Methotrexate-related Epstein-Barr Virus (EBV)-associated lymphoproliferative disorder--so-called "Hodgkin-like lesion"--of the oral cavity in a patient with rheumatoid arthritis.
Patients affected by autoimmune diseases (rheumatoid arthritis, psoriasis, dermatomyositis) who are treated with methotrexate (MTX) sometimes develop lymphoproliferative disorders (LPDs). In approximately 40% of reported cases, the affected sites have been extranodal, and have included the gastrointestinal tract, skin, lung, kidney, and soft tissues. However, MTX-associated LPD (MTX-LPD) is extremely rare in the oral cavity. Here we report a 69-year-old Japanese woman with rheumatoid arthritis (RA) who developed MTX-LPD resembling Hodgkin's disease--so-called "Hodgkin-like lesion"--in the left upper jaw. Histopathologically, large atypical lymphoid cells including Hodgkin or Reed-Sternberg-like cells were found to have infiltrated into granulation tissue in the ulcerative oral mucosa. Immunohistochemistry showed that the large atypical cells were positive for CD20, CD30 and Epstein-Barr virus (EBV)-latent infection membrane protein-1 (LMP-1) and negative for CD15. EBV was detected by in situ hybridization (ISH) with EBV-encoded small RNA (EBER), and polymerase chain reaction (PCR) for LMP-1 and EBNA-2 in material taken from the formalin-fixed, paraffin-embedded specimen. To our knowledge, this is the first reported case of MTX-related EBV-associated LPD (MTX-EBVLPD), "Hodgkin-like lesion", of the oral cavity in a patient with RA.
Kikuchi
Kentaro
K
Division of Pathology, Department of Diagnostic and Therapeutic Sciences, Meikai University School of Dentistry, 1-1 Keyakidai, Sakado, Saitama 350-0283, Japan. k-kikuchi@dent.meikai.ac.jp
Miyazaki
Yuji
Y
Tanaka
Akio
A
Shigematu
Hisao
H
Kojima
Masaru
M
Sakashita
Hideaki
H
Kusama
Kaoru
K
D000368:Aged; D018501:Antirheumatic Agents / Q000009:adverse effects*; D001172:Arthritis, Rheumatoid / Q000188:drug therapy*; D001706:Biopsy; D020031:Epstein-Barr Virus Infections / Q000150:complications* / Q000473:pathology; D005260:Female; D004854:Herpesvirus 4, Human / Q000235:genetics / Q000302:isolation & purification*; D006801:Humans; D008232:Lymphoproliferative Disorders / Q000150:complications / Q000473:pathology / Q000821:virology*; D008727:Methotrexate / Q000009:adverse effects*; D009055:Mouth / Q000473:pathology; D012367:RNA, Viral / Q000235:genetics
D018501:Antirheumatic Agents; D012367:RNA, Viral; D008727:Methotrexate