Unusual Presentation of SET::NUP214-Associated Concomitant Hematological Neoplasm in a Child-Diagnostic and Treatment Struggle

Int J Mol Sci. 2023 Sep 22;24(19):14451. doi: 10.3390/ijms241914451.

Abstract

Simultaneous multilineage hematologic malignancies are uncommon and associated with poorer prognosis than single-lineage leukemia or lymphoma. Here, we describe a concomitant malignant neoplasm in a 4-year-old boy. The child presented with massive lymphoproliferative syndrome, nasal breathing difficulties, and snoring. Morphological, immunocytochemical, and flow cytometry diagnostics showed coexistence of acute myeloid leukemia (AML) and peripheral T-cell lymphoma (PTCL). Molecular examination revealed a rare t(9;9)(q34;q34)/SET::NUP214 translocation as well as common TCR clonal rearrangements in both the bone marrow and lymph nodes. The disease showed primary refractoriness to both lymphoid and myeloid high-dose chemotherapy as well as combined targeted therapy (trametinib + ruxolitinib). Hence, HSCT was performed, and the patient has since been in complete remission for over a year. This observation highlights the importance of molecular techniques for determining the united nature of complex SET::NUP214-positive malignant neoplasms arising from precursor cells with high lineage plasticity.

Keywords: HSCT; NUP214; fusion genes; pediatric leukemia and lymphoma.

Publication types

  • Case Reports

MeSH terms

  • Bone Marrow / pathology
  • Child, Preschool
  • Humans
  • Leukemia, Myeloid, Acute* / complications
  • Lymphoproliferative Disorders* / complications
  • Lymphoproliferative Disorders* / diagnosis
  • Lymphoproliferative Disorders* / genetics
  • Male
  • Nuclear Pore Complex Proteins / genetics
  • Remission Induction
  • Translocation, Genetic

Substances

  • Nuclear Pore Complex Proteins
  • NUP214 protein, human

Grants and funding

Cytogenetics and high-throughput sequencing assessment were supported by the “Nauka—detiam” charity foundation.