- Email: [email protected]
Pulmonary nocardiosis in a patient with idiopathic thrombocytopenic purpura
Journal of Infection (2006) 52, e41–e43
www.elsevierhealth.com/journals/jinf
CASE REPORT
Pulmonary nocardiosis in a patient with idiopathic thrombocytopenic purpura Demosthenes D. Cokkinosa,*, Eleftheria Spanoub, Styliani Giannoua, Elissavet Protopapaa, Loukas Kyriakoub, Dimosthenis Mantzoukisb a
Radiology Department, Athens Hippocration Hospital, 114 Vas. Sofias Ave, 11527 Athens, Greece University of Athens, School of Medicine, 2nd Department of Internal Medicine, Athens, Greece
b
Accepted 16 May 2005 Available online 5 July 2005
KEYWORDS Pulmonary nocardiosis; Idiopathic thrombocytopenic purpura
Summary We present a case of pulmonary nocardiosis in a patient suffering from idiopathic thrombocytopenic purpura (ITP), an autoimmune disorder in which platelets are immunologically destroyed. ITP corticosteroid therapy, as well as the patient’s diabetes mellitus history caused immunosuppression, leading to an incidental lung infection by nocardia asteroides. The combination of pulmonary nocardiosis and ITP is, as far as we know, very rare. Q 2005 The British Infection Society. Published by Elsevier Ltd. All rights reserved.
Introduction
Case report
Idiopathic thrombocytopenic purpura (ITP) is an autoimmune disorder in which platelets (PLT) are immunologically destroyed, resulting in a bleeding tendency. Aiming at restoring PLT count, corticosteroid administration can cause immunosuppression. We present such a case, in which immunodeficiency was further accentuated by diabetes mellitus (DM), with a resulting incidental lung infection due to nocardia asteroides. Pulmonary nocardiosis and ITP combination in recent literature is, to our knowledge, very rare.1
A 52-year-old man with DM history presented with abdominal skin petechiae and cutaneous bruising (day 1). Physical examination revealed a temperature of 36.7 8C, a pulse rate of 63 beats/min, a respiratory rate of 15 breaths/min and a blood pressure of 120/75 mmHg. Blood tests revealed a WBC count of 10 000 cells/ml, a hemoglobin count of 14 g/dl, hematocrit of 45% and a low PLT count (15 000 cells/ml). Bone marrow aspiration established the diagnosis of ITP. Gamma immunoglobulin (30 g IV/day for 5 days) and methylprednisolone (80 mg p.o., three times daily) were administered. The patient was discharged 1 month later (day 31) with a WBC count of 9500 cells/ml, a hemoglobin count of 13.5 g/dl, hematocrit of 42%, a PLT count of 280 000/ml and instructions to continue therapy with 80 mg of methylprednisolone p.o. daily, progressively
* Corresponding author. Tel.: C30 210 643 8058. E-mail address: [email protected] (D.D. Cokkinos).
0163-4453/$30.00 Q 2005 The British Infection Society. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.jinf.2005.05.016
e42
D.D. Cokkinos et al. end-inspiratory rales in the left lung base. A chest X-ray revealed a left lower lung lobe consolidation adjacent to a cavity, confirmed on the next day by computed tomography (CT). He was readmitted for occult pulmonary infection assessment and treatment in a setting of immunodeficiency due to long standing corticosteroid therapy and DM. Multiple sputum and blood cultures, acid fast, Ziehl–Nielsen and Kinyoun stainings, as well as fungograms and antigen control were performed. Bronchoscopy gave brushing and washing specimens, although a biopsy was not possible. The entire panel of tests was negative. A daily sputum culture was also performed which revealed nocardia asteroides. The diagnosis was confirmed by percutaneous biopsy under CT control. Nocardia asteroides was found in the specimen. The patient’s infection was treated with a combination of imipenem/cilastatine (1 g IV three times daily) and trimethoprime/sulphamethoxasole (3 amp 80/400 mg IV four times daily). At the same time G-immunoglobulin boosts (6 g/day for 5 days) and high corticosteroid doses were administered to maintain normal PLT count. On day 105 his clinical condition deteriorated. A chest film showed increase of the consolidation extent and cavity size. A day later a new chest X-ray revealed a large left lung hydropneumothorax, confirmed by CT. An additional right upper lobe small nodule was also observed, consistent with nocardia infiltration of the right lung (Fig. 1). Two milliliters of fetid pus were drained. His blood gas levels deteriorated (PO2: 65 mmHg, PCO2: 30 mmHg, SatO2: 85%, HCOK 3 : 20 mequiv/l,) and he was transferred to the intensive care unit. Blood culture showed septic dissemination of nocardia into the blood. He was intubated, while the PLT count decreased to 10 000/ml. On day 136 he expired with the diagnosis of multiple organ failure and sepsis.
Figure 1 Day 106: Chest CT transverse sections at lung (a) and (b) and mediastinal (c) window: Cavitating lesion in posterior and lateral basal segments of the left lower lobe and extensive left hydropneumothorax are present. A nodular lesion is seen in the right upper lobe, consistent with nocardia infiltration on the right side.
tapering the dose to 48 mg p.o. daily under repeat examination. A month later (day 62), during tapering of corticosteroids, there was an apparent relapse (low PLT count: 30 000 and petechiae). The patient also complained of hemoptysis and was admitted in order to treat his thrombocytopenia, and was also considered for splenectomy. His temperature was 37.1 8C and at physical examination there were
Discussion Cases of pulmonary nocardiosis in a setting of ITP in recent literature are very few.1 Pulmonary nocardiosis is an uncommon opportunistic infection caused by soil-dwelling Gram-positive, branching beaded bacilli.2 Nocardia asteroides is the commonest species associated with human disease, usually affecting immunocompromised patients,2 (i.e. DM in our case) under corticosteroid therapy or with underlying pulmonary pathology3 (silicosis, fibrosis, COPD),2 Parenchymal disease ranges from a small nodule to bilateral infiltrates with cavitation (present in our patient), necrotizing
Pulmonary nocardiosis in a patient with ITP pneumonia eroding bony structures, pleural effusions and bronchopleural fistulae. Treatment includes sulfa-containing regimens, imipenem, ampicillin and minocycline.2 ITP, also known as primary immune or autoimmune thrombocytopenic purpura, is defined as isolated thrombocytopenia of unknown aetiology4 with a normal bone marrow picture. Antibodycoated or immune complex-coated PLTs are destroyed prematurely by the reticuloendothelial system5 mainly in the spleen6 by mononuclear macrophages. If this is not compensated for by increased production by megakaryocytes, thrombocytopenia occurs, resulting in bleeding tendency (purpura)6 as in our patient. Children usually experience a short benign clinical course, but ITP in adults can last more than 6 months (chronic ITP), resisting conventional treatment (corticosteroid, immune globulin intravenously or splenectomy). Aggressive treatment is required in those patients with severe bleeding and/or PLT count !10!109/l.5 The medical management aim is to increase the PLT count to a safe level, avoiding bacterial infection due to splenectomy or prolonged corticosteroid therapy toxicity.6
e43 In our case, the patient’s spleen was not resected. However, although the PLT count initially returned to normal, an incidental infection due to nocardia occurred, as the result of immunosuppression due to corticosteroid administration for ITP treatment combined with DM.
References 1. Ando T, Usa T, Ide A, Abe Y, Sera N, Tominaga T, et al. Pulmonary nocardiosis associated with idiopathic thrombocytopenic purpura. Intern Med 2001;40:246–9. 2. Lederman ER, Crum NF. A case series and focused review of nocardiosis. Clinical and microbiologic aspects. Medicine 2004;83:300–13. 3. Hui CH, Au VW, Rowland K, Slavotinek JP, Gordon DL. Pulmonary nocardiosis re-visited: Experience of 35 patients at diagnosis. Respir Med 2003;97:709–17. 4. Silverman MA. Idiopathic thrombocytopenic purpura 2004 [www.emedicine.com./topic282.htm]. 5. Stasi R, Provan D. Management of immune thrombocytopenic purpura in adults. Mayo Clin Proc 2004;79:504–22 [see also p. 456–7]. 6. Sandler SG. Review: Immune thrombocytopenic purpura: An update for immunohematologists. Immunohematology 2004; 20.
www.elsevierhealth.com/journals/jinf
CASE REPORT
Pulmonary nocardiosis in a patient with idiopathic thrombocytopenic purpura Demosthenes D. Cokkinosa,*, Eleftheria Spanoub, Styliani Giannoua, Elissavet Protopapaa, Loukas Kyriakoub, Dimosthenis Mantzoukisb a
Radiology Department, Athens Hippocration Hospital, 114 Vas. Sofias Ave, 11527 Athens, Greece University of Athens, School of Medicine, 2nd Department of Internal Medicine, Athens, Greece
b
Accepted 16 May 2005 Available online 5 July 2005
KEYWORDS Pulmonary nocardiosis; Idiopathic thrombocytopenic purpura
Summary We present a case of pulmonary nocardiosis in a patient suffering from idiopathic thrombocytopenic purpura (ITP), an autoimmune disorder in which platelets are immunologically destroyed. ITP corticosteroid therapy, as well as the patient’s diabetes mellitus history caused immunosuppression, leading to an incidental lung infection by nocardia asteroides. The combination of pulmonary nocardiosis and ITP is, as far as we know, very rare. Q 2005 The British Infection Society. Published by Elsevier Ltd. All rights reserved.
Introduction
Case report
Idiopathic thrombocytopenic purpura (ITP) is an autoimmune disorder in which platelets (PLT) are immunologically destroyed, resulting in a bleeding tendency. Aiming at restoring PLT count, corticosteroid administration can cause immunosuppression. We present such a case, in which immunodeficiency was further accentuated by diabetes mellitus (DM), with a resulting incidental lung infection due to nocardia asteroides. Pulmonary nocardiosis and ITP combination in recent literature is, to our knowledge, very rare.1
A 52-year-old man with DM history presented with abdominal skin petechiae and cutaneous bruising (day 1). Physical examination revealed a temperature of 36.7 8C, a pulse rate of 63 beats/min, a respiratory rate of 15 breaths/min and a blood pressure of 120/75 mmHg. Blood tests revealed a WBC count of 10 000 cells/ml, a hemoglobin count of 14 g/dl, hematocrit of 45% and a low PLT count (15 000 cells/ml). Bone marrow aspiration established the diagnosis of ITP. Gamma immunoglobulin (30 g IV/day for 5 days) and methylprednisolone (80 mg p.o., three times daily) were administered. The patient was discharged 1 month later (day 31) with a WBC count of 9500 cells/ml, a hemoglobin count of 13.5 g/dl, hematocrit of 42%, a PLT count of 280 000/ml and instructions to continue therapy with 80 mg of methylprednisolone p.o. daily, progressively
* Corresponding author. Tel.: C30 210 643 8058. E-mail address: [email protected] (D.D. Cokkinos).
0163-4453/$30.00 Q 2005 The British Infection Society. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.jinf.2005.05.016
e42
D.D. Cokkinos et al. end-inspiratory rales in the left lung base. A chest X-ray revealed a left lower lung lobe consolidation adjacent to a cavity, confirmed on the next day by computed tomography (CT). He was readmitted for occult pulmonary infection assessment and treatment in a setting of immunodeficiency due to long standing corticosteroid therapy and DM. Multiple sputum and blood cultures, acid fast, Ziehl–Nielsen and Kinyoun stainings, as well as fungograms and antigen control were performed. Bronchoscopy gave brushing and washing specimens, although a biopsy was not possible. The entire panel of tests was negative. A daily sputum culture was also performed which revealed nocardia asteroides. The diagnosis was confirmed by percutaneous biopsy under CT control. Nocardia asteroides was found in the specimen. The patient’s infection was treated with a combination of imipenem/cilastatine (1 g IV three times daily) and trimethoprime/sulphamethoxasole (3 amp 80/400 mg IV four times daily). At the same time G-immunoglobulin boosts (6 g/day for 5 days) and high corticosteroid doses were administered to maintain normal PLT count. On day 105 his clinical condition deteriorated. A chest film showed increase of the consolidation extent and cavity size. A day later a new chest X-ray revealed a large left lung hydropneumothorax, confirmed by CT. An additional right upper lobe small nodule was also observed, consistent with nocardia infiltration of the right lung (Fig. 1). Two milliliters of fetid pus were drained. His blood gas levels deteriorated (PO2: 65 mmHg, PCO2: 30 mmHg, SatO2: 85%, HCOK 3 : 20 mequiv/l,) and he was transferred to the intensive care unit. Blood culture showed septic dissemination of nocardia into the blood. He was intubated, while the PLT count decreased to 10 000/ml. On day 136 he expired with the diagnosis of multiple organ failure and sepsis.
Figure 1 Day 106: Chest CT transverse sections at lung (a) and (b) and mediastinal (c) window: Cavitating lesion in posterior and lateral basal segments of the left lower lobe and extensive left hydropneumothorax are present. A nodular lesion is seen in the right upper lobe, consistent with nocardia infiltration on the right side.
tapering the dose to 48 mg p.o. daily under repeat examination. A month later (day 62), during tapering of corticosteroids, there was an apparent relapse (low PLT count: 30 000 and petechiae). The patient also complained of hemoptysis and was admitted in order to treat his thrombocytopenia, and was also considered for splenectomy. His temperature was 37.1 8C and at physical examination there were
Discussion Cases of pulmonary nocardiosis in a setting of ITP in recent literature are very few.1 Pulmonary nocardiosis is an uncommon opportunistic infection caused by soil-dwelling Gram-positive, branching beaded bacilli.2 Nocardia asteroides is the commonest species associated with human disease, usually affecting immunocompromised patients,2 (i.e. DM in our case) under corticosteroid therapy or with underlying pulmonary pathology3 (silicosis, fibrosis, COPD),2 Parenchymal disease ranges from a small nodule to bilateral infiltrates with cavitation (present in our patient), necrotizing
Pulmonary nocardiosis in a patient with ITP pneumonia eroding bony structures, pleural effusions and bronchopleural fistulae. Treatment includes sulfa-containing regimens, imipenem, ampicillin and minocycline.2 ITP, also known as primary immune or autoimmune thrombocytopenic purpura, is defined as isolated thrombocytopenia of unknown aetiology4 with a normal bone marrow picture. Antibodycoated or immune complex-coated PLTs are destroyed prematurely by the reticuloendothelial system5 mainly in the spleen6 by mononuclear macrophages. If this is not compensated for by increased production by megakaryocytes, thrombocytopenia occurs, resulting in bleeding tendency (purpura)6 as in our patient. Children usually experience a short benign clinical course, but ITP in adults can last more than 6 months (chronic ITP), resisting conventional treatment (corticosteroid, immune globulin intravenously or splenectomy). Aggressive treatment is required in those patients with severe bleeding and/or PLT count !10!109/l.5 The medical management aim is to increase the PLT count to a safe level, avoiding bacterial infection due to splenectomy or prolonged corticosteroid therapy toxicity.6
e43 In our case, the patient’s spleen was not resected. However, although the PLT count initially returned to normal, an incidental infection due to nocardia occurred, as the result of immunosuppression due to corticosteroid administration for ITP treatment combined with DM.
References 1. Ando T, Usa T, Ide A, Abe Y, Sera N, Tominaga T, et al. Pulmonary nocardiosis associated with idiopathic thrombocytopenic purpura. Intern Med 2001;40:246–9. 2. Lederman ER, Crum NF. A case series and focused review of nocardiosis. Clinical and microbiologic aspects. Medicine 2004;83:300–13. 3. Hui CH, Au VW, Rowland K, Slavotinek JP, Gordon DL. Pulmonary nocardiosis re-visited: Experience of 35 patients at diagnosis. Respir Med 2003;97:709–17. 4. Silverman MA. Idiopathic thrombocytopenic purpura 2004 [www.emedicine.com./topic282.htm]. 5. Stasi R, Provan D. Management of immune thrombocytopenic purpura in adults. Mayo Clin Proc 2004;79:504–22 [see also p. 456–7]. 6. Sandler SG. Review: Immune thrombocytopenic purpura: An update for immunohematologists. Immunohematology 2004; 20.