A Case of Hepatoblastoma With Wilson-Mikity Syndrome and Beckwith-Wiedemann Syndrome in an Extremely Low Birth-Weight Infant

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  • Yoshizawa Shinsuke
    Department of Pediatric Surgery, Nihon University School of Medicine
  • Sugito Kiminobu
    Department of Pediatric Surgery, Nihon University School of Medicine
  • Soga Akiko
    Department of Pediatric Surgery, Nihon University School of Medicine
  • Uekusa Shota
    Department of Pediatric Surgery, Nihon University School of Medicine
  • Kaneda Hide
    Department of Pediatric Surgery, Nihon University School of Medicine
  • Furuya Takeshi
    Department of Pediatric Surgery, Nihon University School of Medicine
  • Ohashi Kensuke
    Department of Pediatric Surgery, Nihon University School of Medicine
  • Inoue Mikiya
    Department of Pediatric Surgery, Nihon University School of Medicine
  • Ikeda Taro
    Department of Pediatric Surgery, Nihon University School of Medicine
  • Koshinaga Tsugumichi
    Department of Pediatric Surgery, Nihon University School of Medicine

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Other Title
  • 超低出生体重児にWilson-Mikity症候群とBeckwith-Wiedemann症候群を伴った肝芽腫の1例
  • 症例報告 超低出生体重児にWilson-Mikity症候群とBeckwith-Wiedemann症候群を伴った肝芽腫の1例
  • ショウレイ ホウコク チョウテイシュッショウ タイジュウジ ニ Wilson-Mikity ショウコウグン ト Beckwith-Wiedemann ショウコウグン オ トモナッタ カン ガ シュ ノ 1レイ

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We report a case of hepatoblastoma with Wilson-Mikity syndrome and Beckwith-Wiedemann syndrome (BWS) in an extremely low birth-weight infant. At 8 months of age, she was referred to our hospital due to liver tumor and elevated serum alpha-fetoprotein (AFP) levels upon screening for tumors with BWS. We started chemotherapy (CITA regimen) without an open biopsy, because of unstable conditions due to emphysema and pulmonary hypertension. After two courses of CITA regimen, she received tumor resection. Postoperatively, she received four courses of low CITA regimen. In this case, we carefully observed not only the level of AFP but lectin-reactive AFP (AFP-L3) because the levels of AFP and AFP-L3 showed gradual decreases with transient increases after complete tumor resection. Here we discussed changes of AFP and AFP-L3 levels after tumor resection in hepatoblastomas.

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