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Primary hepatic lymphoma in a patient with rheumatoid arthritis treated with methotrexate.

Tatsumi G, Ukyo N, Hirata H, Tsudo M - Case Rep Hematol (2014)

Bottom Line: MTX was discontinued, and she was carefully monitored thereafter owing to the prolonged history of MTX administration for RA.Rapid progression of PHL was observed; therefore 10 days after the PHL diagnosis, she received 6 cycles of R-THP-COP (rituximab, cyclophosphamide, pirarubicin, vincristine, and prednisolone) therapy and achieved complete remission for more than 1 year.Although MTX-associated lymphoproliferative disorders often show remission after withdrawal of MTX, early diagnosis and treatment are essential for PHL in patients with RA treated with MTX, because of the aggressive nature of the disease.

View Article: PubMed Central - PubMed

Affiliation: Department of Hematology, Osaka Red Cross Hospital, 5-30 Fudegasaki-cho, Tennoji-ku, Osaka 543-8555, Japan.

ABSTRACT
Primary hepatic lymphoma (PHL) has rarely been reported in patients with immunosuppression. We herein describe a case of Epstein-Barr virus- (EBV-) positive PHL in a 67-year-old Japanese woman receiving methotrexate (MTX) treatment for rheumatoid arthritis (RA). The patient, who had been receiving MTX therapy for more than 6 years, presented with low-grade fever and abdominal pain. Initial laboratory tests showed mildly elevated liver enzymes with normal levels of alpha-fetoprotein and carcinoembryonic antigen, and computed tomography scans revealed multiple hepatic tumors with no lymph-node swelling. Examination of liver specimens obtained via ultrasonography-guided needle biopsy indicated EBV-positive diffuse large B cell lymphoma; therefore, she was diagnosed with PHL. MTX was discontinued, and she was carefully monitored thereafter owing to the prolonged history of MTX administration for RA. Rapid progression of PHL was observed; therefore 10 days after the PHL diagnosis, she received 6 cycles of R-THP-COP (rituximab, cyclophosphamide, pirarubicin, vincristine, and prednisolone) therapy and achieved complete remission for more than 1 year. Although MTX-associated lymphoproliferative disorders often show remission after withdrawal of MTX, early diagnosis and treatment are essential for PHL in patients with RA treated with MTX, because of the aggressive nature of the disease.

No MeSH data available.


Related in: MedlinePlus

Clinical course of the patient. The x-axis presents time (days) from admission to our hospital. LDH: lactate dehydrogenase; T-Bil: total bilirubin; and PET/CT: positron emission tomography/computed tomography.
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fig3: Clinical course of the patient. The x-axis presents time (days) from admission to our hospital. LDH: lactate dehydrogenase; T-Bil: total bilirubin; and PET/CT: positron emission tomography/computed tomography.

Mentions: Physical examination at presentation indicated general fatigue; her temperature was 37°C, and her conjunctiva was neither icteric nor anemic. Her performance status was 2. Abdominal tenderness was observed in the right upper quadrant, without any hepatomegaly. The spleen and superficial lymph nodes were not palpable. Her joints of fingers and toes were severely deformed, but she had no pain, tenderness, or swelling in her joints. Laboratory studies on admission showed the following results: total bilirubin content, 2.1 mg/dL (normal range: 0.2–1.0 mg/dL); aspartate aminotransferase level, 153 IU/L (8–38 IU/L); alanine aminotransferase level, 92 IU/L (4–44 IU/L); lactate dehydrogenase (LDH) level, 1646 IU/L (120–260 IU/L); and alkaline phosphatase level, 1991 IU/L (104–338 IU/L). Complete blood counts were normal. Serum interleukin-2 receptor was elevated at 3720 U/mL (145–519 U/mL), but the levels of other tumor markers, such as alpha-fetoprotein (AFP) and carcinoembryonic antigen (CEA), were normal. Serology was negative for hepatitis B virus (HBV) surface antigen, HCV, and human immunodeficiency virus. Computed tomography (CT) revealed multiple hypodense nodules with irregular margins, ranging from 8 to 65 mm, throughout the whole liver (Figure 1(a)). Positron emission tomography (PET)/CT with [18F]-fluorodeoxyglucose (FDG) was performed to further investigate the hepatic lesions and to determine the extent of disease. These lesions demonstrated ring-like hypermetabolic foci, with a maximum standard uptake value of 16.6. No other foci with abnormal FDG uptake were detected elsewhere in the body (Figures 1(b) and 1(c)). A diagnostic ultrasonography-guided fine-needle biopsy was also performed. Histological examination showed diffuse infiltration of medium-to-large atypical lymphoid cells, with necrosis. The cells were positive for CD20 via immunohistochemical analysis and for Epstein-Barr virus- (EBV-) encoded small RNA via in situ hybridization (Figures 2(a)–2(c)). Flow cytometry analysis showed a pathological B-cell phenotype with positivity for CD19 and CD20 and negativity for CD5 and CD10. Cytogenetic analysis detected a complicatedly abnormal karyotype. Bone marrow examination showed no infiltration by the lymphoma. As no other foci of lymphoma were observed, a final diagnosis of MTX-LPD with the features of DLBCL was established. Subsequently, the patient's liver function continued to deteriorate rapidly, despite MTX discontinuation. She then received 6 cycles of R-THP-COP (rituximab, cyclophosphamide, pirarubicin, vincristine, and prednisolone) therapy. Soon after chemotherapy was initiated, her symptoms improved and laboratory measurements of liver function (including LDH levels and total bilirubin content) gradually decreased and returned to normal within 4 weeks (Figure 3). Follow-up FDG-PET/CT was performed 5 weeks after completion of chemotherapy, no FDG uptake by the liver lesions was detected, and only a hypodense area was observed on the CT scan, thus confirming necrotic changes (Figures 1(d) and 1(e)). During the subsequent 1-year follow-up, our patient showed no symptoms or signs of recurrent disease. She also started 500 mg of salazosulfapyridine for RA after the chemotherapy and she experienced no deterioration in her joints.


Primary hepatic lymphoma in a patient with rheumatoid arthritis treated with methotrexate.

Tatsumi G, Ukyo N, Hirata H, Tsudo M - Case Rep Hematol (2014)

Clinical course of the patient. The x-axis presents time (days) from admission to our hospital. LDH: lactate dehydrogenase; T-Bil: total bilirubin; and PET/CT: positron emission tomography/computed tomography.
© Copyright Policy - open-access
Related In: Results  -  Collection

Show All Figures
getmorefigures.php?uid=PMC4290657&req=5

fig3: Clinical course of the patient. The x-axis presents time (days) from admission to our hospital. LDH: lactate dehydrogenase; T-Bil: total bilirubin; and PET/CT: positron emission tomography/computed tomography.
Mentions: Physical examination at presentation indicated general fatigue; her temperature was 37°C, and her conjunctiva was neither icteric nor anemic. Her performance status was 2. Abdominal tenderness was observed in the right upper quadrant, without any hepatomegaly. The spleen and superficial lymph nodes were not palpable. Her joints of fingers and toes were severely deformed, but she had no pain, tenderness, or swelling in her joints. Laboratory studies on admission showed the following results: total bilirubin content, 2.1 mg/dL (normal range: 0.2–1.0 mg/dL); aspartate aminotransferase level, 153 IU/L (8–38 IU/L); alanine aminotransferase level, 92 IU/L (4–44 IU/L); lactate dehydrogenase (LDH) level, 1646 IU/L (120–260 IU/L); and alkaline phosphatase level, 1991 IU/L (104–338 IU/L). Complete blood counts were normal. Serum interleukin-2 receptor was elevated at 3720 U/mL (145–519 U/mL), but the levels of other tumor markers, such as alpha-fetoprotein (AFP) and carcinoembryonic antigen (CEA), were normal. Serology was negative for hepatitis B virus (HBV) surface antigen, HCV, and human immunodeficiency virus. Computed tomography (CT) revealed multiple hypodense nodules with irregular margins, ranging from 8 to 65 mm, throughout the whole liver (Figure 1(a)). Positron emission tomography (PET)/CT with [18F]-fluorodeoxyglucose (FDG) was performed to further investigate the hepatic lesions and to determine the extent of disease. These lesions demonstrated ring-like hypermetabolic foci, with a maximum standard uptake value of 16.6. No other foci with abnormal FDG uptake were detected elsewhere in the body (Figures 1(b) and 1(c)). A diagnostic ultrasonography-guided fine-needle biopsy was also performed. Histological examination showed diffuse infiltration of medium-to-large atypical lymphoid cells, with necrosis. The cells were positive for CD20 via immunohistochemical analysis and for Epstein-Barr virus- (EBV-) encoded small RNA via in situ hybridization (Figures 2(a)–2(c)). Flow cytometry analysis showed a pathological B-cell phenotype with positivity for CD19 and CD20 and negativity for CD5 and CD10. Cytogenetic analysis detected a complicatedly abnormal karyotype. Bone marrow examination showed no infiltration by the lymphoma. As no other foci of lymphoma were observed, a final diagnosis of MTX-LPD with the features of DLBCL was established. Subsequently, the patient's liver function continued to deteriorate rapidly, despite MTX discontinuation. She then received 6 cycles of R-THP-COP (rituximab, cyclophosphamide, pirarubicin, vincristine, and prednisolone) therapy. Soon after chemotherapy was initiated, her symptoms improved and laboratory measurements of liver function (including LDH levels and total bilirubin content) gradually decreased and returned to normal within 4 weeks (Figure 3). Follow-up FDG-PET/CT was performed 5 weeks after completion of chemotherapy, no FDG uptake by the liver lesions was detected, and only a hypodense area was observed on the CT scan, thus confirming necrotic changes (Figures 1(d) and 1(e)). During the subsequent 1-year follow-up, our patient showed no symptoms or signs of recurrent disease. She also started 500 mg of salazosulfapyridine for RA after the chemotherapy and she experienced no deterioration in her joints.

Bottom Line: MTX was discontinued, and she was carefully monitored thereafter owing to the prolonged history of MTX administration for RA.Rapid progression of PHL was observed; therefore 10 days after the PHL diagnosis, she received 6 cycles of R-THP-COP (rituximab, cyclophosphamide, pirarubicin, vincristine, and prednisolone) therapy and achieved complete remission for more than 1 year.Although MTX-associated lymphoproliferative disorders often show remission after withdrawal of MTX, early diagnosis and treatment are essential for PHL in patients with RA treated with MTX, because of the aggressive nature of the disease.

View Article: PubMed Central - PubMed

Affiliation: Department of Hematology, Osaka Red Cross Hospital, 5-30 Fudegasaki-cho, Tennoji-ku, Osaka 543-8555, Japan.

ABSTRACT
Primary hepatic lymphoma (PHL) has rarely been reported in patients with immunosuppression. We herein describe a case of Epstein-Barr virus- (EBV-) positive PHL in a 67-year-old Japanese woman receiving methotrexate (MTX) treatment for rheumatoid arthritis (RA). The patient, who had been receiving MTX therapy for more than 6 years, presented with low-grade fever and abdominal pain. Initial laboratory tests showed mildly elevated liver enzymes with normal levels of alpha-fetoprotein and carcinoembryonic antigen, and computed tomography scans revealed multiple hepatic tumors with no lymph-node swelling. Examination of liver specimens obtained via ultrasonography-guided needle biopsy indicated EBV-positive diffuse large B cell lymphoma; therefore, she was diagnosed with PHL. MTX was discontinued, and she was carefully monitored thereafter owing to the prolonged history of MTX administration for RA. Rapid progression of PHL was observed; therefore 10 days after the PHL diagnosis, she received 6 cycles of R-THP-COP (rituximab, cyclophosphamide, pirarubicin, vincristine, and prednisolone) therapy and achieved complete remission for more than 1 year. Although MTX-associated lymphoproliferative disorders often show remission after withdrawal of MTX, early diagnosis and treatment are essential for PHL in patients with RA treated with MTX, because of the aggressive nature of the disease.

No MeSH data available.


Related in: MedlinePlus