Letters to the Editor / Joint Bone Spine (2015) 287–295
291
chronic inflammatory conditions, the development of amyloidosis is unpredictable and its emergence radically alters prognosis.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
References [1] Pongratz G, Ehrenstein B, Hartung W, et al. A patient with Pfeifer-WeberChristian disease successful therapy with cyclosporin A. Case report. BMC Musculoskelet Disord 2010;27:11–8. [2] Brissaud P, Grossin M. Panniculites systémiques. In: Kahn M.F., Kuntz D., Meyer O., Bardin T., Orcel P., editors. L’actualité rheumatologique Paris: Elsevier; 2000. p. 80–106. [3] Oliveira A, Rodriguez S, Jorge R, et al. Doenc¸a de weber christian paniculite sistémica de etiologia desconhecida. Acta Med Port 2010;23:1113–8. [4] Abramowsky CR. Case 1. Systemic amyloidosis in an infant with panniculitis and pancreatitis. Pediatr Pathol 1984;2:331–6. [5] Pallares R, Sancho S, Nogues R, et al. Amyloidosis (AA type) associated with nodular non suppurative panniculitis. Ann Intern Med 1983;99:488–9. [6] Asauliuk IK, Zagorodni˘ı SI, Karpliuk VI. Pfeifer-Weber-Christian disease with a 20-year course. Lik Sprava 1998;7:154–9. [7] Cuchard P, Cuchard R, Rotman S, et al. Renal amiloidosis. Rev Med Suisse 2012;29:446–51. [8] Wouters C, Martin T, Stichweh D, et al. Infantile onset panniculitis with uveitis and systemic granulomatosis: a new clinicopathologic entity. J Pediatr 2007;151:707–9. [9] Kuroda T, Wada Y, Kobayashi D, et al. Effective anti-TNF-alpha therapy can induce rapid resolution and sustained decrease of gastroduodenal mucosal amyloid deposits in reactive amyloidosis associated with rheumatoid arthritis. J Rheumatol 2009;36:2409–15.
Anne Riveros Frutos ∗ Melania Martínez-Morillo Lourdes Mateo Soria Rheumatology Department, Hospital Universitaris Germans Trias i Pujol, Carretera del Canyet s/n, 08916 Badalona, Spain ∗ Corresponding
author. Tel.: +34 934 651 200 (ext. 8481); fax: +34 934 978 843. E-mail address: [email protected] (A. Riveros Frutos) Accepted 17 November 2014 Available online 24 December 2014
http://dx.doi.org/10.1016/j.jbspin.2014.11.001
Giant cell arteritis occurring during psoriatic arthritis treated by adalimumab
a r t i c l e
i n f o
Keywords: Psoriatic arthritis Giant cell arteritis Positron emission tomography Anti-TNF Vasculitis
We present an observation from one of our patients followed for psoriatic arthritis (PsA) who developed a giant cell arteritis (GCA) under anti-TNF␣ with no specific clinical sign, with the major help of PET-scan.
Fig. 1. FDG uptake all along both subclavian arteries.
Our patient is a 62-year-old male, followed for PsA and ulcerative colitis, treated by adalimumab. After six years, although both diseases were considered in remission, the patient reported hyperthermia up to 38.3 ◦ C, weight loss, and night sweats. The last blood tests showed mild inflammation with a CRP level of 22 mg/L and ESR of 23 mm; no relevant clinical sign of GCA, thromboembolic disease or infection. CT-scan from neck to pelvis found bilateral cervical centimetric adenopathies, but no suggestive sign of tumor or malignant hemopathy. We decided to perform a PET-scan (Fig. 1), which found fluorodesoxyglucose (FDG) uptake all along both subclavian arteries to their humeral portions, compatible with large vessel vasculitis. Temporal artery biopsy confirmed GCA, showing focal granulomatous inflammation with multinucleated giant cells. The patient was treated with glucocorticoids and aspirin. Evolution through 12 months’ follow-up shows a complete regression of the inflammatory syndrome and glucocorticoids are being gradually decreased. A literature review found very few case of PsA developing GCA. Clementz et al. [1] first described in 1989 a 63-year-old woman with PsA who developed GCA associated with pericarditis and pancreatic insufficiency. In a retrospective study based on health claims database, Makredes et al. [2] found an increased prevalence ratio associated with GCA of 4.8 (IC 1.5–15.7). Concerning spondyloarthritis, Verhoeven et al. [3] described a case of aortitis occurring during etanercept therapy for ankylosing spondylitis in a 60-year-old woman. The role of anti-TNF␣ in GCA is controversial: inducing role has been suspected, but we found only two cases of GCA during antiTNF␣ treatment [4,5].
292
Letters to the Editor / Joint Bone Spine (2015) 287–295
Anti-TNF␣ are commonly known to induce leukocytoclastic vasculitis [6], but it seems to be more often observed in rheumatoid arthritis than in PsA. It is still hard to define if the cause of developing GCA is the rheumatism itself or the treatment. In these cases, imputability cannot be proved and the association seems to be more likely coincidental. Nevertheless, anti-TNF␣ and other biological agents are studied as an alternative treatment for GCA. The HECTHOR Study using adalimumab in recently diagnosed GCA showed no difference for remission compared to steroids alone [7]. Finally, the diagnosis of GCA was permitted by the intake of PET-scan, showing arterial FDG uptake. 18-FDG PET-scan appears to have great sensitivity and sensibility in diagnosis of GCA [8]. It appears to be helpful in early diagnosis and evaluation of the activity and extension of the disease. But its usefulness in evaluating treatment response remains unclear. In conclusion, association between PsA, GCA and anti-TNF␣ seems to be rare and probably coincidental.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
References [1] Clementz GL, Gold F, Khaiser N, et al. Giant cell arteritis associated with pericarditis and pancreatic insufficiency in a patient with psoriatic arthritis. J Rheumatol 1989;16:128–9. [2] Makredes M, Robinson D, Bala M, et al. The burden of autoimmune disease: a comparison of prevalence ratios in patients with psoriatic arthritis and psoriasis. J Am Acad Dermatol 2009;61:405–10. [3] Verhoeven F, Bossert M, Lohse-Walliser A, et al. Aortitis during etanercept therapy for ankylosing spondylitis: finding the culprit. Joint Bone Spine 2012;79:524–6. [4] Seton M. Giant cell arteritis in a patient taking etanercept and methotrexate. J Rheumatol 2004;31:1467. [5] Leydet-Quilici H, Luc M, Armingeat T, et al. Giant cell arteritis during adalimumab treatment for rheumatoid arthritis. Joint Bone Spine 2007;74:303–4. [6] Guillevin L, Mouthon L. Tumor necrosis factor-a blockade and the risk of vasculitis. J Rheumatol 2004;31:1885–7. [7] Seror R, Baron G, Hachulla E, et al. Adalimumab for steroid sparing in patients with giant-cell arteritis: results of a multicentre randomized controlled trial. Ann Rheum Dis 2014;73:2074–81. [8] Besson FL, Parienti J-J, Bienvenu B, et al. Diagnostic performance of 18Ffluorodeoxyglucose positron emission tomography in giant cell arteritis: a systematic review and meta-analysis. Eur J Nucl Med Mol Imaging 2011;38: 1764–72.
Justine Corli a Lucie Lemeunier a,∗ Noémie Le Gouellec a,b René-Marc Flipo a a Rhumatologie, Hôpital Roger Salengro, Université de Lille 2, Centre Hospitalier Régional Universitaire de Lille, Lille cedex, France b Néphrologie, hôpital Roger Salengro, université de Lille 2, centre hospitalier régional universitaire de Lille, Lille cedex, France ∗ Corresponding
author. Tel.: +33320446120; fax: +330320445962. E-mail address: [email protected] (L. Lemeunier) Accepted 24 November 2014 Available online 14 February 2015
http://dx.doi.org/10.1016/j.jbspin.2014.11.003
Patients with dual MPO- and PR3-ANCA do not present primary systemic necrotizing vasculitis夽
a r t i c l e
i n f o
Keywords: ANCA Anti-proteinase 3 antibody Anti-myeloperoxydase antibody ANCA associated vasculitis
1. Introduction Antineutrophil cytoplasmic antibodies (ANCA) are good serological markers for primary systemic necrotizing vasculitis (PSNV) [1]. They are detected by indirect immunofluorescence (IIF) microscopy assays on human fixed neutrophils with three different fluorescence patterns: cytoplasmic (cANCA), perinuclear (pANCA), and atypical (aANCA). ANCA specificity is determined by solid-phase assays (ELISA, immunodot and addressable laser bead immunoassay [ALBIA or Luminex® assay]) with two relevant clinical targets (proteinase 3 [PR3] or myeloperoxidase [MPO]); some minor antigens are reported [2], but their detection is not used usually [3]. PR3- and MPO-ANCA with high titres are good serological markers for PSNV: c/PR3-ANCA for granulomatosis with polyangiitis (GPA) [4] (formerly known as Wegener’s granulomatosis) [5], and p/MPO-ANCA for microscopic polyangiitis (MPA) [6]. Usually ANCA are monospecific, but some multispecific ANCA are described, of which some specific for both PR3 and MPO. This study focused on such patients to estimate their frequency among the population of ANCA-positive patients and to evaluate their clinical association. 2. Study population This French study was retrospective and multicenter. The cohort was built using the databases from the Centre Hospitalo-Universitaire (CHU) of Angers and of the CHU of Dijon. The screening period went from May 2004 to May 2012. Each patient presented with a dual MPO-ANCA and PR3-ANCA profile, with at least one of the solid-phase analysis techniques described in Table 1, was enrolled. 3. Results and discussion Over 8 years, we found 28 IIF-ANCA-positive patients with dual PR3 and MPO-ANCA by at least one of the solid-phase test. For the 23 patients with available files, there is no prevalence of sex (13 females, 10 males) and the mean age is 53 years (ranging from 6 to 86 years). These 23 documented patients with both PR3 and MPO-ANCA account for 0.7% of sera and 0.9% of ANCA-positive patients. As shown in Table 1, none of them had a PSNV, neither GPA nor MPA nor EGPA (eosinophilic granulomatosis with polyangiitis). Nevertheless, two cases of secondary vasculitis have been observed among these patients, consequently to an antithyroid agent treatment as previously described [7].
夽 This work was presented as a poster at the 16th International Vasculitis and ANCA Workshop, Paris, April 14th–17th 2013 – Presse Med 2013;42(4, cahier 2):P11: 685–6.
291
chronic inflammatory conditions, the development of amyloidosis is unpredictable and its emergence radically alters prognosis.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
References [1] Pongratz G, Ehrenstein B, Hartung W, et al. A patient with Pfeifer-WeberChristian disease successful therapy with cyclosporin A. Case report. BMC Musculoskelet Disord 2010;27:11–8. [2] Brissaud P, Grossin M. Panniculites systémiques. In: Kahn M.F., Kuntz D., Meyer O., Bardin T., Orcel P., editors. L’actualité rheumatologique Paris: Elsevier; 2000. p. 80–106. [3] Oliveira A, Rodriguez S, Jorge R, et al. Doenc¸a de weber christian paniculite sistémica de etiologia desconhecida. Acta Med Port 2010;23:1113–8. [4] Abramowsky CR. Case 1. Systemic amyloidosis in an infant with panniculitis and pancreatitis. Pediatr Pathol 1984;2:331–6. [5] Pallares R, Sancho S, Nogues R, et al. Amyloidosis (AA type) associated with nodular non suppurative panniculitis. Ann Intern Med 1983;99:488–9. [6] Asauliuk IK, Zagorodni˘ı SI, Karpliuk VI. Pfeifer-Weber-Christian disease with a 20-year course. Lik Sprava 1998;7:154–9. [7] Cuchard P, Cuchard R, Rotman S, et al. Renal amiloidosis. Rev Med Suisse 2012;29:446–51. [8] Wouters C, Martin T, Stichweh D, et al. Infantile onset panniculitis with uveitis and systemic granulomatosis: a new clinicopathologic entity. J Pediatr 2007;151:707–9. [9] Kuroda T, Wada Y, Kobayashi D, et al. Effective anti-TNF-alpha therapy can induce rapid resolution and sustained decrease of gastroduodenal mucosal amyloid deposits in reactive amyloidosis associated with rheumatoid arthritis. J Rheumatol 2009;36:2409–15.
Anne Riveros Frutos ∗ Melania Martínez-Morillo Lourdes Mateo Soria Rheumatology Department, Hospital Universitaris Germans Trias i Pujol, Carretera del Canyet s/n, 08916 Badalona, Spain ∗ Corresponding
author. Tel.: +34 934 651 200 (ext. 8481); fax: +34 934 978 843. E-mail address: [email protected] (A. Riveros Frutos) Accepted 17 November 2014 Available online 24 December 2014
http://dx.doi.org/10.1016/j.jbspin.2014.11.001
Giant cell arteritis occurring during psoriatic arthritis treated by adalimumab
a r t i c l e
i n f o
Keywords: Psoriatic arthritis Giant cell arteritis Positron emission tomography Anti-TNF Vasculitis
We present an observation from one of our patients followed for psoriatic arthritis (PsA) who developed a giant cell arteritis (GCA) under anti-TNF␣ with no specific clinical sign, with the major help of PET-scan.
Fig. 1. FDG uptake all along both subclavian arteries.
Our patient is a 62-year-old male, followed for PsA and ulcerative colitis, treated by adalimumab. After six years, although both diseases were considered in remission, the patient reported hyperthermia up to 38.3 ◦ C, weight loss, and night sweats. The last blood tests showed mild inflammation with a CRP level of 22 mg/L and ESR of 23 mm; no relevant clinical sign of GCA, thromboembolic disease or infection. CT-scan from neck to pelvis found bilateral cervical centimetric adenopathies, but no suggestive sign of tumor or malignant hemopathy. We decided to perform a PET-scan (Fig. 1), which found fluorodesoxyglucose (FDG) uptake all along both subclavian arteries to their humeral portions, compatible with large vessel vasculitis. Temporal artery biopsy confirmed GCA, showing focal granulomatous inflammation with multinucleated giant cells. The patient was treated with glucocorticoids and aspirin. Evolution through 12 months’ follow-up shows a complete regression of the inflammatory syndrome and glucocorticoids are being gradually decreased. A literature review found very few case of PsA developing GCA. Clementz et al. [1] first described in 1989 a 63-year-old woman with PsA who developed GCA associated with pericarditis and pancreatic insufficiency. In a retrospective study based on health claims database, Makredes et al. [2] found an increased prevalence ratio associated with GCA of 4.8 (IC 1.5–15.7). Concerning spondyloarthritis, Verhoeven et al. [3] described a case of aortitis occurring during etanercept therapy for ankylosing spondylitis in a 60-year-old woman. The role of anti-TNF␣ in GCA is controversial: inducing role has been suspected, but we found only two cases of GCA during antiTNF␣ treatment [4,5].
292
Letters to the Editor / Joint Bone Spine (2015) 287–295
Anti-TNF␣ are commonly known to induce leukocytoclastic vasculitis [6], but it seems to be more often observed in rheumatoid arthritis than in PsA. It is still hard to define if the cause of developing GCA is the rheumatism itself or the treatment. In these cases, imputability cannot be proved and the association seems to be more likely coincidental. Nevertheless, anti-TNF␣ and other biological agents are studied as an alternative treatment for GCA. The HECTHOR Study using adalimumab in recently diagnosed GCA showed no difference for remission compared to steroids alone [7]. Finally, the diagnosis of GCA was permitted by the intake of PET-scan, showing arterial FDG uptake. 18-FDG PET-scan appears to have great sensitivity and sensibility in diagnosis of GCA [8]. It appears to be helpful in early diagnosis and evaluation of the activity and extension of the disease. But its usefulness in evaluating treatment response remains unclear. In conclusion, association between PsA, GCA and anti-TNF␣ seems to be rare and probably coincidental.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
References [1] Clementz GL, Gold F, Khaiser N, et al. Giant cell arteritis associated with pericarditis and pancreatic insufficiency in a patient with psoriatic arthritis. J Rheumatol 1989;16:128–9. [2] Makredes M, Robinson D, Bala M, et al. The burden of autoimmune disease: a comparison of prevalence ratios in patients with psoriatic arthritis and psoriasis. J Am Acad Dermatol 2009;61:405–10. [3] Verhoeven F, Bossert M, Lohse-Walliser A, et al. Aortitis during etanercept therapy for ankylosing spondylitis: finding the culprit. Joint Bone Spine 2012;79:524–6. [4] Seton M. Giant cell arteritis in a patient taking etanercept and methotrexate. J Rheumatol 2004;31:1467. [5] Leydet-Quilici H, Luc M, Armingeat T, et al. Giant cell arteritis during adalimumab treatment for rheumatoid arthritis. Joint Bone Spine 2007;74:303–4. [6] Guillevin L, Mouthon L. Tumor necrosis factor-a blockade and the risk of vasculitis. J Rheumatol 2004;31:1885–7. [7] Seror R, Baron G, Hachulla E, et al. Adalimumab for steroid sparing in patients with giant-cell arteritis: results of a multicentre randomized controlled trial. Ann Rheum Dis 2014;73:2074–81. [8] Besson FL, Parienti J-J, Bienvenu B, et al. Diagnostic performance of 18Ffluorodeoxyglucose positron emission tomography in giant cell arteritis: a systematic review and meta-analysis. Eur J Nucl Med Mol Imaging 2011;38: 1764–72.
Justine Corli a Lucie Lemeunier a,∗ Noémie Le Gouellec a,b René-Marc Flipo a a Rhumatologie, Hôpital Roger Salengro, Université de Lille 2, Centre Hospitalier Régional Universitaire de Lille, Lille cedex, France b Néphrologie, hôpital Roger Salengro, université de Lille 2, centre hospitalier régional universitaire de Lille, Lille cedex, France ∗ Corresponding
author. Tel.: +33320446120; fax: +330320445962. E-mail address: [email protected] (L. Lemeunier) Accepted 24 November 2014 Available online 14 February 2015
http://dx.doi.org/10.1016/j.jbspin.2014.11.003
Patients with dual MPO- and PR3-ANCA do not present primary systemic necrotizing vasculitis夽
a r t i c l e
i n f o
Keywords: ANCA Anti-proteinase 3 antibody Anti-myeloperoxydase antibody ANCA associated vasculitis
1. Introduction Antineutrophil cytoplasmic antibodies (ANCA) are good serological markers for primary systemic necrotizing vasculitis (PSNV) [1]. They are detected by indirect immunofluorescence (IIF) microscopy assays on human fixed neutrophils with three different fluorescence patterns: cytoplasmic (cANCA), perinuclear (pANCA), and atypical (aANCA). ANCA specificity is determined by solid-phase assays (ELISA, immunodot and addressable laser bead immunoassay [ALBIA or Luminex® assay]) with two relevant clinical targets (proteinase 3 [PR3] or myeloperoxidase [MPO]); some minor antigens are reported [2], but their detection is not used usually [3]. PR3- and MPO-ANCA with high titres are good serological markers for PSNV: c/PR3-ANCA for granulomatosis with polyangiitis (GPA) [4] (formerly known as Wegener’s granulomatosis) [5], and p/MPO-ANCA for microscopic polyangiitis (MPA) [6]. Usually ANCA are monospecific, but some multispecific ANCA are described, of which some specific for both PR3 and MPO. This study focused on such patients to estimate their frequency among the population of ANCA-positive patients and to evaluate their clinical association. 2. Study population This French study was retrospective and multicenter. The cohort was built using the databases from the Centre Hospitalo-Universitaire (CHU) of Angers and of the CHU of Dijon. The screening period went from May 2004 to May 2012. Each patient presented with a dual MPO-ANCA and PR3-ANCA profile, with at least one of the solid-phase analysis techniques described in Table 1, was enrolled. 3. Results and discussion Over 8 years, we found 28 IIF-ANCA-positive patients with dual PR3 and MPO-ANCA by at least one of the solid-phase test. For the 23 patients with available files, there is no prevalence of sex (13 females, 10 males) and the mean age is 53 years (ranging from 6 to 86 years). These 23 documented patients with both PR3 and MPO-ANCA account for 0.7% of sera and 0.9% of ANCA-positive patients. As shown in Table 1, none of them had a PSNV, neither GPA nor MPA nor EGPA (eosinophilic granulomatosis with polyangiitis). Nevertheless, two cases of secondary vasculitis have been observed among these patients, consequently to an antithyroid agent treatment as previously described [7].
夽 This work was presented as a poster at the 16th International Vasculitis and ANCA Workshop, Paris, April 14th–17th 2013 – Presse Med 2013;42(4, cahier 2):P11: 685–6.
