ISSN 1439-7595 (print), 1439-7609 (online) Mod Rheumatol, 2014; 24(1): 199–205 © 2013 Japan College of Rheumatology DOI 10.3109/14397595.2013.852839


Refractory pseudoseptic arthritis in Behc¸et’s disease successfully treated with infliximab: a case report and literature review Walter Alberto Sifuentes Giraldo • Carlos Antonio Guille´n Astete • Claudia Murillo Romero • Irene Amil Casas • Ana Marı´a Rodrı´guez Garcı´a Francisco Javier Bachiller Corral

Received: 20 April 2012 / Accepted: 9 August 2012 Ó Japan College of Rheumatology 2012

Abstract Arthritis associated with Behc¸et’s disease is typically nonerosive and nondeforming, and most patients respond to colchicine treatment. However, destructive arthritis and refractory arthritis have also been reported on occasion. Elevated white blood cell counts may occur in synovial fluid in BD, but pseudoseptic arthritis is a very rare event in this disease. We report a patient with BD and rapidly progressive deforming pseudoseptic arthritis refractory to colchicine, corticosteroids, and methotrexate, who entered remission after infliximab treatment. Keywords Behc¸et’s disease  Pseudoseptic arthritis  TNF-a blockers  Infliximab

Introduction Behc¸et’s disease (BD) is a chronic, inflammatory, multisystem disease characterized by oral and genital aphthae associated with cutaneous lesions and ophthalmic, neurologic, and rheumatologic manifestations [1]. Although rheumatologic manifestations are not included in the International Study Group (ISG) criteria for BD [2], joint involvement is one of the most frequent manifestations following oral and genital aphthae, occurring in at least one-half of patients, and is the first disease manifestation in 9–16.5 % [3–5]. The reported prevalence of arthritis among different populations varies between 40 and

W. A. Sifuentes Giraldo (&)  C. A. Guille´n Astete  C. Murillo Romero  I. Amil Casas  A. M. Rodrı´guez Garcı´a  F. J. Bachiller Corral Department of Rheumatology, University Hospital Ramo´n y Cajal Ctra, Colmenar Viejo Km. 9,100, 28034 Madrid, Spain e-mail: [email protected] Published online: 07 September 2012

70 % [6–9]. Typically, arthritis associated with BD has an asymmetric mono-oligoarticular pattern and follows a recurrent, self-limited, nonerosive, and nondeforming course [1, 8, 9]. The most frequently affected locations are large joints such as the knees, ankles, wrists, and elbows [8–10]. The occurrence of destructive arthritis in BD has been reported on occasion [11–14]. Pseudoseptic arthritis is a term used to describe arthritis that mimics septic arthritis in both synovial fluid characteristics [white blood cell count (WBC) [50000/mm3, purulent appearance] and clinical expression (fever, malaise, severe joint involvement) [15]. Pseudoseptic arthritis has been associated with several inflammatory conditions, such as rheumatoid arthritis and microcrystalline arthritis [16]. Although high WBC counts may occur in synovial fluid of patients with BD [8, 17], pseudoseptic arthritis has rarely been reported in this disease [18, 19]. Most patients with BD-associated arthritis respond to colchicine treatment [20, 21]. However, in some rare cases of refractory arthritis, treatment with tumor necrosis factor a (TNF-a) blockers has also proven effective [22–24]. We report a patient with BD and refractory, destructive monoarthritis involving the left elbow, with synovial fluid characteristics compatible with pseudoseptic arthritis, and a good clinical response to infliximab treatment. Clinical characteristics and the outcomes of other reported cases of pseudoseptic arthritis associated with BD are also presented and compared with our case.

Case report A 32-year-old Spanish man was admitted in August 2009 to the rheumatology inpatient clinic of an university hospital due to having suffered from arthritis in the left elbow


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for one month. He referred to similar episodes in the same location since the age of 15, characterized by inflammatory pain, swelling and functional limitation lasting 1–2 days spontaneously or with the use of nonsteroidal anti-inflammatory drugs (NSAIDs). He suffered 1–2 episodes per year and stayed asymptomatic between episodes. He did not present fever or constitutional complaints and initially underwent NSAID and low-dose corticosteroid treatment without improvement. Since the age of 10, he had suffered recurrent episodes of oral aphthae, although he had not sought medical advice about these episodes, and had used only antiseptic mouthwashes to treat them. No history of genital aphthae was present but, at the age of 29, he presented a perianal lesion with subsequent development of an abscess and fistula that required surgical management. He also referred to acneiform lesions in the back and folliculitis-like lesions in the inner aspects of both thighs since adolescence. No episodes of ocular inflammation, digestive or other complaints were referred to. There was no family history of spondyloarthritis, psoriasis, inflammatory bowel disease, uveitis, or other rheumatic diseases. Physical examination confirmed the presence of skin lesions, oral aphthae, and arthritis in the left elbow with limited flexion to 100° and extension to 15°. The fundus examination showed no findings of uveitis. Pathergy skin reaction was negative. Blood chemistry was in the normal range and hemogram showed only leukocytosis (15600/ mm3) with neutrophilia (86.4 %) without other alterations. Acute-phase reactants were elevated (CRP 61 mg/L, ESR 44 mm/1st h), but all of the immunologic parameters studied, including rheumatoid factor, anti-cyclic

citrullinated peptide antibodies, antinuclear antibodies, anti-neutrophil cytoplasmic antibodies, complement, and immunoglobulins, were in their normal ranges or were negative. HLA typing showed positivity for the B51 haplotype, but B27 was negative. Serological tests for HBV, HBC, HIV, Brucella and syphilis, as well as the intradermal tuberculin reaction, were also negative. Tests for inflammatory bowel disease, including anti-Saccharomyces cerevisae antibodies, fecal calprotectin, and enteroscopy, were negative. Plain X-ray of left elbow showed only mildly increased density of soft tissue with conserved joint space and no bony lesions (Fig. 1). Ultrasonography showed mild effusion and synovial hypertrophy with a mild power-Doppler signal in the posterior recess. Magnetic resonance (MR) yielded similar findings without osteomyelitis data. Arthrocentesis was attempted twice but fluid for analysis was not obtained. The prednisone dose was increased to 0.5 mg/kg/day (height and weight of the patient were 169 cm and 75 kg, respectively), and significant improvement in the patient’s inflammation and mobility was observed within few weeks. Methotrexate 20 mg/week was added as a corticosteroidsparing agent and colchicine 1 mg/day was added for the oral aphthae. From November 2009 to April 2011, he did not present oral aphthae and the arthritis in his left elbow was controlled, such that he recovered flexion to 130° and extension to 10°. Prednisone and colchicine were withdrawn and methotrexate was maintained. In April 2011, he presented again with oral aphthae, left elbow arthritis, and worsening of his back skin lesions, without fever or other complaints. CRP was increased (30.8 mg/L), but no other analytical alterations were found.

Fig. 1 a X-ray of left elbow in August 2009 showing mildly increased density of soft tissue with conserved joint space and no erosions. b In May 2011, rapid progression was evident, with severe

joint space narrowing, flexion contracture, further increased density of soft tissue, but without apparent erosions

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Fig. 2 Power-Doppler ultrasound scan of the left elbow posterior recess in May 2011 (a) showing significant effusion, synovial hypertrophy, and a moderate power-Doppler signal. The ultrasound

examination performed in January 2012 (b), three months after the beginning of infliximab treatment, showed complete resolution of the synovitis

Ultrasonography of the left elbow showed significant joint effusion, increased synovial hypertrophy, and a moderate power-Doppler signal (Fig. 2). MR confirmed the presence of extensive synovitis distending all the recesses of the left elbow without associated bone marrow edema or muscular alterations. Arthrocentesis did obtain fluid (2, 8, and 10 mL at different times) with a purulent appearance. Fluid analysis showed an elevated WBC count (up to 154,000/mm3) and neutrophilia (92.7 %). Crystals were not evident in polarized light microscopy, and all microbiological studies, including cultures for aerobic and anaerobic bacteria, fungi, and mycobacteria, as well as broad-range polymerase chain reaction, were negative. Prednisone and colchicine were reintroduced, but the patient suffered persistent inflammatory signs in his left elbow and accelerated functional impairment. X-rays showed rapid progression with severe joint space narrowing but without apparent erosions (Fig. 1). A synovial biopsy of the left elbow was performed and histopathologic examination showed only chronic synovitis with an abundant mixed inflammatory infiltrate (neutrophils and lymphocytes), without granulomas, vasculitis signs, or lymphoid follicle formation. All microbiological studies of the biopsy specimen, including cultures for aerobic and anaerobic bacteria, fungi and mycobacteria, Ziehl–Neelsen stain, and broad-range polymerase chain reaction, were also negative. After excluding infection, the antibiotic treatment was retired and an intra-articular (IA) corticosteroid injection in the left elbow was performed, but no improvement was observed. Due to the refractory nature of the arthritis, IFX was started in November 2011. IFX was administered as intravenous infusions (doses of 5 mg/kg); the first infusions were administered at weeks 0, 2, and 6, and every 8 weeks

thereafter. Following the first administration, the patient noticed improvement in the pain and swelling in his left elbow; both were clinically evident at examination after the third dose, and the patient experienced a partial recovery of elbow mobility. Oral aphthae and acneiform lesions responded faster, with these lesions resolving after three weeks of treatment. After three months of IFX administration, inflammatory signs in his left elbow were absent, mobility had recovered to 140° for flexion and 10° for extension, acute-phase reactants were in the normal range (CRP 2.5 mg/L), and ultrasonography showed resolution of the synovitis and no power-Doppler signal in the left elbow recess (Fig. 2). To date, the patient is still receiving IFX treatment, while corticosteroids have been suspended and the methotrexate dose reduced to 10 mg/week. There is no evidence of radiological progression of joint damage in his left elbow, and there have been no new episodes of arthritis nor other manifestations of BD activity.

Discussion Pseudoseptic arthritis is defined as extremely inflammatory arthritis that is not due to infection, with clinical and synovial fluid characteristics that mimic septic arthritis [15]. Patients complain of intense pain, effusion, and erythema of a joint, most commonly a knee. Fever and elevated inflammatory markers are present in some but not all cases [25]. Several diseases are reported to cause pseudoseptic arthritis, including rheumatoid arthritis, juvenile idiopathic arthritis, psoriatic arthritis, reactive arthritis, lupus, gout, pseudogout, transitory synovitis of the hip, neuropathic arthropathy, hemarthrosis, villonodular

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Clinical and US resolution of arthritis with infliximab treatment, but residual mobility limitation IA intra-articular, NA no data available, ref reference, SF synovial fluid, US ultrasonographic, WBC white blood cell count

Methotrexate, corticosteroids, colchicine, IA corticosteroid injection, infliximab 154000/ mm3, 92.7 % Worsening of oral aphthae and skin lesions Left elbow Negative Oral aphthae, perianal involvement, acneiform and folliculitis-like lesions Positive Spanish Male Present case


Complete resolution IA corticosteroid injection and colchicine NA Fever Left knee Positive Oral aphthae Positive TurkishCypriot Male Case 2 [18]


Arthritis improvement and fever disappearance Colchicine, cyclosporine A, corticosteroids Male Case 1 [17]




Oral aphthae and genital ulcers


Both knees


95000/ mm3, 97 %

Outcome Treatment SF WBC count and neutrophil % Systemic complaints Joint involvement Pathergy Extra-articular manifestations HLAB51 Origin Age Sex Case [ref]

synovitis, foreign-body reaction, dialysis-related amyloidosis, sickle cell disease, leukemia, and metastatic carcinoma [15, 16, 26–31]. It has also been reported after accidental intra-articular deposition of pneumococcal vaccine polyvalent, hyaluronan viscosupplementation, and etanercept treatment [32–34]. In our case, after rationally excluding infection or other causes of pseudoseptic arthritis, it was assumed to be a manifestation of BD. This patient was diagnosed with BD based on oral aphthae and skin lesions, along with other BD manifestations such as arthritis, perianal involvement, and HLA-B51 positivity. Although this patient fulfilled only two classification criteria for ISG (one major and one minor), the full clinical picture was compatible with a diagnosis of BD. Elevated WBC counts have been reported in the synovial fluid of patients with BD, with the highest reported values being between 36200 and 39960/mm3, with a predominance of neutrophils [8, 17]. However, there are only two previously published cases of pseudoseptic arthritis in BD (Table 1). The first case, reported by Volpe et al. [18], was a 40-yearold HLA-B51(?) Algerian male with bilateral knee arthritis complicated with a popliteal cyst rupture in the left side. Examination of the synovial fluid revealed an elevated WBC count (83000/mm3) with marked neuthrophilia (95 %), and microbiological studies were negative. The patient was treated with colchicine, cyclosporin, and lowdose corticosteroids, and showed rapid improvement in his arthritis and systemic complaints in a few days. The second case, reported by Humby et al. [19], was a 31-year-old HLA-B51(?) Turkish-Cypriot male with nine-month history of left knee monoarthritis who underwent an arthroscopic synovial biopsy. Seventy-two hours after the procedure, he presented with fever, increased swelling of his knee, and a pathergy reaction at the site of joint aspiration. Arthrocentesis obtained 100 mL of purulent fluid. Microbiological studies of synovial fluid and biopsy as well as his blood were negative. He was treated with an IA corticosteroid injection and colchicine, resulting in complete resolution of the arthritis. The concomitant presentation of pseudoseptic arthritis and an adjacent pathergy skin lesion in this case may support the hypothesis of a similar local pathergy reaction in the synovium, triggered by trauma, as the causal mechanism [19]. Some typical manifestations of BD are characterized by neutrophilic inflammation. This is evident in acneiform lesions, Sweet-like lesions, pyoderma gangrenosum-like lesions, and pathergy reactions [35, 36]. Histopathologic examination of specimens from cutaneous lesions seen in BD cases reveals a neutrophilic vascular reaction or even fully developed leukocytoclastic vasculitis [1]. An abnormally high migration of neutrophils to the skin may be demonstrated in BD using Senn’s skin window technique [35], and a striking neutrophilic infiltration was also

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Table 1 Reported cases of pseudoseptic arthritis in Behc¸et’s disease


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reported in synovial biopsies [37]. Hyperactivation of neutrophils in BD is confirmed by the higher expression of the neutrophil activation markers CD11a, CD10, CD14 on the neutrophil cell surface, and increases in chemotaxis, phagocytosis, superoxide generation, and myeloperoxidase have been also reported. Some proposed mechanisms are the association of HLA B-51 with spontaneous neutrophil activation, high levels of Th1-derived proinflammatory cytokines such as IL8, INF-c, and TNF-a, and increased production of IL17 by Th17 cells [38]. Although BD has long been regarded as a Th1-type autoimmune disease due to the association with HLA-B51 and hyperreactivity against streptococcal antigen, it was recently found that BD shares several clinical features with autoinflammatory diseases. Increased activity of neutrophils and elevated levels of interleukin-1b (IL-1b) are observed in both BD and autoinflammatory diseases [39]. Furthermore, particular variants of the familial Mediterranean fever gene (MEFV) are more common in patients with BD, and colchicine treatment is effective in both diseases [38]. Pseudoseptic arthritis is a manifestation of another rare autoinflammatory disease, named PAPA syndrome (pyogenic sterile arthritis, pyoderma gangrenosum, and acne) [40]. This disease arises from mutations in the PSTPIP1/CD2BP1 gene on chromosome 15q. These mutations produce a hyperphosphorylated PSTPIP1 protein and alter its participation in the activation of the ‘‘inflammasome,’’ leading to IL-1b overproduction [41]. Distinguishing features of PAPA syndrome include early onset, florid and painful flares of recurrent sterile arthritis involving a prominent neutrophilic infiltrate, and autosomal dominant inheritance. Skin involvement is more variable and may present as ulcerations, frank pyoderma gangrenosum, or severe cystic acne. A pathergy reaction may also be present [40, 41]. It is interesting to note that, besides the similar skin manifestations in BD and PAPA syndrome, joint involvement may also coincide in some patients. Destructive arthritis (defined by radiological changes: erosions and/or geodes and/or global narrowing of the joint space and/or ankylosis) has been infrequently reported in BD [11]. Erosive changes are mainly observed in axial joints (sacroiliac), enthesis (calcaneal), and peripheral joints such as metatarsophalangeal and interphalangeal joints of the feet, intercarpal and metacarpophalangeal joints of the hand, knee, wrist and hip joints [12, 13]. In our case, no erosive changes were observed, but the patient developed a severe loss of cartilage and joint space narrowing, leading to mobility impairment. A similar case with nonerosive severe involvement of both elbows leading to ankylosis has been reported [11]. Synovial histopathology in BD-associated arthritis is not specific. Some studies have reported the loss of the

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superficial cell layer and the replacement of synovial lining cells by granulation tissue [42, 43], but another study reported deeper layer involvement, with infiltration by lymphocytes and neutrophils, marked vascularity, and occasional lymphoid follicle formation, whitout evidence of vasculitis or infection [14]. Histopathological studies were not performed in previously reported cases of pseudoseptic arthritis in BD; however, in the case reported by Humby et al. [19], a synovial biopsy prior to pseudoseptic arthritis development showed only nonspecific inflammatory synovitis. Histopathological findings in pseudoseptic arthritis due to other causes also demonstrated nonspecific chronic synovitis, with edematous synovium, congested vessels, fibrin deposits, infiltration by neutrophils, lymphocytes and plasma cells, and some necrotic cells [28, 33]. Colchicine, which is the drug of choice for treating the various manifestations of BD, has shown efficacy for the treatment of nonerosive arthritis [44]. Most patients respond to a colchicine dose of 1–2 mg/day [20, 21]. Colchicine may also be effective in some cases of destructive arthritis, and other useful alternatives are NSAIDs, intra-articular corticosteroid injections, and lowdose systemic corticosteroids [11, 17]. Immunosuppressive agents such as azathioprine and biological therapies such as interferon-a and TNF-a blockers may be tried in rare cases with resistant, longer-lasting, and disabling attacks [44]. TNF-a blocker treatment is recommended in cases of BD-associated arthritis that is recurrent/refractory, has not responded to two immunosuppressive agents (including methotrexate), and requires prednisolone at a dosage of [7.5 mg/day [45]; our case fulfilled these requirements. However, in rare cases, arthritis is the main indication for treatment, and most of the information regarding the therapeutic efficacy of TNF-a blockers derives from prospective studies on the use of infliximab for ocular involvement [46, 47]. Arida et al. [48] reviewed the efficacy and safety aspects of TNF-a blockers for the treatment of several manifestations of BD, including joint involvement, in 369 patients published to March 2010. They found joint involvement improved with infliximab in 94 % (50 of 53 treated patients improved), with etanercept in 100 % (6/6), and with adalimumab in 60 % (3/5). Complete remission was achieved in 41 % and a partial response in 50 %. In patients with arthritis who participated in prospective studies evaluating the effect of repetitive infliximab infusions, a sustained response was evident in 91 %. For these patients, infliximab monotherapy appears be equally as effective as combination therapy with methotrexate and/or cyclosporine-A [48]. Intra-articular administration of infliximab has also been reported to be effective in a patient with severe bilateral knee synovitis and orogenital aphthae [23]. The overall rates of adverse events


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due to TNF-a blocker administration (namely infections, malignancies, and congestive heart failure) in BD were no different from those reported in other diseases, and side effects related to infliximab infusion were mild and did not require the withdrawal of treatment [48].

Conclusion Despite the rarity of pseudoseptic arthritis in BD, this unusual form deserves to be known and considered among the rheumatologic manifestations of BD. Although BDassociated arthritis is typically nonerosive and nondeforming, destructive arthritis may occur in some patients, and this requires rapid identification and aggressive management to prevent the development of sequelae and disability. Finally, TNF-a blockers are a safe and effective alternative in patients with BD-associated arthritis refractory to other treatments. Conflict of interest


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Refractory pseudoseptic arthritis in Behçet's disease successfully treated with infliximab: a case report and literature review.

Arthritis associated with Behçet's disease is typically nonerosive and nondeforming, and most patients respond to colchicine treatment. However, destr...
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