Novel germline SDHD deletion associated with an unusual sympathetic head and neck paraganglioma

Head Neck. 2011 Aug;33(8):1233-40. doi: 10.1002/hed.21384. Epub 2010 Mar 22.

Abstract

Background: Paragangliomas (PGLs) are rare tumors arising either from sympathetic or parasympathetic-associated chromaffin tissue. PGLs can occur either sporadically or as part of a hereditary syndrome. Sympathetic head and neck PGLs are extremely rare tumors and only a few cases have been reported to date.

Methods: We report the pedigree of a patient with a head and neck PGL arising from the right sympathetic trunk. SDHD mutation analysis was performed using standard sequencing, multiplex ligation-dependent probe amplification, chromosome 11-specific comparative genome hybridization, and long-range/short-range polymerase chain reaction (PCR) approaches.

Results: A previously unreported chromosome 11q deletion encompassing 5 annotated genes (SDHD, DLAT, PIH1D2, C11Orf57, and TIMM8B) was detected in the proband.

Conclusion: PGL families considered "mutation-negative" may be attributable to large gene deletions not detectable by standard sequencing methods. Therefore, deletion analysis should be offered to families or individuals at risk for hereditary PGLs.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Angiography, Digital Subtraction / methods
  • Autoantigens / genetics
  • Biopsy, Needle
  • Dihydrolipoyllysine-Residue Acetyltransferase / genetics
  • Female
  • Follow-Up Studies
  • Genetic Testing
  • Germ-Line Mutation
  • Head and Neck Neoplasms / diagnostic imaging
  • Head and Neck Neoplasms / genetics*
  • Head and Neck Neoplasms / surgery
  • Heterozygote
  • Humans
  • Immunohistochemistry
  • Mitochondrial Proteins / genetics
  • Neoplasm Staging
  • Paraganglioma, Extra-Adrenal / diagnostic imaging
  • Paraganglioma, Extra-Adrenal / genetics*
  • Paraganglioma, Extra-Adrenal / surgery
  • Pedigree*
  • Polymerase Chain Reaction
  • Rare Diseases
  • Risk Assessment
  • Sequence Deletion*
  • Succinate Dehydrogenase / genetics*
  • Tomography, X-Ray Computed / methods
  • Treatment Outcome

Substances

  • Autoantigens
  • Mitochondrial Proteins
  • Succinate Dehydrogenase
  • DLAT protein, human
  • Dihydrolipoyllysine-Residue Acetyltransferase