Janus kinase inhibition in the Aicardi–Goutières syndrome

A Vanderver, L Adang, F Gavazzi… - … England Journal of …, 2020 - Mass Medical Soc
JAK Inhibition in the Aicardi–Goutières Syndrome Patients with the Aicardi–Goutières
syndrome, an autosomal recessive disorder that affects the central nervous system, immune …

Genetic, phenotypic, and interferon biomarker status in ADAR1-related neurological disease

GI Rice, N Kitabayashi, M Barth, TA Briggs… - …, 2017 - thieme-connect.com
We investigated the genetic, phenotypic, and interferon status of 46 patients from 37 families
with neurological disease due to mutations in ADAR1. The clinicoradiological phenotype …

Endocrine and Growth Abnormalities in 4H Leukodystrophy Caused by Variants in POLR3A, POLR3B, and POLR1C

F Pelletier, S Perrier, FK Cayami… - The Journal of …, 2021 - academic.oup.com
Context 4H or POLR3-related leukodystrophy is an autosomal recessive disorder typically
characterized by hypomyelination, hypodontia, and hypogonadotropic hypogonadism …

Developmental outcomes of Aicardi Goutières syndrome

L Adang, F Gavazzi, M De Simone… - Journal of child …, 2020 - journals.sagepub.com
Aicardi Goutières syndrome is a monogenic interferonopathy caused by abnormalities in the
intracellular nucleic acid sensing machinery (TREX1, RNASEH2A, RNASEH2B …

[HTML][HTML] X-linked hypomyelination with spondylometaphyseal dysplasia (H-SMD) associated with mutations in AIFM1

N Miyake, NI Wolf, FK Cayami, J Crawford, A Bley… - Neurogenetics, 2017 - Springer
An X-linked condition characterized by the combination of hypomyelinating leukodystrophy
and spondylometaphyseal dysplasia (H-SMD) has been observed in only four families, with …

Neonatal detection of Aicardi Goutières syndrome by increased C26: 0 lysophosphatidylcholine and interferon signature on newborn screening blood spots

T Armangue, JJ Orsini, A Takanohashi… - Molecular genetics and …, 2017 - Elsevier
Abstract Background Aicardi Goutières Syndrome (AGS) is a heritable interferonopathy
associated with systemic autoinflammation causing interferon (IFN) elevation, central …

Aicardi goutières syndrome is associated with pulmonary hypertension

LA Adang, DB Frank, A Gilani, A Takanohashi… - Molecular genetics and …, 2018 - Elsevier
While pulmonary hypertension (PH) is a potentially life threatening complication of many
inflammatory conditions, an association between Aicardi Goutières syndrome (AGS), a rare …

The clinical, biochemical and genetic features associated with RMND1-related mitochondrial disease

YS Ng, CL Alston, D Diodato, AA Morris… - Journal of Medical …, 2016 - jmg.bmj.com
Background Mutations in the RMND1 (Required for Meiotic Nuclear Division protein 1) gene
have recently been linked to infantile onset mitochondrial disease characterised by multiple …

RMND1-related leukoencephalopathy with temporal lobe cysts and hearing loss—another mendelian mimicker of congenital cytomegalovirus infection

N Ulrick, A Goldstein, C Simons, RJ Taft, G Helman… - Pediatric …, 2017 - Elsevier
Background Leukoencephalopathy with temporal lobe cysts may be associated with
monogenetic conditions such as Aicardi–Goutières syndrome or RNASET2 mutations and …

Hepatic involvement in Aicardi-Goutieres syndrome

F Gavazzi, ZM Cross, S Woidill, JM McMann… - …, 2021 - thieme-connect.com
Aicardi-Goutières syndrome (AGS) is a monogenic type-I interferonopathy that results in
neurologic injury. The systemic impact of sustained interferon activation is less well …