title: Atypical focal forms of Whipple's disease seen by rheumatologists.
abstract: We report two atypical cases of focal Whipple's disease with rheumatic presenting symptoms. In one of these cases, the patient presented with chronic intermittent polyarthritis, systemic inflammation, and leukocytosis. Tests were positive for rheumatoid factor and anti-cyclic citrullinated peptide antibodies. There was no structural joint damage. Combined glucocorticoid and methotrexate therapy was only partially effective. Endocarditis requiring emergency valve replacement surgery occurred 4 years later. Evaluation of this event led to the diagnosis of Tropheryma whipplei infection responsible for both the endocarditis and the joint disease. The other patient presented with subacute inflammatory low back pain. His medical history was chiefly remarkable for intermittent inflammatory involvement of the wrists and right knee replacement surgery for osteoarthritis followed by a febrile effusion of the operated knee. Radiographs showed destructive lesions of the wrists. Magnetic resonance imaging findings suggested L2-L3 diskitis. A PCR assay on biopsy specimens from the disk lesions recovered T. whipplei, thus establishing the cause of the diskitis and previous joint manifestations. Combined doxycycline and hydroxychloroquine therapy was followed by full resolution of all clinical and laboratory abnormalities in both patients.
authors.affiliation: Service de Rhumatologie, Centre Hospitalier Universitaire Lyon Sud, 165, rue du Grand-Revoyet, 69495 Pierre-Bénite cedex, France.
authors.forename: Marine
authors.initials: M
authors.lastname: Gaudé
authors.affiliation: Service de Rhumatologie, Centre Hospitalier Universitaire Lyon Sud, 165, rue du Grand-Revoyet, 69495 Pierre-Bénite cedex, France.
authors.forename: Jacques
authors.initials: J
authors.lastname: Tébib
authors.affiliation: Département de Médecine interne, Centre de Référence des Maladies Auto-immunes et Systémiques rares, Hôpital Cochin, Université Paris Descartes, Assistance Publique-Hôpitaux de Paris, Paris, France. Electronic address: xavier.puechal@cch.aphp.fr.
authors.forename: Xavier
authors.initials: X
authors.lastname: Puéchal
mesh_terms: D000369:Aged, 80 and over; D006801:Humans; D008297:Male; D008875:Middle Aged; D008061:Whipple Disease / Q000150:complications / Q000175:diagnosis* / Q000188:drug therapy
keywords: Arthritis; Diskitis; Endocarditis; Tropheryma whipplei; Whipple's disease