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Nocardial epidural abscess of the thoracic spinal cord and review of the literature
J Infect Chemother (2005) 11:169–171 DOI 10.1007/s10156-005-0384-0
© Japanese Society of Chemotherapy and The Japanese Association for Infectious Diseases 2005
CASE REPORT Basar Atalay · Ozlem Azap · Melih Cekinmez Hakan Caner · Mehmet Haberal
Nocardial epidural abscess of the thoracic spinal cord and review of the literature
Received: November 25, 2004 / Accepted: April 20, 2005
Abstract Nocardia is a potential cause of opportunistic infection in organ-transplant recipients. We report an epidural abscess causing acute paraparesis in a patient with a rejected renal transplant and chronic liver disease. Early drainage and treatment with imipenem and trimethoprimsulfamethoxasole improved the patient’s neurologic status. Key words Epidural abscess · Nocardia · Renal transplant · Thoracic spine
Introduction Nocardiosis is a rare opportunistic infection caused by microorganisms of the genus Nocardia. It is associated with significant morbidity and mortality in immunecompromised patients.1 Although the main syndromes caused by the Nocardia species are respiratory tract disease (73%–77%), hematogenous dissemination occurs in about 50% of patients. Primary infection of the central nervous system (CNS) without pulmonary involvement occurs in only 5%–7% of patients, and this invasion may lead to severe neurologic sequelae.1–3 We report an unusual case of a patient with rejected renal transplant and liver failure in whom a gross paravertebral abscess invaded the thoracic spinal epidural space.
B. Atalay · M. Cekinmez · H. Caner Baskent University Department of Neurosurgery, Ankara, Turkey O. Azap Baskent University Department of Infections Disease, Ankara, Turkey M. Haberal Bakent University Department of General Surgery, Ankara, Turkey B. Atalay (*) 16. sokak no 24/4, 06500 Bahcelievler, Ankara, Turkey, Tel. +0090-535-786-99-59; Fax +0090 312 223 73 33 e-mail: [email protected]
Case report A 51-year-old man was admitted to the hospital with acute, increasing neck pain, radiating to both shoulders, and weakness in the lower extremities of 3 days’ duration. He had chronic renal failure and had been undergoing hemodialysis for 5 years after rejection of a renal transplant. He had chronic liver failure due to hepatitis C infection. He also had chronic obstructive pulmonary disease and steroid-induced diabetes mellitus. He was unable to ambulate and was paraparetic and hypoesthetic below the level of the third thoracic vertebrae. Results of all tendon reflex tests were negative. Routine laboratory studies revealed moderate leukocytosis (leucocyte count, 10.6 ¥ 103/ml) and slight anemia (10.6 g/dl) with an increased erythrocyte sedimentation rate (ESR), of 120 mm/h. Liver function test results were abnormal (alkaline phosphatase, 444 U/l; total bilirubin, 3.6 mg/dl; gamma-glutamyl transpeptidase (GGT), 291 U/l; aspartate aminotransferase (AST), 66 U/l; alanine aminotransferase (ALT), 57 U/l). The patient was evaluated using emergent cervical and thoracic magnetic resonance imaging (MRI), which revealed a paravertebral abscess in the C2-T8 muscles that extended to the epidural space over the third thoracic vertebra, compressing the spinal cord (Fig. 1). Computed tomography scans of the abdominal and thoracic regions and abdominal ultrasonography revealed no other abscess. The source of infection could not be identified. As the patient’s liver enzymes were very high, it was decided not to administer general anesthesia and to immediately drain the abscess percutaneously. A percutaneous catheter was introduced to the abscess with the aid of ultrasonography, and 2 l of pus of exudative quality was aspirated. The abscess was continuously drained for 1 week and the patient’s neurologic score subsequently improved. MRI after 1 week revealed subtotal remission of the abscess (Fig. 2). Empiric systemic antibiotics, including intra-venous (IV) imipenem, 250 mg q.i.d.; IV trimethoprim/sulfamethoxasole, 160/ 800 mg b.i.d.; and IV fluconazole, 200 mg/day were started immediately after drainage. Culture of the pus from the epidural abscess revealed an uncommon microorganism,
170
a Fig. 1. a T2-Weighted axial magnetic resonance (MR) image of the third thoracic vertebra, demonstrating a subcutaneous abscess that invaded the epidural space and compressed the spinal cord. b T2-
b weighted sagittal MR image, demonstrating compression over the spinal cord at the level of the third thoracic vertebra
a
b
Fig. 2. a Control MR image 5 days after drainage of the abscess. T2weighted axial MR image of the third thoracic vertebra, demonstrating subtotal remission of the abscess. b T2-weighted sagittal MR image,
demonstrating minimal compression over the spinal cord at the level of the third thoracic vertebra
termed Nocardia asteroides. Fluconazole was stopped; IV imipenem 250 mg q.i.d. was continued for another 6 months in our rehabilitation center’s outpatient clinic. He also continued his rehabilitation program at this center. Trimethoprim/sulfamethoxasole 160/800 mg b.i.d. IV was continued, together with IV imipenem for the first 3 months, and then oral trimethoprim/sulfamethoxasole 160/800 mg b.i.d was continued for the next 3 months. The laboratory values dropped to normal on the day 12 of therapy (leukocyte count, 6.3 ¥ 103 ml; ESR, 31 mm/h; total bilirubin, 1.1 mg/ dl; AST, 31 U/l; ALT, 24 U/l). These values were within the
normal ranges untill the end of therapy. The patient’s general status and neurologic deficit improved, and he was completely ambulatory and pain-free at 9 months’ follow-up.
Discussion Nocardiosis usually presents as an opportunistic infection in immunocompromised individuals. Nocardia spp. are gram-
171
positive, rod-shaped bacteria. They are strict aerobes found in the environment but very rarely cause infection in humans. Patients with AIDS, tuberculosis, lymphoma, those taking immunosuppressant drugs, and those with chronic alcoholism are prone to this opportunistic infection.4–6 Nocardia spp. usually cause infection after inhalation or direct inoculation.6 Hiller and colleagues5 reported a 4% incidence of Nocardia infections in posttransplant patients. Although hematogenous dissemination occurs commonly, blood cultures almost invariably fail to demonstrate the organism,3 and this mandates sampling and examining the material from infectious foci seen on radiologic studies. CNS infection caused by nocardiosis is usually supratentorial, and pathologically it consists of a multiloculated thin-walled abscess(es). Rarely, it involves the cerebellum, spinal cord, or meninges.4,7,8 Nocardial spinal cord abscesses are extremely rare. To our knowledge, only a few cases have been reported in the literature. These patients also had extraneurologic nocardiosis and the abcess(es) were located in the spinal epidural space.6,9,10 Harvey et al.6 reported an epidural abcess at L4 level and treated the abscess with triple agents, consisting of IV amikacin for 8 weeks, IV ceftriaxone for 6 months, and oral trimethoprimsulfamethoxasole for 1 year. Graat et al.10 reported an epidural abcess at T11-L3 level and isolated Nocardia farcinica. They treated this infection with ciprofloxacin (1 g/ day), sulfadiazine (6 g/day), and fluconazole (20 mg/day), for 2 years. Misra et al.9 reported an epidural abcess at T26, but the patient was complicated with Mycobacterium tuberculosis; the Nocardia species was Nocardia brazilensis. Mukunda and colleagues11 have presented a case of an intramedullary spinal abscess caused by Nocardia asteroides without concomitant extraneurologic nocardiosis. The CNS is the most common site of the disseminated disease and disease at this site is usually associated with high morbidity and mortality. In one series, the mortality was 24% in operated patients and 30% in nonoperated patients. In that series, 22% of the cases were diagnosed at autopsy.7 At Baskent University, nocardiosis has been diagnosed in only two unique cases, including our patient. The other case was a disseminated Nocardia asteroides with a brain abscess in a renal transplant recipient. Although an optimal regimen and duration for the treatment of Nocardial infections has not yet been definitively established, Azap et al.12 treated disseminated Nocardia asteroides infection in a renal transplant recipient for 1 year with IV meropenem, together with IV trimethoprimsulfamethoxasole for the first 5 months. In our experience we treated our patient with a spinal epidural abscess with 6 months of IV meropenem together with 3 months of IV and 3 months of oral trimethoprim-sulfamethoxasole. The differential diagnosis of spinal epidural abscess includes infections with Staphylococcus aureus, Actinomycosis, infections with Mycobacterium tuberculosis, fungal infections, and nonspecific osteomyelitis.6,13 Hiller and coworkers5 reported an unusual case of leg paralysis that was caused by Nocardia species in the postoperative course of a renal transplant patient. Our case is unique because the thoracic epidural cord compression occurred by direct inva-
sion from a subcutaneous Nocardial abscess. Simple percutaneous drainage and appropriate antibiotic therapy were effective in our patient.
Conclusion Although life expectancy for patients with chronic diseases is increasing with advances in medicine, incidences of opportunistic infections such as tuberculosis and AIDS are also increasing. Early diagnosis and appropriate antibiotics are important in the management of these infections. Nocardia must be kept in mind as a potential cause of opportunistic infection in organ-transplant recipients. Spinal pain must be accurately evaluated in patients with transplantation and those with chronic diseases. A high degree of suspicion of opportunistic infections, early evaluation with MRI, and proper sampling of the infectious material or biopsy specimen will provide early and accurate diagnosis for this potentially curable infection.
References 1. Queipo-Zaragoza JA, Broseta-Rico E, Alapont-Alacreu JM, Santos-Durantez M, Sanchez-Plumed J, Jimenez-Cruz JF. Nocardial infection in immunosuppressed kidney transplant recipients. Scand J Urol Nephrol 2004;38:168–73. 2. Azap A, Arslan H, Ergin F, Karakayali H, Haberal M. Disseminated Nocardia asteroides and coinfection with Trichophyton rubrum in a renal transplant recipient. Transpl Infect Dis 2002;4:223–5. 3. Koneman EW, Allen SD, Janda WM, Schreckenberger PC, Winn WC. The aerobic gram-positive bacilli. In: Koneman EW, Allen SD, Janda WM, Schreckenberger PC, Winn WC, editors. Color atlas and textbook of diagnostic microbiology. Philadelphia: Lippincott-Raven; 1997. pp. 691–708. 4. Mehta RS, Jain D, Chitnis DS. Nocardial abscess of spinal cord. Neurol India 1999;47:243–4. 5. Hiller R, Singh H, Crone M. Left leg paralysis in a renal transplant. Am J Kidney Dis 1999;33:E4–E6. 6. Harvey AL, Myslinski J, Ortiz F, Ortiz L. A case of Nocardia epidural abscess. J Emerg Med 1998;16:579–81. 7. Mamelak AN, Obana WG, Flaherty JF, Rosenblum ML. Nocardial brain abscess. Treatment strategies and factors influencing outcome. Neurosurgery 1994;35:622–31. 8. Durmaz R, Atasoy MA, Durmaz G, Adapinar B, Arslantas A, Aydinli A, et al. Multiple nocardial abscesses of cerebrum, cerebellum and spinal cord, causing quadriplegia. Clin Neurol Neurosurg 2001;10:59–62. 9. Misra AS, Biswas A, Deb P, Dhibar T, Das SK, Roy T. Compressive myelopathy due to nocardiosis from dermal lesion. J Assoc Physicians India 2003;51:318–21. 10. Graat HC, Van Ooij A, Day GA, McPhee IB. Nocardia farcinica spinal osteomyelitis. Spine 2002;27:E253–7. 11. Mukunda BN, Shekar R, Bass S. Solitary spinal intramedullary abscess caused by Nocardia asteroides. South Med J 1999;92:1223– 4. 12. Azap A, Arslan H, Ergin F, Karakayalı H, Haberal M. Disseminated Nocardia asteroides and coinfection with Tricophyton rubrum in a renal transplant recipient. Transpl Infect Dis 2002; 4:223–5. 13. Oruckaptan HH, Senmevsim O, Soylemezoglu F, Ozgen T. Cervical actinomycosis causing spinal cord compression and multisegmental root failure: case report and review of the literature. Neurosurgery 1998;43:937–40.
© Japanese Society of Chemotherapy and The Japanese Association for Infectious Diseases 2005
CASE REPORT Basar Atalay · Ozlem Azap · Melih Cekinmez Hakan Caner · Mehmet Haberal
Nocardial epidural abscess of the thoracic spinal cord and review of the literature
Received: November 25, 2004 / Accepted: April 20, 2005
Abstract Nocardia is a potential cause of opportunistic infection in organ-transplant recipients. We report an epidural abscess causing acute paraparesis in a patient with a rejected renal transplant and chronic liver disease. Early drainage and treatment with imipenem and trimethoprimsulfamethoxasole improved the patient’s neurologic status. Key words Epidural abscess · Nocardia · Renal transplant · Thoracic spine
Introduction Nocardiosis is a rare opportunistic infection caused by microorganisms of the genus Nocardia. It is associated with significant morbidity and mortality in immunecompromised patients.1 Although the main syndromes caused by the Nocardia species are respiratory tract disease (73%–77%), hematogenous dissemination occurs in about 50% of patients. Primary infection of the central nervous system (CNS) without pulmonary involvement occurs in only 5%–7% of patients, and this invasion may lead to severe neurologic sequelae.1–3 We report an unusual case of a patient with rejected renal transplant and liver failure in whom a gross paravertebral abscess invaded the thoracic spinal epidural space.
B. Atalay · M. Cekinmez · H. Caner Baskent University Department of Neurosurgery, Ankara, Turkey O. Azap Baskent University Department of Infections Disease, Ankara, Turkey M. Haberal Bakent University Department of General Surgery, Ankara, Turkey B. Atalay (*) 16. sokak no 24/4, 06500 Bahcelievler, Ankara, Turkey, Tel. +0090-535-786-99-59; Fax +0090 312 223 73 33 e-mail: [email protected]
Case report A 51-year-old man was admitted to the hospital with acute, increasing neck pain, radiating to both shoulders, and weakness in the lower extremities of 3 days’ duration. He had chronic renal failure and had been undergoing hemodialysis for 5 years after rejection of a renal transplant. He had chronic liver failure due to hepatitis C infection. He also had chronic obstructive pulmonary disease and steroid-induced diabetes mellitus. He was unable to ambulate and was paraparetic and hypoesthetic below the level of the third thoracic vertebrae. Results of all tendon reflex tests were negative. Routine laboratory studies revealed moderate leukocytosis (leucocyte count, 10.6 ¥ 103/ml) and slight anemia (10.6 g/dl) with an increased erythrocyte sedimentation rate (ESR), of 120 mm/h. Liver function test results were abnormal (alkaline phosphatase, 444 U/l; total bilirubin, 3.6 mg/dl; gamma-glutamyl transpeptidase (GGT), 291 U/l; aspartate aminotransferase (AST), 66 U/l; alanine aminotransferase (ALT), 57 U/l). The patient was evaluated using emergent cervical and thoracic magnetic resonance imaging (MRI), which revealed a paravertebral abscess in the C2-T8 muscles that extended to the epidural space over the third thoracic vertebra, compressing the spinal cord (Fig. 1). Computed tomography scans of the abdominal and thoracic regions and abdominal ultrasonography revealed no other abscess. The source of infection could not be identified. As the patient’s liver enzymes were very high, it was decided not to administer general anesthesia and to immediately drain the abscess percutaneously. A percutaneous catheter was introduced to the abscess with the aid of ultrasonography, and 2 l of pus of exudative quality was aspirated. The abscess was continuously drained for 1 week and the patient’s neurologic score subsequently improved. MRI after 1 week revealed subtotal remission of the abscess (Fig. 2). Empiric systemic antibiotics, including intra-venous (IV) imipenem, 250 mg q.i.d.; IV trimethoprim/sulfamethoxasole, 160/ 800 mg b.i.d.; and IV fluconazole, 200 mg/day were started immediately after drainage. Culture of the pus from the epidural abscess revealed an uncommon microorganism,
170
a Fig. 1. a T2-Weighted axial magnetic resonance (MR) image of the third thoracic vertebra, demonstrating a subcutaneous abscess that invaded the epidural space and compressed the spinal cord. b T2-
b weighted sagittal MR image, demonstrating compression over the spinal cord at the level of the third thoracic vertebra
a
b
Fig. 2. a Control MR image 5 days after drainage of the abscess. T2weighted axial MR image of the third thoracic vertebra, demonstrating subtotal remission of the abscess. b T2-weighted sagittal MR image,
demonstrating minimal compression over the spinal cord at the level of the third thoracic vertebra
termed Nocardia asteroides. Fluconazole was stopped; IV imipenem 250 mg q.i.d. was continued for another 6 months in our rehabilitation center’s outpatient clinic. He also continued his rehabilitation program at this center. Trimethoprim/sulfamethoxasole 160/800 mg b.i.d. IV was continued, together with IV imipenem for the first 3 months, and then oral trimethoprim/sulfamethoxasole 160/800 mg b.i.d was continued for the next 3 months. The laboratory values dropped to normal on the day 12 of therapy (leukocyte count, 6.3 ¥ 103 ml; ESR, 31 mm/h; total bilirubin, 1.1 mg/ dl; AST, 31 U/l; ALT, 24 U/l). These values were within the
normal ranges untill the end of therapy. The patient’s general status and neurologic deficit improved, and he was completely ambulatory and pain-free at 9 months’ follow-up.
Discussion Nocardiosis usually presents as an opportunistic infection in immunocompromised individuals. Nocardia spp. are gram-
171
positive, rod-shaped bacteria. They are strict aerobes found in the environment but very rarely cause infection in humans. Patients with AIDS, tuberculosis, lymphoma, those taking immunosuppressant drugs, and those with chronic alcoholism are prone to this opportunistic infection.4–6 Nocardia spp. usually cause infection after inhalation or direct inoculation.6 Hiller and colleagues5 reported a 4% incidence of Nocardia infections in posttransplant patients. Although hematogenous dissemination occurs commonly, blood cultures almost invariably fail to demonstrate the organism,3 and this mandates sampling and examining the material from infectious foci seen on radiologic studies. CNS infection caused by nocardiosis is usually supratentorial, and pathologically it consists of a multiloculated thin-walled abscess(es). Rarely, it involves the cerebellum, spinal cord, or meninges.4,7,8 Nocardial spinal cord abscesses are extremely rare. To our knowledge, only a few cases have been reported in the literature. These patients also had extraneurologic nocardiosis and the abcess(es) were located in the spinal epidural space.6,9,10 Harvey et al.6 reported an epidural abcess at L4 level and treated the abscess with triple agents, consisting of IV amikacin for 8 weeks, IV ceftriaxone for 6 months, and oral trimethoprimsulfamethoxasole for 1 year. Graat et al.10 reported an epidural abcess at T11-L3 level and isolated Nocardia farcinica. They treated this infection with ciprofloxacin (1 g/ day), sulfadiazine (6 g/day), and fluconazole (20 mg/day), for 2 years. Misra et al.9 reported an epidural abcess at T26, but the patient was complicated with Mycobacterium tuberculosis; the Nocardia species was Nocardia brazilensis. Mukunda and colleagues11 have presented a case of an intramedullary spinal abscess caused by Nocardia asteroides without concomitant extraneurologic nocardiosis. The CNS is the most common site of the disseminated disease and disease at this site is usually associated with high morbidity and mortality. In one series, the mortality was 24% in operated patients and 30% in nonoperated patients. In that series, 22% of the cases were diagnosed at autopsy.7 At Baskent University, nocardiosis has been diagnosed in only two unique cases, including our patient. The other case was a disseminated Nocardia asteroides with a brain abscess in a renal transplant recipient. Although an optimal regimen and duration for the treatment of Nocardial infections has not yet been definitively established, Azap et al.12 treated disseminated Nocardia asteroides infection in a renal transplant recipient for 1 year with IV meropenem, together with IV trimethoprimsulfamethoxasole for the first 5 months. In our experience we treated our patient with a spinal epidural abscess with 6 months of IV meropenem together with 3 months of IV and 3 months of oral trimethoprim-sulfamethoxasole. The differential diagnosis of spinal epidural abscess includes infections with Staphylococcus aureus, Actinomycosis, infections with Mycobacterium tuberculosis, fungal infections, and nonspecific osteomyelitis.6,13 Hiller and coworkers5 reported an unusual case of leg paralysis that was caused by Nocardia species in the postoperative course of a renal transplant patient. Our case is unique because the thoracic epidural cord compression occurred by direct inva-
sion from a subcutaneous Nocardial abscess. Simple percutaneous drainage and appropriate antibiotic therapy were effective in our patient.
Conclusion Although life expectancy for patients with chronic diseases is increasing with advances in medicine, incidences of opportunistic infections such as tuberculosis and AIDS are also increasing. Early diagnosis and appropriate antibiotics are important in the management of these infections. Nocardia must be kept in mind as a potential cause of opportunistic infection in organ-transplant recipients. Spinal pain must be accurately evaluated in patients with transplantation and those with chronic diseases. A high degree of suspicion of opportunistic infections, early evaluation with MRI, and proper sampling of the infectious material or biopsy specimen will provide early and accurate diagnosis for this potentially curable infection.
References 1. Queipo-Zaragoza JA, Broseta-Rico E, Alapont-Alacreu JM, Santos-Durantez M, Sanchez-Plumed J, Jimenez-Cruz JF. Nocardial infection in immunosuppressed kidney transplant recipients. Scand J Urol Nephrol 2004;38:168–73. 2. Azap A, Arslan H, Ergin F, Karakayali H, Haberal M. Disseminated Nocardia asteroides and coinfection with Trichophyton rubrum in a renal transplant recipient. Transpl Infect Dis 2002;4:223–5. 3. Koneman EW, Allen SD, Janda WM, Schreckenberger PC, Winn WC. The aerobic gram-positive bacilli. In: Koneman EW, Allen SD, Janda WM, Schreckenberger PC, Winn WC, editors. Color atlas and textbook of diagnostic microbiology. Philadelphia: Lippincott-Raven; 1997. pp. 691–708. 4. Mehta RS, Jain D, Chitnis DS. Nocardial abscess of spinal cord. Neurol India 1999;47:243–4. 5. Hiller R, Singh H, Crone M. Left leg paralysis in a renal transplant. Am J Kidney Dis 1999;33:E4–E6. 6. Harvey AL, Myslinski J, Ortiz F, Ortiz L. A case of Nocardia epidural abscess. J Emerg Med 1998;16:579–81. 7. Mamelak AN, Obana WG, Flaherty JF, Rosenblum ML. Nocardial brain abscess. Treatment strategies and factors influencing outcome. Neurosurgery 1994;35:622–31. 8. Durmaz R, Atasoy MA, Durmaz G, Adapinar B, Arslantas A, Aydinli A, et al. Multiple nocardial abscesses of cerebrum, cerebellum and spinal cord, causing quadriplegia. Clin Neurol Neurosurg 2001;10:59–62. 9. Misra AS, Biswas A, Deb P, Dhibar T, Das SK, Roy T. Compressive myelopathy due to nocardiosis from dermal lesion. J Assoc Physicians India 2003;51:318–21. 10. Graat HC, Van Ooij A, Day GA, McPhee IB. Nocardia farcinica spinal osteomyelitis. Spine 2002;27:E253–7. 11. Mukunda BN, Shekar R, Bass S. Solitary spinal intramedullary abscess caused by Nocardia asteroides. South Med J 1999;92:1223– 4. 12. Azap A, Arslan H, Ergin F, Karakayalı H, Haberal M. Disseminated Nocardia asteroides and coinfection with Tricophyton rubrum in a renal transplant recipient. Transpl Infect Dis 2002; 4:223–5. 13. Oruckaptan HH, Senmevsim O, Soylemezoglu F, Ozgen T. Cervical actinomycosis causing spinal cord compression and multisegmental root failure: case report and review of the literature. Neurosurgery 1998;43:937–40.