PMID- 21135294
OWN - NLM
STAT- MEDLINE
DCOM- 20110105
LR  - 20130625
IS  - 1539-3704 (Electronic)
IS  - 0003-4819 (Linking)
VI  - 153
IP  - 11
DP  - 2010 Dec 7
TI  - The immune reconstitution inflammatory syndrome in whipple disease: a cohort 
      study.
PG  - 710-7
LID - 10.7326/0003-4819-153-11-201012070-00004 [doi]
AB  - BACKGROUND: Whipple disease, which is caused by infection with Tropheryma 
      whipplei, can be treated effectively with antimicrobials. Occasionally, 
      inflammation reappears after initial improvement; this is often interpreted as 
      refractory or recurrent disease. However, polymerase chain reaction for T. 
      whipplei in tissue is sometimes negative during reinflammation, indicating 
      absence of vital bacteria, and this reinflammation does not respond to 
      antimicrobials but does respond to steroids. OBJECTIVE: To demonstrate that the 
      immune reconstitution inflammatory syndrome (IRIS) occurs in patients treated for 
      Whipple disease. DESIGN: Cohort study. (International Standard Randomised 
      Controlled Trial Number Register registration number: ISRCTN45658456) SETTING: 2 
      academic medical centers in Germany. METHODS: 142 patients treated for Whipple 
      disease out of a cohort of 187 were observed for reappearance of inflammatory 
      signs after effective antibiotic therapy. Definitions of IRIS in HIV infection, 
      tuberculosis, and leprosy were adapted for application to Whipple disease. 
      RESULTS: On the basis of study definitions, IRIS was diagnosed in 15 of 142 
      patients. Symptoms included fever, arthritis, pleurisy, erythema nodosum, 
      inflammatory orbitopathy, small-bowel perforation, and a hypothalamic syndrome. 
      Two patients died. There was a positive correlation with previous 
      immunosuppressive treatment and a negative correlation with previous diarrhea and 
      weight loss. LIMITATIONS: The study was observational and thus has inherent 
      weaknesses, such as incomplete and potentially selective data recording. 
      CONCLUSION: The immune reconstitution inflammatory syndrome was diagnosed in 
      about 10% of patients with Whipple disease in the study cohort; the outcome 
      varied from mild to fatal. Patients who had had previous immunosuppressive 
      therapy were at particular risk. An immune reconstitution syndrome should be 
      considered in patients with Whipple disease in whom inflammatory symptoms recur 
      after effective treatment. Early diagnosis and treatment with steroids may be 
      beneficial; prospective studies are needed. PRIMARY FUNDING SOURCE: European 
      Commission and Deutsche Forschungsgemeinschaft.
FAU - Feurle, Gerhard E
AU  - Feurle GE
AD  - DRK Krankenhaus Neuwied, Neuwied, Germany. g.e.feurle@t-online.de
FAU - Moos, Verena
AU  - Moos V
FAU - Schinnerling, Katina
AU  - Schinnerling K
FAU - Geelhaar, Anika
AU  - Geelhaar A
FAU - Allers, Kristina
AU  - Allers K
FAU - Biagi, Federico
AU  - Biagi F
FAU - Blaker, Hendrik
AU  - Blaker H
FAU - Moter, Annette
AU  - Moter A
FAU - Loddenkemper, Christoph
AU  - Loddenkemper C
FAU - Jansen, Andreas
AU  - Jansen A
FAU - Schneider, Thomas
AU  - Schneider T
LA  - eng
SI  - ISRCTN/ISRCTN45658456
PT  - Journal Article
PT  - Multicenter Study
PT  - Randomized Controlled Trial
PT  - Research Support, Non-U.S. Gov't
PL  - United States
TA  - Ann Intern Med
JT  - Annals of internal medicine
JID - 0372351
RN  - 0 (Anti-Bacterial Agents)
SB  - IM
MH  - Aged
MH  - Anti-Bacterial Agents/therapeutic use
MH  - Arthritis/etiology
MH  - Central Nervous System Diseases/etiology
MH  - Cohort Studies
MH  - Female
MH  - Fever/etiology
MH  - Humans
MH  - Immune Reconstitution Inflammatory Syndrome/diagnosis/drug therapy/*etiology
MH  - Intestinal Perforation/etiology
MH  - Intestine, Small
MH  - Male
MH  - Middle Aged
MH  - Orbital Diseases/etiology
MH  - Prospective Studies
MH  - Recurrence
MH  - Risk Factors
MH  - Skin Diseases/etiology
MH  - Whipple Disease/drug therapy/*immunology
EDAT- 2010/12/08 06:00
MHDA- 2011/01/06 06:00
CRDT- 2010/12/08 06:00
PHST- 2010/12/08 06:00 [entrez]
PHST- 2010/12/08 06:00 [pubmed]
PHST- 2011/01/06 06:00 [medline]
AID - 153/11/710 [pii]
AID - 10.7326/0003-4819-153-11-201012070-00004 [doi]
PST - ppublish
SO  - Ann Intern Med. 2010 Dec 7;153(11):710-7. doi: 
      10.7326/0003-4819-153-11-201012070-00004.