Concurrent langerhans cell histiocytosis and B-lineage lymphoid proliferation in the bone marrow

Korean J Lab Med. 2009 Oct;29(5):402-5. doi: 10.3343/kjlm.2009.29.5.402.

Abstract

We present three cases of concurrent Langerhans cell histiocytosis (LCH) and B-lineage lymphoid cell infiltrations and/or nodules in the bone marrow. The first patient was a 25-month-old boy who presented with LCH on the right shoulder and multiple osteolytic lesions. Bone marrow biopsy showed the presence of LCH and two large lymphoid nodules of B-lineage, which were located in the paratrabecular region. Both LCH and the lymphoid nodules resolved after treatment with prednisone, vinblastine, methotrexate, and cyclophosphamide. The second patient was a 7-month-old girl who presented with LCH in the scalp and bone marrow. In spite of the treatment, a follow-up bone marrow analysis performed after 16 months showed LCH and increased B-lineage lymphoid cells in the interstitial area. The third patient was a 26-month-old girl, and imaging studies revealed reddish skin lesions and multiple osteolytic lesions. Skin biopsy and bone marrow biopsy did not show the presence of LCH; however, we initiated the treatment on the basis of the results of imaging studies. The follow-up study after 6 months showed the presence of LCH and large, patchy infiltration of B-lymphoid cells. We report three rare cases of concurrent bone marrow involvement of LCH and B-lineage lymphoid proliferation, which strongly suggest lymphoid malignancy. Further, clonal changes should be studied to elucidate the common pathogenic mechanism between the two diseases.

Publication types

  • Case Reports

MeSH terms

  • Antineoplastic Agents / therapeutic use
  • B-Lymphocytes / immunology
  • B-Lymphocytes / pathology*
  • Bone Marrow / immunology
  • Bone Marrow / pathology*
  • Cell Proliferation
  • Child
  • Child, Preschool
  • Cyclophosphamide / therapeutic use
  • Drug Therapy, Combination
  • Female
  • Histiocytosis, Langerhans-Cell / diagnosis*
  • Histiocytosis, Langerhans-Cell / drug therapy
  • Histiocytosis, Langerhans-Cell / pathology
  • Humans
  • Male
  • Methotrexate / therapeutic use
  • Prednisone / therapeutic use
  • Vinblastine / therapeutic use

Substances

  • Antineoplastic Agents
  • Vinblastine
  • Cyclophosphamide
  • Prednisone
  • Methotrexate