopmental disorders.

The Journal of clinical endocrinology and metabolism
2026

Methylphenidate use in hyperphagic Prader-Willi syndrome: A clinical note.

Indian journal of psychiatry
2026

Cross-Species Upregulation of MAGED2 in Liver Cancer Suggests a Role in Obesity-Driven Tumor Progression.

Current issues in molecular biology
2026

Possibilities and Limitations of Prenatal Diagnosis of Rare Imprinting Syndromes: Prader-Willi Syndrome.

Children (Basel, Switzerland)
2026

Genetic and Clinical Characteristics of Chromosome 15q11-q13 Duplication Syndrome in Chinese Children.

American journal of medical genetics. Part A
2026

Dampened surge in heart rate at respiratory event termination in children with Prader-Willi syndrome.

Sleep medicine
2026

Patient advocacy group perspectives on treatment priorities and clinical trials for the rare neurodevelopmental condition, Prader-Willi syndrome.

Orphanet journal of rare diseases
2026

Hypercoagulability in Prader-Willi Syndrome: A case-control study exploring coagulation profiles and thrombotic risk.

Genetics in medicine : official journal of the American College of Medical Genetics
2026

Clinical Presentation, Genetics, and Laboratory Testing with Integrated Genetic Analysis of Molecular Mechanisms in Prader-Willi and Angelman Syndromes: A Review.

International journal of molecular sciences
2026

Understanding the burden of endocrine and metabolic disorders in Prader-Willi syndrome: data from the Italian registry.

Journal of endocrinological investigation
2026

Genomic Imprinting, Epigenetic Dysregulation, and Neuropsychiatric Mechanisms in Prader-Willi Syndrome: A Multi-Level Integrative Review.

Cells
2026

Unravelling Narcolepsy: A Series of Complex Pediatric Cases.

Neurology. Clinical practice
2026

Management of Pathological Dental Attrition in Prader-Willi Syndrome: A Case Report Using the Personalized Radboud Strategy.

Special care in dentistry : official publication of the American Association of Hospital Dentists, the Academy of Dentistry for the Handicapped, and the American Society for Geriatric Dentistry
2026

Early onset scoliosis in syndromic and neuromuscular disorders: A multidisciplinary approach.

Journal of clinical orthopaedics and trauma
2026

Advances and challenges of precision epigenetic therapy in treating genomic imprinting diseases.

Translational pediatrics
2026

Divergent epigenetic profile underlie pubertal disorders in MKRN3-associated central precocious puberty and Prader-Willi syndrome: insights from a frameshift variant.

World journal of pediatrics : WJP
2026

Oxytocin in infants with Prader-Willi syndrome to improve dysphagia and disease trajectory.

Orphanet journal of rare diseases
2026

Markedly Low Prevalence of Fatty Liver Despite Obesity in Prader-Willi Syndrome: A Search for Protective Genetic Markers.

Journal of clinical and experimental hepatology
2026

Beyond Genetic Protection: Revisiting Hepatic Resilience in Prader-Willi Syndrome.

Journal of clinical and experimental hepatology
2026

Severity and phenotype of sleep-disordered breathing in Prader-Willi syndrome compared to obstructive sleep apnea syndrome in children.

Respiratory medicine
2025

Oxytocin Deficiency in Childhood and Adolescence: Clinical Features, Diagnostic Challenges and Therapeutic Perspectives.

Current issues in molecular biology
2026

Dual Genetic Diagnosis of Prader-Willi Syndrome and TMC1-Related Severe Congenital Hearing Loss: Diagnostic Challenges and Cochlear Implant Outcomes.

Diagnostics (Basel, Switzerland)
2026

Tubo-ovarian abscess in a young child with suspected Prader-Willi syndrome: a complication of childhood obesity.

BMJ case reports
2026

Age Does Not Affect Respiratory Characteristics in Children With Prader-Willi Syndrome Before and After Growth Hormone Therapy.

Acta paediatrica (Oslo, Norway : 1992)
2026

The Discovery of RGH-706, a Highly Efficacious MCH1 Receptor Antagonist, for the Treatment of Obesity and Insatiable Hunger.

Journal of medicinal chemistry
2026

Assessment of Nutrition Quality in People With Prader-Willi Syndrome in Australia.

Journal of human nutrition and dietetics : the official journal of the British Dietetic Association
2025

Pharmacogenomic testing for Prader-Willi syndrome: a mixed methods analysis of caregiver experiences and utilization.

Pharmacogenomics
2026

Experiences and Support Needs of Siblings of Individuals With Prader-Willi Syndrome: An Integrative Systematic Review.

Journal of applied research in intellectual disabilities : JARID
2025

Neuropeptides and the Autonomic Nervous System in Prader-Willi Syndrome.

International journal of molecular sciences
2026

Diazoxide Choline Extended-Release Tablets in Prader-Willi Syndrome: A Randomized, Double-Blind, Withdrawal Period Study.

The Journal of clinical endocrinology and metabolism
2025

GABAergic regulation of Locus coeruleus activity in necdin-deficient mice, an animal model of Prader-Willi syndrome.

Journal of neurodevelopmental disorders
2025

Barriers, Limitations, and Experiences with Clinical Trials-Treatment in Rare Diseases with Prader-Willi Syndrome as an Example.

Genes
2026

Targeted Next-Generation Sequencing of the Leptin-Melanocortin Pathway in Severe Obesity.

Obesity (Silver Spring, Md.)
2025

Transcriptomic signatures in brain and blood related to cognitive and psychiatric phenotypes of Prader-Willi syndrome.

Scientific reports
2025

Prader-Willi syndrome: A rare genetic disorder with complex clinical manifestations.

Journal of family medicine and primary care
2026

Functional Independence in Adults With Prader-Willi Syndrome: First Report Using the FIM Instrument.

American journal of medical genetics. Part A
2025

Internal diseases and molecular mechanisms causing slipped capital femoral epiphysis in children.

World journal of orthopedics
2025

Attention skills, learning and academic abilities in children and adolescents with genetic disorders: a systematic review.

Frontiers in psychology
2025

A scoping review of dietary interventions to treat obesity among Prader-Willi syndrome individuals.

Intractable &amp; rare diseases research
2025

Diagnostic Value of Exome Sequencing in Isolated Polyhydramnios.

Prenatal diagnosis
2025

EndoCompass Project: Research Roadmap for Growth Disorders.

Hormone research in paediatrics
2025

Magel2 in hypothalamic POMC neurons influences the impact of stress on anxiety-like behavior and spatial learning associated with a food reward in male mice.

Frontiers in neural circuits
2025

Delayed Diagnosis of 48XXYY Syndrome: A Case Report Highlighting the Role of G-Banding Cytogenetics.

Journal of UOEH
2025

Myokine Levels in Relation to Bone Markers and Adipokines in Children with Prader-Willi Syndrome During Growth Hormone Therapy and Dietary Intervention.

International journal of molecular sciences
2026

Multi-targeting zinc finger nuclease vector unsilences paternal UBE3A in a mouse model of Angelman syndrome.

Gene therapy
2025

The spectrum of cytogenetics and clinical profile in Robertsonian translocations: An experience of two decades from tertiary referral center in India.

Medical journal, Armed Forces India
2025

Comparing evidence-based telemental health treatments for caregivers of children with Prader Willi and Williams syndromes: feasibility, acceptability, and preliminary outcomes.

Cognitive behaviour therapy
2025

Atypical case of Rett syndrome with concurrent MECP2 gene mutation and del(15)(q22qter) karyotype: A case report and review of literature.

World journal of clinical pediatrics
2026

Prader-Willi syndrome corrected in human hypothalamic organoids.

Nature reviews. Endocrinology
2025

Prenatal diagnosis of Prader-Willi syndrome via maternal UPD15 with placental mosaicism: incidental discovery of fetal DMD carrier status.

Frontiers in genetics
2026

Recommendations for real-world evidence of efficacy and safety of GLP-1 agonists in Prader-Willi syndrome: Report of a workshop held by the Foundation for Prader-Willi Research and International Prader Willi Syndrome Organisation.

Diabetes, obesity &amp; metabolism
2025

Harnessing the microbiota-gut-brain axis to prevent and treat pediatric neurodevelopmental disorders: translational insights and strategies.

Journal of translational medicine
2025

Long-term growth hormone effects in Prader-Willi syndrome: A systematic review and meta-analysis.

Saudi medical journal
2025

Oxytocin neurons drive melanocortin circuit maturation via vesicle release during a neonatal critical period.

PLoS biology
2025

Food responsiveness, addiction, and hyperphagia in Prader-Willi syndrome: a cross-sectional study of 210 Chinese patients.

Frontiers in endocrinology
2025

Sleep-disordered breathing in Prader-Willi syndrome: Two illustrative examples.

Respiratory medicine case reports
2025

Effects of probiotics on patients with Prader-Willi syndrome: a systematic review and meta-analysis of randomized controlled trials.

Frontiers in nutrition
2025

Clinical Case of Comorbid Course of Metabolically Associated Fatty Liver and Pancreas Disease in a Child with Prader-Willi Syndrome.

Journal of mother and child
2025

Eating behaviour and eating disorders in individuals with rare neurodevelopmental variants: current knowledge and future research directions.

Frontiers in psychiatry
2025

In-depth behavioral characterization of a rat model of Schaaf-Yang syndrome.

Scientific reports
2025

Effective Conservative Management of Severe Scoliosis in a Girl with Prader-Willi Syndrome: A 20-Year Case Study Follow-Up.

Journal of clinical medicine
2025

Imprinting Disorders and Epigenetic Alterations in Children Conceived by Assisted Reproductive Technologies: Mechanisms, Clinical Outcomes, and Prenatal Diagnosis.

Genes
2025

Rescue of imprinted genes by epigenome editing in human cellular models of Prader-Willi syndrome.

Nature communications
2025

Growth hormone treatment in adults with Prader-Willi syndrome: an update.

Expert review of endocrinology &amp; metabolism
2025

Systematic Review of Intervention Programs Designed to Improve the Socioemotional Skills of Children and Adolescents With Prader-Willi Syndrome.

American journal on intellectual and developmental disabilities
2025

Prader-Willi Syndrome in Adulthood: A Case Report of Dermatologic and Ophthalmic Features Not Well Documented in the Literature.

Cureus
2026

Clinical significance and association with pregnancy outcome of positive non-invasive prenatal screening for trisomy 15 in singleton pregnancy: prospective cohort study, systematic review and meta-analysis.

Ultrasound in obstetrics &amp; gynecology : the official journal of the International Society of Ultrasound in Obstetrics and Gynecology
2026

Codesigning a Neurocognitive Assessment Protocol for Hyperphagia: Perspectives From Stakeholders in Prader-Willi Syndrome.

Journal of intellectual disability research : JIDR
2025

Long-term intranasal oxytocin therapy in patients with hypothalamic syndrome: case series and literature review.

Endocrine connections
2025

Management of a complex tibial fracture in a patient with Prader-Willi syndrome and severe obesity.

Journal of Yeungnam medical science
2026

Modern Luque Trolley technique in the surgical management of early onset scoliosis: a case report of a patient followed to maturity and final fusion.

Spine deformity
2026

Evaluating DNA methylation episignatures as a first-tier diagnostic test in individuals with suspected genetic disorders.

European journal of human genetics : EJHG
2025

Multidimensional Characterisation of Eating Behaviour in Genetic Obesity: A Systematic Review.

Obesity facts
2025

Weight Loss Effect of Lisdexamfetamine in Children with Severe Obesity: A Case Series.

Childhood obesity (Print)
2025

Late Diagnosis of Prader-Willi Syndrome in an Adolescent With Significant Complications of Type 2 Diabetes.

JCEM case reports
2025

Beyond the usual suspects: neonatal presentation of Prader-Willi syndrome.

BMJ case reports
2025

Experiences and Support Needs of Siblings of Individuals With Prader-Willi Syndrome- Findings From a Two-Stage Qualitative Study.

Journal of applied research in intellectual disabilities : JARID
2025

Preliminary Report on Temperature Dysregulation in a Cohort of Youth with Prader-Willi Syndrome.

Reports (MDPI)
2025

Glycemic and renal effects of SGLT2 Inhibitors in Prader-Willi syndrome: Benefits and risks.

Diabetes &amp; metabolism
2026

High Rate of Dysphagia and Silent Aspiration in Infants With Prader-Willi Syndrome-Considering Laryngeal Clefts.

American journal of medical genetics. Part A
2025

Ovarian Sex Cord Tumor With Annular Tubules (SCTAT) Harbor Recurrent Copy Number Alterations, Including Monosomy 22.

Modern pathology : an official journal of the United States and Canadian Academy of Pathology, Inc
2025

Chromosome 15q Structural Variants Associated with Syndromic Autism Spectrum Disorder: Clinical and Genomic Insights from Three Case Reports in a Brazilian Reference Center.

International journal of molecular sciences
2025

Pitolisant may lessen not only sleepiness but improve hyperphagia and behavior problems in Prader-Willi syndrome.

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

Long-term impact of growth hormone therapy on mortality and type 2 diabetes in Prader-Willi syndrome: a nationwide cohort study.

Frontiers in endocrinology
2025

Bariatric Surgery in Patients With Prader-Willi Syndrome.

Journal of metabolic and bariatric surgery
2026

Sleep disorder assessment in children and adolescents with neurodevelopmental disorders.

Jornal de pediatria
2025

Development of disease-specific growth charts for Argentine Prader-Willi syndrome without growth hormone treatment.

Annals of human biology
2025

Pharmacotherapy and metabolic/bariatric surgery: either or both?

International journal of obesity (2005)
2026

Loss of Necdin causes social deficit and aberrant synaptic function through destabilization of SynGAP.

Molecular psychiatry
2025

Maternal uniparental disomy of chromosome 15 with concurrent paternal non-chromosome 15 marker chromosome: a rare presentation of prader-willi syndrome.

Molecular cytogenetics
2025

Altered Behavior and Neuronal Activity with Paternal Snord116 Deletion.

Genes
2025

Atypical Prader-Willi Syndrome Deletions: Insights Into the Complex Regulation and Phenotypic Variability.

Molecular genetics &amp; genomic medicine
2025

High-throughput assessment of FMR1 and SNRPN methylation-based newborn screening using IsoPure and QIAcube HT systems.

Epigenomics
2025

Sex Hormone Replacement Therapy and Bleeding Patterns among Adolescents and Young Adult Females with Prader-Willi Syndrome.

Journal of pediatric and adolescent gynecology
2025

Transoral outlet reduction (TORe) for treatment of weight regain after biliopancreatic diversion in a patient with Prader-Willi syndrome and super-super obesity: report of the first case.

Updates in surgery
2026

The Diagnostic and Therapeutic Challenges of Schaaf-Yang Syndrome: A Brazilian Case Report.

Journal of child neurology
2025

Relationship between body mass index and nutritional status across genetic subtypes of Prader-Willi syndrome.

Clinical nutrition ESPEN
2025

Molecular characterization of imprinting disorders: Beckwith-Wiedemann, Silver-Russell, and Prader-Willi syndromes in Egyptian patients.

BMC pediatrics
2025

The burden of illness in Prader-Willi syndrome: a systematic literature review.

Orphanet journal of rare diseases
2025

A questionnaire-based survey on hyperphagia in individuals with Prader-Willi syndrome in Japan.

Endocrine journal
2025

Growth Hormone Treatment in Patients With KBG Syndrome: Novel Insights, Challenges and Recommendations From Six New Patients and Literature Review.

American journal of medical genetics. Part A
2025

Light and sex modify Snord116 genotype effects on metabolism, behavior, and imprinted gene networks following circadian entrainment.

bioRxiv : the preprint server for biology
2025

Assessing Metabolic Syndrome Risk in Children and Adolescents with Prader-Willi Syndrome: A Comparison of Index Performance.

Journal of clinical medicine
2025

Prenatal Phenotype in a Neonate with Prader-Willi Syndrome and Literature Review.

Diagnostics (Basel, Switzerland)
2025

GNB1 haploinsufficiency presents as monogenic obesity syndrome.

Journal of pediatric endocrinology &amp; metabolism : JPEM
2025

Mechanism of EHMT2-mediated genomic imprinting associated with Prader-Willi syndrome.

Nature communications
2025

A proof-of-concept study of pitolisant for excessive daytime sleepiness in patients with Prader-Willi syndrome.

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

Mixed Segmental Uniparental Disomy of Chromosome 15q11-q1 Coexists with Homozygous Variant in GNB5 Gene in Child with Prader-Willi and Lodder-Merla Syndrome.

Genes
2025

Efficacy and safety of pitolisant in children above 6 years with narcolepsy.

Expert opinion on pharmacotherapy
2025

Biallelic variants in SREK1 downregulating SNORD115 and SNORD116 cause a Prader-Willi-like syndrome.

The Journal of clinical investigation
2025

Sensorineural deafness in a child with Prader-Willi Syndrome-A rare case report.

Journal of family medicine and primary care
2025

Assessment of hypothalamic-pituitary-adrenal axis impairment and effects of hydrocortisone treatment in adults with Prader-Willi syndrome.

Frontiers in endocrinology
2025

Role of mitochondrial function in the oxidative stress profile of children with Prader-Willi syndrome.

Free radical biology &amp; medicine
2025

Biallelic pathogenic variants in POMC can cause combined pituitary hormonal deficiency associated with severe obesity.

European journal of endocrinology
2025

Sertraline-Induced Mood and Behavioral Activation in Two Adults With Prader-Willi Syndrome.

Case reports in psychiatry
2025

Health outcomes of children with Prader-Willi or Angelman syndromes: a European population-based multicentre study.

Archives of disease in childhood
2025

New drug approved for hyperphagia in Prader-Willi syndrome.

The lancet. Diabetes &amp; endocrinology
2025

Hexasomy of the 15q11q13 region: a detailed report and review of the literature.

European journal of medical genetics
2025

Improvement in body composition of Japanese participants with Prader-Willi syndrome following somatropin treatment: an open-label, multi cohort Phase 3 study.

Endocrine journal
2025

Effects of microbiota-based interventions on depression and anxiety in children and adolescents-A systematic review.

Journal of pediatric gastroenterology and nutrition
2025

Prader Willi syndrome: advances in genetics.

Advances in genetics
2025

High Rates of Dysphagia and Silent Aspiration in Infants With Prader-Willi Syndrome.

American journal of medical genetics. Part A
2025

Serum Lipoprotein(a) and High-Sensitivity C-reactive Protein Correlate With Somatic Parameters Including MLPA Subgroups in Children With Prader-Willi Syndrome.

Journal of the Endocrine Society
2025

A Randomized Double-Blind Placebo-Controlled Trial of Guanfacine Extended Release for Aggression and Self-Injurious Behavior Associated With Prader-Willi Syndrome.

American journal of medical genetics. Part B, Neuropsychiatric genetics : the official publication of the International Society of Psychiatric Genetics
2025

Targeting Histone H3K9 Methyltransferase G9a as a Potential Therapeutic Strategy for Neuropsychiatric Disorders.

Medicinal research reviews
2025

Hyperactive Catatonia in an Adolescent With Prader-Willi Syndrome.

Cureus
2025

[Sleep disorders in imprinting disorders].

Zhurnal nevrologii i psikhiatrii imeni S.S. Korsakova
2025

Association of ring chromosome 18 and Prader-Willi syndrome: the first described case report.

Pediatric endocrinology, diabetes, and metabolism
2026

Autonomic Control of Heart Rate During Sleep Is Depressed in Young Children With Prader-Willi Syndrome.

Journal of sleep research
2025

A new homozygous pathogenic LEPR variant causing severe, early onset obesity in a Senegalese child.

Obesity research &amp; clinical practice
2025

Variant pubertal development in Prader-Willi syndrome: early and slow progression of pubarche with normal age at gonadarche.

Frontiers in endocrinology
2025

Depression, Anxiety of Death, and Fear of Death in Family Caregivers of People With Prader-Willi Syndrome: A Mixed Study.

Global advances in integrative medicine and health
2025

Comparison of Body Composition, Basal Metabolic Rate and Metabolic Outcomes of Adults with Prader-Willi Syndrome and Age- and BMI-Matched Patients with Essential Obesity.

Journal of clinical medicine
2025

Effectiveness of topiramate in the treatment of behavioural disorders in Prader-Willi syndrome.

Journal of psychiatry &amp; neuroscience : JPN
2025

Diazoxide choline (Vykat XR) for Prader-Willi syndrome-associated hyperphagia.

The Medical letter on drugs and therapeutics
2025

Mom genes and dad genes: genomic imprinting in the regulation of social behaviors.

Epigenomics
2025

Anesthesia management for patients with Prader-Willi syndrome undergoing bariatric surgery: a single-center retrospective case series study.

BMC anesthesiology
2025

Height loss with age in adults with Prader-Willi syndrome may result in artifactual increases in BMI.

Scientific reports
2025

Cytokine response to resistance exercise in children with excess adiposity and Prader-Willi syndrome.

Physiological reports
2025

A Case of Prader-Willi Syndrome With a Deletion Including MAGEL2 , NDN , and MKRN3 , but Excluding SNRPN and SNORD116.

American journal of medical genetics. Part A
2025

A case report of Prader-Willi syndrome in a child with metabolic disorders and severe obstructive sleep apnea treated effectively with continuous positive airway pressure.

Translational pediatrics
2025

Life Satisfaction, Global Health and Mood in Prader-Willi Syndrome: Use of PROMIS and Glasgow Depression Scales.

Journal of applied research in intellectual disabilities : JARID
2025

Classic Prader-Willi Syndrome Phenotype Caused by an Atypical Deletion in the 15q11 Region Not Involving the SNORD Genes.

Clinical genetics
2025

Long-Term Efficacy and Safety of Growth Hormone in Children Suffering from Short Stature in China (CGLS): An Open-Label, Multicenter, Prospective and Retrospective, Observational Study.

Advances in therapy
2025

Sleep-Disordered Breathing and Central Respiratory Control in Children: A Comprehensive Review.

Children (Basel, Switzerland)
2025

Neuroglia in eating disorders (obesity, Prader-Willi syndrome and anorexia nervosa).

Handbook of clinical neurology
2025

A dual effect of FUBP1 on SPA lncRNA maturation.

RNA (New York, N.Y.)
2025

The Role of the Arcuate Nucleus in Regulating Hunger and Satiety in Prader-Willi Syndrome.

Current issues in molecular biology
2025

Loss of Snord116 protects cardiomyocyte kinetics during ischemic stress.

Journal of molecular and cellular cardiology plus
2025

Personalized endpoints in Prader-Willi syndrome: a case study with goal attainment scaling.

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

Fetal cardiac rhabdomyoma incidentally associated with Prader-Willi syndrome: A case report.

European journal of obstetrics, gynecology, and reproductive biology
2025

The prevalence and surgical outcome of late diagnosed hip dysplasia in children with Prader-Willi syndrome: a retrospective study.

BMC musculoskeletal disorders
2025

Prader-Willi syndrome gene expression profiling of obese and non-obese patients reveals transcriptional changes in CLEC4D and ANXA3.

Journal of pediatric endocrinology &amp; metabolism : JPEM
2025

Footprints in the Sno: investigating the cellular and molecular mechanisms of SNORD116.

Open biology
2025

Epigenetic Age in Prader-Willi Syndrome and Essential Obesity: A Comparison with Chronological and Vascular Ages.

Journal of clinical medicine
2025

A Transcriptomic Signature of Depressive Symptoms in Late Life.

Biological psychiatry global open science
2025

Neglected Chronically Dislocated Hip in a Prader-Willi Child: A Case Report and Literature Review.

Journal of orthopaedic case reports
2025

Adenotonsillectomy success for treating obstructive sleep apnea in children with Prader-Willi syndrome.

International journal of pediatric otorhinolaryngology
2025

Early-onset growth hormone treatment in Prader-Willi syndrome attenuates transition to severe obesity.

Journal of pediatric endocrinology &amp; metabolism : JPEM
2025

Management of obstructive sleep apnea-hypopnea syndrome in children: what is the role of orthodontics? A scoping review.

Sleep &amp; breathing = Schlaf &amp; Atmung
2025

Gynecological issues in children and adolescents seen at rare-disease referral centers: an observational retrospective cohort study.

Orphanet journal of rare diseases
2025

The Effects of Bilingualism on the Executive Control Abilities of the Prader-Willi Syndrome Population.

Journal of psycholinguistic research
2025

Selective changes in vasopressin neurons and astrocytes in the suprachiasmatic nucleus of Prader-Willi syndrome subjects.

Journal of neuroendocrinology
2025

Investigation of a mouse model of Prader-Willi Syndrome with combined disruption of Necdin and Magel2.

JCI insight
2025

Vocabulary and reading skills in adults with Prader-Willi syndrome.

Journal of communication disorders
2025

Fiber Intervention Study in Prader-Willi Syndrome: Insights into Metabolic and Microbiota Shifts.

The Journal of clinical endocrinology and metabolism
2025

Early oxytocin treatment in infants with Prader-Willi syndrome is safe and is associated with better endocrine, metabolic and behavioral outcomes.

Orphanet journal of rare diseases
2025

NDUFB7 mutations cause brain neuronal defects, lactic acidosis, and mitochondrial dysfunction in humans and zebrafish.

Cell death discovery
2025

Circadian rhythm defects in Prader-Willi syndrome neurons.

HGG advances
2025

Comorbidities, Endocrine Medications, and Mortality in Prader-Willi Syndrome-A Swedish Register Study.

Journal of clinical medicine
2025

Efficacy and safety of once-weekly semaglutide monotherapy in a young subject with Prader-Willi syndrome, obesity, and type 2 diabetes: a case report.

Frontiers in endocrinology
2025

Functional analysis of a novel homozygous missense IVD gene variant: a case report with dual genetic diagnoses.

Frontiers in pediatrics
2025

Anxiety, Depression and Stress in Parents and Siblings of People Who Have Prader-Willi Syndrome: Morbidity Prevalence and Mitigating Factors.

Journal of intellectual disability research : JIDR
2025

Parents' Experiences and Views About Use of Wearable Technology for Research and Treatment Monitoring of Children with Neurodevelopmental Disorders.

Journal of developmental and behavioral pediatrics : JDBP
2025

Activation of the imprinted Prader-Willi syndrome locus by CRISPR-based epigenome editing.

Cell genomics
2025

Perception of four intellectual and developmental disabilities based on search engine and news portrayal.

PloS one
2025

Validation of the Food Safe Zone questionnaire for families of individuals with Prader-Willi syndrome.

Journal of neurodevelopmental disorders
2025

Management of Obesity-Related Genetic Disorders.

Endocrinology and metabolism clinics of North America
2024

Case report: Long-term efficacy and safety of semaglutide in the treatment of syndromic obesity in Prader Willi syndrome - case series and literature review.

Frontiers in endocrinology
2025

Deep brain stimulation of the hypothalamic region: a systematic review.

Acta neurochirurgica
2025

Wearable sensors in paediatric neurology.

Developmental medicine and child neurology
2025

Targeting of retrovirus-derived Rtl8a/8b causes late-onset obesity, reduced social response and increased apathy-like behaviour.

Open biology
2025

Pharmacological Aspects in the Management of Children and Adolescents with Prader-Willi Syndrome.

Paediatric drugs
2025

Modulation of respiration and hypothalamus.

Vitamins and hormones
2024

GH Therapy in Non-Growth Hormone-Deficient Children.

Children (Basel, Switzerland)
2025

Assessment of Quality of Life and Psychological Well-Being in Italian Adult Subjects with Prader-Willi Syndrome Using the Health Survey Short Form and the Psychological General Well-Being Index Questionnaires.

Healthcare (Basel, Switzerland)
2025

A review of Prader-Willi syndrome.

JAAPA : official journal of the American Academy of Physician Assistants
2025

Current practices in MRI screening in early onset scoliosis.

Spine deformity
Ver todos os 2.895 no EuropePMC

Associações

Organizações que acompanham esta doença — pra ter apoio e orientação

Centro de Genética Humana , Instituto de Ciências Biológicas, Universidade Federal de Goiás (CEGH-ICB/UFG)

Comunidades

Grupos ativos de quem convive com esta doença aqui no Raras

Ainda não existe comunidade no Raras para Síndrome Prader-Willi

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Perguntas, dicas e experiências compartilhadas aqui na página

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Doenças relacionadas

Doenças com sintomas parecidos — ajudam quem ainda está buscando diagnóstico

Ordenadas pelo número de sintomas em comum.

Referências e fontes

Bases de dados externas citadas neste artigo

Publicações científicas

Artigos indexados no PubMed ligados a esta doença no grafo RarasNet — título, periódico e PMID direto da fonte, sem intermediação de IA.

  1. Understanding the impact of growth hormone on ventilatory control stability in children with Prader-Willi syndrome.
    European journal of pediatrics· 2026· PMID 41857417mais citado
  2. Hypercoagulability in Prader-Willi Syndrome: A case-control study exploring coagulation profiles and thrombotic risk.
    Genetics in medicine : official journal of the American College of Medical Genetics· 2026· PMID 41685566mais citado
  3. Clinical Presentation, Genetics, and Laboratory Testing with Integrated Genetic Analysis of Molecular Mechanisms in Prader-Willi and Angelman Syndromes: A Review.
    International journal of molecular sciences· 2026· PMID 41683698mais citado
  4. Genomic Imprinting, Epigenetic Dysregulation, and Neuropsychiatric Mechanisms in Prader-Willi Syndrome: A Multi-Level Integrative Review.
    Cells· 2026· PMID 41677631mais citado
  5. Unravelling Narcolepsy: A Series of Complex Pediatric Cases.
    Neurology. Clinical practice· 2026· PMID 41669756mais citado
  6. [Neurocognitive profile and vulnerability for mental health problems in selected genetic syndromes with disorders of intellectual development].
    Nervenarzt· 2026· PMID 41979662recente
  7. Case Report: Schaaf-Yang Syndrome Milder Phenotype Due to Potential Pathogenic Novel Missense Variant as an Unusual Cause of Obesity in a Pediatric Patient.
    Appl Clin Genet· 2026· PMID 41937924recente
  8. Early neurodevelopmental brain perfusion abnormalities and functional connectivity findings in infants with Prader-Willi syndrome.
    J Neurodev Disord· 2026· PMID 41937185recente
  9. The pharmacological management of obesity in Prader-Willi syndrome: what does the future hold?
    Expert Opin Pharmacother· 2026· PMID 41925226recente
  10. Two siblings with Schaaf-Yang syndrome treated with growth hormone.
    Clin Pediatr Endocrinol· 2026· PMID 41923794recente

Bases de dados e fontes oficiais

Identificadores e referências canônicas usadas para montar este verbete.

  1. ORPHA:739(Orphanet)
  2. OMIM OMIM:176270(OMIM)
  3. MONDO:0008300(MONDO)
  4. GARD:5575(GARD (NIH))
  5. Variantes catalogadas(ClinVar)
  6. Busca completa no PubMed(PubMed)
  7. Artigo Wikipedia(Wikipedia)
  8. Q594013(Wikidata)

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

Conteúdo mantido por Agente Raras · Médicos e pesquisadores podem colaborar

Síndrome Prader-Willi
Compêndio · Raras BR

Síndrome Prader-Willi

ORPHA:739 · MONDO:0008300
Prevalência
1-9 / 100 000
Herança
Autosomal dominant, Not applicable
CID-10
Q87.1 · Síndromes com malformações congênitas associadas predominantemente com nanismo
CID-11
Ensaios
26 ativos
Medicamentos
5 registrados
Início
Antenatal, Neonatal
Prevalência
0.0 (Worldwide)
MedGen
UMLS
C0032897
EuropePMC
Wikidata
Wikipedia
Papers 10a
Evidência
🥈 Ensaio clínico
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