- Email: [email protected]
Nocardia discitis in an immunocompetent patient
Letters to the editor / Joint Bone Spine 74 (2007) 205e211 [3] Narvaez J, Domingo-Domenech E, Narvaez JA, Nolla JM, Valverde J. Acquired hypophosphatemic osteomalacia associated with multiple myeloma. Joint Bone Spine 2005;72:424e6. [4] Barutcuoglu B, Parildar Z, Mutaf I, Habif S, Bayindir O. Spuriously elevated inorganic phosphate level in a multiple myeloma patient. Clin Lab Haematol 2003;25:271e4. [5] Busse JC, Gelbard MA, Byrnes JJ, Hellman R, Vaamonde CC. Pseudohyperphosphatemia and dysproteinemia. Arch Intern Med 1987;147:2045e6. [6] Jarchovsky I, Beitner H, Plavnick L. Spurious hyperphosphatemia in multiple myeloma. Harefuah 1991;121:515e6. [7] Marcu CB, Hotchkiss M. Pseudohyperphosphatemia in a patient with multiple myeloma. Conn Med 2004;68:71e2. [8] Sonnenblick M, Eylath U, Brisk R, Eldad C, Hershk C. Paraprotein interference with colorimetry of phosphate in serum of some patients with multiple myeloma. Clin Chem 1986;32:1537e9. [9] Oren S, Feldman A, Turkot S, Lugassy G. Hyperphosphatemia in multiple myeloma. Ann Hematol 1994;69:41e3.
Imane El Bouchti* Ahlam Belkhou Rachid Younsi Selma El Asan Rheumatology Department, Ibn Tofail Hospital, Mohammed VI Teaching Hospitals, Marrakech, Morocco *Corresponding author. Service de Rhumatologie, Hopital Ibn Tofail, CHU Mohammed VI, 42000, Marrakech, Morocco Tel.: þ212 6743 5921. E-mail address: [email protected] (I. El Bouchti) 21 March 2006 Available online 6 February 2007 1297-319X/$ - see front matter Ó 2007 Published by Elsevier Masson SAS. doi:10.1016/j.jbspin.2006.07.004
Nocardia discitis in an immunocompetent patient
207
At admission, his body temperature was normal. His body weight was 145 kg, yielding a body mass index of 44.8. He had severe back pain that made him unable to sit or stand. Motor function and sensation were normal, and no pyramidal signs were found at the lower limbs. A thorough physical examination for foci of infection identified a large maxillary abscess with a painless swelling of the palate. There was no evidence of pulmonary, gastrointestinal, or genitourinary infection. Laboratory tests showed severe inflammation (15,900 leukocytes/mm3; C-reactive protein, 168 mg/L; and fibrinogen, 7 g/L). Blood cultures were negative. The lumbosacral junction was not clearly visible on standard radiographs. CT of the lumbar spine disclosed loss of L5eS1 disk height, severe kissing erosions of the lower L5 and upper S1 endplate, and reactive sclerosis of S1 suggesting discitis (Fig. 1). In addition, epidural involvement was noted along L5 and S1, and an abscess was seen in each of the ilio-psoas muscles. The severe pain and CT evidence of epidural involvement prompted a percutaneous needle biopsy of the disk followed immediately by probabilistic antibiotic therapy with oxacillin, rifampin, and netromycin. No organisms were recovered from the biopsy specimens. After 5 days, no improvements were noted in the clinical symptoms or laboratory parameters. The patient was transferred to the septic orthopedic surgery unit. Percutaneous drainage of the ilio-psoas abscesses was not feasible, given the severe obesity. A surgical biopsy of the L5eS1 disk and vertebras was performed under general anesthesia. A large amount of purulent fluid was drained from the L5eS1 space. Nocardia asteroides was identified in the biopsy specimen. Subsequently, the cultures of the percutaneous biopsy done in the rheumatology department grew the same organism. Imipenem and amikacin were given based on the antibiotic susceptibility test results. The CRP level declined initially but subsequently increased starting on the 15th treatment day, and
Keywords: Spinal osteomyelitis; Nocardia asteroides; Immunocompetent
1. Introduction Disseminated nocardiosis occurs chiefly in immunocompromised patients. Osteoarticular foci are uncommon and, among them, spinal infection is exceedingly rare. 2. Case-report A 46-year-old man was admitted for severe bilateral low back pain and sciatica unresponsive to morphine. He was obese and had been experiencing low back pain for several years. Over the last few months, the pain had worsened, becoming continuous and preventing sleep. There was a decline in general health but no fever or chills. He had morbid obesity and sleep apnea syndrome, as well as a past history of smoking and alcohol abuse. His obesity precluded outpatient computed tomography (CT).
Fig. 1. Computed tomography of the spine, sagittal section: combined osteolysis and sclerotic lesions of the L5 and S1 endplates.
208
Letters to the editor / Joint Bone Spine 74 (2007) 205e211
Fig. 2. Computed tomography of the abdomen, transverse section: sacral osteolysis and large abscesses in the ilio-psoas muscles.
the pain started worsening shortly afterward. CT showed marked enlargement of the ilio-psoas muscle abscesses. The abscess on the left extended to the renal compartment and the abscess on the right to the lesser trochanter (Figs. 2 and S1; see the supplementary material associated with thus article online). The worsening infection despite appropriate antibiotic treatment prompted surgical treatment, which was performed 7 days later via bilateral lumbar incisions. Drainage of the two abscesses recovered 4.5 L of pus, whose culture confirmed the diagnosis of monobacterial N. asteroides infection. The antibiotic susceptibility pattern was unchanged. Imipemen therapy was continued, in combination with amikacin for 10 days followed by oral rifampin. After 80 days of imipenem therapy, the outcome was favorable with a CRP level at 7 mg/L. Oral linezolid was substituted for the imipenem. Follow-up CT showed complete resolution of the abscesses and stabilization of the vertebral lesions. The maxillary abscess resolved. The antibiotics were stopped after a total of 4 months. The patient was transferred to the rehabilitation department for further walking and self-sufficiency rehabilitation.
or skeleton. Septic arthritis and indolent osteomyelitis are the two main patterns of osteoarticular involvement in patients with disseminated nocardiosis [3]. Disseminated nocardiosis occurs chiefly in immunocompromised patients, most notably those with hematological malignancies, HIV infection, or immunosuppressant therapy. Long-term glucocorticoid therapy is a major and independent risk factor for nocardiosis [4]. The expanding population of patients with immunodepression may explain the increase in reported nocardiosis cases in recent years. About 10 cases of N. asteroides discitis have been reported [5]. The thoracic and cervical spine were predominantly involved. Pulmonary involvement and immunodepression were found in only half the cases. In our patient, the maxillary abscess was a possible starting point for the infection. It was not drained but resolved with antibiotic therapy. No risk factors were identified, although morbid obesity and past alcohol abuse may have promoted the development of the infection. The high morbidity and mortality rates of disseminated nocardiosis indicate a need for an early diagnosis. However, blood cultures are usually negative, and local specimens must therefore be obtained. Our case illustrates the usefulness of vertebral biopsy in patients with spinal involvement. Antimicrobial therapy should be started intravenously and continued for several months. Surgery is often performed, both for cleansing and for reconstruction [6]. The need for surgery is well illustrated by our case, in which appropriate antibiotic therapy was inadequate despite the absence of acquired resistance or co-infection with other organisms. Cotrimoxazole is the antimicrobial of choice in N. asteroides infections. Either amikacin or imipenem should be given also. Our patient was not given cotrimoxazole, because antibiotic susceptibility testing showed intermediate susceptibility to this antimicrobial. Linezolid is a recently introduced oxazolidinone that is used chiefly to treat staphylococcal infections. Advantages of linezolid in patients with Nocardia discitis include efficacy via the oral route, good penetration within bone tissue, and efficacy on the full range of Nocardia species [7]. Supplementary material
3. Discussion Nocardia are aerobic, filamentous, Gram-positive actinomycetes. The dominant species is N. asteroides complex, which includes N. asteroides stricto sensu, N. farcinica, and N. nova. Nocardia are ubiquitous in soil [1]. Local traumatic inoculation can lead to cutaneous or subcutaneous infection, which may progress to chronic soft tissue infection known as mycetoma. Mycetoma may spread to adjacent osteoarticular structures [2]. Inhalation is the most common mode of infection. The subsequent pleuropulmonary infection may go unnoticed. If hematogenous dissemination occurs, foci may develop in the brain, meninges, subcutaneous tissue, kidneys,
Supplementary material (Fig. S1) associated with this article can be found at http://www.sciencedirect.com, at doi:10.1016/ j.jbspin.2006.08.003.
References [1] Saubolle MA, Sussland D. Nocardiosis: review of clinical and laboratory experience. J Clin Microbiol 2003;41:4497e501. [2] Carlisle JT, Greer DL, Hyslop NE. Actinomycetoma of the hand caused by Nocardia asteroides. J Infect Dis 1988;158:244e6. [3] Corti ME, Villafane Fioti MF. Nocardiosis: a review. Int J Infect Dis 2003;7:243e50.
Letters to the editor / Joint Bone Spine 74 (2007) 205e211 [4] Lederman ER, Crum NF. A case series and focused review of nocardiosis: clinical and microbiologic aspects. Medicine 2004;83:300e13. [5] Laurin JM, Resnik CS, Wheeler D. Vertebral osteomyelitis caused by Nocardia. J Rheumatol 1991;18:455e8. [6] Harm C, Graat HC, Van Ooij A, Day JA, Mac Phee IB. Nocardia farcinica spinal osteomyelitis. Spine 2002;27:253e7. [7] Moylett EH, Pacheco SE, Brown-Elliott AE. Clinical experience with linezolid for treatment of Nocardia infection. Clin Infect Dis 2003;36:313e8.
Emmanuel Chatelus* Rose-Marie Javier Jean Sibilia Jean-Louis Kuntz Rheumatology Department, Hautepierre Teaching Hospital, CHU de Hautepierre, Avenue Moliere, 67098 Strasbourg Cedex, France *Corresponding author. E-mail address: [email protected] (E. Chatelus) Emmanuel Forestier Internal Medicine and Infectious and Tropical Diseases Department, Strasbourg Teaching Hospital, Strasbourg, France Jeannot Gaudias Septic Surgery Department, Illkirch Center for Traumatology and Orthopedic Surgery, Strasbourg, France 28 March 2006 Available online 2 February 2007 1297-319X/$ - see front matter Ó 2007 Elsevier Masson SAS. All rights reserved. doi:10.1016/j.jbspin.2006.08.003
209
heart failure, carpal tunnel syndrome and neuropathy [4]. Amyloid infiltration in vital organs can be fatal if left untreated. Amyloid deposition in joints can be detected in the course of systemic myeloma complicating with the restriction of movements of infiltrated regions. Most common involved joints are shoulders, wrists, knees and metacarpophalangeal joints. Generally, arthropathy presents with bilateral symmetric polyarthritis. However, amyloid deposition in bilateral knees associated with other joint invasion has been reported very rarely [5]. We hereby present a case with the diagnosis of MM complicated with amyloid deposition in bilateral knee and ankle joints under the treatment.
2. Case report This fifty four years old male patient was referred to our hospital because of anemia, generalized bone pain and elevated erythrocyte sedimentation rate (ESR). He complained restriction in movements of bilateral wrist joints and diagnosed as carpal tunnel syndrome few months ago without further investigation and treatment. On his physical examination, there was no finding except pallor. Laboratory analysis showed anemia (haemoglobin as 7.9 g/dl), hyperuricemia (8.2 mg/dl) and elevated ESR (67 mm/h). There was rouleaux formation on peripheral blood smear. Urinalysis revealed proteinuria more than 1 gr/day and immunofixation of urine and blood showed monoclonal gammopathy of lambda-light chain. No lythic lesions were detected on his plain bone radiograms. Bone marrow aspiration and biopsy demonstrated 30e40% lambda-positive atypical plasma cells, confirming the diagnosis of lambda light chain myeloma, stage IIA, according to Durie-Salmon classification. Standard dose of VAD regimen was administered as vincristine 0.4 mg/day D1e4, adriablastina 9 mg/m2/day
Amyloid deposition in knee and ankle joints in the course of multiple myeloma Keywords: Amyloidosis; Multiple myeloma; Knee and ankle joints
1. Introduction Multiple myeloma (MM) is a malignant hematological disease, which is characterized by the production of monoclonal M protein and abnormal plasma cell infiltration of bone marrow and other tissues in some cases [1,2]. MM is sometimes complicated by amyloidosis [3,4]. Amyloidosis may be systemic or localized. Although the current classification of the disease is based on the biochemical composition of the precursor protein, unrelated to the biochemical features, clinical features are similar and may vary correlated with the involved tissue and organs. The most common symptoms are fatigue, weight loss, macroglossia, orthostatic hypotension, diarrhea, cardiomegaly, nephrotic syndrome, renal failure, congestive
Fig. 1. Right knee T2-weighted sagittal plane MRI: Low signal intensity and signal void areas are present in effusion of knee joint posteriorly.
Imane El Bouchti* Ahlam Belkhou Rachid Younsi Selma El Asan Rheumatology Department, Ibn Tofail Hospital, Mohammed VI Teaching Hospitals, Marrakech, Morocco *Corresponding author. Service de Rhumatologie, Hopital Ibn Tofail, CHU Mohammed VI, 42000, Marrakech, Morocco Tel.: þ212 6743 5921. E-mail address: [email protected] (I. El Bouchti) 21 March 2006 Available online 6 February 2007 1297-319X/$ - see front matter Ó 2007 Published by Elsevier Masson SAS. doi:10.1016/j.jbspin.2006.07.004
Nocardia discitis in an immunocompetent patient
207
At admission, his body temperature was normal. His body weight was 145 kg, yielding a body mass index of 44.8. He had severe back pain that made him unable to sit or stand. Motor function and sensation were normal, and no pyramidal signs were found at the lower limbs. A thorough physical examination for foci of infection identified a large maxillary abscess with a painless swelling of the palate. There was no evidence of pulmonary, gastrointestinal, or genitourinary infection. Laboratory tests showed severe inflammation (15,900 leukocytes/mm3; C-reactive protein, 168 mg/L; and fibrinogen, 7 g/L). Blood cultures were negative. The lumbosacral junction was not clearly visible on standard radiographs. CT of the lumbar spine disclosed loss of L5eS1 disk height, severe kissing erosions of the lower L5 and upper S1 endplate, and reactive sclerosis of S1 suggesting discitis (Fig. 1). In addition, epidural involvement was noted along L5 and S1, and an abscess was seen in each of the ilio-psoas muscles. The severe pain and CT evidence of epidural involvement prompted a percutaneous needle biopsy of the disk followed immediately by probabilistic antibiotic therapy with oxacillin, rifampin, and netromycin. No organisms were recovered from the biopsy specimens. After 5 days, no improvements were noted in the clinical symptoms or laboratory parameters. The patient was transferred to the septic orthopedic surgery unit. Percutaneous drainage of the ilio-psoas abscesses was not feasible, given the severe obesity. A surgical biopsy of the L5eS1 disk and vertebras was performed under general anesthesia. A large amount of purulent fluid was drained from the L5eS1 space. Nocardia asteroides was identified in the biopsy specimen. Subsequently, the cultures of the percutaneous biopsy done in the rheumatology department grew the same organism. Imipenem and amikacin were given based on the antibiotic susceptibility test results. The CRP level declined initially but subsequently increased starting on the 15th treatment day, and
Keywords: Spinal osteomyelitis; Nocardia asteroides; Immunocompetent
1. Introduction Disseminated nocardiosis occurs chiefly in immunocompromised patients. Osteoarticular foci are uncommon and, among them, spinal infection is exceedingly rare. 2. Case-report A 46-year-old man was admitted for severe bilateral low back pain and sciatica unresponsive to morphine. He was obese and had been experiencing low back pain for several years. Over the last few months, the pain had worsened, becoming continuous and preventing sleep. There was a decline in general health but no fever or chills. He had morbid obesity and sleep apnea syndrome, as well as a past history of smoking and alcohol abuse. His obesity precluded outpatient computed tomography (CT).
Fig. 1. Computed tomography of the spine, sagittal section: combined osteolysis and sclerotic lesions of the L5 and S1 endplates.
208
Letters to the editor / Joint Bone Spine 74 (2007) 205e211
Fig. 2. Computed tomography of the abdomen, transverse section: sacral osteolysis and large abscesses in the ilio-psoas muscles.
the pain started worsening shortly afterward. CT showed marked enlargement of the ilio-psoas muscle abscesses. The abscess on the left extended to the renal compartment and the abscess on the right to the lesser trochanter (Figs. 2 and S1; see the supplementary material associated with thus article online). The worsening infection despite appropriate antibiotic treatment prompted surgical treatment, which was performed 7 days later via bilateral lumbar incisions. Drainage of the two abscesses recovered 4.5 L of pus, whose culture confirmed the diagnosis of monobacterial N. asteroides infection. The antibiotic susceptibility pattern was unchanged. Imipemen therapy was continued, in combination with amikacin for 10 days followed by oral rifampin. After 80 days of imipenem therapy, the outcome was favorable with a CRP level at 7 mg/L. Oral linezolid was substituted for the imipenem. Follow-up CT showed complete resolution of the abscesses and stabilization of the vertebral lesions. The maxillary abscess resolved. The antibiotics were stopped after a total of 4 months. The patient was transferred to the rehabilitation department for further walking and self-sufficiency rehabilitation.
or skeleton. Septic arthritis and indolent osteomyelitis are the two main patterns of osteoarticular involvement in patients with disseminated nocardiosis [3]. Disseminated nocardiosis occurs chiefly in immunocompromised patients, most notably those with hematological malignancies, HIV infection, or immunosuppressant therapy. Long-term glucocorticoid therapy is a major and independent risk factor for nocardiosis [4]. The expanding population of patients with immunodepression may explain the increase in reported nocardiosis cases in recent years. About 10 cases of N. asteroides discitis have been reported [5]. The thoracic and cervical spine were predominantly involved. Pulmonary involvement and immunodepression were found in only half the cases. In our patient, the maxillary abscess was a possible starting point for the infection. It was not drained but resolved with antibiotic therapy. No risk factors were identified, although morbid obesity and past alcohol abuse may have promoted the development of the infection. The high morbidity and mortality rates of disseminated nocardiosis indicate a need for an early diagnosis. However, blood cultures are usually negative, and local specimens must therefore be obtained. Our case illustrates the usefulness of vertebral biopsy in patients with spinal involvement. Antimicrobial therapy should be started intravenously and continued for several months. Surgery is often performed, both for cleansing and for reconstruction [6]. The need for surgery is well illustrated by our case, in which appropriate antibiotic therapy was inadequate despite the absence of acquired resistance or co-infection with other organisms. Cotrimoxazole is the antimicrobial of choice in N. asteroides infections. Either amikacin or imipenem should be given also. Our patient was not given cotrimoxazole, because antibiotic susceptibility testing showed intermediate susceptibility to this antimicrobial. Linezolid is a recently introduced oxazolidinone that is used chiefly to treat staphylococcal infections. Advantages of linezolid in patients with Nocardia discitis include efficacy via the oral route, good penetration within bone tissue, and efficacy on the full range of Nocardia species [7]. Supplementary material
3. Discussion Nocardia are aerobic, filamentous, Gram-positive actinomycetes. The dominant species is N. asteroides complex, which includes N. asteroides stricto sensu, N. farcinica, and N. nova. Nocardia are ubiquitous in soil [1]. Local traumatic inoculation can lead to cutaneous or subcutaneous infection, which may progress to chronic soft tissue infection known as mycetoma. Mycetoma may spread to adjacent osteoarticular structures [2]. Inhalation is the most common mode of infection. The subsequent pleuropulmonary infection may go unnoticed. If hematogenous dissemination occurs, foci may develop in the brain, meninges, subcutaneous tissue, kidneys,
Supplementary material (Fig. S1) associated with this article can be found at http://www.sciencedirect.com, at doi:10.1016/ j.jbspin.2006.08.003.
References [1] Saubolle MA, Sussland D. Nocardiosis: review of clinical and laboratory experience. J Clin Microbiol 2003;41:4497e501. [2] Carlisle JT, Greer DL, Hyslop NE. Actinomycetoma of the hand caused by Nocardia asteroides. J Infect Dis 1988;158:244e6. [3] Corti ME, Villafane Fioti MF. Nocardiosis: a review. Int J Infect Dis 2003;7:243e50.
Letters to the editor / Joint Bone Spine 74 (2007) 205e211 [4] Lederman ER, Crum NF. A case series and focused review of nocardiosis: clinical and microbiologic aspects. Medicine 2004;83:300e13. [5] Laurin JM, Resnik CS, Wheeler D. Vertebral osteomyelitis caused by Nocardia. J Rheumatol 1991;18:455e8. [6] Harm C, Graat HC, Van Ooij A, Day JA, Mac Phee IB. Nocardia farcinica spinal osteomyelitis. Spine 2002;27:253e7. [7] Moylett EH, Pacheco SE, Brown-Elliott AE. Clinical experience with linezolid for treatment of Nocardia infection. Clin Infect Dis 2003;36:313e8.
Emmanuel Chatelus* Rose-Marie Javier Jean Sibilia Jean-Louis Kuntz Rheumatology Department, Hautepierre Teaching Hospital, CHU de Hautepierre, Avenue Moliere, 67098 Strasbourg Cedex, France *Corresponding author. E-mail address: [email protected] (E. Chatelus) Emmanuel Forestier Internal Medicine and Infectious and Tropical Diseases Department, Strasbourg Teaching Hospital, Strasbourg, France Jeannot Gaudias Septic Surgery Department, Illkirch Center for Traumatology and Orthopedic Surgery, Strasbourg, France 28 March 2006 Available online 2 February 2007 1297-319X/$ - see front matter Ó 2007 Elsevier Masson SAS. All rights reserved. doi:10.1016/j.jbspin.2006.08.003
209
heart failure, carpal tunnel syndrome and neuropathy [4]. Amyloid infiltration in vital organs can be fatal if left untreated. Amyloid deposition in joints can be detected in the course of systemic myeloma complicating with the restriction of movements of infiltrated regions. Most common involved joints are shoulders, wrists, knees and metacarpophalangeal joints. Generally, arthropathy presents with bilateral symmetric polyarthritis. However, amyloid deposition in bilateral knees associated with other joint invasion has been reported very rarely [5]. We hereby present a case with the diagnosis of MM complicated with amyloid deposition in bilateral knee and ankle joints under the treatment.
2. Case report This fifty four years old male patient was referred to our hospital because of anemia, generalized bone pain and elevated erythrocyte sedimentation rate (ESR). He complained restriction in movements of bilateral wrist joints and diagnosed as carpal tunnel syndrome few months ago without further investigation and treatment. On his physical examination, there was no finding except pallor. Laboratory analysis showed anemia (haemoglobin as 7.9 g/dl), hyperuricemia (8.2 mg/dl) and elevated ESR (67 mm/h). There was rouleaux formation on peripheral blood smear. Urinalysis revealed proteinuria more than 1 gr/day and immunofixation of urine and blood showed monoclonal gammopathy of lambda-light chain. No lythic lesions were detected on his plain bone radiograms. Bone marrow aspiration and biopsy demonstrated 30e40% lambda-positive atypical plasma cells, confirming the diagnosis of lambda light chain myeloma, stage IIA, according to Durie-Salmon classification. Standard dose of VAD regimen was administered as vincristine 0.4 mg/day D1e4, adriablastina 9 mg/m2/day
Amyloid deposition in knee and ankle joints in the course of multiple myeloma Keywords: Amyloidosis; Multiple myeloma; Knee and ankle joints
1. Introduction Multiple myeloma (MM) is a malignant hematological disease, which is characterized by the production of monoclonal M protein and abnormal plasma cell infiltration of bone marrow and other tissues in some cases [1,2]. MM is sometimes complicated by amyloidosis [3,4]. Amyloidosis may be systemic or localized. Although the current classification of the disease is based on the biochemical composition of the precursor protein, unrelated to the biochemical features, clinical features are similar and may vary correlated with the involved tissue and organs. The most common symptoms are fatigue, weight loss, macroglossia, orthostatic hypotension, diarrhea, cardiomegaly, nephrotic syndrome, renal failure, congestive
Fig. 1. Right knee T2-weighted sagittal plane MRI: Low signal intensity and signal void areas are present in effusion of knee joint posteriorly.