@article{PAR00011796, title = {{A} severe {W}hipple disease with an immune reconstitution inflammatory syndrome : an additional case of thalidomide efficiency}, author = {{L}e {B}lay, {P}. and {R}akotonirainy, {H}. and {L}agier, {J}. {C}. and {R}aoult, {D}idier and {P}uechal, {X}. and {P}ers, {Y}. {M}.}, editor = {}, language = {{ENG}}, abstract = {{W}e report the case of a 38-year-old man who presented with severe diarrhea, weight loss of 10 kg, ankles paresthesia and severe motor weakness in the left fibular nerve territory after introduction of azathioprine and corticosteroid for proteinuria. {C}oloscopy and gastroscopy revealed a typical aspect of {W}hipple disease ({WD}), associated with both positive {PAS} staining and specific immunohistochemistry. {T}. whipplei {PCR} results were positive in blood, faeces, saliva and duodenal biopsy specimens. {D}iagnosis of {WD} with systemic manifestations was retained and doxycycline plus hydroxychloroquine therapy were started. {T}his treatment improved joint pain, and skin and intestinal symptoms. {O}ne month later, our patient presented with fever and an important inflammatory syndrome ({CRP} 150 mg/d{L} and 16.8 10(9)/{L} leukocytes), while no infection was found despite a thorough review. {W}e concluded it was an immune reconstitution inflammatory syndrome ({IRIS}). {M}anifestations persisted despite increasing corticosteroids and thalidomide (200 mg/day) was introduced with good efficacy on these symptoms. {WD} may be revealed by non-specific symptoms such as weight loss or arthralgia, but also by many other misleading signs. {O}ur observation illustrates the highly polymorphic clinical presentation of {WD}, and the diagnostic difficulties that may arise. {T}his is also a new report of thalidomide effectiveness in {IRIS} in {WD}.}, keywords = {{W}hipple disease ; {P}roteinuria ; {M}otor weakness ; {I}mmune reconstitution ; inflammatory syndrome ; {T}halidomide}, booktitle = {}, journal = {{J}oint {B}one {S}pine}, volume = {81}, numero = {3}, pages = {260--262}, ISSN = {1297-319{X}}, year = {2014}, DOI = {10.1016/j.jbspin.2013.10.007}, URL = {https://www.documentation.ird.fr/hor/{PAR}00011796}, }