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1. Unbiased phenotype and genotype matching maximizes gene discovery and diagnostic yield

2. A loss-of-function mutation in human Oxidation Resistance 1 disrupts the spatial–temporal regulation of histone arginine methylation in neurodevelopment

5. Nociception and pain in humans lacking a functional TRPV1 channel

6. Neurodevelopmental and synaptic defects in DNAJC6 parkinsonism, amenable to gene therapy

10. Clinical and experimental evidence suggest a link between KIF7 and C5orf42-related ciliopathies through Sonic Hedgehog signaling

11. WiTNNess: An international natural history study of infantile‐onset TNNT1 myopathy.

12. Point mutations in IMPDH2 which cause early-onset neurodevelopmental disorders disrupt enzyme regulation and filament structure

13. SPTSSA variants alter sphingolipid synthesis and cause a complex hereditary spastic paraplegia

19. Infantile SOD1 deficiency syndrome caused by a homozygous SOD1 variant with absence of enzyme activity

23. Loss-of-function mutation in humanOxidation Resistance gene 1disrupts the spatial-temporal regulation of histone arginine methylation in early brain development

24. Contributors

26. Mutation-Specific Effects on Thin Filament Length in Thin Filament Myopathy

31. Infantile SOD1 deficiency syndrome caused by a homozygous SOD1 variant with absence of enzyme activity

33. Mutations in SLC1A4, encoding the brain serine transporter, are associated with developmental delay, microcephaly and hypomyelination

34. Infantile cerebral and cerebellar atrophy is associated with a mutation in the MED17 subunit of the transcription preinitiation mediator complex

35. Joubert syndrome 2 (JBTS2) in Ashkenazi Jews is associated with a TMEM216 mutation

38. C6ORF66 is an assembly factor of mitochondrial complex I

48. A deleterious variant of INTS1 leads to disrupted sleep–wake cycles

49. Leukoencephalopathy with accumulated succinate is indicative of SDHAF1 related complex II deficiency

50. Pathogenic Variants in NUP214 Cause “Plugged” Nuclear Pore Channels and Acute Febrile Encephalopathy

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