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Colestase intra-hepática recorrente benigna
ORPHA:65682CID-10 · K83.1CID-11 · 5C58.04DOENÇA RARA

A colestase intra-hepática recorrente benigna (BRIC) é uma doença hepática hereditária caracterizada por episódios intermitentes de colestase intra-hepática, geralmente sem progressão para lesão hepática crônica. Acredita-se agora que o BRIC pertence a um espectro clínico de distúrbios colestáticos intra-hepáticos que varia desde ataques intermitentes leves no BRIC até a colestase grave, crônica e progressiva observada na colestase intra-hepática familiar progressiva (PFIC).

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Introdução

O que você precisa saber de cara

📋

A colestase intra-hepática recorrente benigna (BRIC) é uma doença hepática hereditária caracterizada por episódios intermitentes de colestase intra-hepática, geralmente sem progressão para lesão hepática crônica. Acredita-se agora que o BRIC pertence a um espectro clínico de distúrbios colestáticos intra-hepáticos que varia desde ataques intermitentes leves no BRIC até a colestase grave, crônica e progressiva observada na colestase intra-hepática familiar progressiva (PFIC).

Publicações científicas
249 artigos
Último publicado: 2026 Apr
Medicamentos
1 registrados
ODEVIXIBAT

Tem tratamento?

1 medicamento registrado
Ver detalhes, fases e interações →
ODEVIXIBAT

Escala de raridade

CLASSIFICAÇÃO ORPHANET · BRASIL 2024
Unknown
Ultra-rara
<1/50k
Muito rara
1/20k
Rara
1/10k
Pouco freq.
1/5k
Incomum
1/2k
Prevalência
0.0
Worldwide
Início
All ages
🏥
SUS: Sem cobertura SUSScore: 0%
CID-10: K83.1
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Entender a doença

Do básico ao detalhe, leia no seu ritmo

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Sinais e sintomas

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

Partes do corpo afetadas

🫃
Digestivo
13 sintomas
📏
Crescimento
3 sintomas
👂
Ouvidos
1 sintomas
🦴
Ossos e articulações
1 sintomas

+ 10 sintomas em outras categorias

Características mais comuns

90%prev.
Perda de peso
Muito frequente (99-80%)
90%prev.
Doença hepática colestática
Muito frequente (99-80%)
90%prev.
Concentração elevada de transaminase hepática circulante
Muito frequente (99-80%)
90%prev.
Prurido
Muito frequente (99-80%)
90%prev.
Fezes acólicas
Muito frequente (99-80%)
90%prev.
Fadiga
Muito frequente (99-80%)
28sintomas
Muito frequente (8)
Frequente (1)
Ocasional (7)
Sem dados (12)

Os sintomas variam de pessoa para pessoa. Abaixo estão as 28 características clínicas mais associadas, ordenadas por frequência.

Perda de pesoWeight loss
Muito frequente (99-80%)90%
Doença hepática colestáticaCholestatic liver disease
Muito frequente (99-80%)90%
Concentração elevada de transaminase hepática circulanteElevated circulating hepatic transaminase concentration
Muito frequente (99-80%)90%
PruridoPruritus
Muito frequente (99-80%)90%
Fezes acólicasAcholic stools
Muito frequente (99-80%)90%

Linha do tempo da pesquisa

Publicações por ano — veja quando o interesse científico cresceu
Anos de pesquisa1desde 2026
Total histórico249PubMed
Últimos 10 anos82publicações
Pico202114 papers
Linha do tempo
2026Hoje · 2026🧪 2010Primeiro ensaio clínico📈 2021Ano 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

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

ABCB11Bile salt export pumpDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Catalyzes the transport of the major hydrophobic bile salts, such as taurine and glycine-conjugated cholic acid across the canalicular membrane of hepatocytes in an ATP-dependent manner, therefore participates in hepatic bile acid homeostasis and consequently to lipid homeostasis through regulation of biliary lipid secretion in a bile salts dependent manner (PubMed:15791618, PubMed:16332456, PubMed:18985798, PubMed:19228692, PubMed:20010382, PubMed:20398791, PubMed:22262466, PubMed:24711118, Pub

LOCALIZAÇÃO

Apical cell membraneRecycling endosome membraneEndosomeCell membrane

VIAS BIOLÓGICAS (2)
Synthesis of bile acids and bile salts via 7alpha-hydroxycholesterolRecycling of bile acids and salts
MECANISMO DE DOENÇA

Cholestasis, progressive familial intrahepatic, 2

A disorder characterized by early onset of cholestasis that progresses to hepatic fibrosis, cirrhosis, and end-stage liver disease before adulthood. PFIC2 inheritance is autosomal recessive.

OUTRAS DOENÇAS (3)
progressive familial intrahepatic cholestasis type 2benign recurrent intrahepatic cholestasis type 2intrahepatic cholestasis of pregnancy
HGNC:42UniProt:O95342
ATP8B1Phospholipid-transporting ATPase ICDisease-causing germline mutation(s) inTolerante
FUNÇÃO

Catalytic component of a P4-ATPase flippase complex which catalyzes the hydrolysis of ATP coupled to the transport of phospholipids, in particular phosphatidylcholines (PC), from the outer to the inner leaflet of the plasma membrane (PubMed:17948906, PubMed:25315773). May participate in the establishment of the canalicular membrane integrity by ensuring asymmetric distribution of phospholipids in the canicular membrane (By similarity). Thus may have a role in the regulation of bile acids transpo

LOCALIZAÇÃO

Cell membraneApical cell membraneCell projection, stereociliumEndoplasmic reticulumGolgi apparatus

VIAS BIOLÓGICAS (1)
Ion transport by P-type ATPases
MECANISMO DE DOENÇA

Cholestasis, progressive familial intrahepatic, 1

A disorder characterized by early onset of cholestasis that progresses to hepatic fibrosis, cirrhosis, and end-stage liver disease before adulthood. PFIC1 inheritance is autosomal recessive.

OUTRAS DOENÇAS (4)
cholestasis, intrahepatic, of pregnancy, 1progressive familial intrahepatic cholestasis type 1benign recurrent intrahepatic cholestasis type 1intrahepatic cholestasis of pregnancy
HGNC:3706UniProt:O43520

Medicamentos e terapias

ODEVIXIBATPhase 3

Mecanismo: Ileal bile acid transporter inhibitor

Ver mais no OpenTargets

Variantes genéticas (ClinVar)

700 variantes patogênicas registradas no ClinVar.

🧬 ATP8B1: GRCh38/hg38 18q11.1-23(chr18:20966775-80255845)x3 ()
🧬 ATP8B1: GRCh37/hg19 18q21.2-22.2(chr18:52640210-68070259)x4 ()
🧬 ATP8B1: GRCh37/hg19 18q21.2-23(chr18:53564430-74587425)x1 ()
🧬 ATP8B1: NM_001374385.1(ATP8B1):c.1473+1G>C ()
🧬 ATP8B1: NM_001374385.1(ATP8B1):c.637C>A (p.Leu213Ile) ()
Ver todas no ClinVar

Classificação de variantes (ClinVar)

Distribuição de 267 variantes classificadas pelo ClinVar.

254
13
Patogênica (95.1%)
VUS (4.9%)
VARIANTES MAIS SIGNIFICATIVAS
ALPK2: Single allele [Pathogenic]
ATP8B1: NM_001374385.1(ATP8B1):c.958_967del (p.Met320fs) [Likely pathogenic]
ABCB11: NM_003742.4(ABCB11):c.1549C>T (p.Arg517Cys) [Likely pathogenic]
ABCB11: NM_003742.4(ABCB11):c.3355A>G (p.Lys1119Glu) [Likely pathogenic]
ABCB11: NM_003742.4(ABCB11):c.3422G>T (p.Gly1141Val) [Likely pathogenic]

Diagnóstico

Os sinais que médicos procuram e os exames que confirmam

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Tratamento e manejo

Remédios, cuidados de apoio e o que precisa acompanhar

Pipeline de tratamentos
Pipeline regulatório — de medicamentos já aprovados a drogas em pesquisa exploratória.
3Fase 31
·Pré-clínico1
Medicamentos catalogadosEnsaios clínicos· 1 medicamento · 1 ensaio
Carregando informações de tratamento...

Onde tratar no SUS

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

🇧🇷 Atendimento SUS — Colestase intra-hepática recorrente benigna

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Ensaios clínicos abertos e novidades científicas recentes

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Publicações mais relevantes

Timeline de publicações
83 papers (10 anos)
#1

Baseline analysis of patient reported outcomes in the progressive familial intrahepatic cholestasis patient registry.

Journal of pediatric gastroenterology and nutrition2026 Jan 29

Progressive familial intrahepatic cholestasis (PFIC) is an overarching term for rare monogenic defects that result in cholestatic liver disease. Larger consortia-based registries have begun to address the challenges of PFIC as a rare disease with variable phenotype, but patient-reported outcomes (PROs) have not been extensively described. Here, we report baseline analysis from the PFIC network patient registry (PNPR), an effort to report outcome measures identified as meaningful and impactful to those living with the disease. The PNPR is a prospective, international, voluntary patient registry collecting longitudinal PROs relevant to PFIC and its complications, including diagnosis, symptoms, surgeries, medications as well as validated measures related to itch, sleep, and general health (patient-reported outcomes measurement information system [PROMIS] measures), disease impact on family quality of life (QoL), and financial burden of the disease. Baseline data from 161 international patients were included. Registrants included patients affected by several subtypes and benign recurrent intrahepatic cholestasis, and 19 participants with an unknown or missing diagnosis. Pruritus is an important contributor to morbidity with severity of itch positively correlating with sleep disturbance and sleep impairment and negatively correlating with family QoL and overall health. The financial burden of disease was reflected by higher out-of-pocket medical costs and more reported challenges arranging medical care compared to the general US population. The PNPR fills a previously identified gap in PFIC research-the lack of PROs-and reveals the negative impact of disease and pruritus on patient and family function, QoL indicators, finances, and measures of general health.

#2

Enigmatic functions of ATP8B1: cholestasis, inflammation, phosphoinositide flipping, and cellular homeostasis.

Cell cycle (Georgetown, Tex.)2025

Mutations in ATP8B1 cause a spectrum of cholestatic liver disease, ranging from Progressive-Familial-Intrahepatic-Cholestasis type-1 (PFIC1) to Benign-Recurrent-Intrahepatic-Cholestasis type-1 (BRIC1). Manifestations of PFIC1 include severe pruritus, jaundice, and liver damage. Extrahepatic features sometimes observed in PFIC1 include sensorineural hearing loss, diarrhea, pancreatitis, and short stature. ATP8B1 was shown to translocate phospholipids across the plasma membrane; however, expression of ATP8B1 in many tissues and the range of pathological manifestations in ATP8B1 deficiency suggest diverse physiological functions of ATP8B1, and pleiotropic mechanisms regulating its activity. Recent studies suggest that phosphoinositides, including PIP2 and PIP3, can function as regulators, substrates, and binding partners of ATP8B1. New research shows that ATP8B1 modulates host immune system by regulating cleavage of pyroptotic-executioner Gasdermin D (GSDMD), and inflammation-resolution pathways such as phagocytosis/efferocytosis. Further mechanistic insights can accelerate development of new therapies for restoring membrane integrity, reducing inflammasome activity, and correcting metabolic imbalances caused by ATP8B1 dysfunction.

#3

Benign Recurrent Intrahepatic Cholestasis Type 1 with Novel Gene Mutation Complicated by Distal Renal Tubular Acidosis: A Case Report.

Case reports in gastroenterology2025

Benign recurrent intrahepatic cholestasis (BRIC) is a rare autosomal recessive inherited disorder characterized by intermittent jaundice episodes. We report a 25-year-old male patient with numerous hospitalizations for jaundice. The diagnosis of BRIC type 1 was established by liver biopsy, genetic analysis, and the exclusion of alternative etiologies of cholestasis. A novel missense heterozygous variant was identified in the ATP8B1 gene (c.2081T>A). The acute cholestatic attack, along with associated complications such as distal renal tubular acidosis and acute pancreatitis, was successfully managed with plasmapheresis and rifampicin. Novel mutations that differ from those documented in the literature in combination with renal tubular acidosis may enhance our comprehension of this topic.

#4

Plasmapheresis can improve clinical outcomes in patients with therapy resistant benign recurrent intrahepatic cholestasis: A case report.

Transfusion and apheresis science : official journal of the World Apheresis Association : official journal of the European Society for Haemapheresis2025 Jun

Due to the rarity of benign recurrent intrahepatic cholestasis (BRIC), limited literature exists regarding the therapeutic benefit of plasmapheresis, particularly in the context of severe hyperbilirubinemia. A 25-year-old female presented with jaundice and severe pruritus. Laboratory findings revealed a total bilirubin of 37 mg/dL and a direct bilirubin of 27 mg/dL. Alanine aminotransferase (ALT) was 31 U/L, aspartate aminotransferase (AST) was 35 U/L, and alkaline phosphatase was 175 U/L. The patient had a five-year history of recurrent episodes, previously resolving spontaneously or with medical treatment. Following liver biopsy diagnosis and failure of conventional therapy, she underwent plasmapheresis on alternate days. Four sessions of one plasma volume exchange, using albumin, were performed concurrently with medical treatment. The patient demonstrated rapid and significant improvement in pruritus and urine color, with no adverse events or need for fresh frozen plasma. Total bilirubin decreased progressively to 5 mg/dL, and direct bilirubin to 4 mg/dL. Plasmapheresis appears to be a beneficial adjunct therapy for refractory BRIC, effectively reducing bilirubin levels and alleviating pruritus. This intervention may improve patient quality of life and potentially prevent renal complications associated with hyperbilirubinemia, serving as a bridge to liver transplantation, if necessary.

#5

A sporadic case of benign recurrent intrahepatic cholestasis in a growth-impaired young male.

Science progress2025

Benign recurrent intrahepatic cholestasis (BRIC) is a rare genetic liver disorder characterized by recurrent episodes of jaundice and severe pruritus without significant liver damage. Here, we present the case of a man in his early 20s, standing 143 cm tall, who has experienced recurrent jaundice and pruritus over an 18-year period. Laboratory investigations and liver biopsy examination indicated typical intrahepatic cholestasis with normal gamma-glutamyl transpeptidase levels, excluding viral, metabolic, and autoimmune causes. Further genetic analysis confirmed the diagnosis of BRIC with two compound heterozygous mutations in ATP8B1 gene on chromosome 18: NM_001374385.1: c.2081T > C p. (Ile694Thr) (rs541474497) in exon 18 and NC_000018.10 (NM_001374385.1): c.1631-2A > G in intron 15. To our knowledge, the latter has not been reported. Family lineage analysis and Sanger sequencing showed that these mutations were de novo rather than hereditary. Treatment with ursodeoxycholic acid and cholestyramine resulted in complete resolution of jaundice and pruritus, with normalization of serum bilirubin level after six months of therapy.

Publicações recentes

Ver todas no PubMed

📚 EuropePMC141 artigos no totalmostrando 81

2026

Baseline analysis of patient reported outcomes in the progressive familial intrahepatic cholestasis patient registry.

Journal of pediatric gastroenterology and nutrition
2025

Case Report: Mild BRIC-like cholestasis despite a gross USP53 deletion-novel findings and literature review.

Frontiers in genetics
2025

Enigmatic functions of ATP8B1: cholestasis, inflammation, phosphoinositide flipping, and cellular homeostasis.

Cell cycle (Georgetown, Tex.)
2025

Benign Recurrent Intrahepatic Cholestasis Type 1 with Novel Gene Mutation Complicated by Distal Renal Tubular Acidosis: A Case Report.

Case reports in gastroenterology
2025

Next-Generation Sequencing in Diagnosis of Monogenic Cholestatic Liver Disorders: A Single-Center Experience.

Molecular syndromology
2025

Odevixibat for Episodic Intrahepatic Cholestasis due to Biallelic Mutations in ATP8B1: A Case Series.

Liver international : official journal of the International Association for the Study of the Liver
2025

Use of maralixibat in severe and recurrent intrahepatic cholestasis of pregnancy with concurrent benign recurrent intrahepatic cholestasis.

Journal of hepatology
2025

Molecular Advances in Cholestatic Liver Diseases.

Advances in anatomic pathology
2025

Early-onset intrahepatic cholestasis of pregnancy resulting from a genetic mutation: A case report.

Case reports in women's health
2025

Plasmapheresis can improve clinical outcomes in patients with therapy resistant benign recurrent intrahepatic cholestasis: A case report.

Transfusion and apheresis science : official journal of the World Apheresis Association : official journal of the European Society for Haemapheresis
2025

A sporadic case of benign recurrent intrahepatic cholestasis in a growth-impaired young male.

Science progress
2025

Early plasmapheresis in type 2 benign recurrent intrahepatic cholestasis: A case report and review of literature.

World journal of hepatology
2025

Hepatoprotective Effects of Cilnidipine in Cholestatic Liver Disease: Role of FXR and NRF2 Signalling.

Journal of experimental pharmacology
2024

Recurrent Jaundice Unraveled: A Case of Benign Recurrent Intrahepatic Cholestasis (BRIC) in an Indian Patient.

Cureus
2024

A Novel Compound Heterozygous Mutation in Progressive Familial Intrahepatic Cholestasis (PFIC) 4: A Rare Case Report With Literature Review.

Cureus
2023

Case Report: Intrahepatic cholestasis: a diagnostic dilemma.

Wellcome open research
2024

An Adult Case of Benign Recurrent Intrahepatic Cholestasis Due to MYO5B Deficiency.

The Tokai journal of experimental and clinical medicine
2024

A novel case report of benign recurrent intrahepatic cholestasis-associated USP53 genetic mutation in a Pakistani girl.

SAGE open medical case reports
2024

Diagnosis and management of benign recurrent intrahepatic cholestasis and psychosocial stressors in an adolescent: A case report.

World journal of clinical cases
2024

A Rare Case of Benign Recurrent Intrahepatic Cholestasis Initially Diagnosed in Middle-age.

Alternative therapies in health and medicine
2023

A novel homozygous mutation in the USP53 gene as the cause of benign recurrent intrahepatic cholestasis in children: a case report.

The Turkish journal of pediatrics
2023

Clinicopathologic Features, Genetics, Treatment, and Long-Term Outcomes in Japanese Children and Young Adults with Benign Recurrent Intrahepatic Cholestasis: A Multicenter Study.

Journal of clinical medicine
2023

Case report: A rare case of pyruvate kinase deficiency and Crigler-Najjar syndrome type II with a novel pathogenic variant of PKLR and UGT1A1 mutation.

Frontiers in genetics
2023

[One case of benign recurrent intrahepatic cholestasis type 2].

Zhonghua gan zang bing za zhi = Zhonghua ganzangbing zazhi = Chinese journal of hepatology
2023

Improvement of cholestatic episodes in patients with benign recurrent intrahepatic cholestasis (BRIC) treated with rifampicin. A long-term follow-up.

Scandinavian journal of gastroenterology
2022

Case Report: A Rare Heterozygous ATP8B1 Mutation in a BRIC1 Patient: Haploinsufficiency?

Frontiers in medicine
2022

Case Report: A Rare Case of Benign Recurrent Intrahepatic Cholestasis-Type 1 With a Novel Heterozygous Pathogenic Variant of ATP8B1.

Frontiers in medicine
2022

Benign Recurrent Intrahepatic Cholestasis Type 1 with Novel Nonsense Mutations in the ATP8B1 Gene.

Case reports in gastroenterology
2022

Coronavirus disease 2019 (COVID-19) as a potential trigger for benign recurrent intrahepatic cholestasis.

Clinical case reports
2021

Prenatal Noninvasive Trio-WES in a Case of Pregnancy-Related Liver Disorder.

Diagnostics (Basel, Switzerland)
2021

Triggers of benign recurrent intrahepatic cholestasis and its pathophysiology: a review of literature.

Acta gastro-enterologica Belgica
2021

Benign Recurrent Intrahepatic Cholestasis in Pregnancy: Fetal Death at 36 Weeks of Gestation.

Case reports in obstetrics and gynecology
2021

Case Report: A Novel Homozygous Variant Identified in a Chinese Patient With Benign Recurrent Intrahepatic Cholestasis-Type 1.

Frontiers in medicine
2021

Case Report: A Novel Single Variant TJP2 Mutation in a Case of Benign Recurrent Intrahepatic Cholestasis.

JPGN reports
2021

Benign recurrent intrahepatic cholestasis - 2 (BRIC-2)/ABCB11 deficiency in a young child - Report from a tertiary care center in South India.

Indian journal of pathology &amp; microbiology
2021

Benign recurrent intrahepatic cholestasis type 2 in a child: A case report and novel mutation.

Turkish archives of pediatrics
2021

Benign Recurrent Intrahepatic Cholestasis - Unravelleing the Paradox.

Indian pediatrics
2021

The Mystery of Episodic Recurrent Jaundice in a Young Male: Cholestasis With a Normal Gamma-Glutamyl Transferase.

Cureus
2021

The Bile Salt Export Pump: Molecular Structure, Study Models and Small-Molecule Drugs for the Treatment of Inherited BSEP Deficiencies.

International journal of molecular sciences
2021

Assessment of Adenosine Triphosphatase Phospholipid Transporting 8B1 (ATP8B1) Function in Patients With Cholestasis With ATP8B1 Deficiency by Using Peripheral Blood Monocyte-Derived Macrophages.

Hepatology communications
2021

Long-Term Colestyramine Treatment Prevents Cholestatic Attacks in Refractory Benign Recurrent Intrahepatic Cholestasis Type 1 Disease.

Hepatology (Baltimore, Md.)
2021

Cholestasis Due to USP53 Deficiency.

Journal of pediatric gastroenterology and nutrition
2020

ABCB11 deficiency presenting as transient neonatal cholestasis: Correlation with genotypes and BSEP expression.

Liver international : official journal of the International Association for the Study of the Liver
2020

Hyperthyroidism as a Potential Trigger for Benign Recurrent Intrahepatic Cholestasis.

ACG case reports journal
2021

New paradigms of USP53 disease: normal GGT cholestasis, BRIC, cholangiopathy, and responsiveness to rifampicin.

Journal of human genetics
2020

Cholestasis in Benign Recurrent Intrahepatic Cholestasis 2.

ACG case reports journal
2020

Benign Recurrent Intrahepatic Cholestasis (BRIC): An African Case Report.

Case reports in gastrointestinal medicine
2020

Nonsense variant of ATP8B1 gene in heterozygosis and benign recurrent intrahepatic cholestasis: A case report and review of literature.

World journal of hepatology
2020

Successful treatment of a patient with benign recurrent intrahepatic cholestasis type 1 with albumin dialysis.

Artificial organs
2019

A Physiology-Based Model of Human Bile Acid Metabolism for Predicting Bile Acid Tissue Levels After Drug Administration in Healthy Subjects and BRIC Type 2 Patients.

Frontiers in physiology
2019

Genetic Causes of Liver Disease: When to Suspect a Genetic Etiology, Initial Lab Testing, and the Basics of Management.

The Medical clinics of North America
2020

Genetic contribution of ABCC2 to Dubin-Johnson syndrome and inherited cholestatic disorders.

Liver international : official journal of the International Association for the Study of the Liver
2019

Clinical characteristics and genetic profiles of young and adult patients with cholestatic liver disease.

Revista espanola de enfermedades digestivas
2019

Expanding etiology of progressive familial intrahepatic cholestasis.

World journal of hepatology
2019

A rare cause of a cholestatic jaundice in a North African teenager.

Liver international : official journal of the International Association for the Study of the Liver
2019

Beyond an Obvious Cause of Cholestasis in a Toddler: Compound Heterozygosity for ABCB11 Mutations.

Pediatrics
2019

Benign Recurrent Intrahepatic Cholestasis Type 2 in Siblings with Novel ABCB11 Mutations.

Pediatric gastroenterology, hepatology &amp; nutrition
2018

[Clinical and pathological features of inherited metabolic liver disease in adults].

Zhonghua gan zang bing za zhi = Zhonghua ganzangbing zazhi = Chinese journal of hepatology
2019

Percutaneous transgastral biliodigestive diversion as treatment option for benign recurrent intrahepatic cholestasis.

Liver international : official journal of the International Association for the Study of the Liver
2018

[Clinical characteristics and diagnosis of patients with benign recurrent intrahepatic cholestasis].

Zhonghua gan zang bing za zhi = Zhonghua ganzangbing zazhi = Chinese journal of hepatology
2018

New Insights in Genetic Cholestasis: From Molecular Mechanisms to Clinical Implications.

Canadian journal of gastroenterology &amp; hepatology
2018

Clinical phenotype and molecular analysis of a homozygous ABCB11 mutation responsible for progressive infantile cholestasis.

Journal of human genetics
2018

Can genetic testing guide the therapy of cholestatic pruritus? A case of benign recurrent intrahepatic cholestasis type 2 with severe nasobiliary drainage-refractory itch.

Hepatology communications
2018

Benign recurrent intrahepatic cholestasis (BRIC)-like episode associated with ATP8B1 variation underlying protracted cholestatic course of acute hepatitis E virus infection.

Digestive and liver disease : official journal of the Italian Society of Gastroenterology and the Italian Association for the Study of the Liver
2017

Genetic Testing in Liver Disease: What to Order, in Whom, and When.

Clinics in liver disease
2018

Genetic determinants of cholangiopathies: Molecular and systems genetics.

Biochimica et biophysica acta. Molecular basis of disease
2017

Early Diagnosis of ABCB11 Spectrum Liver Disorders by Next Generation Sequencing.

Pediatric gastroenterology, hepatology &amp; nutrition
2017

Endoscopic nasobiliary drainage: an effective treatment option for benign recurrent intrahepatic cholestasis (BRIC).

BMJ case reports
2017

Current and future therapies for inherited cholestatic liver diseases.

World journal of gastroenterology
2017

Clinical Variability After Partial External Biliary Diversion in Familial Intrahepatic Cholestasis 1 Deficiency.

Journal of pediatric gastroenterology and nutrition
2016

Nasobiliary Drainage for Benign Recurrent Intrahepatic Cholestasis in Patients Refractory to Standard Therapy.

Clinical and investigative medicine. Medecine clinique et experimentale
2016

Benign Recurrent Intrahepatic Cholestasis in a Young Adult.

Journal of clinical and diagnostic research : JCDR
2016

Molecular Genetic Dissection and Neonatal/Infantile Intrahepatic Cholestasis Using Targeted Next-Generation Sequencing.

The Journal of pediatrics
2015

Genetics and Molecular Modeling of New Mutations of Familial Intrahepatic Cholestasis in a Single Italian Center.

PloS one
2015

Safety and efficacy of long-term nasobiliary drainage to treat intractable pruritus in cholestatic liver disease.

Frontline gastroenterology
2016

Successful treatment with 4-phenylbutyrate in a patient with benign recurrent intrahepatic cholestasis type 2 refractory to biliary drainage and bilirubin absorption.

Hepatology research : the official journal of the Japan Society of Hepatology
2015

Pathophysiology and Diseases of the Proximal Pathways of the Biliary System.

Archives of pathology &amp; laboratory medicine
2015

[P4-ATP-ase Atp8b1/FIC1: structural properties and (patho)physiological functions].

Bioorganicheskaia khimiia
2015

ATP8B1 and ATP11C: Two Lipid Flippases Important for Hepatocyte Function.

Digestive diseases (Basel, Switzerland)
2014

Progressive familial intrahepatic cholestasis.

Journal of clinical and experimental hepatology
2015

Compound heterozygous mutations of ABCB11 responsible for benign recurrent intrahepatic cholestasis.

Journal of digestive diseases
Ver todos os 141 no EuropePMC

Associações

Organizações que acompanham esta doença — pra ter apoio e orientação

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Grupos ativos de quem convive com esta doença aqui no Raras

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Referências e fontes

Bases de dados externas citadas neste artigo

Publicações científicas

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

  1. Baseline analysis of patient reported outcomes in the progressive familial intrahepatic cholestasis patient registry.
    Journal of pediatric gastroenterology and nutrition· 2026· PMID 41609050mais citado
  2. Enigmatic functions of ATP8B1: cholestasis, inflammation, phosphoinositide flipping, and cellular homeostasis.
    Cell cycle (Georgetown, Tex.)· 2025· PMID 41084973mais citado
  3. Benign Recurrent Intrahepatic Cholestasis Type 1 with Novel Gene Mutation Complicated by Distal Renal Tubular Acidosis: A Case Report.
    Case reports in gastroenterology· 2025· PMID 40880677mais citado
  4. Plasmapheresis can improve clinical outcomes in patients with therapy resistant benign recurrent intrahepatic cholestasis: A case report.
    Transfusion and apheresis science : official journal of the World Apheresis Association : official journal of the European Society for Haemapheresis· 2025· PMID 40483043mais citado
  5. A sporadic case of benign recurrent intrahepatic cholestasis in a growth-impaired young male.
    Science progress· 2025· PMID 40261314mais citado
  6. Case Report: Mild BRIC-like cholestasis despite a gross USP53 deletion-novel findings and literature review.
    Front Genet· 2025· PMID 41356217recente
  7. Erratum to "A sporadic case of benign recurrent intrahepatic cholestasis in a growth-impaired young male".
    Sci Prog· 2025· PMID 40981406recente

Bases de dados e fontes oficiais

Identificadores e referências canônicas usadas para montar este verbete.

  1. ORPHA:65682(Orphanet)
  2. MONDO:0019008(MONDO)
  3. GARD:12185(GARD (NIH))
  4. Variantes catalogadas(ClinVar)
  5. Busca completa no PubMed(PubMed)
  6. Q2449937(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

Colestase intra-hepática recorrente benigna
Compêndio · Raras BR

Colestase intra-hepática recorrente benigna

ORPHA:65682 · MONDO:0019008
Prevalência
Unknown
Herança
Autosomal dominant, Autosomal recessive
CID-10
K83.1 · Obstrução de via biliar
CID-11
Medicamentos
1 registrados
Início
All ages
Prevalência
0.0 (Worldwide)
MedGen
UMLS
C0149841
EuropePMC
Wikidata
Papers 10a
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