Case Reports / Journal of Clinical Neuroscience 18 (2011) 425–428
the lower levels of the spine may lead to spinal cord compression syndromes or to symptoms of either acute nerve compression or chronic spinal stenosis.4,5 To treat CPPD in the acute phase, oral non-steroidal anti-inﬂammatory drugs or oral colchicine is preferred. Alternatively oral or intravenous glucocorticoids can be used. There is no effective therapy to prevent progressive CPPD crystal deposition disease. In conclusion, CPPD should be considered in patients with non-speciﬁc spinal lesions. References 1. Jones AC, Chuck AJ, Arie EA, et al. Diseases associated with calcium pyrophosphate deposition disease. Semin Arthritis Rheum 1992;22:188–202. 2. Wilkins E, Dieppe P, Maddison P, et al. Osteoarthritis and articular chondrocalcinosis in the elderly. Ann Rheum Dis 1983;42:280–4. 3. Ryan LM. The ank gene story. Arthritis Res 2001;3:77–9. 4. Dieppe P, Alexander GJM, Jones HE, et al. Pyrophosphate arthropathy: a clinical and radiological study of 105 cases. Ann Rheum Dis 1982;41:371–6. 5. Muthukumar N, Karuppaswamy U. Tumoral calcium pyrophosphate dihydrate deposition disease of the ligamentum ﬂavum. Neurosurgery 2003;53:103–8.
Fig. 2. Coronal CT scan of the cervical spine showing a ‘‘crowned dens’’, which is typical for calcium pyrophosphate dihydrate crystal deposits in pyrophosphate gout (arrow).
Brainstem abscess caused by Haemophilus inﬂuenza and Peptostreptococcus species M. Stein a,⇑, I. Schirotzek b, M. Preuss a, W. Scharbrodt a, M. Oertel a a b
Department of Neurosurgery, University Hospital Gießen, Klinikstrasse 29, 35385 Gießen, Germany Department of Neurology, University Hospital Gießen, Gießen, Germany
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Article history: Received 20 March 2010 Accepted 23 March 2010
Keywords: Brainstem abscess Haemophilus inﬂuenza type b Stereotactic aspiration
a b s t r a c t We present a patient with a solitary brainstem abscess caused by Haemophilus inﬂuenza type b (Hib) and Peptostreptococcus species. This is the ﬁrst report of a Hib brainstem abscess in the English literature. Hib has been mainly associated with respiratory or nasal infections, and a few cases of intracerebral abscesses, but no brainstem abscesses have been described. The literature on solitary brainstem abscesses was reviewed and an overview of the literature between January 1984 and May 2009 is presented. Ó 2010 Elsevier Ltd. All rights reserved.
⇑ Corresponding author. Tel.: +49 6419945530; fax: +49 6419945509. E-mail address: [email protected]
Abscesses of the brainstem are rare and represent fewer than 1% of all brain abscesses.1,2 A few patients with Haemophilus inﬂuenza type b (Hib) brain abscesses have been reported in the literature3–7 but to our knowledge none have been brainstem abscesses. In this
Case Reports / Journal of Clinical Neuroscience 18 (2011) 425–428
paper we report a patient with a solitary brainstem abscess caused by Hib and Peptostreptococcus species and review the relevant literature. 2. Case report A 47-year-old female presented to the University hospital’s Department of Neurology with a 15-day history of bronchitis, and intense headache and neck pain for the past 3 days. She had a medical history of hypertension and had undergone surgery for a phaeochromocytoma 7 years previously. On admission, she was alert and oriented. She suffered from headache and nausea without vomiting. No fever was detected. A spontaneous nystagmus to the left side was observed. Overall, the motor exam was unremarkable except for an ataxic gait on the right side. The complete blood count revealed: red blood count, 87 1012/L; haemoglobin level, 87 g/L; platelet count, 257 106/L; white blood count, 6.1 106/L and an elevated C-reactive protein level of 253.7 mg/L. A CT scan of the brain with contrast showed a hypodense lesion without enhancement in the left lateral midbrain. Intravenous therapy with ceftriaxone, ampicillin and metronidazole was started. Under antibiotic treatment, the C-reactive protein level decreased to 14.9 mg/L. After 3 days the patient developed rightsided hemiparesis and pyramidal signs. Neurological deterioration occurred and the patient developed tetraparesis. An urgent MRI scan showed an encapsulated, ring-enhancing, left-sided brainstem abscess (Fig. 1a). On day 5 after admission, the solitary brainstem abscess was drained stereotactically. The center of the brainstem abscess was targeted by a suboccipital transcerebellar approach with the patient in prone position. At the target point, 7 mL of pus was drained. Cultures grew Hib and Peptostreptococcus species. Intravenous antibiotic therapy was continued with vancomycin and ceftriaxone. A repeated MRI of the brain on day 5 after surgery showed resolution of the brainstem abscess (Fig. 1b). The neurological status, however, improved only minimally. CT imaging of the abdomen and the chest revealed no original source of the infection. Her immunoglobulin analysis revealed a decreased level of immunoglobulin G (IgG) (5.0 g/L). Two months after admission, the patient was discharged to a neurologic rehabilitation center. On the day of discharge the patient was awake and alert. She suffered from improving tetraparesis, worse in the legs. Active ﬁst closure with slight weakness was possible bilaterally.
Fig. 1. (a) Axial post-gadolinium T1-weighted MRI showing left-sided brainstem abscess prior to stereotactical aspiration and (b) the brainstem abscess 3 weeks after stereotactic aspiration.
Her clinical situation improved slowly during neurologic rehabilitation. Six months after stereotactic treatment, the patient was discharged home with moderate deﬁcit. She was mobilizing in a wheelchair and was able to use both hands in daily activities.
3. Literature review The detection of brainstem abcesses was difﬁcult until MRI was introduced to clinical radiology. In the era of brain CT imaging, brainstem abscesses were diagnosed inconsistently because of bone artefacts in the posterior fossa. Consequently, this review includes publications on solitary brainstem abcesses between January 1984 and May 2009. A MedLine search with the keywords ‘‘solitary brainstem abscess’’ provided 23 results, of which three articles were excluded as not relevant. Without the term ‘‘solitary’’, 361 items were found. Out of these 361 articles, 56 were found on solitary brainstem abscesses. Next, we substituted ‘‘brainstem’’ with ‘‘brain stem’’ and found 241 articles. Out of these 241, another 6 new articles were detected. After excluding the items published before 1984, a total of 35 articles on the relevant topic of solitary brainstem abscess were accepted for this review. The results have been presented in Supplementary Tables 1 and 2.8–42 Overall, 39 patients with solitary brainstem abcesses were reported in 35 articles. The mean age was 37 years (range: 2–71 years). The female to male ratio was 1 to 1.2. The most common initial symptom was headache (51%), followed by diplopia (41%), hemiparesis (31%) and nausea or vomiting (26%). The onset of these symptoms varied from 1 day to 4 months prior to admission. Fever was documented in 32% of patients on admission. The most common abscess region was the pons (64%). White blood cell (WBC) count was unknown in 22 patients. In nine patients, the WBC count was elevated and in eight patients the WBC count was normal. MRI was used for neuroimaging in most patients (54%). At the end of the 1980s, before the routine use of MRI, the most commonly used imaging was CT scanning (46%). The size of the brainstem abscess was documented in 31% (n = 12/39) of the patients. Surgical treatment was performed in 26 patients and the organism that was identiﬁed most often (in 39% of patients) was Streptococcus species. In three patients, a gram-positive organism was identiﬁed but not speciﬁed. No organism could be identiﬁed in 12% of patients. Stereotactical aspiration was performed in 14 patients (54%). A secondary microsurgical incision of the brainstem was required for one patient and in three patients a second stereotactic abscess aspiration was performed. Open procedures such as surgical incision or aspiration were performed in 46% of patients. On admission, mild, moderate and severe neurological deﬁcits were recorded in 9%, 54% and 27% of patients, respectively. After surgical treatment, no neurologic deﬁcit was observed in 15% of patients, mild in 42%, moderate in 39% and a severe neurologic deficit occurred in 4% of patients. Medical treatment without surgery occured in 13 patients. In seven of these patients (54%) no possible source of infection could be identiﬁed. The medical treatment ranged from monotherapy with ampicillin to a combination of seven different antibiotics. The neurological examination on admission revealed: a mild neurologic deﬁcit in 46% of patients; moderate neurologic deﬁcit in 39% of patients; and a severe deﬁcit in 15% of patients. In one patient the outcome was unknown. After medical treatment the neurologic deﬁcit was absent in 23% of patients, mild in 46% of patients, moderate in 15% of patients and severe in 8%.
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4. Discussion Our patient developed a brainstem abscess after a history of bronchitis. A brainstem abscess caused by Hib has not been described in the reviewed literature. No other source of infection in the presented case was known. Initial medical treatment with antibiotics failed although on antibiotic sensitivity testing after abscess drainage, in vitro-sensitivity was observed. The most common clinical presentations of invasive Hib infection in adults are: pneumonia (22%), bacteraemia (21%), epiglottitis (16%), and meningitis (7%), together responsible for two-thirds of all reports.43 In one report, infection with Hib was combined with other bacteria in 71% of patients.44 In the presented patient the development of the brainstem abscess was possibly promoted by a deﬁciency of immunoglobulin G (IgG), which had been newly diagnosed. At the moment no guidelines exist for the treatment of solitary brainstem abscesses. Possible treatment options include stereotactic abscess drainage, open microsurgical excision or medical therapy alone. Between January 1984 and May 2009, 54% of the published cases of operatively treated solitary brainstem abscess were treated stereotactically. Stereotactic drainage and biopsy of brain lesions is considered a safe and established procedure.1 The surgically treated patients presented more frequently with moderate and severe neurologic deﬁcits compared to the patients in the medically treated cohort (54% versus [vs.] 39% and 27% vs. 15%) and the patients who were treated by medical therapy alone had primarily mild neurological deﬁcits on admission (46% vs. 9%). As a result, the literature tends to show that better neurologic outcomes are achieved in medically treated patients. However, if medical therapy fails, the identiﬁcation of an organism is important for further therapy. The size of the brainstem abscess may have a role in the decision to treat medically compared to surgically. Depending on the patients’ clinical status, if the abscess is close to the surface of the brainstem, a surgical approach seems reasonable. In contrast, a deep-seated and small lesion would prompt medical treatment regardless of the clinical condition. 5. Conclusion Overall, no general recommendations can be made on the basis of the existing literature. Medical therapy alone is a treatment option for awake patients with minor neurologic deﬁcits. If neurological deterioration occurs, or if patients have major a neurologic deﬁcit, stereotactic drainage is a safe procedure in the treatment of solitary brainstem abscess. Appendix A. Supplementary data Supplementary data associated with this article can be found, in the online version, at doi:10.1016/j.jocn.2010.03.054. References 1. Rajshekhar V, Chandy MJ. Computerized tomography-guided stereotactic surgery for brainstem masses: a risk-beneﬁt analysis in 71 patients. J Neurosurg 1995;82:976–81. 2. Russell JA, Shaw MD. Chronic abscess of the brain stem. J Neurol Neurosurg Psychiatry 1977;4:625–9. 3. Cardelle G, Picaza JA, Jordan JR. Brain abscess due to Hemophilus inﬂuenzae in an infant seven month old. Revista Cubana Pediatria 1950;22:9–19. 4. Mihancea N, Dobre M, Diaconescu C, et al. Acute rhinopharyngitis, acute interstitial pneumonia and parieto-frontal brain abscess with H. inﬂuenzae type B. Pneumoftiziologia 1992;41:56–7.
5. Feldman WE, Schwartz J. Haemophilus inﬂuenzae type b brain abscess complicating meningitis: case report. Pediatrics 1983;72:473–5. 6. Amlin KM. Inﬂuenzal meningitis: report of two cases, one complicated by brain abscess. Hawaii Med J 1946;6:98–100. 7. Hanna R. Cerebral abscess and paradoxic embolism associated with congenital heart review: report of seven cases with review of the literature. Am J Dis Child 1941;62:555–67. 8. Wait SD, Beres EJ, Nakaji P. Bacterial abscess of the medulla oblongata. J Clin Neurosci 2009;16:1082–4. 9. Sclar G, Boardman P, Prestigiacomo CJ. Solitary brainstem abscess. Neurology 2007;68:1231. 10. Müller-Richter UD, Bele S, Roldan JC, et al. The importance of dental-based treatment shown on the case report of a pontine abscess caused by Streptococcus viridans. Mund Kiefer Gesichtschir 2007;11:161–6. 11. Kniss MS, Sivakumar K. Solitary pyogenic abscess of the medulla oblongata: survival after aspiration and antibiotics. Neurology 2006;66:1836. 12. Suzer T, Coskun E, Cirak B, et al. Brain stem abscesses in childhood. Childs Nerv Syst 2005;21:27–31. 13. Moorthy RK, Rajshekhar V. Isolated ring-enhancing lesion of the brainstem in a patient with cyanotic heart disease: role of stereotactic intervention. Neurology India 2003;51:404–6. 14. Fuentes S, Bouillot P, Regis J, et al. Management of brain stem abscess. Br J Neurosurg 2001;15:57–62. 15. Sarma S, Sekhar LN. Brain-stem abscess successfully treated by microsurgical drainage: a case report. Neurol Res 2001;23:855–61. 16. Kagawa R, Okada Y, Shima T, et al. Neuroimaging ﬁndings of the development and resolution of solitary brainstem abscess: characteristics of neuroimagings in the early stage of brainstem abscess and importance of surgical management for brainstem abscess – case report. Neurol Med Chir 1999;39:621–4. 17. Kalarostaghi AH, Pell MF, Turner J, et al. Polymicrobial brain stem abscess due to Streptococcus anginosus and Actinomyces species. J Clin Neurosci 1999;6:415–8. 18. Nakajima H, Iwai Y, Yamanaka K, et al. Successful treatment of brainstem abscess with stereotactic aspiration. Surg Neurol 1999;52:445–8. 19. Bavetta S, Paterakis K, Srivatsa SR, et al. Brainstem abscess: preoperative MRI appearance and survival following stereotactic aspiration. J Neurosurg Sci 1996;40:139–43. 20. Imai H, Ono N, Zama A, et al. Diagnosis and treatment of brainstem abscess using magnetic resonance imaging and microsurgical aspiration – case report. Neurol Med Chir 1995;35:160–4. 21. Rajshekhar V, Chandy MJ. Successful stereotactic management of a large cardiogenic brain stem abscess. Neurosurgery 1994;34:368–71 discussion 371. 22. Wabi G. Brain stem abscess. Indian Pediatr 1993;30:547–8. 23. Jamjoom ZA. Solitary brainstem abscess successfully treated by microsurgical aspiration. Br J Neurosurg 1992;6:249–53. 24. Fujino H, Kobayashi T, Goto I, et al. Cure of a man with solitary abscess of the brain-stem. J Neurol 1990;237:265–6. 25. Prakash B, Mehta G, Gondal R, et al. Tuberculous abscess of the brain stem. Surg Neurol 1989;32:445–8. 26. Rossitch Jr E, Alexander 3rd E, Schiff SJ, et al. The use of computed tomographyguided stereotactic techniques in the treatment of brain stem abscesses. Clin Neurol Neurosurg 1988;90:365–8. 27. Kashiwagi S, Abiko S, Aoki H. Brainstem abscess. Surg Neurol 1987;28:63–6. 28. Nauta HJ, Contreras FL, Weiner RL, et al. Brain stem abscess managed with computed tomography-guided stereotactic aspiration. Neurosurgery 1987;20:476–80. 29. Dake MD, McMurdo SK, Rosenblum ML, et al. Pyogenic abscess of the medulla oblongata. Neurosurgery 1986;18:370–2. 30. Ramalho J, Castillo M. Case of the season: brainstem abscess. Semin Roentgenol 2008;43:168–70. 31. Arias M, Dapena D, Arias-Rivas S, et al. Widespread brainstem abscess resolved with medical therapy. Neurologia (Barcelona, Spain) 2007;22:118–21. 32. Payne SJ, Hickman SJ, Howard RS. Abscess of the medulla oblongata following endotracheal intubation. J Neurol Neurosurg Psychiatry 2006;77:563–4. 33. Bekar A, Kocaeli H, Yilmaz E, et al. Trigeminal neuralgia caused by a pontine abscess: case report. Neurosurgery 2004;55:1434. 34. Pandian JD, Moosa NV, Cherian PJ, et al. Brainstem abscess complicating tetralogy of Fallot successfully treated with antibiotics alone. Neurology India 2000;48:272–5. 35. Garcia-Monco JC, Gomez Beldarrain M, Fernandez Canton G, et al. Resolution of a brainstem abscess through antituberculous therapy. Neurology 1997;49:265–7. 36. Fulgham JR, Wijdicks EF, Wright AJ. Cure of a solitary brainstem abscess with antibiotic therapy: case report. Neurology 1996;46:1451–4. 37. Adachi J, Uki J, Kazumoto K, et al. Diagnosis of brainstem abscess in the cerebritis stage by magnetic resonance imaging – case report. Neurol Med Chir (Tokyo) 1995;35:467–70. 38. Carpenter JL. Brain stem abscesses: cure with medical therapy, case report, and review. Clin Infect Dis 1994;18:219–26. 39. Wang HS, Kuo MF, Huang SC. Medical cure of a brainstem abscess and serial brainstem auditory evoked potentials. Dev Med Child Neurol 1992;34: 911–5. 40. Ruelle A, Zerbi D, Zuccarello M, et al. Brain stem abscess treated successfully by medical therapy. Neurosurgery 1991;28:742–5 discussion 745–746.
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41. Outin HD, Merrer J, Molho M, et al. Solitary listerial abscess of the brain stem. Cure with antibiotic treatment. Revue neurologique 1989;145: 153–6. 42. Bertoldi RV, Sperling MR. Nocardia brain stem abscess: diagnosis and response to medical therapy. Bull Clin Neurosci 1984;49:99–104.
43. McVernon J, Trotter CL, Slack MP, et al. Trends in Haemophilus inﬂuenzae type b infections in adults in England and Wales: surveillance study. BMJ 2004;329:655–8. 44. Simon HB, Southwick FS, Moellering Jr RC, et al. Hemophilus inﬂuenzae in hospitalized adults: current perspectives. Am J Med 1980;69:219–26.
A patient with brucellar cervical spondylodiscitis complicated by epidural abscess _ Alper Eker a, Ilkay Uzunca b, Özlem Tansel c,⇑, Murat Birtane d a
Department of Infectious Diseases and Clinical Microbiology, Edirne State Hospital, 22030 Edirne, Turkey Department of Neurology, Edirne State Hospital, 22030 Edirne, Turkey c Department of Infectious Diseases and Clinical Microbiology, Medical Faculty of Trakya University, 22030 Edirne, Turkey d Department of Physical Medicine and Rehabilitation, Medical Faculty of Trakya Universtiy, 22030 Edirne, Turkey b
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Article history: Received 24 May 2010 Accepted 21 June 2010
Keywords: Brucellosis Cervical vertebrae Epidural abscess Spondylitis
a b s t r a c t Brucellar cervical spondylodiscitis and epidural abscess are serious medical conditions that can cause permanent neurological deﬁcits. Fortunately, they are rare. We report a 34-year-old male patient, complaining of fever and neck pain and stiffness, with increased deep tendon reﬂexes. A lumbar puncture was normal. Brucella species organisms were isolated from blood cultures, and the Rose-Bengal test and the standard tube agglutination (STA) test were positive. The diagnosis was made on MRI. The patient was treated with doxycycline and rifampin daily for 16 weeks. On day 51 of treatment, the patient had no symptoms and his physical and neurological examinations were normal. His repeat cervical MRI was almost normal. The STA test was negative at week 20. It is important to consider brucellar cervical spondylodiscitis with epidural abscess in endemic regions. Ó 2010 Elsevier Ltd. All rights reserved.
1. Introduction Brucellosis is a systemic infection caused by a facultative intracellular, non-encapsulated, non-motile, gram-negative coccobacillus. Brucellosis is a zoonosis, and can occur in all regions of the world.1,2 Brucella can infect many organs and tissues, and consequently, there are a variety of clinical manifestations of brucellosis. Osteoarticular disease is the most common complication of brucellosis. The spectrum of bone and joint lesions includes arthritis, bursitis, tenosynovitis, sacroiliitis, spondylitis, and osteomyelitis.2 Spinal epidural abscesses (SEA) due to Brucella are rare and develop from previous spondylitis. Abscesses tend to locate in the thoracic and lumbar areas of the spine, and less frequently, in the cervical spine.3 Vertebral involvement is the most difﬁcult form to diagnose and treat. We present a patient with brucellar cervical spondylodiscitis complicated by an epidural abscess. We discuss diagnosis and treatment of the condition in the light of previous reports. 2. Case report A 34 year-old male patient was admitted to our hospital complaining of a fever beginning 20 days prior that rose by the end of the day, as well as neck pain that increased with motion. His occupation was stockbreeding. He stated that he was a consumer of unpasteurized milk and milk products, and he had recently eaten cheese made of unpasteurized milk. During the clinical examination, his fever was measured as 38.3 °C. He had pain at cervical vertebrae 4/5/6 that increased with local palpation. He had neck stiffness, and his deep tendon reﬂexes were increased. On the date ⇑ Corresponding author. Tel.: + 90 284 235 76 41x4815, fax: + 90 284 235 39 33. E-mail address: [email protected]
of admission, his laboratory ﬁndings were: urea, 27 mg/dL; creatine, 0.9 mg/dL; aspartate aminotransferase, 72 U/L; alanine transaminase, 55 U/L; C-reactive protein, 4.7 mg/dL; erythrocyte sedimentation rate, 30 mm/hour; peripheral white blood cell count, 9570/mm3 (47% polymorphonuclear leukocytes, 47% lymphocytes, 6% monocytes). A lumbar puncture yielded colorless cerebrospinal ﬂuid (CSF) under normal pressure with; 57 mg/dL glucose (blood glucose value was 89 mg/dL) and 46 mg/dL protein. The lymphocyte count was 4/mm3 of CSF. Serological tests for Brucella and Ehrlich-Ziehl-Neelsen staining were negative. CSF cultures were negative for bacteria, fungi and bacilli. Brucella spp. was isolated from blood cultures (BacT/ALERTÒ 3D, bioMérieux, Marcy l’Etoile, France), the Rose-Bengal test was positive, and the standard tube agglutination (STA) test was 1/320. He was given 200 mg doxycycline and 600 mg rifampin daily. A cervical MRI detected narrowing of the C4/5 intervertebral disc space and subchondral congestion on the joint surfaces. T2weighted MRI revealed increased heterogeneous signal intensity, suggesting spondylodiscitis. A heterogeneous and septated ﬂuid collection with peripheral enhancement was identiﬁed in the ventral epidural area along the dorsal contours of the C4/5 vertebral bodies, consistent with an epidural abscess. There was marked spinal cord compression on the right. A heterogeneously contrast-enhanced mass that caused root compression was also observed at the same vertebra (Fig. 1). Within 48 hours of the start of antibiotic therapy, the patient’s fever decreased. Starting on day 7 of treatment, his neck pain subsided. On day 51 of treatment, he had no symptoms and his physical and neurological examinations were normal. On his repeat cervical MRI, there were ﬁndings related to the prior spondylodiscitis at the C4/5 level, and the abscess had almost completely regressed (Fig. 2). Treatment continued for 16 weeks. At week 20, the STA test was negative.