Platelet-derived growth factor receptor-beta in Gorham's disease

Nat Clin Pract Oncol. 2006 Dec;3(12):693-7. doi: 10.1038/ncponc0660.

Abstract

Background: A 17-year-old male presented with pain in his lower-left chest. He had no significant medical history and was previously in good health. He had a fractured ninth left anterior rib and the tenth, eleventh and twelfth ribs were absent, which was thought to be a congenital anomaly. Several months later, he presented again with back pain, an enlarging mass in the lower-left chest wall, erosion of the lateral pedicles of the lower thoracic vertebrae and pleural effusion.

Investigations: Physical examination, chest X-ray, MRI of the spine, incisional biopsy, serial CT imaging of the hemithorax, immunohistochemistry, flow cytometry, and enzyme-linked immunosorbent assays.

Diagnosis: Gorham's lymphangiomatosis with expression of platelet-derived growth factor receptor-beta and elevated circulating platelet-derived growth factor-BB.

Management: Spine stabilization, thalidomide, celecoxib, interferon-alpha2b, pamidronate, zoledronate, thoracotomy, pleurectomy, talc pleurodesis, and imatinib mesylate.

Publication types

  • Case Reports
  • Research Support, N.I.H., Extramural

MeSH terms

  • Adolescent
  • Chylothorax / etiology
  • Fatal Outcome
  • Humans
  • Lymphangiosarcoma / etiology*
  • Lymphangiosarcoma / metabolism
  • Lymphangiosarcoma / pathology
  • Male
  • Osteolysis, Essential / complications*
  • Osteolysis, Essential / diagnosis
  • Proto-Oncogene Proteins c-sis / metabolism*

Substances

  • Proto-Oncogene Proteins c-sis