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Joubert syndrome with hepatic defect

ORPHA:1454DiseaseAutosomal recessiveNeonatal

Ассоциированные гены (4)

TMEM67
transmembrane protein 67
Disease-causing germline mutation(s) in
OMIM: 609884
RPGRIP1L
RPGRIP1 like
Disease-causing germline mutation(s) in
OMIM: 610937
CC2D2A
coiled-coil and C2 domain containing 2A
Disease-causing germline mutation(s) in
OMIM: 612013
INPP5E
inositol polyphosphate-5-phosphatase E
Disease-causing germline mutation(s) in
OMIM: 613037

Фенотипы (48)

Очень частый (80–99%)12
HP:0000657Oculomotor apraxia
HP:0001251Ataxia
HP:0001252Hypotonia
HP:0001320Cerebellar vermis hypoplasia
HP:0002104Apnea
HP:0002240Hepatomegaly
HP:0002342Intellectual disability, moderate
HP:0002612Congenital hepatic fibrosis
HP:0002793Abnormal pattern of respiration
HP:0002910Elevated circulating hepatic transaminase concentration
HP:0005248Intrahepatic biliary atresia
HP:0007360Aplasia/Hypoplasia of the cerebellum
Частый (30–79%)11
HP:0000112Nephropathy
HP:0000276Long face
HP:0000505Visual impairment
HP:0000567Chorioretinal coloboma
HP:0000588Optic disc coloboma
HP:0000612Iris coloboma
HP:0000639Nystagmus
HP:0001288Gait disturbance
HP:0001347Hyperreflexia
HP:0004422Biparietal narrowing
HP:0008872Feeding difficulties in infancy
Периодический (5–29%)25
HP:0000003Multicystic kidney dysplasia
HP:0000023Inguinal hernia
HP:0000083Renal insufficiency
HP:0000202Orofacial cleft
HP:0000238Hydrocephalus
HP:0000256Macrocephaly
HP:0000369Low-set ears
HP:0000426Prominent nasal bridge
HP:0000463Anteverted nares
HP:0000486Strabismus
HP:0000508Ptosis
HP:0000864Abnormality of the hypothalamus-pituitary axis
HP:0001162Postaxial hand polydactyly
HP:0001250Seizure
HP:0001337Tremor
HP:0001394Cirrhosis
HP:0001409Portal hypertension
HP:0001744Splenomegaly
HP:0002085Occipital encephalocele
HP:0002269Abnormality of neuronal migration
HP:0002553Highly arched eyebrow
HP:0002650Scoliosis
HP:0002896Neoplasm of the liver
HP:0007370Aplasia/Hypoplasia of the corpus callosum
HP:0100626Chronic hepatic failure

Эпидемиология (2)

Cases/families
Worldwide
Point prevalence
<1 / 1 000 000
Worldwide

Лекарства и терапия

Источник: Open Targets Platform (в реальном времени)

Клинические исследования

Источник: ClinicalTrials.gov (в реальном времени)

Внешние ресурсы