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Benign recurrent intrahepatic cholestasis type 1(BRIC1)

MedGen UID:
1637492
Concept ID:
C4551899
Disease or Syndrome
Synonyms: Recurrent familial intrahepatic cholestasis 1; Summerskill syndrome
 
Gene (location): ATP8B1 (18q21.31)
 
Monarch Initiative: MONDO:0009469
OMIM®: 243300
Orphanet: ORPHA99960

Definition

The phenotypic spectrum of ATP8B1 deficiency ranges from severe through moderate to mild. Severe ATP8B1 deficiency is characterized by infantile-onset cholestasis that progresses to cirrhosis, hepatic failure, and early death. Although mild-to-moderate ATP8B1 deficiency initially was thought to involve intermittent symptomatic cholestasis with a lack of hepatic fibrosis, it is now known that hepatic fibrosis may be present early in the disease course. Furthermore, in some persons with ATP8B1 deficiency the clinical findings can span the phenotypic spectrum, shifting over time from the mild end of the spectrum (episodic cholestasis) to the severe end of the spectrum (persistent cholestasis). Sensorineural hearing loss (SNHL) is common across the phenotypic spectrum. [from GeneReviews]

Additional description

From OMIM
Benign recurrent intrahepatic cholestasis is characterized by intermittent episodes of cholestasis without extrahepatic bile duct obstruction. There is initial elevation of serum bile acids, followed by cholestatic jaundice which generally spontaneously resolves after periods of weeks to months (Summerskill and Walshe, 1959; Schapiro and Isselbacher, 1963; Brenard et al., 1989). Tygstrup et al. (1999) stated that referring to this disorder as 'benign' is a misnomer, because the disease has an impact on the quality of life in some patients. They preferred the term 'recurrent familial intrahepatic cholestasis.' Genetic Heterogeneity of Benign Recurrent Intrahepatic Cholestasis See also BRIC2 (605479), caused by mutation in the ABCB11 gene (603201) on chromosome 2q24.  http://www.omim.org/entry/243300

Clinical features

From HPO
Hepatomegaly
MedGen UID:
42428
Concept ID:
C0019209
Finding
Abnormally increased size of the liver.
Intrahepatic cholestasis with episodic jaundice
MedGen UID:
870571
Concept ID:
C4025019
Disease or Syndrome
Intermittent jaundice
MedGen UID:
871317
Concept ID:
C4025805
Pathologic Function
Jaundice that is sometimes present, sometimes not.
Hearing impairment
MedGen UID:
235586
Concept ID:
C1384666
Disease or Syndrome
A decreased magnitude of the sensory perception of sound.
Pancreatitis
MedGen UID:
14586
Concept ID:
C0030305
Disease or Syndrome
The presence of inflammation in the pancreas.
Conjugated hyperbilirubinemia
MedGen UID:
82787
Concept ID:
C0268307
Disease or Syndrome
Abnormally high level of conjugated bilirubin in the blood.
Increased serum bile acid concentration
MedGen UID:
868605
Concept ID:
C4023004
Finding
An increase in the concentration of bile acid in the blood.
Pruritus
MedGen UID:
19534
Concept ID:
C0033774
Sign or Symptom
Pruritus is an itch or a sensation that makes a person want to scratch. This term refers to an abnormally increased disposition to experience pruritus.

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVBenign recurrent intrahepatic cholestasis type 1

Recent clinical studies

Diagnosis

Mizutani A, Sabu Y, Naoi S, Ito S, Nakano S, Minowa K, Mizuochi T, Ito K, Abukawa D, Kaji S, Sasaki M, Muroya K, Azuma Y, Watanabe S, Oya Y, Inomata Y, Fukuda A, Kasahara M, Inui A, Takikawa H, Kusuhara H, Bessho K, Suzuki M, Togawa T, Hayashi H
Hepatol Commun 2021 Jan;5(1):52-62. Epub 2020 Sep 26 doi: 10.1002/hep4.1605. PMID: 33437900Free PMC Article
Naik J, de Waart DR, Utsunomiya K, Duijst S, Mok KH, Oude Elferink RP, Bosma PJ, Paulusma CC
Dig Dis 2015;33(3):314-8. Epub 2015 May 27 doi: 10.1159/000371665. PMID: 26045263
Mizuochi T, Kimura A, Tanaka A, Muto A, Nittono H, Seki Y, Takahashi T, Kurosawa T, Kage M, Takikawa H, Matsuishi T
Clin Chim Acta 2012 Aug 16;413(15-16):1301-4. Epub 2012 Apr 14 doi: 10.1016/j.cca.2012.04.011. PMID: 22525741

Therapy

Koukoulioti E, Ziagaki A, Weber SN, Lammert F, Berg T
Hepatology 2021 Jul;74(1):522-524. Epub 2021 Jun 28 doi: 10.1002/hep.31671. PMID: 33277690
Naik J, de Waart DR, Utsunomiya K, Duijst S, Mok KH, Oude Elferink RP, Bosma PJ, Paulusma CC
Dig Dis 2015;33(3):314-8. Epub 2015 May 27 doi: 10.1159/000371665. PMID: 26045263
Mizuochi T, Kimura A, Tanaka A, Muto A, Nittono H, Seki Y, Takahashi T, Kurosawa T, Kage M, Takikawa H, Matsuishi T
Clin Chim Acta 2012 Aug 16;413(15-16):1301-4. Epub 2012 Apr 14 doi: 10.1016/j.cca.2012.04.011. PMID: 22525741

Prognosis

Mizutani A, Sabu Y, Naoi S, Ito S, Nakano S, Minowa K, Mizuochi T, Ito K, Abukawa D, Kaji S, Sasaki M, Muroya K, Azuma Y, Watanabe S, Oya Y, Inomata Y, Fukuda A, Kasahara M, Inui A, Takikawa H, Kusuhara H, Bessho K, Suzuki M, Togawa T, Hayashi H
Hepatol Commun 2021 Jan;5(1):52-62. Epub 2020 Sep 26 doi: 10.1002/hep4.1605. PMID: 33437900Free PMC Article

Clinical prediction guides

Naik J, de Waart DR, Utsunomiya K, Duijst S, Mok KH, Oude Elferink RP, Bosma PJ, Paulusma CC
Dig Dis 2015;33(3):314-8. Epub 2015 May 27 doi: 10.1159/000371665. PMID: 26045263
Paulusma CC, Folmer DE, Ho-Mok KS, de Waart DR, Hilarius PM, Verhoeven AJ, Oude Elferink RP
Hepatology 2008 Jan;47(1):268-78. doi: 10.1002/hep.21950. PMID: 17948906

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