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Rheumatology Advance Access originally published online on August 25, 2006
Rheumatology 2007 46(3):484-489; doi:10.1093/rheumatology/kel268
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© The Author 2006. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For Permissions, please email: journals.permissions@oxfordjournals.org

Poncet's disease: reactive arthritis accompanying tuberculosis. Two case reports and a review of the literature

E. J. A. Kroot1,2, J. M. W. Hazes2, E. M. Colin2 and R. J. E. M. Dolhain2

1Department of Rheumatology, St Franciscus Hospital, Rotterdam and 2Department of Rheumatology, Erasmus MC, Rotterdam, The Netherlands.

Correspondence to: R. J. E. M. Dolhain, MD, PhD, Erasmus MC, University Medical Center, Department of Rheumatology, Z-712, PO Box 2040, 3000 CA Rotterdam, The Netherlands. E-mail: r.dolhain{at}erasmusmc.nl


   Abstract
Top
 Abstract
Introduction
 Patients and methods
 Results
 Discussion
 Acknowledgements
 References
 
Objective. Reactive arthritis (ReA) in tuberculosis (TB) is known as Poncet's disease. It is a rare aseptic form of arthritis observed in patients with active TB. We present two such patients and review the literature on Poncet's disease.

Methods. Two patients who were identified with Poncet's disease at the Department of Rheumatology of Erasmus MC, Rotterdam University Hospital, during the last 5 yrs are reported. In addition, a review of the literature on Poncet's disease is given: the PubMed/MEDLINE database was studied up to December 2005 using the term ‘Poncet's disease’ and the terms ‘arthritis’, ‘reactive’ and ‘tuberculosis’.

Results. After careful work-up, the polyarthritis and erythema nodosum in both presented patients with active TB could be diagnosed as Poncet's disease. Resolution of the arthritis with anti-TB drugs occurred in just a few days.

Reviewing the literature, 50 case reports were found. In most reports ‘Poncet's disease’ was described as an aseptic polyarthritis, presumably ReA arthritis developing in the presence of active TB elsewhere. However, no uniform characterization of the term ‘Poncet's disease’ could be abstracted from these reports.

Conclusion. Both presented patients and the review of the literature demonstrate that active TB may be complicated by ReA known as Poncet's disease. Early recognition of this rare complication of TB is of major importance to avoid delayed initiation of appropriate treatment.

KEY WORDS: Tuberculosis, Poncet's disease, Arthritis, Reactive


   Introduction
Top
 Abstract
 Introduction
Patients and methods
 Results
 Discussion
 Acknowledgements
 References
 
As a result of the recent worldwide resurgence of tuberculosis (TB), extrapulmonary manifestations of TB including arthritis will show an increase as well. This is of major importance as musculoskeletal manifestations are the most common form of extrapulmonary TB, accounting for 10–19% of the cases [1–3]. Besides septic TB arthritis, rare examples of non-suppurative reactive arthritis (ReA) have been described in association with TB [4–41]. This clinical entity became known as Poncet's disease [3, 41]. Since not all clinicians are aware of Poncet's disease, this entity is likely to be underdiagnosed.

We report two patients who presented with arthritis and erythema nodosum, in which a diagnosis of Poncet's disease was made. In addition, a review of the literature on Poncet's disease is presented.


   Patients and methods
Top
 Abstract
 Introduction
 Patients and methods
Results
 Discussion
 Acknowledgements
 References
 
Case reports
During the last 5 yrs two cases of Poncet's disease were identified at the Department of Rheumatology of Erasmus MC, Rotterdam University Medical Center. These cases are presented.

Review
The PubMed/MEDLINE database was studied up to December 2005 using the term ‘Poncet's disease’ and the terms ‘arthritis’, ‘reactive’ and ‘tuberculosis’. Articles in all languages were included. Only cases of presumed non-septic arthritis in the presence of active TB were included. In addition, articles were only included when disease characteristics could be abstracted from the case reports.


   Results
Top
 Abstract
 Introduction
 Patients and methods
 Results
Discussion
 Acknowledgements
 References
 
Case 1
A 45-yr-old man born in Suriname was admitted with relapsing fever for 11/2 weeks, muscle weakness and fatigue. Physical examination revealed temperature of 38.8°C, arthritis of the second metacarpophalangeal joint of the right hand, right knee and both ankles and erythema nodosum on both lower legs. Laboratory testing showed leucocytes of 13.8 x 109 E/l and an erythrocyte sedimentation rate (ESR) of 128 mm/h. IgM-RF and anti-cyclic citrullinated peptide (anti-CCP) antibodies, were negative. On an X-ray of the chest infiltration of the lingula of the left lung was observed. X-rays of ankles, hands and feet showed no abnormalities. Standard cultures and cultures for TB of blood and mucus were negative. Analysis of synovial fluid of the right knee and both ankles revealed leucocytes between 4.0 and 8.2 x 109 E/l and no crystals. Standard cultures and cultures for TB of all obtained synovial fluid samples were negative.

Because of the infiltration in the lingua, treatment with clarithromycin and amoxicillin/clavulanic acid was started. Hereupon, the patient improved. Nevertheless, the arthritis persisted. On a control chest X-ray, performed 8 weeks after admission, the infiltration had remained unaltered. For that reason, additional investigations were performed including bronchoscopy to obtain specimen for histology and culture. After 4 weeks, one culture of bronchial mucus revealed Mycobacterium tuberculosis and Poncet's disease was diagnosed. Therapy with isoniazide, ethambutol, pyrazinamide and rifampicin was started after which the arthritis resolved in a few days.

Case 2
A 22-yr-old Nigerian man was admitted for persistent fever for several months and generalized joint pains. A week before presentation, the patient had experienced swelling of both hands. Examination of the joints revealed arthritis of both elbows and the left wrist, dactylitis of both second and third digits, tendinitis of both achilles tendons and arthritis of the right ankle. Further physical examination revealed lymphadenopathy in the right armpit and erythema nodosum on both lower legs. Laboratory testing, including ESR, values of IgM-RF and anti-CCP, showed no abnormalities. Sexual transmitted diseases were excluded. On X-rays of the thorax, hands, feet and ankles, no abnormalities were observed. Synovial fluid analysis from the right ankle revealed leucocytes of 5.2 x 109 E/l and no crystals. Standard cultures and cultures for TB of the synovial fluid were negative. Since no diagnosis could be made, the lymph node in the right armpit was extirpated. Histological examination of the obtained tissue revealed granulomas, including epithelioid cells and multinucleated giant cells. Cultures from the lymph node revealed M. tuberculosis. The polyarthritis was diagnosed as Poncet's disease. Therapy with ethambutol, pyrazinamide, isoniazide and rifampicin was started after which the arthritis resolved in a few days.

Review
By studying the PubMed/MEDLINE database, 35 reports containing disease characteristics of 57 patients, were found [7–41]. Seven cases were not included in the analysis: six patients suffered only from athralgias [8, 16, 21] and in one case, arthritis was reported after 6-month therapy with anti-tuberculous (anti-TB) drugs [35]. From the review of 102 patients with suspected Poncet's disease, disease characteristics of only three patients could be abstracted [11]. So finally, 50 cases were included. Data extracted from these cases are presented in Table 1. Twenty-nine cases (68%) involved males. Although by definition, Poncet's disease is a non-septic arthritis, only in 15 cases (30%) was septic tuberculous arthritis ruled out by culture or histology. In five patients (10%) [7, 13, 17, 19, 23], septic tuberculous arthritis of one joint was demonstrated in addition to presumed non-septic arthritis of other joints. Septic tuberculous arthritis was demonstrated either by histology [7, 17, 23], culture [13] or characteristic X-ray findings [7, 17, 19]. In two of these patients, septic polyarticular tuberculous arthritis was ruled out by culture of other joints [13, 19] suggesting that these cases represent a mixture of septic tuberculous arthritis and Poncet's disease. In the other three patients [7, 17, 23], no additional joint cultures were performed. Besides the three patients with characteristic X-ray findings due to tuberculous destruction of bone and joints, no irreversible joint damage was observed in the 50 case reports. Fifteen patients (30%) presented with an oligoarthritis of less than four joints and the other patients presented with polyarthritis. In eight patients (16%), no localization of polyarthritis was described. In the other 42 cases, knees (62%) and ankles (57%) were the most commonly involved joints followed by the wrists (48%). In 12 patients (29%), small joints of hand or feet were involved as well. In 10 (20%) patients, the diagnosis of TB was based on the clinical picture only. In 48% of the patients, the TB was localized extrapulmonarily. In 50% of the patients, abnormalities on standard chest X-rays were observed. Only 6% of the patients presented with erythema nodosum [12, 27, 36]. Resolution of the arthritis with anti-TB drugs ranged from 1 week to 4 months.


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  		TABLE 1. Characteristics of 50 cases of Poncet's disease

 

   Discussion
Top
 Abstract
 Introduction
 Patients and methods
 Results
 Discussion
Acknowledgements
 References
 
The two presented cases and the review demonstrate that active tuberculosis may be complicated by ReA known as Poncet's disease. It is widely known that tuberculous septic monoarthritis, in which M. tuberculosis can be isolated from the joint, may complicate tuberculous infection; that active TB may be complicated by a sterile ReA is less known and therefore often missed.

The diagnosis ‘Poncet's disease’ is used to indicate an aseptic polyarthritis, presumably a ReA, developing in the presence of active TB elsewhere [42]. It has been suggested that Poncet's disease mainly occurs in patients with extrapulmonary TB [3] and that the presence of erythema nodosum is an important hallmark of this disease. However, in the reviewed cases, extrapulmonary TB was only present in half of the patients and only 6% of the patients presented with erythema nodosum.

TB was formerly constituted the most important cause of erythema nodosum [43]. But at present, the prevalence of primary TB among patients presenting with erythema nodosum has decreased markedly [44]. Therefore, currently the differential diagnosis of patients presenting with erythema nodosum and arthritis should consist of systemic disorders (sarcoidosis and inflammatory bowel disease), infections (Streptococcal pharyngitis, TB, histoplasmosis, coccidioidomycosis, Chlamydophila pneumoniae and enteric infections) and use of particular drugs (sulfonamides, oral contraceptives and penicillin) [44, 45]. In our patients, the diagnosis Lofgren's syndrome, a sarcoidosis associated disorder defined as the triad of hilar adenopathy, acute arthritis and erythema nodosum was considered as well, but seemed less likely because Lofgren's syndrome is usually a self-limiting disorder rapidly resolving with symptomatic therapy [45].

Five patients with oligo- or polyarthritis were described that demonstrated a septic tuberculous arthritis of one joint in addition to presumed non-septic arthritis of other joints [7, 13, 17, 19, 23]. In two of these patients, the non-septic nature of the arthritis of the other joints was underscored by culture and histology [13, 19]. Therefore, it has been suggested that septic tuberculous arthritis can be accompanied by a ReA in other joints [7, 13, 17], as has been proposed for gonococcal arthritis [46]. For some authors, however, these case reports are a reason to question the existence of Poncet's disease and state that all cases of TB-associated arthritis require extensive histological examination and culture of synovial tissue [23]. We did not perform synovial tissue analysis in our patients. First, synovial tissue analysis should only be performed when therapeutic consequences are implied. This was not the case. Secondly, synovial tissue biopsies of TB arthritis may be complicated by fistulae [1]. And thirdly, the negative cultures for TB of multiple joint aspirates from both patients support a diagnosis of Poncet's disease, since in septic TB arthritis 79% of the synovial fluid cultures have been reported to be positive [1]. Finally, septic tuberculous arthritis responds rather slowly to anti-TB drug treatment [1], whereas the arthritis in the presented patients resolved in just a few days, as has been reported in most reviewed cases.

Although Poncet's disease is considered a ReA, the clinical presentation of Poncet's disease differs from the classical pattern of ReA [47, 48]. In contrast to ReA, the onset of symptoms in Poncet's disease before the start of arthritis is much longer than just a few weeks, whereas resolution of arthritis upon starting of adequate anti-tuberculous therapy is mostly within a few weeks. Chronic arthritis has never been reported in Poncet's disease. Furthermore, Poncet's disease is generally, except in two reports [13, 24], not associated with sacroiliitis.

Despite the difference in clinical presentation, the pathogenetic mechanism is considered to be similar. It has been hypothesized that after infection, as a result of systemic immunization, sensitized CD4+ cells together with bacterial antigens migrate to the joints and cause arthritis. This hypothesis is supported by the animal model of adjuvant arthritis in which injection of heat-killed desiccated M. Tuberculosis (complete Freund's adjuvant) results in arthritis [1]. The human counterpart of this model is observed in patients with bladder cancer receiving immunotherapy by means of intravesical instillation of attenuated M. bacillus Calmette-Guérin. In 0.5% of these patients, a reactive polyarthritis is observed [1].

In conclusion, the differential diagnosis of patients at risk for TB presenting with arthritis should definitely include Poncet's disease, even if these patients do not demonstrate erythema nodosum. Correct identification of this rare complication of TB may avoid delayed initiation of appropriate treatment.


   Acknowledgements
Top
 Abstract
 Introduction
 Patients and methods
 Results
 Discussion
 Acknowledgements
References
 
We are grateful to Dr Manuel Castro Cabezas, Dr Alena Uss, Marjolein van Eersel and Dr Tetsuro Namba for translating the case reports that were not published in English.

The authors have declared no conflicts of interest.


   References
Top
 Abstract
 Introduction
 Patients and methods
 Results
 Discussion
 Acknowledgements
 References
 

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Submitted 3 February 2006; revised version accepted 4 July 2006.
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