Refractory invasive aspergillosis controlled with posaconazole and pulmonary surgery in a patient with chronic granulomatous disease: case report

Italian Journal of Pediatrics - Tập 40 - Trang 1-4 - 2014
Eda Kepenekli1, Ahmet Soysal1,2, Canan Kuzdan1, Nezih Onur Ermerak3, Mustafa Yüksel3, Mustafa Bakır1
1Division of Pediatric Infectious Diseases, Marmara University School of Medicine, Istanbul, Turkey
2Department of Pediatrics, Division of Pediatric Infectious Diseases, Marmara University Pendik Training and Research Hospital, Istanbul, Turkey
3Department of Chest Surgery, Marmara University School of Medicine, Istanbul, Turkey

Tóm tắt

Invasive aspergillosis is an important cause of morbidity and mortality in immunocompromised patients. Among primary immunodefiencies, chronic granulomatous disease (CGD) has the highest prevalence of invasive fungal diseases. Voriconazole is recommended for the primary treatment of invasive aspergillosis in most patients. In patients whose aspergillosis is refractory to voriconazole, therapeutic options include changing class of antifungal, for example using an amphotericin B formulation, an echinocandin, combination therapy, or further use of azoles. Posaconazole is a triazole derivative which is effective in Aspergillosis prophylaxis and treatment. Rarely, surgical therapy may be needed in some patients. Lesions those are contiguous with the great vessels or the pericardium, single cavitary lesion that cause hemoptysis, lesions invading the chest wall, aspergillosis that involves the skin and the bone are the indications for surgical therapy. Chronic granulomatous disease (CGD) is an inherited immundeficiency caused by defects in the phagocyte nicotinamide adenine dinucleotidephosphate (NADPH) oxidase complex which is mainstay of killing microorganisms. CGD is characterized by recurrent life-threatening bacterial and fungal infections and by abnormally exuberant inflammatory responses leading to granuloma formation, such as granulomatous enteritis, genitourinary obstruction, and wound dehiscence. The diagnosis is made by neutrophil function testing and the genotyping. Herein, we present a case with CGD who had invasive pulmonary aspergillosis refractory to voriconazole and liposomal amphotericine B combination therapy that was controlled with posaconazole treatment and pulmonary surgery.

Tài liệu tham khảo

Heyworth PG, Cross AR, Curnutte JT: Chronic granulomatous disease. Curr Opin Immunol. 2003, 15: 578-584. 10.1016/S0952-7915(03)00109-2. Seger RA: Modern management of chronic granulomatous disease. Br J Haematol. 2003, 140: 255-266. Segal BH, Leto TL, Gallin JI, Malech HL, Holland SM: Genetic, biochemical and clinical features of chronic granulomatous disease. Medicine. 2000, 79: 170-200. 10.1097/00005792-200005000-00004. Seger RA, Gungor T, Belohradsky BH: Treatment of chronic granulomatous disease with myeloablative conditioning and an unmodified hemopoietic allograft: a survey of the European experience, 1985–2000. Blood. 2002, 100: 4344-4350. 10.1182/blood-2002-02-0583. Horwitz ME, Barret AJ, Brown MR: Treatment of chronic granulomatous disease with myeloablative conditioning and t-cell depleted hematopoietic allograft. N Engl J Med. 2001, 344: 881-888. 10.1056/NEJM200103223441203. Martire B, Rondelli R, Soresina A: Clinical features, long-term follow-up and outcome of a large cohort of patients with chronic granulomatous disease: an Italian multicenter study. Clin Immunol. 2008, 126: 155-164. 10.1016/j.clim.2007.09.008. Blumental S, Mouy R, Mahlaoui N, Bougnoux ME, Debré M, Beauté J, Lortholary O, Blanche S, Fischer A: Invasive mold infections in chronic granulomatous disease: a 25-year retrospective survey. Clin Infect Dis. 2011, 53 (12): e159-e169. 10.1093/cid/cir731. Mouy R, Veber F, Blanche S: Long-term itraconazole prophylaxis against Aspergillus infections in thirty-two patients with chronic granulomatous disease. J Pediatr. 1994, 125: 998-1003. 10.1016/S0022-3476(05)82023-2. Gallin JI, Alling DW, Malech HL: Itraconazole to prevent fungal infections in chronic granulomatous disease. N Engl J Med. 2003, 348: 2416-2422. 10.1056/NEJMoa021931. Patterson TF, Kirkpatrick WR, White M: Invasive aspergillosis: disease spectrum, treatment practices, and outcomes. Medicine. 2000, 79: 250-260. 10.1097/00005792-200007000-00006. Marr KA, Patterson T, Denning D: Aspergillosis: pathogenesis, clinical manifestations, and therapy. Infect Dis Clin North Am. 2002, 16: 875-894. 10.1016/S0891-5520(02)00035-1. Falcone EL, Holland SM: Invasive fungal infection in chronic granulomatous disease: insights into pathogenesis and management. Curr Opin Infect Dis. 2012, 25 (6): 658-669. 10.1097/QCO.0b013e328358b0a4. Beaute J, Obenga G, Le Mignot L, Mahlaoui N, Bougnoux ME, Mouy R: Epidemiology and outcome of invasive fungal diseases in patients with chronic granulomatous disease. Pediatr Infect Dis J. 2011, 30 (1): 57-62. 10.1097/INF.0b013e3181f13b23. Walsh TJ, Anaissie EJ, Denning DW, Herbrecht R, Kontoyiannis DP, Marr KA: Treatment of aspergillosis: clinical practice guidelines of the infectious diseases society of America. Clin Infect Dis. 2008, 46: 327-360. 10.1086/525258. Walsh TJ, Raad I, Patterson TF, Chandrasekar P, Donowitz GR, Graybill R: Treatment of invasive aspergillozis with posaconazole in patients who are refractory to or intolerant of conventional therapy: an externally controlled trial. Clin Infect Dis. 2007, 44: 2-22. 10.1086/508774. Segal BH, Barnhart LA, Anderson VL, Walsh TJ, Malech HL, Holland SM: Posaconazole as salvage therapy in patients with chronic granulomatous disease and invasive filamentous fungal infection. Clin Infect Dis. 2005, 40: 1684-1688. 10.1086/430068.