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. 2018 Aug;9(2):187-191.
doi: 10.3892/mco.2018.1654. Epub 2018 Jun 14.

Lymphoproliferative disorder with pathological fracture of the femur in a patient with rheumatoid arthritis treated with methotrexate: A case report

Naoto Oebisu  1 , Manabu Hoshi  1 , Makoto Ieguchi  1 , Tadashi Iwai  1 , Sayaka Tanaka  2 , Masahiko Osawa  2 , Hiroaki Nakamura  1
Affiliations

Lymphoproliferative disorder with pathological fracture of the femur in a patient with rheumatoid arthritis treated with methotrexate: A case report

Naoto Oebisu et al. Mol Clin Oncol. 2018 Aug.

Abstract

Methotrexate (MTX) is the key drug for the treatment of rheumatoid arthritis (RA). MTX-treated RA has been associated with the development of lymphoproliferative disorders (LPDs). Notably, the hyperimmune state of RA itself or the immunosuppressive state induced by MTX administration may contribute to development of LPD. Furthermore, Epstein-Barr virus (EBV) has been indicated to contribute to the development of MTX-LPD. MTX-associated LPD (MTX-LPD) may affect nodal or extranodal sites, including the gastrointestinal tract, skin, lungs, kidneys, and soft tissues, at an almost equal frequency. However, it is rare for MTX-LPD to manifest as multiple bone tumors with a pathological fracture. The present study reported the case of a 46-year-old Japanese woman with RA who had complications of EBV-positive MTX-LPD during an approximate 5-year course of MTX therapy. The present study indicated a rare case in which the LPD had spread to multiple bones in a patient with a pathologic fracture. Notably, the LPD was subclassified as diffuse large B-cell lymphoma (DLBCL).

Keywords: diffuse large B-cell lymphoma; methotrexate-associated lymphoproliferative disorder; multiple bone tumors; pathological fracture; rheumatoid arthritis.

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Figures

Figure 1.
Figure 1.
Initial hip X-ray image. Ill-defined osteolytic lesion and pathological fracture of the right femoral trochanter were detected. (A) Anteroposterior view; (B) lateral view. The white arrows indicate pathological fracture of femoral neck.
Figure 2.
Figure 2.
Initial magnetic resonance imaging. The mass was detected diffusely at the bilateral femur and iliac. (A) T1-weighted image; (B) T2-weighted image; (C) contrast-enhanced T1-weighted image.
Figure 3.
Figure 3.
18F-fluoro-deoxy-glucose positron emission tomography (PET). (A) Whole-body PET showed multiple abnormal uptakes. (B) PET-computed tomography (CT) of the thoracic level. The SUVmax was 21.6 at the right axillary lymph node. (C) PET-CT of the iliac bone. The SUVmax was 10.2 at the left iliac bone. (D) PET-CT of the ribs showed abnormal uptakes at some ribs. The SUVmax was 5.46-7.82.
Figure 4.
Figure 4.
Histological findings. (A) H&E staining showed diffuse infiltration of monotonous lymphoid cells. (B-D) Immunohistological studies. Infiltrating mononuclear cells were predominantly positive for CD20 (B) and MUM-1 (C), but negative for B-cell lymphoma 6 (D).
Figure 5.
Figure 5.
At the final follow-up, X-ray shows sclerotic change of bilateral proximal femurs. (A) Anteroposterior view; (B) Lateral view.
See this image and copyright information in PMC

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