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4 - Pantothenate Kinase-Associated Neurodegeneration (Hallervorden–Spatz Syndrome)

from Section 1 - Bilateral Predominantly Symmetric Abnormalities

Published online by Cambridge University Press:  05 August 2013

Andrea Rossi
Affiliation:
Children’s Research Hospital, Genoa, Italy
Zoran Rumboldt
Affiliation:
Medical University of South Carolina
Mauricio Castillo
Affiliation:
University of North Carolina, Chapel Hill
Benjamin Huang
Affiliation:
University of North Carolina, Chapel Hill
Andrea Rossi
Affiliation:
G. Gaslini Children's Research Hospital
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Summary

Specific Imaging Findings

In pantothenate kinase-associated neurodegeneration (PKAN, formerly known as Hallervorden–Spatz syndrome), MRI shows markedly hypointense globi pallidi on T2-weighted images, with a small hyperintense central or anteromedial area. This finding has been labelled the “eye-of-the-tiger” sign and is highly characteristic of PKAN; it is visible on both axial and coronal images. Gradient-echo T2*-weighted images show more profound hypointensity owing to paramagnetic effects. T1-weighted images may show a corresponding high signal intensity of the pallida. There is no contrast enhancement. CT may reveal symmmetrically increased attenuation, primarily in the anteromedial globus pallidus.

Pertinent Clinical Information

This rare autosomal recessive disorder is a part of a group of diseases called “neurodegeneration with brain iron accumulation” (NBIA) which also includes aceruloplasminemia and neuroferritinopathy. PKAN typically presents in older children or adolescents with oromandibular dystonia, mental deterioration, pyramidal signs, and retinal degeneration. Most patients die within 10 years of the clinical onset, although longer survival into early adulthood is possible.

Differential Diagnosis

HARP Syndrome (hypopre-β-lipoproteinemia, acanthocytosis, retinitis pigmentosa, and pallidal degeneration)

• may be indistinguishable

Other Forms of NBIA

• “eye-of-the-tiger” sign absent

Toxic Encephalopathies (CO poisoning) (3)

• globus pallidus T2 hyperintensity without hypointense portion

Kernicterus

• globus pallidus T2 hyperintensity without hypointense portion

Methylmalonic Acidemia

• globus pallidus T2 hyperintensity without hypointense portion

Normal Iron Deposition

• iron starts accumulating in the pallidi during later childhood and adolescence and is usually seen on MRI from approximately 25 years of age onwards

Type
Chapter
Information
Brain Imaging with MRI and CT
An Image Pattern Approach
, pp. 9 - 10
Publisher: Cambridge University Press
Print publication year: 2012

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References

1. Gordon, N. Pantothenate kinase-associated neurodegeneration (Hallervorden–Spatz syndrome). Eur J Pediatr Neurol 2002;6:243–7.CrossRefGoogle ScholarPubMed
2. Angelini, L, Nardocci, N, Rumi, V. Hallervorden–Spatz disease: clinical and MRI study of 11 cases diagnosed in life. J Neurol 1992;239:417–25.CrossRefGoogle ScholarPubMed
3. Zhou, B, Westaway, SK, Levinson, B, et al.A novel pantothenate kinase gene (PANK2) is defective in Hallervorden–Spatz syndrome. Nature Genet 2001;28:345–9.CrossRefGoogle ScholarPubMed
4. Savoiardo, M, Halliday, WC, Nardocci, N, et al.Hallervorden–Spatz disease: MR and pathologic findings. AJNR 1993;14:155–62.Google ScholarPubMed
5. Ching, KH, Westaway, SK, Gitschier, J, et al.HARP syndrome is allelic with pantothenate kinase-associated neurodegeneration. Neurology 2002;58:1673–4.CrossRefGoogle ScholarPubMed

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