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Living-donor liver transplantation providing an adequate chemotherapy for a pediatric patient with anaplastic large cell lymphoma complicated with liver failure due to the aggravation of biliary hepatopathy by secondary hemophagocytic lymphohistiocytosis

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Abstract

Anaplastic large cell lymphoma (ALCL) accounts for 10–15% of childhood non-Hodgkin lymphoma cases; it is generally chemo-sensitive and is one of the most curable pediatric cancers. We report here a case of pediatric ALCL complicated with acute liver failure due to the aggravation of pre-existing biliary hepatopathy by lymphoma-associated hemophagocytic lymphohistiocytosis (HLH). Although the initial treatment response against ALCL was very good, poor and irreversible liver function due to biliary cirrhosis worsening by lymphoma-associated HLH prevented the patient from receiving further consolidation chemotherapies. To make matters worse, his condition was accompanied with intrahepatic fungal pseudoaneurysm and invasive fungal infection. Thus, we decided to perform an urgent living-donor liver transplantation from his father to correct the patient’s liver function and make it possible to proceed with further ALCL therapy. After the living-donor liver transplantation, the patient successfully received consolidation therapy with brentuximab vedotin. To our knowledge, this may be an early reported case of a pediatric patient undergoing liver transplantation during treatment for ALCL. In most patients with HLH-associated ALCL, liver function improves when ALCL is controlled. However, acute liver failure is occasionally observed in HLH cases with pre-existing liver dysfunction. In such cases, liver transplantation should be considered to correct liver dysfunctions if the disease control of HLH is satisfactory.

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Acknowledgements

We would like to thank Kaoru Yoshida of the Department of Pediatric Hematology and Oncology Research, National Center for Child Health and Development for performing the RT-PCR and direct sequencing of NPM-ALK.

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Authors and Affiliations

  1. Children’s Cancer Center, National Center for Child Health and Development, Tokyo, Japan

    Kenichi Sakamoto, Tomoo Osumi, Satoshi Yoshimura, Motohiro Kato, Daisuke Tomizawa, Takao Deguchi & Kimikazu Matsumoto

  2. Department of Pediatric Hematology and Oncology Research, National Center for Child Health and Development, Tokyo, Japan

    Nobutaka Kiyokawa

  3. Organ Transplantation Center, National Center for Child Health and Development, Tokyo, Japan

    Seiichi Shimizu, Akinari Fukuda, Seisuke Sakamoto & Mureo Kasahara

  4. Department of Pathology, National Center for Child Health and Development, Tokyo, Japan

    Noriyuki Nakano & Takako Yoshioka

  5. Department of Radiology, National Center for Child Health and Development, Tokyo, Japan

    Osamu Miyazaki & Syunsuke Nosaka

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  1. Kenichi Sakamoto
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  2. Tomoo Osumi
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Contributions

KS collected data and provided medical care for this case, and wrote the manuscript supported by MK and DT. TO, SY, SS, MK, DT, SS, OM, SN, MK, and KM provided medical care for this case. TD and NK performed flow-cytometric analysis. NN, TY, and RI performed pathological diagnostic procedures. All authors commented on the manuscript and contributed to the final manuscript.

Corresponding author

Correspondence to Kenichi Sakamoto.

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The authors declare no competing financial interests.

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12185_2020_2949_MOESM1_ESM.pdf

Supplementary material 1 (PDF 223 kb) Figure 1. Image findings at 12 months post-liver transplantation and at 8 months after cessation of chemotherapy. A, B Abdominal computerized tomography (CT) shows no hepatosplenomegaly nor lymphadenopathy; C, D positron emission tomography with CT (PET-CT) reveals no high radiotracer accumulation at lymph nodes.

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Sakamoto, K., Osumi, T., Yoshimura, S. et al. Living-donor liver transplantation providing an adequate chemotherapy for a pediatric patient with anaplastic large cell lymphoma complicated with liver failure due to the aggravation of biliary hepatopathy by secondary hemophagocytic lymphohistiocytosis. Int J Hematol 112, 900–905 (2020). https://doi.org/10.1007/s12185-020-02949-z

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  • DOI: https://doi.org/10.1007/s12185-020-02949-z

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