Normal-appearing white matter in patients with phenylketonuria: water content, myelin water fraction, and metabolite concentrations

Radiology. 2007 Jan;242(1):236-43. doi: 10.1148/radiol.2421051758.

Abstract

Purpose: To prospectively assess relative water content (RWC), myelin water fraction (MWF), and hydrogen 1 magnetic resonance (MR) spectroscopy findings in the white matter (WM) of patients with phenylketonuria (PKU).

Materials and methods: This study was approved by the institution's investigational review board, and informed consent was obtained. T2 water relaxation data were acquired by using a 48-echo measurement in a transverse plane through the genu and splenium of the corpus callosum in 16 patients (six men, 10 women; age range, 18-40 years) with PKU and 16 age- and sex-matched control subjects. MR spectroscopy was performed in a voxel (94x70x15 mm) above the ventricles. WM in control subjects (defined as normal WM) was compared with normal-appearing WM (NAWM) and diffuse WM lesions in patients with PKU by using a Student t test.

Results: Patients with PKU had two forms of NAWM: (a) areas that looked normal on intermediate-weighted (IW) and T2-weighted MR images and long T2 maps and (b) areas that looked normal on IW and T2-weighted MR images but were hyperintense on long T2 maps. Both forms of NAWM showed increased RWC (up to 2.5%, P<.001) and reduced MWF (up to 56%, P<.001) relative to normal WM; these changes paralleled those seen in diffuse WM lesions. Approximately 9% of the water in diffuse WM lesions was in a reservoir with a long T2 time of 200-800 msec. Myoinositol concentrations were reduced by 14% (P=.003) in patients with PKU.

Conclusion: In patients with PKU, NAWM and diffuse WM lesions have altered RWC and MWF relative to normal WM, and diffuse WM lesions show a redistribution of water into an extracellular reservoir with a long T2 time.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Body Water / metabolism*
  • Corpus Callosum / metabolism*
  • Corpus Callosum / pathology*
  • Female
  • Humans
  • Magnetic Resonance Spectroscopy / methods
  • Male
  • Nerve Fibers, Myelinated / metabolism*
  • Nerve Fibers, Myelinated / pathology*
  • Phenylketonurias / metabolism*
  • Phenylketonurias / pathology*
  • Protons
  • Water / analysis

Substances

  • Protons
  • Water