Multiple childhood osteosarcomas in an american indian family with erythroid macrocytosis and skeletal anomalies

John J. Mulvihill, Harvey R. Gralnick, Jacqueline Whang‐Peng, Brigid G. Leventhal

研究成果: 雜誌貢獻文章同行評審

25 引文 斯高帕斯(Scopus)

摘要

Three of nine children of possibly consanguineous American Indian parents developed typical osteosarcoma in a 2‐year period. Etiologic investigations detected limb anomalies and elevated mean corpuscular volumes (98‐109 μm3) in the surviving tumor patient, several of her sibs, and her father. Limb anomalies included simple clinodactyly with brachymesophalangy, absence of one digital ray of the foot, and bilateral radioulnar synostosis. The red cell macrocytosis was not accompanied by anemia or explained by the usual causes. No unusual environmental exposures were found and screening for possible oncogenic viruses by culture, electron microscopy, and serology was negative. All family members had elevated antibody titers to Epstein‐Barr viral antigens. The proband and her father had excessive chromosomal breaks in the bone marrow. This unusual familial pattern of osseous malignancy and malformation and defective erythropoiesis, tentatively called OSLAM syndrome, may represent impaired regulation of bone development. Cancer 40:3115‐3122, 1977.
原文英語
頁(從 - 到)3115-3122
頁數8
期刊Cancer
40
發行號6
DOIs
出版狀態已發佈 - 1月 1 1977
對外發佈

ASJC Scopus subject areas

  • 腫瘤科
  • 癌症研究

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