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Nonketotic hyperglycinemia (NKH)

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A newborn presents with severe hypotonia, apnea, and intractable seizures within the first days of life. CSF glycine is markedly elevated with an elevated CSF:plasma glycine ratio.

AR; glycine cleavage system genes

  • GLDC (~70%), AMT (~20%), GCSH
  • Severe neonatal encephalopathy
  • Hypotonia, apnea, hiccups
  • Intractable seizures
  • Elevated CSF:plasma glycine ratio (>0.08)
  • Burst suppression on EEG
  • Most severe form: neonatal, lethal
  • Attenuated forms exist
  • Not on standard newborn screening (glycine is not part of routine acylcarnitine/amino-acid profiles flagged for NKH)
  • Elevated plasma and CSF glycine with paired CSF:plasma glycine ratio >0.08 (the key biochemical hallmark)
  • Confirm with molecular testing of glycine cleavage system genes (GLDC, AMT, GCSH); exclude secondary causes (e.g., valproate, ketosis)
  • Limited options; sodium benzoate, dextromethorphan, ketogenic diet (variable efficacy)

"NKH is BaD": treatment is sodium Benzoate and Dextromethorphan (an NMDA antagonist that prevents glycine from binding its receptor).

Glycine cleavage system has 3 proteins: P protein (GLDC, ~70% of mutations), T protein (AMT, ~20%), and H protein (GCSH, <1%). Glycine builds up in the CSF, causing symptoms.

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