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Eosinophilic granulomatosis with polyangiitis and IgA nephropaty in a patient with HIV infection
G Model
ARTICLE IN PRESS Med Clin (Barc). 2016;xxx(xx):xxx–xxx
www.elsevier.es/medicinaclinica
Letter to the Editor Eosinophilic granulomatosis with polyangiitis and IgA nephropaty in a patient with HIV infection夽 Granulomatosis eosinofílica con poliangitis y nefropatía IgA en paciente con infección por VIH Dear Sir, Eosinophilic granulomatosis with polyangiitis (Churg-Strauss Syndrome [CSS]) is a necrotising vasculitis of small and medium vessel positive ANCA, whose association, both with the infection due to human immunodeficiency virus (HIV), and with IgA nephropathy, is exceptional. Below is the case of a patient with HIV, CSS and mesangial glomerulonephritis with IgA deposits. A 37-year-old male with a history of bronchial asthma, chronic sinusitis and seronegative polyarthritis of seven years’ evolution, treated with methylprednisolone 4 mg/24 h methotrexate 10 mg/week until one year before being diagnosed with HIV. Since then, in antiretroviral treatment with good results. He visits because he had been experiencing pain in his calf for one month, gastrocnemius gait, widespread musculoskeletal pain, fatigue, anorexia and intensification of his usual rhinorrhea. For three days he had cyanosis of the fourth finger of the right hand, and purpuric lesions on his ankles, fingers, toes and gluteal area, some with necrotic sores. Upon physical examination, moderate to severe bronchospasm was also observed. Laboratory tests revealed leukocytosis (31.7 × 109 ) with eosinophilia of 58%, PCR 83.75 mg/l, neutrophil cytoplasmic antibodies with perinuclear pattern (p-ANCA) and positive antimyeloperoxidase (anti-MPO). Chest radiography and CT scan showed bilateral alveolar infiltrates consistent with diffuse alveolar haemorrhage. In the fibrobronchoscopy, blood remnants were observed, 36% eosinophils and the microbiological study were negative. To complete the study, a gastrocnemius biopsy showed necrotising vasculitis with thrombosis and eosinophilia. The diagnosis of eosinophilic granulomatosis with polyangiitis was concluded since it complied with ACR 19901 criteria and treatment was initiated with three pulses of methylprednisolone 500 mg intravenously (iv) and then 1 mg/kg/day of prednisone per day gradually falling off. In addition, alprostadil IV was administered as treatment for the ischaemia of the fourth finger. Four months after 0.9 g proteinuria was observed in 24 h urine without microhematuria, and his renal function was preserved, so a renal biopsy was performed which showed results of IgA nephropathy (mesangial glomerulonephritis with IgA deposits and an Oxford 2008 classification score of: M1 S1 E0 T0), treating with enalapril at 5 mg/day. After treatment, the patient experienced clinical and
夽 Please cite this article as: Morante I, Guelvenzu B, Marco H, Olivé A. Granulomatosis eosinofílica con poliangitis y nefropatía IgA en paciente con infección por VIH. Med Clin (Barc). 2016. http://dx.doi.org/10.1016/j.medcli.2016.01.025
laboratory improvement, and has remained in remission for four months. A variety of rheumatic manifestations in patients with HIV infection have previously been reported, such as diffuse infiltrative lymphocytosis syndrome or HIV-associated arthritis. Other disorders that appear frequently are vasculitis, reactive and psoriatic arthritis, polymyositis and infections of the locomotor system.2 The medical literature has reported two cases of patients with HIV and CSS.3,4 Cooper and Patterson3 reported the case of a patient who had a clinical visit, compatible with CSS and was simultaneously diagnosed with HIV. The second case appears in the literature published by Nguyen et al.4 The patient is also diagnosed with CSS and HIV in parallel, although the patient previously had had asthma and chronic sinusitis for several years. The latter suggest a possible link between the hypereosinophilic favoured by HIV infection and the development of CSS. The incidence of kidney disease in CSS is much lower (25%), and is usually less severe than in the rest of ANCA-associated vasculitis. The most typical renal involvement is in the form of focal and segmental pauci-immune necrotising glomerulonephritis.1 In the medical literature only two cases with IgA nephropathy and CSS have been reported.5,6 Richer et al.5 reported the case of a patient with asthma, nasal polyps and chronic sinusitis of 10 years’ evolution. After admission because of a sepsis episode the patient undergoes a deterioration of renal function and has multiple mononeuritis, diagnosing CSS and mesangial deposits of IgA. De Stefano et al.6 report the case of a patient who, after being diagnosed with CSS 16 years earlier, had impaired renal function and urine sediment, and by renal biopsy, mesangial proliferative glomerulonephritis with IgA deposits was diagnosed. It may have been a chance finding in both cases as this is the most common primary renal disease, but the existence of a link between ANCA vasculitis nephropathy cannot be completely ruled out. Although renal glomerular involvement because of HIV infection is very heterogeneous, IgA nephropathy has been identified in a large number of patients.7–9 In the study by Nebuloni et al.7 after 73 renal biopsies performed over 20 years in HIV patients, four of them had IgA nephropathy. Given the high frequency in the general population, it could be a chance finding rather than proving an aetiological connection. Until now a possible causal relationship between the above three diseases has not been identified, so we attribute its appearance in patients as random.
References 1. Greco A, Rizzo MI, de Virgilio A, Gallo A, Fusconi M, Ruoppolo G, et al. ChurgStrauss syndrome. Autoimmun Rev. 2015;14:341–8. 2. Reveille JD, Williams FM. Rheumatic complications of HIV infection. Best Pract Res Clin Rheumatol. 2006;20:1159–79. 3. Cooper LM, Patterson JA. Allergic granulomatosis and angiitis of Churg-Strauss. Case report in a patient with antibodies to human immunodeficiency virus and hepatitis B virus. Int J Dermatol. 1989;28:597–9.
˜ S.L.U. All rights reserved. 2387-0206/© 2016 Elsevier Espana,
MEDCLE-3531; No. of Pages 2
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ARTICLE IN PRESS Letter to the Editor / Med Clin (Barc). 2016;xxx(xx):xxx–xxx
4. Nguyen H, Ferentz K, Patel A, Le C. Churg-Strauss syndrome associated with HIV infection. J Am Board Fam Pract. 2005;18:140–2. 5. Richer C, Mouthon L, Cohen P, Royer I, Guettier C, Guillevin L. IgA glomerulonephritis associated with microscopic polyangiitis or Churg-Strauss syndrome. Clin Nephrol. 1999;52:47–50. 6. De Stefano R, Frati E, Brugnano R, Covarelli C, Pittavini L, Sidoni A. IgA glomerulonephritis associated with Churg-Strauss syndrome. Clin Exp Rheumatol. 2011;29:141–2. 7. Nebuloni M, Barbiano di Belgiojoso G, Genderini A, Tosoni A, Landriani N, Heidempergher M, et al. Glomerular lesions in HIV-positive patients: a 20-year biopsy experience from Northern Italy. Clin Nephrol. 2009;72:38–45. 8. Gutiérrez E, Morales E, Gutiérrez Martínez E, Manzanares MJ, Rosello G, Mérida ˜ E, et al. Glomerulopatías asociadas a infección por VIH: una perspectiva espanola. Nefrologia. 2007;27:439–47. 9. Kimmel PL. The nephropaties of VIH infection: pathogenesis and treatment. Curr Opin Nephrol Hypertens. 2000;9:117–22.
Isla Morante a,∗ , Berenice Guelvenzu b , Helena Marco b , Alejandro Olivé c a
Servicio de Reumatología, Hospital Universitario Central de Asturias, Oviedo, Asturias, Spain b Servicio de Nefrología, Hospital Universitario Germans Trias i Pujol, Badalona, Barcelona, Spain c Servicio de Reumatología, Hospital Universitario Germans Trias i Pujol, Badalona, Barcelona, Spain ∗ Corresponding author. E-mail address: [email protected] (I. Morante).
ARTICLE IN PRESS Med Clin (Barc). 2016;xxx(xx):xxx–xxx
www.elsevier.es/medicinaclinica
Letter to the Editor Eosinophilic granulomatosis with polyangiitis and IgA nephropaty in a patient with HIV infection夽 Granulomatosis eosinofílica con poliangitis y nefropatía IgA en paciente con infección por VIH Dear Sir, Eosinophilic granulomatosis with polyangiitis (Churg-Strauss Syndrome [CSS]) is a necrotising vasculitis of small and medium vessel positive ANCA, whose association, both with the infection due to human immunodeficiency virus (HIV), and with IgA nephropathy, is exceptional. Below is the case of a patient with HIV, CSS and mesangial glomerulonephritis with IgA deposits. A 37-year-old male with a history of bronchial asthma, chronic sinusitis and seronegative polyarthritis of seven years’ evolution, treated with methylprednisolone 4 mg/24 h methotrexate 10 mg/week until one year before being diagnosed with HIV. Since then, in antiretroviral treatment with good results. He visits because he had been experiencing pain in his calf for one month, gastrocnemius gait, widespread musculoskeletal pain, fatigue, anorexia and intensification of his usual rhinorrhea. For three days he had cyanosis of the fourth finger of the right hand, and purpuric lesions on his ankles, fingers, toes and gluteal area, some with necrotic sores. Upon physical examination, moderate to severe bronchospasm was also observed. Laboratory tests revealed leukocytosis (31.7 × 109 ) with eosinophilia of 58%, PCR 83.75 mg/l, neutrophil cytoplasmic antibodies with perinuclear pattern (p-ANCA) and positive antimyeloperoxidase (anti-MPO). Chest radiography and CT scan showed bilateral alveolar infiltrates consistent with diffuse alveolar haemorrhage. In the fibrobronchoscopy, blood remnants were observed, 36% eosinophils and the microbiological study were negative. To complete the study, a gastrocnemius biopsy showed necrotising vasculitis with thrombosis and eosinophilia. The diagnosis of eosinophilic granulomatosis with polyangiitis was concluded since it complied with ACR 19901 criteria and treatment was initiated with three pulses of methylprednisolone 500 mg intravenously (iv) and then 1 mg/kg/day of prednisone per day gradually falling off. In addition, alprostadil IV was administered as treatment for the ischaemia of the fourth finger. Four months after 0.9 g proteinuria was observed in 24 h urine without microhematuria, and his renal function was preserved, so a renal biopsy was performed which showed results of IgA nephropathy (mesangial glomerulonephritis with IgA deposits and an Oxford 2008 classification score of: M1 S1 E0 T0), treating with enalapril at 5 mg/day. After treatment, the patient experienced clinical and
夽 Please cite this article as: Morante I, Guelvenzu B, Marco H, Olivé A. Granulomatosis eosinofílica con poliangitis y nefropatía IgA en paciente con infección por VIH. Med Clin (Barc). 2016. http://dx.doi.org/10.1016/j.medcli.2016.01.025
laboratory improvement, and has remained in remission for four months. A variety of rheumatic manifestations in patients with HIV infection have previously been reported, such as diffuse infiltrative lymphocytosis syndrome or HIV-associated arthritis. Other disorders that appear frequently are vasculitis, reactive and psoriatic arthritis, polymyositis and infections of the locomotor system.2 The medical literature has reported two cases of patients with HIV and CSS.3,4 Cooper and Patterson3 reported the case of a patient who had a clinical visit, compatible with CSS and was simultaneously diagnosed with HIV. The second case appears in the literature published by Nguyen et al.4 The patient is also diagnosed with CSS and HIV in parallel, although the patient previously had had asthma and chronic sinusitis for several years. The latter suggest a possible link between the hypereosinophilic favoured by HIV infection and the development of CSS. The incidence of kidney disease in CSS is much lower (25%), and is usually less severe than in the rest of ANCA-associated vasculitis. The most typical renal involvement is in the form of focal and segmental pauci-immune necrotising glomerulonephritis.1 In the medical literature only two cases with IgA nephropathy and CSS have been reported.5,6 Richer et al.5 reported the case of a patient with asthma, nasal polyps and chronic sinusitis of 10 years’ evolution. After admission because of a sepsis episode the patient undergoes a deterioration of renal function and has multiple mononeuritis, diagnosing CSS and mesangial deposits of IgA. De Stefano et al.6 report the case of a patient who, after being diagnosed with CSS 16 years earlier, had impaired renal function and urine sediment, and by renal biopsy, mesangial proliferative glomerulonephritis with IgA deposits was diagnosed. It may have been a chance finding in both cases as this is the most common primary renal disease, but the existence of a link between ANCA vasculitis nephropathy cannot be completely ruled out. Although renal glomerular involvement because of HIV infection is very heterogeneous, IgA nephropathy has been identified in a large number of patients.7–9 In the study by Nebuloni et al.7 after 73 renal biopsies performed over 20 years in HIV patients, four of them had IgA nephropathy. Given the high frequency in the general population, it could be a chance finding rather than proving an aetiological connection. Until now a possible causal relationship between the above three diseases has not been identified, so we attribute its appearance in patients as random.
References 1. Greco A, Rizzo MI, de Virgilio A, Gallo A, Fusconi M, Ruoppolo G, et al. ChurgStrauss syndrome. Autoimmun Rev. 2015;14:341–8. 2. Reveille JD, Williams FM. Rheumatic complications of HIV infection. Best Pract Res Clin Rheumatol. 2006;20:1159–79. 3. Cooper LM, Patterson JA. Allergic granulomatosis and angiitis of Churg-Strauss. Case report in a patient with antibodies to human immunodeficiency virus and hepatitis B virus. Int J Dermatol. 1989;28:597–9.
˜ S.L.U. All rights reserved. 2387-0206/© 2016 Elsevier Espana,
MEDCLE-3531; No. of Pages 2
G Model 2
ARTICLE IN PRESS Letter to the Editor / Med Clin (Barc). 2016;xxx(xx):xxx–xxx
4. Nguyen H, Ferentz K, Patel A, Le C. Churg-Strauss syndrome associated with HIV infection. J Am Board Fam Pract. 2005;18:140–2. 5. Richer C, Mouthon L, Cohen P, Royer I, Guettier C, Guillevin L. IgA glomerulonephritis associated with microscopic polyangiitis or Churg-Strauss syndrome. Clin Nephrol. 1999;52:47–50. 6. De Stefano R, Frati E, Brugnano R, Covarelli C, Pittavini L, Sidoni A. IgA glomerulonephritis associated with Churg-Strauss syndrome. Clin Exp Rheumatol. 2011;29:141–2. 7. Nebuloni M, Barbiano di Belgiojoso G, Genderini A, Tosoni A, Landriani N, Heidempergher M, et al. Glomerular lesions in HIV-positive patients: a 20-year biopsy experience from Northern Italy. Clin Nephrol. 2009;72:38–45. 8. Gutiérrez E, Morales E, Gutiérrez Martínez E, Manzanares MJ, Rosello G, Mérida ˜ E, et al. Glomerulopatías asociadas a infección por VIH: una perspectiva espanola. Nefrologia. 2007;27:439–47. 9. Kimmel PL. The nephropaties of VIH infection: pathogenesis and treatment. Curr Opin Nephrol Hypertens. 2000;9:117–22.
Isla Morante a,∗ , Berenice Guelvenzu b , Helena Marco b , Alejandro Olivé c a
Servicio de Reumatología, Hospital Universitario Central de Asturias, Oviedo, Asturias, Spain b Servicio de Nefrología, Hospital Universitario Germans Trias i Pujol, Badalona, Barcelona, Spain c Servicio de Reumatología, Hospital Universitario Germans Trias i Pujol, Badalona, Barcelona, Spain ∗ Corresponding author. E-mail address: [email protected] (I. Morante).