Malignant head and neck paragangliomas in SDHB mutation carriers

Otolaryngol Head Neck Surg. 2007 Jul;137(1):126-9. doi: 10.1016/j.otohns.2007.01.015.

Abstract

Objective: Three of four paraganglioma syndromes (PGLs) have been characterized on a molecular genetic basis. PGL 1 is associated with mutations of the succinate dehydrogenase subunit D (SDHD) gene, PGL 3 is caused by SDHC gene mutations, and PGL 4 is caused by SDHB gene mutations. The objective of this study was to investigate whether PGLs are associated with malignant head and neck paragangliomas (HNPs).

Study design and setting: Through November 2005, we screened 195 HNP patients for mutations of the genes SDHB, SDHC, and SDHD.

Results: We detected 5 SDHC, 13 SDHB, and 45 SDHD gene mutations. In seven SDHB mutation carriers, there were distant metastases. No signs of metastases were found in SDHC and SDHD patients. One patient with a sporadic HNP presented with locally metastatic disease.

Conclusions: SDHB mutations are associated with a high rate of malignant HNPs.

Significance: In SDHB patients, a three-body region imaging and scintigraphy or DOPA-PET must be performed to exclude metastases.

MeSH terms

  • Adult
  • Aged
  • Bone Neoplasms / secondary
  • Carotid Body Tumor / genetics
  • Exons / genetics
  • Female
  • Glomus Jugulare Tumor / genetics
  • Head and Neck Neoplasms / genetics*
  • Humans
  • Introns / genetics
  • Iron-Sulfur Proteins / genetics*
  • Lymphatic Metastasis / diagnosis
  • Male
  • Membrane Proteins / genetics
  • Middle Aged
  • Mutation / genetics*
  • Mutation, Missense / genetics
  • Paraganglioma / genetics*
  • Paraganglioma / secondary
  • Polymorphism, Single-Stranded Conformational
  • Prospective Studies
  • Succinate Dehydrogenase / genetics*

Substances

  • Iron-Sulfur Proteins
  • Membrane Proteins
  • SDHC protein, human
  • SDHD protein, human
  • SDHB protein, human
  • Succinate Dehydrogenase