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A Novel Mutation in PNPLA2 Leading to Neutral Lipid Storage Disease With Myopathy

Daniel B. Ash, BS; Dimitra Papadimitriou, MD; Arthur P. Hays, MD; Salvatore DiMauro, MD; Michio Hirano, MD
Arch Neurol. 2012;69(9):1190-1192. doi:10.1001/archneurol.2011.2600.
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Background  Mutations in PNPLA2, a gene encoding adipose triglyceride lipase, lead to neutral lipid storage disease with myopathy.

Objective  To report the clinical and molecular features of a case of neutral lipid storage disease with myopathy resulting from a novel mutation in PNPLA2.

Design  Case report.

Setting  University hospital.

Patient  A 65-year-old man with progressive muscle weakness and high serum creatine kinase levels.

Intervention  Direct sequencing of the PNPLA2 gene.

Results  Identification of a novel homozygous mutation in the patient's PNPLA2 gene confirmed the suspected diagnosis of neutral lipid storage disease with myopathy.

Conclusion  Screening of the PNPLA2 gene should be considered for patients presenting with high levels of creatine kinase, progressive muscle weakness, and systemic lipid accumulation. The presence of Jordans anomaly can be a strong diagnostic clue.

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Figures

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Grahic Jump Location

Figure 1. Vacuolization of granulocytes (Jordans anomaly) in a peripheral blood smear from the patient.

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Grahic Jump Location

Figure 2. Electropherogram showing the patient's homozygous c.1081G>T mutation in exon 9 of the PNPLA2 gene.

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Grahic Jump Location

Figure 3. Pedigree of the family based on molecular data. Half-filled symbols are PNPLA2 mutation carriers, and the black symbol denotes homozygous mutation status of the proband (P). U indicates untested.

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