- Email: [email protected]
Brucella Case That Led to Atlantoaxial Spinal Instability in Childhood
Case Report
Brucella Case That Led to Atlantoaxial Spinal Instability in Childhood _ ¨ kten1, Yurdal Gezercan1, Anıl Atmis¸2, U ¨ mit C¸elik2 Ali Arslan1, Go¨khan C¸avus¸1, Emre Bilgin1, Ali Ihsan O
Key words Atlantoaxial - Brucella - Pediatrics - Spine - Surgical treatment - Tuberculosis -
From the Departments of 1Neurosurgery and 2Pediatric Infectious Diseases and Neurology, Adana City Training and Research Hospital, Adana, Turkey To whom correspondence should be addressed: Gökhan Çavusx, M.D. [E-mail: [email protected]] Citation: World Neurosurg. (2019) 131:108-111. https://doi.org/10.1016/j.wneu.2019.07.089 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
INTRODUCTION Spinal instability is defined as “inability of the spine to maintain the abnormal displacement pattern under the physiologic load and, as a result of this, the loss of ability to prevent neurologic deficit, major deformity, and pain.”1 Degenerative, traumatic, infectious, and iatrogenic causes of the spine may lead to spinal instability. The rate of infections causing instability are low, particularly in childhood. In the pediatric age group, atlantoaxial dislocation or subluxation related to infection is usually reported after retropharyngeal abscess, pharyngitis, nasopharyngitis, adenotonsillitis, tonsillar abscess, parotitis, cervical abscess, and otitis media; however, these problems are rarely seen.2-4 Tuberculosis and brucellosis are the primary granulomatous diseases with infection origin that involve the spine. In brucellosis cases, early surgical decompression and stabilization are advised in the presence of spinal vertebra involvement, spinal cord compression, and instability.5,6 One case with atlantoaxial subluxation was reported in the literature and was treated medically.2 Our case is the first one in the literature in which
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- BACKGROUND:
Atlantoaxial subluxation caused by Brucella infection is rarely seen in children.
- CASE
DESCRIPTION: C1-C2 dislocation, erosion in the odontoid bone, and compression to the brainstem were detected on cervical computed tomography and magnetic resonance imaging in a 6-year-old patient who suffered neck pain, deviation to the right in the neck, fever, and pain in his extremities. He was hospitalized in the Clinic of Pediatric Infectious Diseases. Positive Brucella agglutination tests were obtained, so C1-C2 stabilization and fusion were performed. Antibrucellosis antibiotic treatment was administered for 6 months. It was suggested that the dislocation was related to odontoid erosion and laxity of the atlantoaxial ligamentous structures during Brucella infection.
- CONCLUSIONS:
Atlantoaxial dislocation and instability develops secondarily to paravertebral abscesses, only rarely. Like brucellosis of childhood, granulomatous infectious diseases rarely cause atlantoaxial subluxation or dislocation. Torticollis and neck pains should be taken seriously for the purpose of early diagnosis of patients at risk in endemic regions. Stabilization and fusion should be performed when instability is detected, and these patients should be assessed with both pediatric infectious diseases and neurosurgery clinics. Our case is the first one in the literature in which atlantoaxial instability developed due to Brucella infection and stabilization was performed.
atlantoaxial dislocation developed in relation to brucellosis infection and in which posterior C1-C2 stabilization was achieved via the Harms technique.7 CASE REPORT A 6-year-old male patient was examined in a pediatric clinic at another center 1 and a half month ago due to complaints of intermittent fever, pain in the neck, deviation to the right in the neck, and restriction in motion. Because sedimentation was found to be high (300 mm/hour), brucellosis 1/320 and latex agglutination were detected as positive (þ) in serologic tests. Antibrucellosis treatment (rifampicin as 300 mg/day, cotrimethoxazole as 200 mg/day) was begun. He received brucellosis treatment for 21 days. He was forwarded to the pediatric clinic at our hospital for advanced diagnosis and treatment. Because sedimentation was found to be high (80 mm/ hour) and brucellosis 1/320 and latex
agglutination were detected as positive (þ) in serologic tests at the pediatric infectious disease clinic, triple antibrucellosis treatment (aminoglycoside as 40 mg/day, rifampicin 300 mg/day, cotrimethoxazole 200 mg/day) was initiated for the patient. Because erosion in the C2 odontoid bone, C1-2 dislocation, and compression to the medulla were detected on cervical computed tomography and magnetic resonance imaging, the patient was taken to our clinic (Figure 1). The neurologic examination of the patient was assessed as normal. Torticollis in which the head deviated toward the right was detected. The upper cervical instability of the patient was confirmed, and surgical intervention was planned. C1-2 stabilization with the Harms technique with the posterior approach and bone fusion were performed (Figure 2). An intraoperative neuromonitor was used during surgery. A pediatric polyaxial _ screw (Piron Medical, Izmir, Turkey) was used during the surgery. An autograft
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.07.089
CASE REPORT CHILD'S SPINAL INSTABILITY FROM BRUCELLA
ALI ARSLAN ET AL.
Figure 1. (A) Preoperative cervical computed tomography (CT) sagittal section. (B) Preoperative 3-dimensional cervical CT. (C) Preoperative T2 cervical magnetic resonance imaging sagittal section.
was taken from the occipital bone outer tabula with the osteotome technique, and 10 cc cancellous allograft (Matchstick, ilium) were used for bone fusion. The postoperative neurologic examination was assessed as normal. Brucellosis treatment (rifampicin 300 mg/day, cotrimethoxazole 200 mg/day) was continued for 6 months at the advice of the pediatric infectious diseases clinic. At the 6-month follow-up, the patient’s neurologic examination was normal, sedimentation was 6 mm/hour, and brucellosis was detected as 1/80. In the follow-up 24 months later, there was no
problem in stabilization and the bone fusion seemed to be ensured (Figures 3 and 4).
DISCUSSION Brucellosis is found worldwide. Although it has been eradicated in many European countries and North America, it is an important public health problem, particularly in the Mediterranean region containing Turkey, Iran, Arabian Peninsula, India, Mexico, and some regions of Middle and South America.8-12
Figure 2. Postoperative cervical computed tomography of (A) sagittal, (B) axial, and (C) postoperative T2 cervical magnetic resonance imaging sagittal sections.
WORLD NEUROSURGERY 131: 108-111, NOVEMBER 2019
Brucellosis is a systemic zoonotic disease that can affect many organ systems caused by the aerobic, gram-negative, coccobacillus that cannot be encapsulated.6 Brucella abortus, Brucella suis, Brucella canis, and Brucella melitensis may cause human diseases. B. melitensis is the most lethal and commonly seen one among all types.9 Natural reservoirs of brucellosis are domestic animals, such as sheep, goats, cattle, camels, dogs, and swine. Humans are infected via the direct contact of infected animals or materials and by eating the animal products, particularly raw milk and cheese.10,12-15 It is a systemic disease in which several tissues and organs related to the reticuloendothelial and muscular system can be affected. The diagnosis and treatment of vertebral involvement are the most difficult of all musculoskeletal system diseases.2,16-20 Gonzales-Gay et al21 have reported that spondylitis is the most commonly seen evidence. They discovered spondylitis in 126 of 158 patients. Among these, spondylitis cases have been rarely implicated with spinal epidural abscess or paraspinal purulent mass, and they have usually been seen in the lumbar region. Osteomyelitis is revealed in the vertebra, particularly in the lumbosacral region. Osteoarticular involvement is the most commonly seen complication and has been reported between 10% and 85% in many series.13-16,22 In terms of spinal involvement frequency, it is ranked as lumbar, thoracal, and cervical.14,15,23 Cervical brucellosis is seen less than other localizations. Colmenero22 reported a 7.3% cervical localization in 105, cases and Colmenero et al24 found 3% for cervical localization in another study. In the literature, cervical brucellosis has rarely been reported, and a brucellosis case causing atlantoaxial subluxation has been encountered only once in the literature.2 Atlantoaxial subluxation and/or dislocation is usually seen in children due to otitis media, pharyngitis, viral infections, retropharyngeal abscess, tonsillectomy, pharyngoplasty, retropharyngeal inflammation, and trauma, and sometimes the etiologies are unknown.2-4 Three theories have been suggested to describe how atlantoaxial subluxation occurs because of pharyngeal inflammation
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CASE REPORT CHILD'S SPINAL INSTABILITY FROM BRUCELLA
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Figure 3. Postoperative cervical computed tomography acquired at the 24th month: sagittal (A) and axial (B) sections.
or infection.25,26 According to the first theory, metastatic effusion originating from an inflammatory procedure in the head and neck may cause subluxation in the joints. According to the second theory, a muscle spasm caused by inflammation may lead to atlantoaxial subluxation. According to the third theory, regional hyperemia associated with the inflammatory process may lead to decalcification of the spine, which causes ligamentous avulsion and then subluxation. Serkan et al2 suggest that the inflammatory process along the upper cervical region causes effusion in the atlantoaxial joint and that this leads to loosening of the transverse and lateral ligaments, which causes subluxation. Our opinion about this case is that the weakening of ligaments was due to inflammation and
development of subluxation, parallel to the opinion of Serkan et al.2 The clinical signs of cervical brucellosis abscess are spinal pain, local sensitivity, and fever.2,8,18-21 There is no specific symptomatology or clinical sign of the spinal form of brucellosis; thus a higher suspicion index is required for correct and early diagnosis.27 Apart from the systemic findings of the brucellosis infection, neck pain and torticollis are the most important clinic complaints and findings in patients with subluxation. The diagnosis of the brucellosis is usually established with positive blood and serologic tests. Treatment of these patients is usually medical, but there is an indication for surgical treatment in the presence of the spinal paravertebral abscess, potential neurologic deficit, and
instability.8,23 In spinal infections, there are 2 most important indications for surgical intervention: potential neurologic deficit risk and mechanic instability.23,28 In brucellosis cases, early surgical decompression and stabilization are made in the presence of spinal vertebra involvement, spinal cord compression, and instability.5 Long-term brucellosisspecific antibiotic treatment should be given to these patients at the same time.2,9,13 In our case, there was pain in the neck, deviation in the neck toward the right, and restriction in motion apart from the systemic findings of the brucellosis. Because C1-C2 subluxation and odontoid erosion compression to the brainstem related to instability were detected on cervical computed tomography and magnetic resonance imaging, surgical intervention indication was established and C1-C2 stabilization was done via the Harms technique. Brucellosis-specific antibiotic treatment was administrated to the patient for 6 months. In the control made 6 months later, sedimentation was detected as 6 mm/hour and a brucellosis agglutination test was detected as 1/80. Bone fusion was detected in the control made 24 months later. One case that presented with acute torticollis associated with brucellosis has been reported in the literature, and it was also treated medically. Our case is the first case in the literature that gives manifestation torticollis with late diagnosis and treatment in which we detected atlantoaxial subluxation and instability, and thus we performed C1-C2 stabilization via the Harms technique. CONCLUSION
Figure 4. Postoperative 3-dimensional cervical computed tomography acquired at the 24th month: sagittal (A) and posterior (B) views.
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Granulomatous diseases of childhood such as brucellosis may give manifestations with several complaints and signs. Atlantoaxial subluxation or instability should be suggested, particularly in patients with torticollis in terms of endemic regions in terms of the risk factors. Pediatric patients with these complaints should be assessed in a neurosurgery clinic with assistance from a pediatric infectious diseases clinic. The most important issue is that a delay in diagnosis and treatment may lead to instability. We suggest that if there is instability in the spinal brucellosis, fusion results will be
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.07.089
CASE REPORT CHILD'S SPINAL INSTABILITY FROM BRUCELLA
ALI ARSLAN ET AL.
almost perfect with the necessary and sufficient instrumentation. This will ensure complete stabilization and longterm medical treatment in conjunction with antibiotherapy. REFERENCES 1. Pope MH, Panjabi M. Biomechanical definitions of spinal instability. Spine. 1985;10:255-256. 2. Simsek S, Yigitkanli K, Kazanci A, Belen D, Bavbek M. Medically treated paravertebral Brucella abscess presenting with acute torticollis: case report. Surg Neurol. 2007;67:207-210. 3. Fielding JW, Francis WR, Hawkins RJ, Henisinger RN. Atlantoaxial rotary deformities. Semin Spine Surg. 1991;3:33-38. 4. Welinder NR, Hoffmann P, Håkansson S. Pathogenesis of non-traumatic atlanto-axial subluxation (Grisel’s syndrome). Eur Arch Otorhinolaryngol. 1997;254:251-254. 5. Tezer M, Ozturk C, Aydogan M, Camurdan K, Erturer E, Hamzaoglu A. Noncontiguous dual segment thoracic brucellosis with neurological deficit. Spine J. 2006;6:321-324. 6. Hantzidis P, Papadopoulos A, Kalabakos C, Boursinos L, Dimitriou CG. Brucella cervical spondylitis complicated by spinal cord compression: a case report. Cases J. 2009;2:6698. 7. Harms J, Melcher RP. Posterior C1-C2 fusion with polyaxial screw and rod fixation. Spine (Phila Pa 1976). 2001;26:2467-2471. 8. Mandell GL, Bennett JE, Dolin R. Principles and Practice of Infectious Disease. Philadelphia, PA: Churchill Livingstone; 2000:2669-2674.
11. Hasanjani Roushan MR, Mohrez M, Smailnejad Gangi SM, Soleimani Amiri MJ, Hajiahmadi M. Epidemiological features and clinical manifestations in 469 adult patients with brucellosis in Babol, Northern Iran. Epidemiol Infect. 2004;132: 1109-1114. 12. Buzgan T, Karahocagil MK, Irmak H, et al. Clinical manifestations and complications in 1028 cases of brucellosis: a retrospective evaluation and review of the literature. Int J Infect Dis. 2010;14: e469-e478. 13. Alp E, Doganay M. Current therapeutic strategy in spinal brucellosis. Int J Infect Dis. 2008;12:573-577. 14. Chelli Bouaziz M, Ladeb MF, Chakroun M, Chaabane S. Spinal brucellosis: a review. Skel Radiol. 2008;37:785-790. 15. Pourbagher A, Pourbagher MA, Savas L, et al. Epidemiologic, clinical, and imaging findings in brucellosis patients with osteoarticular involvement. Am J Roentgenol. 2006;187:873-880. 16. Tekkök IH, Berker M, Özcan OE, et al. Brucellosis of the spine. Thinking. 1993;33:838. 17. Gür A, Geyik MF, Dikici B, Nas K, Cevik R, Sarac JHS. Complications of brucellosis in different age groups: a study of 283 cases in southeastern Anatolia of Turkey. Yonsei Med J. 2003:33-44. 18. Basaranoglu M, Mert A, Tabak F, Kanberoglu KAY. A case of cervical Brucella spondylitis with paravertebral abscess and neurological deficits. Scand J Infect Dis. 1999;31:214-215. 19. Namiduru M, Karaoglan I, Gursoy S, Bayazit N, Sirikci A. Brucellosis of the spine: Evaluation of the clinical, laboratory, and radiological findings of 14 patients. Rheumatol Int. 2004;24:125-129.
22. Colmenero JD, Reguera JM, Fernandeznebro A, et al. Osteoarticular complications of brucellosis. Ann Rheum Dis. 1991;50:23-26. 23. Ekici MA, Özbek Z, Gökoǧlu A, Menkü A. Surgical management of cervical spinal epidural abscess caused by Brucella melitensis: report of two cases and review of the literature. J Kor Neurosurg Soc. 2012;51: 383-387. 24. Colmenero JD, Jiménez-Mejías ME, SánchezLora FJ, et al. Pyogenic, tuberculous, and brucellar vertebral osteomyelitis: a descriptive and comparative study of 219 cases. Ann Rheum Dis. 1997;56:709-715. 25. Dhaon BK, Jaiswal A, Nigam V, Jain V. Atlantoaxial rotatory fixation secondary to tuberculosis of occiput: a case report. Spine (Phila Pa 1976). 2003;28:E203-E205. 26. Morita T, Fujiki N, Shiomi Y, Kurata K, Oda N. Atlantoaxial subluxation in an adult secondary to retropharyngeal abscess. Auris Nasus Larynx. 2001; 28:177-179. 27. Nas K, Tasdemir N, Cakmak E, Kemaloglu MS, Bukte Y, Geyik MF. Cervical intramedullary granuloma of Brucella: a case report and review of the literature. Eur Spine J. 2007;16(suppl 3):255-259. 28. Lee MC, Wang MY, Fessler RG, Liauw J, Kim DH. Instrumentation in patients with spinal infection. Neurosurg Focus. 2004;17:E7.
Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 7 May 2019; accepted 9 July 2019
9. Bingol A, Yucemen N, Meco O. Medically treated intraspinal/Brucella/granuloma. Surg Neurol. 1999; 52:570-576.
20. Pina MA, Modrego PJ, Uroz JJ, Cobeta JC, Lerin FJ, Baiges JJ. Brucellar spinal epidural abscess of cervical location: report of four cases. Eur Neurol. 2001;45:249-253.
10. Geyik MF, Gür A, Nas K, et al. Musculoskeletal involvement in brucellosis in different age groups: a study of 195 cases. Swiss Med Wkly. 2002;132: 98-104.
21. González-Gay MA, García-Porrúa C, Ibañez DGPM. Osteoarticular complications of brucellosis in an Atlantic area of Spain. J Rheumatol. 1999;26: 141-145.
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Citation: World Neurosurg. (2019) 131:108-111. https://doi.org/10.1016/j.wneu.2019.07.089 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
www.journals.elsevier.com/world-neurosurgery
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Brucella Case That Led to Atlantoaxial Spinal Instability in Childhood _ ¨ kten1, Yurdal Gezercan1, Anıl Atmis¸2, U ¨ mit C¸elik2 Ali Arslan1, Go¨khan C¸avus¸1, Emre Bilgin1, Ali Ihsan O
Key words Atlantoaxial - Brucella - Pediatrics - Spine - Surgical treatment - Tuberculosis -
From the Departments of 1Neurosurgery and 2Pediatric Infectious Diseases and Neurology, Adana City Training and Research Hospital, Adana, Turkey To whom correspondence should be addressed: Gökhan Çavusx, M.D. [E-mail: [email protected]] Citation: World Neurosurg. (2019) 131:108-111. https://doi.org/10.1016/j.wneu.2019.07.089 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
INTRODUCTION Spinal instability is defined as “inability of the spine to maintain the abnormal displacement pattern under the physiologic load and, as a result of this, the loss of ability to prevent neurologic deficit, major deformity, and pain.”1 Degenerative, traumatic, infectious, and iatrogenic causes of the spine may lead to spinal instability. The rate of infections causing instability are low, particularly in childhood. In the pediatric age group, atlantoaxial dislocation or subluxation related to infection is usually reported after retropharyngeal abscess, pharyngitis, nasopharyngitis, adenotonsillitis, tonsillar abscess, parotitis, cervical abscess, and otitis media; however, these problems are rarely seen.2-4 Tuberculosis and brucellosis are the primary granulomatous diseases with infection origin that involve the spine. In brucellosis cases, early surgical decompression and stabilization are advised in the presence of spinal vertebra involvement, spinal cord compression, and instability.5,6 One case with atlantoaxial subluxation was reported in the literature and was treated medically.2 Our case is the first one in the literature in which
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- BACKGROUND:
Atlantoaxial subluxation caused by Brucella infection is rarely seen in children.
- CASE
DESCRIPTION: C1-C2 dislocation, erosion in the odontoid bone, and compression to the brainstem were detected on cervical computed tomography and magnetic resonance imaging in a 6-year-old patient who suffered neck pain, deviation to the right in the neck, fever, and pain in his extremities. He was hospitalized in the Clinic of Pediatric Infectious Diseases. Positive Brucella agglutination tests were obtained, so C1-C2 stabilization and fusion were performed. Antibrucellosis antibiotic treatment was administered for 6 months. It was suggested that the dislocation was related to odontoid erosion and laxity of the atlantoaxial ligamentous structures during Brucella infection.
- CONCLUSIONS:
Atlantoaxial dislocation and instability develops secondarily to paravertebral abscesses, only rarely. Like brucellosis of childhood, granulomatous infectious diseases rarely cause atlantoaxial subluxation or dislocation. Torticollis and neck pains should be taken seriously for the purpose of early diagnosis of patients at risk in endemic regions. Stabilization and fusion should be performed when instability is detected, and these patients should be assessed with both pediatric infectious diseases and neurosurgery clinics. Our case is the first one in the literature in which atlantoaxial instability developed due to Brucella infection and stabilization was performed.
atlantoaxial dislocation developed in relation to brucellosis infection and in which posterior C1-C2 stabilization was achieved via the Harms technique.7 CASE REPORT A 6-year-old male patient was examined in a pediatric clinic at another center 1 and a half month ago due to complaints of intermittent fever, pain in the neck, deviation to the right in the neck, and restriction in motion. Because sedimentation was found to be high (300 mm/hour), brucellosis 1/320 and latex agglutination were detected as positive (þ) in serologic tests. Antibrucellosis treatment (rifampicin as 300 mg/day, cotrimethoxazole as 200 mg/day) was begun. He received brucellosis treatment for 21 days. He was forwarded to the pediatric clinic at our hospital for advanced diagnosis and treatment. Because sedimentation was found to be high (80 mm/ hour) and brucellosis 1/320 and latex
agglutination were detected as positive (þ) in serologic tests at the pediatric infectious disease clinic, triple antibrucellosis treatment (aminoglycoside as 40 mg/day, rifampicin 300 mg/day, cotrimethoxazole 200 mg/day) was initiated for the patient. Because erosion in the C2 odontoid bone, C1-2 dislocation, and compression to the medulla were detected on cervical computed tomography and magnetic resonance imaging, the patient was taken to our clinic (Figure 1). The neurologic examination of the patient was assessed as normal. Torticollis in which the head deviated toward the right was detected. The upper cervical instability of the patient was confirmed, and surgical intervention was planned. C1-2 stabilization with the Harms technique with the posterior approach and bone fusion were performed (Figure 2). An intraoperative neuromonitor was used during surgery. A pediatric polyaxial _ screw (Piron Medical, Izmir, Turkey) was used during the surgery. An autograft
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.07.089
CASE REPORT CHILD'S SPINAL INSTABILITY FROM BRUCELLA
ALI ARSLAN ET AL.
Figure 1. (A) Preoperative cervical computed tomography (CT) sagittal section. (B) Preoperative 3-dimensional cervical CT. (C) Preoperative T2 cervical magnetic resonance imaging sagittal section.
was taken from the occipital bone outer tabula with the osteotome technique, and 10 cc cancellous allograft (Matchstick, ilium) were used for bone fusion. The postoperative neurologic examination was assessed as normal. Brucellosis treatment (rifampicin 300 mg/day, cotrimethoxazole 200 mg/day) was continued for 6 months at the advice of the pediatric infectious diseases clinic. At the 6-month follow-up, the patient’s neurologic examination was normal, sedimentation was 6 mm/hour, and brucellosis was detected as 1/80. In the follow-up 24 months later, there was no
problem in stabilization and the bone fusion seemed to be ensured (Figures 3 and 4).
DISCUSSION Brucellosis is found worldwide. Although it has been eradicated in many European countries and North America, it is an important public health problem, particularly in the Mediterranean region containing Turkey, Iran, Arabian Peninsula, India, Mexico, and some regions of Middle and South America.8-12
Figure 2. Postoperative cervical computed tomography of (A) sagittal, (B) axial, and (C) postoperative T2 cervical magnetic resonance imaging sagittal sections.
WORLD NEUROSURGERY 131: 108-111, NOVEMBER 2019
Brucellosis is a systemic zoonotic disease that can affect many organ systems caused by the aerobic, gram-negative, coccobacillus that cannot be encapsulated.6 Brucella abortus, Brucella suis, Brucella canis, and Brucella melitensis may cause human diseases. B. melitensis is the most lethal and commonly seen one among all types.9 Natural reservoirs of brucellosis are domestic animals, such as sheep, goats, cattle, camels, dogs, and swine. Humans are infected via the direct contact of infected animals or materials and by eating the animal products, particularly raw milk and cheese.10,12-15 It is a systemic disease in which several tissues and organs related to the reticuloendothelial and muscular system can be affected. The diagnosis and treatment of vertebral involvement are the most difficult of all musculoskeletal system diseases.2,16-20 Gonzales-Gay et al21 have reported that spondylitis is the most commonly seen evidence. They discovered spondylitis in 126 of 158 patients. Among these, spondylitis cases have been rarely implicated with spinal epidural abscess or paraspinal purulent mass, and they have usually been seen in the lumbar region. Osteomyelitis is revealed in the vertebra, particularly in the lumbosacral region. Osteoarticular involvement is the most commonly seen complication and has been reported between 10% and 85% in many series.13-16,22 In terms of spinal involvement frequency, it is ranked as lumbar, thoracal, and cervical.14,15,23 Cervical brucellosis is seen less than other localizations. Colmenero22 reported a 7.3% cervical localization in 105, cases and Colmenero et al24 found 3% for cervical localization in another study. In the literature, cervical brucellosis has rarely been reported, and a brucellosis case causing atlantoaxial subluxation has been encountered only once in the literature.2 Atlantoaxial subluxation and/or dislocation is usually seen in children due to otitis media, pharyngitis, viral infections, retropharyngeal abscess, tonsillectomy, pharyngoplasty, retropharyngeal inflammation, and trauma, and sometimes the etiologies are unknown.2-4 Three theories have been suggested to describe how atlantoaxial subluxation occurs because of pharyngeal inflammation
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CASE REPORT CHILD'S SPINAL INSTABILITY FROM BRUCELLA
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Figure 3. Postoperative cervical computed tomography acquired at the 24th month: sagittal (A) and axial (B) sections.
or infection.25,26 According to the first theory, metastatic effusion originating from an inflammatory procedure in the head and neck may cause subluxation in the joints. According to the second theory, a muscle spasm caused by inflammation may lead to atlantoaxial subluxation. According to the third theory, regional hyperemia associated with the inflammatory process may lead to decalcification of the spine, which causes ligamentous avulsion and then subluxation. Serkan et al2 suggest that the inflammatory process along the upper cervical region causes effusion in the atlantoaxial joint and that this leads to loosening of the transverse and lateral ligaments, which causes subluxation. Our opinion about this case is that the weakening of ligaments was due to inflammation and
development of subluxation, parallel to the opinion of Serkan et al.2 The clinical signs of cervical brucellosis abscess are spinal pain, local sensitivity, and fever.2,8,18-21 There is no specific symptomatology or clinical sign of the spinal form of brucellosis; thus a higher suspicion index is required for correct and early diagnosis.27 Apart from the systemic findings of the brucellosis infection, neck pain and torticollis are the most important clinic complaints and findings in patients with subluxation. The diagnosis of the brucellosis is usually established with positive blood and serologic tests. Treatment of these patients is usually medical, but there is an indication for surgical treatment in the presence of the spinal paravertebral abscess, potential neurologic deficit, and
instability.8,23 In spinal infections, there are 2 most important indications for surgical intervention: potential neurologic deficit risk and mechanic instability.23,28 In brucellosis cases, early surgical decompression and stabilization are made in the presence of spinal vertebra involvement, spinal cord compression, and instability.5 Long-term brucellosisspecific antibiotic treatment should be given to these patients at the same time.2,9,13 In our case, there was pain in the neck, deviation in the neck toward the right, and restriction in motion apart from the systemic findings of the brucellosis. Because C1-C2 subluxation and odontoid erosion compression to the brainstem related to instability were detected on cervical computed tomography and magnetic resonance imaging, surgical intervention indication was established and C1-C2 stabilization was done via the Harms technique. Brucellosis-specific antibiotic treatment was administrated to the patient for 6 months. In the control made 6 months later, sedimentation was detected as 6 mm/hour and a brucellosis agglutination test was detected as 1/80. Bone fusion was detected in the control made 24 months later. One case that presented with acute torticollis associated with brucellosis has been reported in the literature, and it was also treated medically. Our case is the first case in the literature that gives manifestation torticollis with late diagnosis and treatment in which we detected atlantoaxial subluxation and instability, and thus we performed C1-C2 stabilization via the Harms technique. CONCLUSION
Figure 4. Postoperative 3-dimensional cervical computed tomography acquired at the 24th month: sagittal (A) and posterior (B) views.
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Granulomatous diseases of childhood such as brucellosis may give manifestations with several complaints and signs. Atlantoaxial subluxation or instability should be suggested, particularly in patients with torticollis in terms of endemic regions in terms of the risk factors. Pediatric patients with these complaints should be assessed in a neurosurgery clinic with assistance from a pediatric infectious diseases clinic. The most important issue is that a delay in diagnosis and treatment may lead to instability. We suggest that if there is instability in the spinal brucellosis, fusion results will be
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.07.089
CASE REPORT CHILD'S SPINAL INSTABILITY FROM BRUCELLA
ALI ARSLAN ET AL.
almost perfect with the necessary and sufficient instrumentation. This will ensure complete stabilization and longterm medical treatment in conjunction with antibiotherapy. REFERENCES 1. Pope MH, Panjabi M. Biomechanical definitions of spinal instability. Spine. 1985;10:255-256. 2. Simsek S, Yigitkanli K, Kazanci A, Belen D, Bavbek M. Medically treated paravertebral Brucella abscess presenting with acute torticollis: case report. Surg Neurol. 2007;67:207-210. 3. Fielding JW, Francis WR, Hawkins RJ, Henisinger RN. Atlantoaxial rotary deformities. Semin Spine Surg. 1991;3:33-38. 4. Welinder NR, Hoffmann P, Håkansson S. Pathogenesis of non-traumatic atlanto-axial subluxation (Grisel’s syndrome). Eur Arch Otorhinolaryngol. 1997;254:251-254. 5. Tezer M, Ozturk C, Aydogan M, Camurdan K, Erturer E, Hamzaoglu A. Noncontiguous dual segment thoracic brucellosis with neurological deficit. Spine J. 2006;6:321-324. 6. Hantzidis P, Papadopoulos A, Kalabakos C, Boursinos L, Dimitriou CG. Brucella cervical spondylitis complicated by spinal cord compression: a case report. Cases J. 2009;2:6698. 7. Harms J, Melcher RP. Posterior C1-C2 fusion with polyaxial screw and rod fixation. Spine (Phila Pa 1976). 2001;26:2467-2471. 8. Mandell GL, Bennett JE, Dolin R. Principles and Practice of Infectious Disease. Philadelphia, PA: Churchill Livingstone; 2000:2669-2674.
11. Hasanjani Roushan MR, Mohrez M, Smailnejad Gangi SM, Soleimani Amiri MJ, Hajiahmadi M. Epidemiological features and clinical manifestations in 469 adult patients with brucellosis in Babol, Northern Iran. Epidemiol Infect. 2004;132: 1109-1114. 12. Buzgan T, Karahocagil MK, Irmak H, et al. Clinical manifestations and complications in 1028 cases of brucellosis: a retrospective evaluation and review of the literature. Int J Infect Dis. 2010;14: e469-e478. 13. Alp E, Doganay M. Current therapeutic strategy in spinal brucellosis. Int J Infect Dis. 2008;12:573-577. 14. Chelli Bouaziz M, Ladeb MF, Chakroun M, Chaabane S. Spinal brucellosis: a review. Skel Radiol. 2008;37:785-790. 15. Pourbagher A, Pourbagher MA, Savas L, et al. Epidemiologic, clinical, and imaging findings in brucellosis patients with osteoarticular involvement. Am J Roentgenol. 2006;187:873-880. 16. Tekkök IH, Berker M, Özcan OE, et al. Brucellosis of the spine. Thinking. 1993;33:838. 17. Gür A, Geyik MF, Dikici B, Nas K, Cevik R, Sarac JHS. Complications of brucellosis in different age groups: a study of 283 cases in southeastern Anatolia of Turkey. Yonsei Med J. 2003:33-44. 18. Basaranoglu M, Mert A, Tabak F, Kanberoglu KAY. A case of cervical Brucella spondylitis with paravertebral abscess and neurological deficits. Scand J Infect Dis. 1999;31:214-215. 19. Namiduru M, Karaoglan I, Gursoy S, Bayazit N, Sirikci A. Brucellosis of the spine: Evaluation of the clinical, laboratory, and radiological findings of 14 patients. Rheumatol Int. 2004;24:125-129.
22. Colmenero JD, Reguera JM, Fernandeznebro A, et al. Osteoarticular complications of brucellosis. Ann Rheum Dis. 1991;50:23-26. 23. Ekici MA, Özbek Z, Gökoǧlu A, Menkü A. Surgical management of cervical spinal epidural abscess caused by Brucella melitensis: report of two cases and review of the literature. J Kor Neurosurg Soc. 2012;51: 383-387. 24. Colmenero JD, Jiménez-Mejías ME, SánchezLora FJ, et al. Pyogenic, tuberculous, and brucellar vertebral osteomyelitis: a descriptive and comparative study of 219 cases. Ann Rheum Dis. 1997;56:709-715. 25. Dhaon BK, Jaiswal A, Nigam V, Jain V. Atlantoaxial rotatory fixation secondary to tuberculosis of occiput: a case report. Spine (Phila Pa 1976). 2003;28:E203-E205. 26. Morita T, Fujiki N, Shiomi Y, Kurata K, Oda N. Atlantoaxial subluxation in an adult secondary to retropharyngeal abscess. Auris Nasus Larynx. 2001; 28:177-179. 27. Nas K, Tasdemir N, Cakmak E, Kemaloglu MS, Bukte Y, Geyik MF. Cervical intramedullary granuloma of Brucella: a case report and review of the literature. Eur Spine J. 2007;16(suppl 3):255-259. 28. Lee MC, Wang MY, Fessler RG, Liauw J, Kim DH. Instrumentation in patients with spinal infection. Neurosurg Focus. 2004;17:E7.
Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 7 May 2019; accepted 9 July 2019
9. Bingol A, Yucemen N, Meco O. Medically treated intraspinal/Brucella/granuloma. Surg Neurol. 1999; 52:570-576.
20. Pina MA, Modrego PJ, Uroz JJ, Cobeta JC, Lerin FJ, Baiges JJ. Brucellar spinal epidural abscess of cervical location: report of four cases. Eur Neurol. 2001;45:249-253.
10. Geyik MF, Gür A, Nas K, et al. Musculoskeletal involvement in brucellosis in different age groups: a study of 195 cases. Swiss Med Wkly. 2002;132: 98-104.
21. González-Gay MA, García-Porrúa C, Ibañez DGPM. Osteoarticular complications of brucellosis in an Atlantic area of Spain. J Rheumatol. 1999;26: 141-145.
WORLD NEUROSURGERY 131: 108-111, NOVEMBER 2019
Citation: World Neurosurg. (2019) 131:108-111. https://doi.org/10.1016/j.wneu.2019.07.089 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
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