In neurologically intact patients with isolated tuberculosis of the posterior spinal elements, surgical intervention can be of benefit in select patients by reducing instability, deformity and late complications

Al-Khudairi, N.

Meir, A.

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Case ReportIsolated tuberculosis of the posterior spinal elements:case report and discussion of managementNaji Al-Khudairi and Adam MeirVictor Horsley Department of Neurosurgery, The National Hospital for Neurology and Neurosurgery, University College London,London WC1N 3BG, UKCorresponding author: Adam Meir. Email: adam.meir@uclh.nhs.ukLessonIn neurologically intact patients with isolated tuberculosisof the posterior spinal elements, surgical intervention canbe of benefit in select patients by reducing instability,deformity and late complications.KeywordsSpinal tuberculosis, posterior elements, atypicaltuberculosisIntroductionTuberculosis (TB) has been described as an ancientinfectious disease, with evidence being discovered incenturies-old skeletal remains.1 In recent decades,there has been a signiﬁcant resurgence of TB, caus-ing 2–3 million deaths annually worldwide.2According to the Health Protection AgencyEnhanced TB Surveillance Report, in 2011, theincidence of active TB in the UK was 14.4 per100,000 population.3Spinal TB is a relatively uncommon condition;although its true incidence is uncertain, it isthought to occur in 1.7% of the population world-wide.4 It is a disease of young active adults withconcomitant issues of deformity, and hence has aneconomic inﬂuence. It typically presents with backpain and/or neurological deﬁcit and deformity. Theinfection begins in the vertebral body and spreadsto adjacent vertebra by extending beneath theanterior longitudinal ligament.5 Involvement of theposterior elements is considered rare, particularly inisolation, infection is usually localised to the verte-bral body. In spinal TB patients, the incidenceof isolated posterior elements disease is between2 and 3%.6,7There is great controversy as to the optimal man-agement of spinal TB. Currently, surgical treatment isgenerally only indicated in cases of instability,deformity or neurological deﬁcit.8 As such, ambula-tory chemotherapy remains the mainstay of treat-ment for spinal TB.9Despite this, we report a case of isolated TB of theposterior spinal elements in a neurologically intactpatient, on whom surgery was performed.Case reportA 34-year-old Indian woman presented to her generalpractitioner with a one-month history of a painfulmidline lump over the lumbar region of her back.She was suﬀering from a sore throat and headache.The patient immigrated to the UK from India in2009. She had no medical history and was HIV-negative. On examination, the lump was ﬁrm with acentral groove, and was tender. The diﬀerential diag-nosis at the time was a soft tissue lump/lipoma. Therewas progressive growth of the lump with increasingpain over the following months. The patient wasreferred for an ultrasound scan in January 2011,which showed a bilobed cystic lesion. She also hada chest X-ray (CXR) and computed tomography(CT) chest scan, which revealed a lesion on the ﬁfthleft rib. In February 2011, the patient underwentmagnetic resonance imaging (MRI) of the wholespine. The results conﬁrmed a bilobed cystic lesion,extending from the spinal canal posteriorly withinvolvement of the L1 spinous process on eitherside of the midline, spanning T12/L1 to mid-L3levels (Figure 1). The MRI scan was reported as apossible complex meningocele. The patient wasreferred to neurosurgery at this time.On further questioning it was found that thepatient had no history of chest symptoms such asbreathlessness, cough or chest pain. On a visual ana-logue scale from 0 to 10, with 0 representing ‘no pain’and 10 representing the ‘worst pain imaginable’, heraverage low back pain (LBP) was 8/10 and her worstLBP was 9/10. She had an Oswestry Disability Index(ODI) of 50%. On examination, she was not obese.She had a normal gait and normal overall sagittal andcoronal balance of the spine. Upper and lower limbmotor and sensory examinations were normal with! The Author(s), 2014Creative Commons CC-BY-NC: This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 3.0 License (http://www.creativecommons.org/licenses/by-nc/3.0/) which permits non-commercial use, reproduction and distribution of the work without further permission providedthe original work is attributed as specified on the SAGE and Open Access page (http://www.uk.sagepub.com/aboutus/openaccess.htm).Journal of the Royal Society of Medicine Open;5(9) 1–6DOI: 10.1177/2054270414543396no pathological reﬂexes. The patient did have anobvious swelling on the lower back, in the midline,which was ﬁrm, had a central groove and did ﬂuctu-ate. Laboratory tests revealed a C-reactive protein(CRP) of 27.5mg/L (normal range 0–10mg/L) andan alkaline phosphatase of 78 IU/L (normal range20–140 IU/L). The patient underwent a CT scan ofher lumbar spine (Figure 2). The results of this, aswell as the previous chest CT and CXR, in combin-ation with the patient’s ethnic background, suggestedTB as the probable diagnosis. At this stage, thepatient had no deformity of the spine or any neuro-logical symptoms.Surgery for the L1 posterior paraspinal abscesswas undertaken in April 2011. This was carried outunder general anaesthesia with the patient in theprone position on a Montreal mattress. The abscesswas aspirated prior to opening; 120mL of straw-coloured ﬂuid was extracted. Upon incision, ﬁndingsincluded pus and a granular abscess bed. The L1spinous process was deep in the bed. A washoutwith 3L of normal saline was performed. Fluid andFigure 1. (a) Sagittal and (b) transverse view of MRI scan showing a bilobed cystic lesion spanning T12/L1 to mid-L3, withinvolvement of the L1 spinous process. The lesion measured 9.6 3.8 5.6 cm.Figure 2. (a) Sagittal and (b) transverse view of CT scan showing bony involvement of the L1 spinous process.2 Journal of the Royal Society of Medicine Open 5(9)bone were sent for biopsy. There were no surgicalcomplications and the patient was discharged homesix days later. Her cultures were found to be positivefor TB, and on advice from microbiology, ambulantchemotherapy was commenced.At six weeks’ follow-up, her MRI images showedresolution of the cystic lesion (Figure 3). Her averageLBP was now 1/10 and worst LBP was 1/10. Her ODIwas 16%, and CRP 7.5mg/L. There was a markedimprovement in her functional ability, judged by herself-assessment in performing activities of daily living(Table 1). At latest follow-up in October 2012, thepatient was found to have no deformity of the spineand no neurological deﬁcit. By that time she hadreceived her 12-month course of anti-TB chemother-apy. Her CRP in July 2012 was 6.3mg/L. She did havesymptoms of pain in her chest and legs. At this stage,there was no evidence of worsening of the spinal con-dition. The senior author thought in view of her recentvegetarian diet and her racial origin this may be due toa deﬁciency in vitamin D. Her blood results latershowed vitamin D levels to be 8 nmol/L, she wasstarted on vitamin D supplements. She also had alow thyroid stimulating hormone, 7.24 mLU/L, andnormal free T4, 13.8 mLU/L. A referral to an endo-crinologist was recommended. The patient remainedunder spinal review for two years following her surgi-cal intervention.DiscussionWe presented a case of a neurologically intact patientwith TB of the spine. In such patients, surgicaltreatment is rarely indicated, ambulant chemotherapyforms the mainstay of treatment.9 However, due tothe atypical anatomical location of her lesion, as wellas her symptomatology of pain and loss of function,we decided that the best mode of management for ourpatient would be surgery.Vertebral TB is usually located in the anteriorbody, involvement of the posterior elements is con-sidered rare, particularly in isolation.6,7 In this case,the infection was seemingly centred on the spinousprocess and lamina at one level with a soft tissuemass passing posteriorly, breaching the lumbarfascia into the abscess cavity. Two hypotheses existexplaining this atypical presentation: (1) Spread ofthe mycobacterium from a primary focus throughthe Batson venous plexus.6,7,10 This network ofveins located in the epidural space contains novalves to control the ﬂow of blood. Increased pres-sure in the body cavities can lead to retrograde ﬂowof blood from the infected viscera to the vertebralvenous plexi. The posterior external venous plexianastomose freely with the other vertebral venousplexi and hence infection could pass to the posteriorveins and hence to the posterior spinal structures bythis mechanism. (2) Infection of immunocomprom-ised patients. In recent decades, atypical forms ofTB have been increasingly reported in HIV-positivepatients.11,12 Narlawar et al.7 tested 21 of theirpatients with isolated TB of the posterior elementsfor HIV antibodies, and found seven (33.3%) to bepositive. Spread through the lymphatic system10and multidrug resistant TB13 are less-widely acceptedtheories for atypical spinal TB.Figure 3. (a) Sagittal and (b) transverse view of MRI scan showing resolution of cystic lesion, with some residual inflammation inthe abscess bed.Al-Khudairi and Meir 3Few studies have reported isolated TB of the pos-terior spinal elements (Table 2). The majority of thesepatients presented with severe neurological deﬁcits,and were treated with antitubercular therapy aloneor in combination with surgery. It was only in theArora et al.14 study that ﬁve patients had no neuro-logical deﬁcits, and were therefore treated only withantitubercular therapy.There remains great controversy regardingthe management of a neurologically intact, non-deformed patient with spinal TB. The MedicalResearch Council studies recommended ambulantchemotherapy as the gold standard management forsuch patients. However, detailed analysis of theresults later revealed that in certain patients surgerycan reduce long-term kyphotic deformity and oﬀerfaster resolution.16 Severe kyphosis can lead toneurological deﬁcits and late complications, whichcan be diﬃcult to treat. Early surgical interventioncould potentially stop this progression.17Tuli18 proposed the ‘middle-path’ regime for thetreatment of spinal TB. Patients should initially betreated with anti-TB chemotherapy; surgery shouldbe reserved for those who are refractory to treatment.However, he believed that lesions aﬀecting the pos-terior elements of the spine are an indication forimmediate surgery, due to the dangerous develop-ment of superﬁcial abscesses or sinuses and secondaryinfection of the meninges. In the case of our patient,we performed relatively minor surgery, and ambulantchemotherapy was later commenced on the advice ofmicrobiology.The majority of patients with TB respond well tochemotherapy; however, up to 10% of patients withcentral nervous system disease report a paradoxicalresponse, deﬁned as a worsening of pre-existing TBand/or the development of new lesions, in a patientwho initially responded well to therapy.19,20 This per-centage can rise to 30% in HIV-positive patients, thesame patients who are likely to have atypical spinalTB, further supporting our decision for early surgicalintervention.Due to its rarity and unfamiliarity, isolated TB ofthe posterior elements tends to be a delayed diagno-sis,15 as in this case. This delay makes simultaneousinvolvement of both the posterior and anterior elem-ents via anterior spread more likely. Dual involve-ment leads to severe spinal instability. For pureanterior ﬁxation to be successful, an intact posteriortension band is biomechanically advantageous, whichis improbable if there is posterior involvement.13Therefore, hypothesised early surgical interventioncan reduce the risk of deformity, instability andneurological deﬁcit.Our patient showed signiﬁcant improvement infunctional state following surgery (Table 1). HerODI dropped from 50% to 16%, representing a sig-niﬁcant clinical improvement.21 This can probably beattributed to the drainage of the abscess, whichrelieved the pressure compressing the paraspinalTable 1. Preoperative and postoperative assessment of activities of daily living.Activities Preoperative PostoperativeVigorous activities, such as running, lifting heavy objects,participating in strenuous sportsLimited a lot Limited a littleModerate activities, such as moving a table, pushing a vacuumcleaner, bowling or playingLimited a lot Limited a littleLifting or carrying groceries Limited a lot Limited a littleClimbing several flights of stairs Limited a lot Limited a littleClimbing one flight of stairs Limited a little Not limited at allBending, kneeling or stooping Limited a lot Limited a littleWalking more than a mile Limited a lot Limited a littleWalking several blocks Limited a lot Limited a littleWalking one block Limited a lot Not limited at allBathing or dressing yourself Limited a lot Not limited at allPatient was required to indicate whether she was ‘limited a lot’, ‘limited a little’ or ‘not limited at all’ when performing the aboveactivities of daily living.4 Journal of the Royal Society of Medicine Open 5(9)muscles hence allowing more normal function andreduced pain. The paraspinal muscles are crucial formobility and range of movement. We chose not toperform a laminectomy in this patient, to maintainnatural anatomical barriers preventing the spread ofinfection anteriorly and into the epidural space and/or anterior column. Also, a full laminectomy causesdisruption of the tension band increasing the likeli-hood of deformity. It is thought that since TB infec-tion can form chronic sinuses that incision anddrainage of TB abscesses can be problematic. Wedid not ﬁnd this in our case.Early surgical management in certain patients canreduce instability, deformity and late complications.We recommend patients with isolated TB of the pos-terior elements be reviewed on a case-by-case basiswhen determining management.DeclarationsCompeting interests: None declaredFunding: None declaredEthical approval: Written informed consent for the publicationwas obtained from the patient.Guarantor: AMContributorship: NA drafted the manuscript and provided intel-lectual input; AM provided the concept for the paper, the spinalsurgical intellectual and clinical input, and revision of the paper.Acknowledgements: NoneProvenance: Not commissioned; peer-reviewed by Ian Coulter.References1. Hajdu T, Donoghue HD, Bernert Z, Fothi E, Kovari Iand Marcsik A. A case of spinal tuberculosis from themiddle ages in Transylvania (Romania). Spine. (Phila Pa1976) 2012; 37: E1598–E1601.2. Ruiz Manzano J. Tuberculosis and immigration. MedClin (Barc) 2000; 114: 257–258.3. HPA enhanced TB surveillance of England and Wales,2012, London, UK: Health Protection Agency.4. Fuentes Ferrer M, Gutierrez Torres L, Ayala RamirezO, Rumayor Zarzuelo M and del Prado Gonzalez N.Tuberculosis of the spine. A systematic review of caseseries. Int Orthop 2012; 36: 221–231.5. Boachie-Adjei O and Squillante RG. Tuberculosis ofthe spine. Orthop Clin North Am 1996; 27: 95–103.6. Yalniz E, Pekindil G and Aktas S. Atypical tubercu-losis of the spine. Yonsei Med J 2000; 41: 657–661.7. Narlawar RS, Shah JR, Pimple MK, Patkar DP,Patankar T and Castillo M. Isolated tuberculosis ofposterior elements of spine: magnetic resonance ima-ging findings in 33 patients. Spine (Phila Pa 1976)2002; 27: 275–281.8. Bhojraj S and Nene A. Lumbar and lumbosacral tuber-culous spondylodiscitis in adults. Redefining the indi-cations for surgery. J Bone Joint Surg Br 2002; 84:530–534.9. Medical Research Council Working Party onTuberculosis of the Spine. A 15-year assessment of con-trolled trials of the management of tuberculosis of thespine in Korea and Hong Kong. Thirteenth Report ofthe Medical Research Council Working Party onTuberculosis of the Spine. J Bone Joint Surg Br 1998;80: 456–462.10. Naim-ur-Rahman N. Atypical forms of spinal tubercu-losis. J Bone Joint Surg Br 1980; 62: 162–165.11. Mallolas J, Gatell JM, Rovira M, Conget JI, Trilla Aand Soriano E. Vertebral arch tuberculosis in twohuman immunodeficiency virus-seropositive heroinaddicts. Arch Intern Med 1988; 148: 1125–1127.12. Shafer RW and Edlin BR. Tuberculosis in patientsinfected with human immunodeficiency virus: perspec-tive on the past decade. Clin Infect Dis 1996; 22:683–704.Table 2. Studies reporting cases of isolated TB of the posterior spinal elements in the last 33 years.Authors Cases Symptoms ManagementArora et al.14 24 Group 1 – rapid onset weakness orevidence of epidural abscessGroup 1 – emergency decompressivelaminectomyGroup 2 – no neurological deficit orepidural abscessGroup 2 – antitubercular therapyYazdi et al.15 1 History of back pain, progressive weaknessof lower extremitiesSurgery – midline incision and dissection.Followed by antitubercular therapyNarlawar et al.7 33 Pain, weakness, tingling, loss of sensation,bladder and bowel problems, fever,swelling, weight loss, coughAntitubercular therapyNaim-ur-Rahman10 5 Backache, weakness, sensory loss. Surgery – laminectomyAl-Khudairi and Meir 513. Wellons JC, Zomorodi AR, Villaviciencio AT, WoodsCW, Lawson WT and Eastwood JD. Sacral tubercu-losis: a case report and review of the literature. SurgNeurol 2004; 61: 136–139; discussion 139–141.14. Arora S, Sabat D, Maini L, et al. Isolated involvementof the posterior elements in spinal tuberculosis: areview of twenty-four cases. J Bone Joint Surg Am2012; 94: e151.15. Yazdi T and Rahimizadeh A. An illustrative caseIsolated posterior spinal arch tuberculosis. WorldSpinal Column J 2012; 3: 98–101.16. Mak KC and Cheung KM. Erratum to: surgical treat-ment of acute TB spondylitis: indications and out-comes. Eur Spine J 2013; 22: 211.17. Cheung WY and Luk KD. Clinical and radiologicaloutcomes after conservative treatment of TBspondylitis: is the 15 years’ follow-up in the MRCstudy long enough? Eur Spine J 2013; 22: 594–602.18. Tuli SM. Results of treatment of spinal tuberculosis by‘‘middle-path’’ regime. J Bone Joint Surg Br 1975; 57:13–23.19. Gupta M, Bajaj BK and Khwaja G. Paradoxicalresponse in patients with CNS tuberculosis. J AssocPhysicians India 2003; 51: 257–260.20. Breen RA, Smith CJ, Bettinson H, et al. Paradoxicalreactions during tuberculosis treatment in patients withand without HIV co-infection. Thorax 2004; 59:704–707.21. Fairbank JC and Pynsent PB. The Oswestry disabilityindex. Spine (Phila Pa 1976) 2000; 25: 2940–2952; dis-cussion 2952.6 Journal of the Royal Society of Medicine Open 5(9)

Isolated tuberculosis of the posterior spinal elements: case report and discussion of management

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Isolated tuberculosis of the posterior spinal elements: case report and discussion of management

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