Impact of Diagnosis and Treatment of Tropheryma Whipplei Infection in Patients with Pre-existing Chronic Inflammatory Rheumatic Diseases: Data from the National Tw-IRD Registry

Damien CAILLET PORTILLLO¹, Xavier Puéchal², Yannick Degboe¹, Marie Kostine³, Alexia Michaut⁴, Andre Ramon⁵, Daniel Wendling⁶, Nathalie Costedoat-Chalumeau⁷, Pascal Richette⁸, Hubert Marotte⁹, Justine VIX¹⁰, Jean Jacques Dubost¹¹, Sebastien Ottaviani¹², Gaël Mouterde¹³, Anne Grasland¹⁴, Aline Frazier-Mironer¹⁵, Vincent GERMAIN¹⁶, Fabienne COURY¹⁷, Anne Tournadre¹¹, Martin Soubrier¹⁸, Pauline Brevet¹⁹, Laurent Cavalie²⁰, Laurent Arnaud²¹, Christophe Richez²², Adeline Ruyssen-Witrand²³ and Arnaud Constantin²⁴, ¹Pierre-Paul Riquet University Hospital, Toulouse & Toulouse III University - Paul Sabatier, Rheumatology, Toulouse, France, ²National Referral Center for Rare Systemic Autoimmune Diseases, Cochin Hospital, Paris, France, ³Pellegrin Hospital, University Hospital of Bordeaux, Rheumatology, Bordeaux, France, ⁴Hospital center, Loire Vendée Ocean, Rheumatology, La Roche-sur-Yon, France, La Roche-sur-Yon, France, ⁵University Hospital of DIJON, Rheumatology, Dijon, France, ⁶CHU, University Teaching Hospital, Besançon, France, ⁷Inserm DR Paris 5, Paris, France, ⁸Department of Rheumatology, Hôpital Lariboisière, Assistance Publique-Hôpitaux de Paris, Paris, France, ⁹INSERM 1059, Saint-Etienne, France, ¹⁰University Hospital of poitiers, Rheumatology, Poitiers, France, Poitiers, France, ¹¹University Hospital of Clermont Ferrand, Rheumatology, Clermont-Ferrand, France, ¹²Hopital Bichat-Claude Bernard, Paris, France, ¹³Lapeyronie Hospital, Montpellier, France, ¹⁴Louis-Mourier Hospital, AP-HP, Colombes, Rheumatology, Colombes, France, ¹⁵APHP Hôpital Lariboisire, Paris, France, ¹⁶Pau Hospital, Rheumatology, Pau, France, Pau, France, ¹⁷Pierre-Bénite Hospital, University Hospital Lyon, Rheumatology, Lyon, France, ¹⁸Gabriel-Montpied Hospital, Clermont-Ferrand, France, ¹⁹University Hospital of Rouen, Rheumatology, Rouen, France., Le Havre, France, ²⁰Purpan Hospital, Federal Institute of Biology (IFB), University Hospital, Toulouse, Biology, Toulouse, France, ²¹Hautepierre Hospital, University Hospital of Strasbourg, Rheumatology, Strasbourg, France, ²²Université de Bordeaux, Bordeaux, France, ²³CHU de Toulouse, Toulouse, France, ²⁴Toulouse University Hospital, Toulouse, France

Meeting: ACR Convergence 2022

Keywords: Arthritis, Infectious, Disease-Modifying Antirheumatic Drugs (Dmards), Infection

Session Information

Date: Sunday, November 13, 2022

Title: Infection-related Rheumatic Disease Poster

Session Type: Poster Session B

Session Time: 9:00AM-10:30AM

Background/Purpose: Tropheryma whipplei (Tw) infection is a rare condition, characterized by inflammatory joint symptoms in more than 75% of the cases, which can lead the physician to diagnose chronic inflammatory rheumatic diseases (IRD) and to initiate DMARDs. DMARDs are often ineffective and may reveal digestive signs, systemic manifestations or involvement of other organs. We hypothesized that treatment of Tw infection has a favorable impact on rheumatologic and extra-rheumatologic manifestations attributed to IRD.

To validate this hypothesis, we initiated a registry with the objectives to describe the characteristics of IRD and their treatments, the diagnostic and therapeutic modalities of Tw infections and the impact of the treatment of Tw infection on the evolution of IRD and associated DMARDs.

Methods: We initiated a French National registry including patients with pre-existing IRD, treated with DMARDs, later diagnosed with Tw infection. Cases were identified through a call for observation via the “Club Rhumatismes et inflammations” website. We collected clinical and biological data about the characteristics of IRD and their treatments, the diagnostic and therapeutic modalities of Tw infections, and the impact of the treatment of Tw infection on the evolution of IRD and associated DMARDs.

Results: Seventy-three IRD patients were included. Mean age at diagnosis was 49 years (SD +/- 10.9), with 78% of men, median IRD duration was 79 months (IQR 36; 140), including rheumatoid arthritis (31), spondyloarthritis (14), psoriatic arthritis (6) and other IRDs (22). All IRD patients were treated with DMARDs, with no therapeutic response in 51% of cases, worsening of rheumatologic symptoms in 34% of cases, and occurrence of extra-articular manifestations in 27% of cases. Screening for Tw infection mainly involved saliva and stool PCR, while diagnostic modalities involved organ specific PCR and biopsies, in particular duodenal biopsies (PCR positive in 87% of cases and histology in only 38% of cases). At the time of Tw infection diagnosis, mean age was 58 years (SD +/- 10.1), all patients had joint involvement, 33% axial involvement, 11% entheseal involvement, 84% extra-articular manifestations, 93% elevated CRP, 86% hypoalbuminemia and 67% anemia. Tw infection treatment modalities (median follow-up of 22 months) mainly involved a combination of doxycycline (95%) and hydroxychloroquine (96%), with complete recovery in 79% of cases and Tw-related deaths in 2 cases. At the same time, Tw infection treatment was associated with IRD remission in 93% of cases, with a median time to remission of 2 months (IQR 1; 4.25), leading to DMARD withdrawal in 94% of cases and corticosteroid therapy withdrawal in 65% of cases.

Conclusion: A Tw infection should be considered in IRD patients with peripheral joint involvement and inadequate response to DMARDs, particularly in the presence of extra-articular manifestations, high CRP and hypoalbuminemia. In such patients, positive results of screening and diagnostic tests for Tw infection may lead to the initiation of Tw infection treatment which is associated with complete recovery of Tw infection and rapid remission of the IRD, allowing DMARD and corticosteroid therapy withdrawal in most the cases.

Disclosures: D. CAILLET PORTILLLO, None; X. Puéchal, Roche; Y. Degboe, None; M. Kostine, Bristol-Myers Squibb(BMS), Merck/MSD, Novartis, Janssen, Biogen; A. Michaut, None; A. Ramon, None; D. Wendling, AbbVie/Abbott, Bristol-Myers Squibb(BMS), Merck/MSD, Pfizer, Roche, Amgen, Nordic Pharma, UCB, Novartis, Janssen, Eli Lilly, Sandoz, Galapados, Grunenthal; N. Costedoat-Chalumeau, UCB, Roche; P. Richette, AbbVie, Amgen, Biogen, Bristol Myers Squibb, Celgene, Eli Lilly, Janssen, Novartis, Pfizer, Roche, Sanofi-Aventis, UCB; H. Marotte, AbbVie/Abbott, Amgen, Biogen, Bristol-Myers Squibb(BMS), CellTrion HealthCare, Fresenius Kabi, HealthCare, Janssen, Eli Lilly, Nordic Pharma, Novartis, Medac, Pfizer, Merck/MSD, Galapagos, UCB; J. VIX, None; J. Dubost, None; S. Ottaviani, None; G. Mouterde, None; A. Grasland, None; A. Frazier-Mironer, None; V. GERMAIN, None; F. COURY, None; A. Tournadre, AbbVie, Eli Lilly, Fresenius-Kabi, Novartis, Pfizer, Sanofi, UCB; M. Soubrier, None; P. Brevet, None; L. Cavalie, None; L. Arnaud, AstraZeneca, Novartis, GlaxoSmithKlein(GSK), Bristol-Myers Squibb(BMS), Bristol-Myers Squibb(BMS); C. Richez, AbbVie/Abbott, Amgen, AstraZeneca, Biogen, Bristol-Myers Squibb(BMS), Galapados, GlaxoSmithKlein(GSK), Eli Lilly, Novartis, Pfizer; A. Ruyssen-Witrand, Pfizer, AbbVie/Abbott, Novartis, Eli Lilly, Janssen, Bristol-Myers Squibb(BMS), galapagos, fresenius kabi, Merck/MSD, UCB, Pfizer, Roche, Sanofi; A. Constantin, None.

To cite this abstract in AMA style:

CAILLET PORTILLLO D, Puéchal X, Degboe Y, Kostine M, Michaut A, Ramon A, Wendling D, Costedoat-Chalumeau N, Richette P, Marotte H, VIX J, Dubost J, Ottaviani S, Mouterde G, Grasland A, Frazier-Mironer A, GERMAIN V, COURY F, Tournadre A, Soubrier M, Brevet P, Cavalie L, Arnaud L, Richez C, Ruyssen-Witrand A, Constantin A. Impact of Diagnosis and Treatment of Tropheryma Whipplei Infection in Patients with Pre-existing Chronic Inflammatory Rheumatic Diseases: Data from the National Tw-IRD Registry [abstract]. Arthritis Rheumatol. 2022; 74 (suppl 9). https://acrabstracts.org/abstract/impact-of-diagnosis-and-treatment-of-tropheryma-whipplei-infection-in-patients-with-pre-existing-chronic-inflammatory-rheumatic-diseases-data-from-the-national-tw-ird-registry/. Accessed .

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