Whipple's disease diagnosed during biological treatment for joint disease.
OBJECTIVE
Increased susceptibility to infections is among the main safety concerns raised by biological agents. We describe five cases of Whipple's disease diagnosed during treatment with biological agents.


METHODS
We retrospectively identified five cases of Whipple's disease diagnosed between 2003 and 2009 in patients treated with TNFalpha antagonists in five French hospitals.


RESULTS
Five patients (four male; mean age: 50.4 years; range: 38-67) underwent biological therapy according to prior diagnoses of rheumatoid arthritis (n=2), ankylosing spondylitis (n=2), or spondyloarthropathy (n=1). Biological therapy failed to control the disease, which responded to appropriate antibiotics for Whipple's disease. Retrospectively, clinical symptoms before biological therapy were consistent with Whipple's disease. All five patients had favorable outcomes (mean follow-up, 29 months [13-71]).


CONCLUSIONS
Biological therapy probably worsened preexisting Whipple's disease, triggering the visceral disorders. Whipple's disease must be ruled out in patients with joint disease, as patients with this spontaneously fatal condition should not receive immunosuppressive agents.
Hoppé
Emmanuel
E
Service de rhumatologie, Inserm U922, centre hospitalier universitaire d'Angers, 4, rue Larrey, 49933 Angers cedex 9, France. emhoppe@chu-angers.fr
Masson
Charles
C
Audran
Maurice
M
Drillon
Marie
M
Andreu
Marita
M
Saraux
Alain
A
Berthelot
Jean-Marie
JM
Maugars
Yves
Y
Hmamouchi
Ihsane
I
Morel
Jacques
J
D000068879:Adalimumab; D000328:Adult; D000368:Aged; D000900:Anti-Bacterial Agents / Q000627:therapeutic use; D000911:Antibodies, Monoclonal / Q000009:adverse effects / Q000627:therapeutic use; D061067:Antibodies, Monoclonal, Humanized; D001172:Arthritis, Rheumatoid / Q000188:drug therapy*; D004318:Doxycycline / Q000009:adverse effects / Q000627:therapeutic use; D000068800:Etanercept; D005260:Female; D006801:Humans; D006886:Hydroxychloroquine / Q000627:therapeutic use; D007074:Immunoglobulin G / Q000009:adverse effects / Q000627:therapeutic use; D007166:Immunosuppressive Agents / Q000009:adverse effects / Q000627:therapeutic use*; D000069285:Infliximab; D008297:Male; D008875:Middle Aged; D018124:Receptors, Tumor Necrosis Factor / Q000627:therapeutic use; D012189:Retrospective Studies; D025242:Spondylarthropathies / Q000188:drug therapy*; D013167:Spondylitis, Ankylosing / Q000188:drug therapy*; D014409:Tumor Necrosis Factor-alpha / Q000037:antagonists & inhibitors*; D008061:Whipple Disease / Q000175:diagnosis* / Q000188:drug therapy
D000900:Anti-Bacterial Agents; D000911:Antibodies, Monoclonal; D061067:Antibodies, Monoclonal, Humanized; D007074:Immunoglobulin G; D007166:Immunosuppressive Agents; D018124:Receptors, Tumor Necrosis Factor; D014409:Tumor Necrosis Factor-alpha; D006886:Hydroxychloroquine; D000069285:Infliximab; D000068879:Adalimumab; D004318:Doxycycline; D000068800:Etanercept