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Pontocerebellar hypoplasia type 1

ORPHA:2254Malformation syndromeAutosomal recessiveNeonatal

Ассоциированные гены (6)

AGTPBP1
ATP/GTP binding carboxypeptidase 1
Disease-causing germline mutation(s) in
OMIM: 606830
VRK1
VRK serine/threonine kinase 1
Disease-causing germline mutation(s) in
OMIM: 602168
EXOSC3
exosome component 3
Disease-causing germline mutation(s) in
OMIM: 606489
EXOSC8
exosome component 8
Disease-causing germline mutation(s) in
OMIM: 606019
SLC25A46
solute carrier family 25 member 46
Disease-causing germline mutation(s) (loss of function) in
OMIM: 610826
EXOSC9
exosome component 9
Disease-causing germline mutation(s) in
OMIM: 606180

Фенотипы (29)

Очень частый (80–99%)9
HP:0001252Hypotonia
HP:0001263Global developmental delay
HP:0001265Hyporeflexia
HP:0001270Motor delay
HP:0001324Muscle weakness
HP:0002398Degeneration of anterior horn cells
HP:0002878Respiratory failure
HP:0003202Skeletal muscle atrophy
HP:0007360Aplasia/Hypoplasia of the cerebellum
Частый (30–79%)7
HP:0000253Progressive microcephaly
HP:0000529Progressive visual loss
HP:0000639Nystagmus
HP:0000648Optic atrophy
HP:0001508Failure to thrive
HP:0011968Feeding difficulties
HP:0012110Hypoplasia of the pons
Периодический (5–29%)10
HP:0000486Strabismus
HP:0001250Seizure
HP:0001257Spasticity
HP:0001308Tongue fasciculations
HP:0001347Hyperreflexia
HP:0002120Cerebral cortical atrophy
HP:0002350Cerebellar cyst
HP:0003477Peripheral axonal neuropathy
HP:0004886Congenital laryngeal stridor
HP:0033725Thin corpus callosum
Очень редкий (1–4%)3
HP:0000565Esotropia
HP:0001251Ataxia
HP:0002804Arthrogryposis multiplex congenita

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