Concordance for neuroblastoma in monozygotic twins: case report and review of the literature

J Pediatr Surg. 2010 Dec;45(12):2312-6. doi: 10.1016/j.jpedsurg.2010.08.025.

Abstract

The patients were infant male twins born by cesarean delivery following a healthy pregnancy at 36 weeks' gestation to unrelated parents. At 4 months of age, twin 2 presented with hepatomegaly and a right suprarenal mass. Resection of an adrenal tumor and a liver tumor biopsy were performed. Twin 1 had no symptoms at 4 months of age. Screening by abdominal ultrasonography showed multiple masses in the liver but no adrenal mass. Metaiodobenzylguanidine scintigraphy showed positive findings in multiple liver masses. A laparoscopic biopsy for a liver tumor was performed. All primary tumor and liver tumor specimens from twin 2 and the liver tumor of twin 1 had the same histologic classification of neuroblastoma and nearly identical genetic aberrations, including a chromosome gain or loss using array-comparative genomic hybridization. From these clinical and pathologic findings and genetic analyses, we strongly demonstrate the transplacental metastatic spread from twin 2 to twin 1. In the literature, 9 pairs of concordant twin neuroblastomas, including the current twin, have been presented; and the clinical findings of 5 twin pairs may represent placental metastases from one twin with congenital neuroblastoma to the other twin. This study is the first report presenting the possibility of twin-to-twin metastasis in monozygotic twins with neuroblastoma based on an analysis of the clinical features and genetic aberrations.

Publication types

  • Case Reports
  • Review

MeSH terms

  • 3-Iodobenzylguanidine
  • Adrenal Gland Neoplasms / diagnostic imaging
  • Adrenal Gland Neoplasms / drug therapy
  • Adrenal Gland Neoplasms / embryology*
  • Adrenal Gland Neoplasms / genetics
  • Adrenal Gland Neoplasms / pathology
  • Adrenal Gland Neoplasms / surgery
  • Adrenalectomy
  • Antineoplastic Combined Chemotherapy Protocols / therapeutic use
  • Chromosome Aberrations
  • Combined Modality Therapy
  • Comparative Genomic Hybridization
  • Cyclophosphamide / administration & dosage
  • Diseases in Twins / embryology*
  • Diseases in Twins / epidemiology
  • Diseases in Twins / genetics
  • Female
  • Fetofetal Transfusion*
  • Humans
  • Infant
  • Iodine Radioisotopes
  • Liver Neoplasms / chemistry
  • Liver Neoplasms / diagnostic imaging
  • Liver Neoplasms / drug therapy
  • Liver Neoplasms / secondary*
  • Male
  • Neuroblastoma / chemistry
  • Neuroblastoma / diagnostic imaging
  • Neuroblastoma / drug therapy
  • Neuroblastoma / embryology*
  • Neuroblastoma / epidemiology
  • Neuroblastoma / genetics
  • Neuroblastoma / secondary*
  • Neuroblastoma / surgery
  • Placenta / pathology*
  • Pregnancy
  • Radionuclide Imaging
  • Twins, Monozygotic*
  • Ultrasonography
  • Vincristine / administration & dosage

Substances

  • Iodine Radioisotopes
  • 3-Iodobenzylguanidine
  • Vincristine
  • Cyclophosphamide