Hepatoblastoma relapses with a normal level of alpha-fetoprotein: report of two cases

Hepatoblastoma (HB) is the most common malignant liver tumor in children. The level of alpha-fetoprotein (AFP) is used for monitoring the response to antitumor therapy and for diagnosing relapses. The occurrence of HB relapses with normal AFP levels in patients who had elevated levels of this tumor marker at disease onset is considered to be an uncommon situation. The aim of this study was to describe cases in which AFP-negative hepatoblastoma relapses developed. The study participants included two HB patients who were initially stratified into high-risk and standard-risk groups and who received treatment at the Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology and the Petrovsky National Research Center of Surgery inMoscow. At the moment of relapse acknowledgement these patients had normal AFP levels, which was confirmed by serial measurements. Immunohistochemistry (IHC) tests were performed on paired samples of the tumor at disease onset and at disease relapse respectively to evaluate AFP expression. The presented clinical cases demonstrate that normal AFP levels, when accompanied by suspicious clinical symptoms, do not allow to exclude an HB relapse. The possibility of relapse with a normal AFP level reinforces the importance of following the current plan of screening tests, which includes not only an evaluation of AFP levels, but also other tests such as a chest X-ray and an abdominal ultrasound. Parents gave their consent to use information about the child in the article.

[1]  C. Rodríguez-Galindo,et al.  Upfront window vincristine/irinotecan treatment of high‐risk hepatoblastoma: A report from the Children's Oncology Group AHEP0731 study committee , 2017, Cancer.

[2]  I. Leuschner,et al.  Small cell undifferentiated (SCUD) hepatoblastomas: All malignant rhabdoid tumors? , 2016, Genes, chromosomes & cancer.

[3]  S. Miyano,et al.  Abstract 3294: Clonal evolution and integral analysis of hepatoblastoma , 2015 .

[4]  С. Р. Варфоломеева,et al.  Диспансерное наблюдение пациентов c гепатобластомой , 2015 .

[5]  R. P. Guillerman,et al.  Relapse surveillance in AFP-positive hepatoblastoma: re-evaluating the role of imaging , 2014, Pediatric Radiology.

[6]  B. Morland,et al.  Dose-dense cisplatin-based chemotherapy and surgery for children with high-risk hepatoblastoma (SIOPEL-4): a prospective, single-arm, feasibility study , 2013, The Lancet. Oncology.

[7]  G. Perilongo,et al.  Relapses in hepatoblastoma patients: clinical characteristics and outcome--experience of the International Childhood Liver Tumour Strategy Group (SIOPEL). , 2013, European journal of cancer.

[8]  B. Morland,et al.  Prognostic stratification for children with hepatoblastoma: the SIOPEL experience. , 2012, European journal of cancer.

[9]  J. Birch Epidemiology of Pediatric Liver Tumors , 2011 .

[10]  B. Morland,et al.  Cisplatin versus cisplatin plus doxorubicin for standard-risk hepatoblastoma. , 2009, The New England journal of medicine.

[11]  G. Tomlinson,et al.  Small cell undifferentiated variant of hepatoblastoma: Adverse clinical and molecular features similar to rhabdoid tumors , 2009, Pediatric blood & cancer.

[12]  G. Perilongo,et al.  Hepatoblastoma with a low serum alpha-fetoprotein level at diagnosis: the SIOPEL group experience. , 2008, European journal of cancer.

[13]  K. McHugh,et al.  2005 PRETEXT: a revised staging system for primary malignant liver tumours of childhood developed by the SIOPEL group , 2006, Pediatric Radiology.

[14]  C. Jones,et al.  The hook effect: a need for constant vigilance , 2006, Annals of clinical biochemistry.

[15]  M. B. McCarville,et al.  Imaging recommendations for malignant liver neoplasms in children , 2006, Pediatric blood & cancer.

[16]  D. V. von Schweinitz,et al.  Pretreatment prognostic factors and treatment results in children with hepatoblastoma , 2002, Cancer.

[17]  U. Göbel,et al.  DIAGNOSTIC VALUE OF ALPHA1-FETOPROTEIN AND BETA-HUMAN CHORIONIC GONADOTROPIN IN INFANCY AND CHILDHOOD , 2001, Pediatric hematology and oncology.

[18]  C. Herzog,et al.  Childhood cancers: hepatoblastoma. , 2000, The oncologist.

[19]  J. Krischer,et al.  Ifosfamide/Carboplatin/Etoposide (ICE) for Recurrent Malignant Solid Tumors of Childhood: A Pediatric Oncology Group Phase I/II Study , 1995, Journal of pediatric hematology/oncology.