- Email: [email protected]
Unusual Presentation of Isolated Sacral Tuberculosis
Joint Bone Spine 73 (2006) 751–752 http://france.elsevier.com/direct/BONSOI/
Case report
Unusual Presentation of Isolated Sacral Tuberculosis Ashok Kumar *, Manish Kumar Varshney, Vivek Trikha Department of Orthopedics, All India Institute of Medical Sciences, New Delhi 110029, India Received 5 November 2005; accepted 30 January 2006 Available online 19 April 2006
Abstract Tuberculosis is as old as mankind. Vertebral tuberculosis is fairly common form of bone and joint tuberculosis. However, isolated sacral tuberculosis is rare. It may present as an atypical sacral lesion having epidural granuloma without destruction of sacrum on MRI and plain Xrays. This atypical presentation may lead to delay in diagnosis and treatment. This case report intends to emphasize that spinal tuberculosis should be the first and foremost differential diagnosis in the presence of atypical clinical and radiological features of a sacral lesion particularly in developing countries. Early diagnosis and treatment can prevent or minimize the neurological morbidity in such cases. © 2006 Elsevier Masson SAS. All rights reserved. Keywords: Spinal tuberculosis; Sacral tuberculosis; Bone tumor
1. Introduction Vertebral tuberculosis is the commonest form of skeletal tuberculosis constituting about 50% of all cases of tuberculosis of bones and joints [1]. Literature regarding isolated sacral tuberculosis is scanty and it is usually reported as a part of lumbo-sacral tuberculosis (3–7%) [1]. It commonly presents with chronic low backache, discharging sinuses or abscess with or without neurological deficit [2]. Isolated sacral tuberculosis may also have atypical presentation of epidural soft tissue mass [2] or prolapsed intervertebral disc. The patient in our case had both these rare presentations making the diagnosis a difficult one. 2. Case report A 35-year-old female patient presented with 1 year history of recurrent episodes of mild to moderate pain and stiffness of the lower back. She had no history of low-grade fever, decreased appetite, weight loss, involvement of bowel and bladder. She also had no history of trauma to the back. Pain used to radiate to both lower leg and ankles, it was aggravated by walking and relieved by rest. Physical examination revealed paravertebral muscle spasm and tenderness over the lower lumbar and sacral spine. Bilateral straight leg raising test was re* Corresponding author. Present address: Type 3, Flat number 317, Pocket 2, Sector 12, Dwarka, Delhi, India. E-mail address: [email protected] (A. Kumar).
1297-319X/$ - see front matter © 2006 Elsevier Masson SAS. All rights reserved. doi:10.1016/j.jbspin.2006.01.016
stricted to 60° and forward bending of the spine was painful. Bilateral planter flexors and evertors were having grade 4/5 muscle power and bilateral ankle jerks were absent with sensory loss along the lateral border of feet. Hematological parameters were within normal limits barring erythrocyte sedimentation rate of 40 mm/hr. Repeated plain radiographs of the lumbo-sacral spine and pelvis did not reveal any bony or soft tissue abnormality. Chest X-ray was normal and Montoux test was found positive. MRI of the lumbo-sacral spine revealed diffuse marrow signal alternation from the first sacral to fifth sacral vertebrae and left iliac blade appearing hypointense on T1 and hyperintense on T2 weighted images suggestive of bone marrow edema with presacral, parasacral and epidural soft tissue extending from the sacral second to fifth sacral vertebrae (Fig. 1). No abnormality of cord or destruction of the cortex of sacrum was seen. Broad based posterior disc bulge with posterocentral disc protrusion between fifth lumbar and first sacral vertebra indenting the thecal sac was also seen on MRI. Gadolinium contrast enhanced MRI (Fig. 2) showed enhancement of the epidural soft tissue on T2 weighted images. Aspirated material from the parasacral collection was sent for staining for bacteria, fungi and Mycobacterium tuberculosis and culture sensitivity. Stained smears for bacteria, fungi and M. tuberculosis were found negative. Myco-3 DNA polymerase chain reaction (PCR) identified the species to be M. tuberculosis. Culture was found positive for M. tuberculosis at 6 weeks. Patient was given antitubercular treatment of 3 months of four drugs (isoniazid 300 mg; rifampicin 450 mg; pyrazinamide 1500 mg; ethambutol 800 mg) fol-
752
A. Kumar et al. / Joint Bone Spine 73 (2006) 751–752
Fig. 1. MRI (non-contrast) of the lumbo-sacral spine showing diffuse marrow signal alternation from S1 to S5 vertebrae appearing hypointense on T1 and hyperintense on T2 weighted images suggestive of bone marrow edema with presacral, parasacral and epidural soft tissue extending from S2 to S5 level. No abnormality of cord or destruction of the cortex of sacrum is seen.
Fig. 2. Coronal and sagittal sections of Gadolinium enhanced contrast MRI showing enhancement of epidural soft tissue and bone marrow edema.
lowed by 9 months of two drugs (isonazid 300 mg; rifampicin 450 mg) which is the standard antitubercular drug regimen at our institute. Patient had complete recovery from antitubercular drug and bed rest in the initial period and was asymptomatic at the last follow-up of 3 years. 3. Discussion India has one fifth of world’s tuberculosis suffering population, out of which skeletal tuberculosis constitutes 3% [3]. Fifty percent of these have tuberculosis of spine [3]. Isolated sacral tuberculosis is rare [2,4]. It usually presents as chronic back pain in adults and discharging sinuses or abscess in children [2]. Neurological deficit is relatively uncommon in isolated sacral tuberculosis [2]. Radiologically pre and parasacral collection with destruction of the sacrum is seen on CT scan. MRI of the sacrum usually reveals diffuse marrow edema which is hypointense on T1 and hyperintense on T2 weighted images [2]. Gadolinium contrast enhanced MRI show the enhancement of granulation tissue. Various atypical features of the spinal tuberculosis of spine have been described in literature including single vertebrae in-
volvement [4–6], non-contiguous involvement at more than one level (skip lesion) [4–6], neural arch tuberculosis [4–6] sacral [2] and cervical tuberculosis [2]. Tuberculosis of dorsolumbar or lumbo-sacral spine may also have an uncommon presentation where conventional Xrays do not reveal any obvious osseous lesion but MRI show the presence of extradural granuloma or peridural fibrosis causing compression of spinal cord [1,4]. Isolated sacral tuberculosis with similar presentation has been reported by Patankar et al. [2]. Patient in our report also had extradural soft tissue without evidence of bony destruction on plain radiographs and MRI. Enhancement of epidural soft tissue on Gadolinium contrast enhanced MRI suggested the possibility of an infection or tumor. MRI documentation of the posterior broad based disc bulge with posterocentral disc protrusion between lumbar fifth and sacral first level without significant compression of thecal sac did not explain the clinical presentation completely. Despite conflicting clinical and radiological features, first differential diagnosis was atypical tuberculosis of sacral spine due to high incidence and prevalence of tuberculosis in our country. This was substantiated by the positive PCR for tuberculosis [7]. Diagnosis was confirmed by positive culture for M. tuberculosis from the aspirated material from parasacral collection. Diagnosis by culture usually takes a longer time (3–8 weeks) while it can be established rapidly by histopathological diagnosis of tissue obtained by percutaneous biopsy guided by CT scan or ultrasound. Patient had a complete recovery with antitubercular treatment and bed rest. It is hence imperative to bear in mind the possibility of such atypical presentations of tuberculosis even in non-endemic areas for making a rapid and correct diagnosis and providing adequate treatment. Furthermore, one has to keep in mind that anti-TNF treatments are a further predisposing condition [8]. References [1] Tuli SM. Neurological complications. In: Tuli SM, editor. Tuberculosis of skeletal system 2nd ed. New Delhi: Jaypee Brothers; 1997. p. 217–33. [2] Patankar T, Krishnan A, Kate H, Patkar D, Kale H, Prasad S, et al. Imaging in isolated sacral tuberculosis: a review of 15 cases. Skeletal Radiology 2000;29:392–6. [3] Rajasekaran S, Dheenadhyalan J. Tuberculosis of spine. In: Bulstrode C, Buckwalter J, Carr A, Marsh L, Fairbank J, MacDonald JW, Bowden G, editors. Oxford textbook of orthopedics and trauma, 1st ed. UK: Oxford university press; 2002. p. 1532–60. [4] Rahman NU, El-Bakry A, Jamjoom A, Jamjoom ZA, Kolawole TM. Atypical forms of spinal tuberculosis: case report and review of literature. Surg Neurol 1999;51:602–7. [5] Rahman NU. Atypical forms of spinal tuberculosis. J Bone Joint Surg Br 1980;62:162–5. [6] Weaver P, Lifeso RM. The radiological diagnosis of tuberculosis of the adult spine. Skeletal Radiol 1984;12:178–86. [7] Chakravorty S, Sen MK, Tyagi JS. Diagnosis of extrapulmonary tuberculosis by smear, culture, and PCR using universal sample processing technology. J Clin Microbiol 2005;43:4357–62. [8] Tubach F, Salmon-Céron D, Ravaud P. Mariette X and for the RATIO Study Group. The RATIO observatory: French registry of opportunistic infections, severe bacterial infections, and lymphomas complicating antiTnFα therapy. Joint Bone Spine 2005;72:456–60.
Case report
Unusual Presentation of Isolated Sacral Tuberculosis Ashok Kumar *, Manish Kumar Varshney, Vivek Trikha Department of Orthopedics, All India Institute of Medical Sciences, New Delhi 110029, India Received 5 November 2005; accepted 30 January 2006 Available online 19 April 2006
Abstract Tuberculosis is as old as mankind. Vertebral tuberculosis is fairly common form of bone and joint tuberculosis. However, isolated sacral tuberculosis is rare. It may present as an atypical sacral lesion having epidural granuloma without destruction of sacrum on MRI and plain Xrays. This atypical presentation may lead to delay in diagnosis and treatment. This case report intends to emphasize that spinal tuberculosis should be the first and foremost differential diagnosis in the presence of atypical clinical and radiological features of a sacral lesion particularly in developing countries. Early diagnosis and treatment can prevent or minimize the neurological morbidity in such cases. © 2006 Elsevier Masson SAS. All rights reserved. Keywords: Spinal tuberculosis; Sacral tuberculosis; Bone tumor
1. Introduction Vertebral tuberculosis is the commonest form of skeletal tuberculosis constituting about 50% of all cases of tuberculosis of bones and joints [1]. Literature regarding isolated sacral tuberculosis is scanty and it is usually reported as a part of lumbo-sacral tuberculosis (3–7%) [1]. It commonly presents with chronic low backache, discharging sinuses or abscess with or without neurological deficit [2]. Isolated sacral tuberculosis may also have atypical presentation of epidural soft tissue mass [2] or prolapsed intervertebral disc. The patient in our case had both these rare presentations making the diagnosis a difficult one. 2. Case report A 35-year-old female patient presented with 1 year history of recurrent episodes of mild to moderate pain and stiffness of the lower back. She had no history of low-grade fever, decreased appetite, weight loss, involvement of bowel and bladder. She also had no history of trauma to the back. Pain used to radiate to both lower leg and ankles, it was aggravated by walking and relieved by rest. Physical examination revealed paravertebral muscle spasm and tenderness over the lower lumbar and sacral spine. Bilateral straight leg raising test was re* Corresponding author. Present address: Type 3, Flat number 317, Pocket 2, Sector 12, Dwarka, Delhi, India. E-mail address: [email protected] (A. Kumar).
1297-319X/$ - see front matter © 2006 Elsevier Masson SAS. All rights reserved. doi:10.1016/j.jbspin.2006.01.016
stricted to 60° and forward bending of the spine was painful. Bilateral planter flexors and evertors were having grade 4/5 muscle power and bilateral ankle jerks were absent with sensory loss along the lateral border of feet. Hematological parameters were within normal limits barring erythrocyte sedimentation rate of 40 mm/hr. Repeated plain radiographs of the lumbo-sacral spine and pelvis did not reveal any bony or soft tissue abnormality. Chest X-ray was normal and Montoux test was found positive. MRI of the lumbo-sacral spine revealed diffuse marrow signal alternation from the first sacral to fifth sacral vertebrae and left iliac blade appearing hypointense on T1 and hyperintense on T2 weighted images suggestive of bone marrow edema with presacral, parasacral and epidural soft tissue extending from the sacral second to fifth sacral vertebrae (Fig. 1). No abnormality of cord or destruction of the cortex of sacrum was seen. Broad based posterior disc bulge with posterocentral disc protrusion between fifth lumbar and first sacral vertebra indenting the thecal sac was also seen on MRI. Gadolinium contrast enhanced MRI (Fig. 2) showed enhancement of the epidural soft tissue on T2 weighted images. Aspirated material from the parasacral collection was sent for staining for bacteria, fungi and Mycobacterium tuberculosis and culture sensitivity. Stained smears for bacteria, fungi and M. tuberculosis were found negative. Myco-3 DNA polymerase chain reaction (PCR) identified the species to be M. tuberculosis. Culture was found positive for M. tuberculosis at 6 weeks. Patient was given antitubercular treatment of 3 months of four drugs (isoniazid 300 mg; rifampicin 450 mg; pyrazinamide 1500 mg; ethambutol 800 mg) fol-
752
A. Kumar et al. / Joint Bone Spine 73 (2006) 751–752
Fig. 1. MRI (non-contrast) of the lumbo-sacral spine showing diffuse marrow signal alternation from S1 to S5 vertebrae appearing hypointense on T1 and hyperintense on T2 weighted images suggestive of bone marrow edema with presacral, parasacral and epidural soft tissue extending from S2 to S5 level. No abnormality of cord or destruction of the cortex of sacrum is seen.
Fig. 2. Coronal and sagittal sections of Gadolinium enhanced contrast MRI showing enhancement of epidural soft tissue and bone marrow edema.
lowed by 9 months of two drugs (isonazid 300 mg; rifampicin 450 mg) which is the standard antitubercular drug regimen at our institute. Patient had complete recovery from antitubercular drug and bed rest in the initial period and was asymptomatic at the last follow-up of 3 years. 3. Discussion India has one fifth of world’s tuberculosis suffering population, out of which skeletal tuberculosis constitutes 3% [3]. Fifty percent of these have tuberculosis of spine [3]. Isolated sacral tuberculosis is rare [2,4]. It usually presents as chronic back pain in adults and discharging sinuses or abscess in children [2]. Neurological deficit is relatively uncommon in isolated sacral tuberculosis [2]. Radiologically pre and parasacral collection with destruction of the sacrum is seen on CT scan. MRI of the sacrum usually reveals diffuse marrow edema which is hypointense on T1 and hyperintense on T2 weighted images [2]. Gadolinium contrast enhanced MRI show the enhancement of granulation tissue. Various atypical features of the spinal tuberculosis of spine have been described in literature including single vertebrae in-
volvement [4–6], non-contiguous involvement at more than one level (skip lesion) [4–6], neural arch tuberculosis [4–6] sacral [2] and cervical tuberculosis [2]. Tuberculosis of dorsolumbar or lumbo-sacral spine may also have an uncommon presentation where conventional Xrays do not reveal any obvious osseous lesion but MRI show the presence of extradural granuloma or peridural fibrosis causing compression of spinal cord [1,4]. Isolated sacral tuberculosis with similar presentation has been reported by Patankar et al. [2]. Patient in our report also had extradural soft tissue without evidence of bony destruction on plain radiographs and MRI. Enhancement of epidural soft tissue on Gadolinium contrast enhanced MRI suggested the possibility of an infection or tumor. MRI documentation of the posterior broad based disc bulge with posterocentral disc protrusion between lumbar fifth and sacral first level without significant compression of thecal sac did not explain the clinical presentation completely. Despite conflicting clinical and radiological features, first differential diagnosis was atypical tuberculosis of sacral spine due to high incidence and prevalence of tuberculosis in our country. This was substantiated by the positive PCR for tuberculosis [7]. Diagnosis was confirmed by positive culture for M. tuberculosis from the aspirated material from parasacral collection. Diagnosis by culture usually takes a longer time (3–8 weeks) while it can be established rapidly by histopathological diagnosis of tissue obtained by percutaneous biopsy guided by CT scan or ultrasound. Patient had a complete recovery with antitubercular treatment and bed rest. It is hence imperative to bear in mind the possibility of such atypical presentations of tuberculosis even in non-endemic areas for making a rapid and correct diagnosis and providing adequate treatment. Furthermore, one has to keep in mind that anti-TNF treatments are a further predisposing condition [8]. References [1] Tuli SM. Neurological complications. In: Tuli SM, editor. Tuberculosis of skeletal system 2nd ed. New Delhi: Jaypee Brothers; 1997. p. 217–33. [2] Patankar T, Krishnan A, Kate H, Patkar D, Kale H, Prasad S, et al. Imaging in isolated sacral tuberculosis: a review of 15 cases. Skeletal Radiology 2000;29:392–6. [3] Rajasekaran S, Dheenadhyalan J. Tuberculosis of spine. In: Bulstrode C, Buckwalter J, Carr A, Marsh L, Fairbank J, MacDonald JW, Bowden G, editors. Oxford textbook of orthopedics and trauma, 1st ed. UK: Oxford university press; 2002. p. 1532–60. [4] Rahman NU, El-Bakry A, Jamjoom A, Jamjoom ZA, Kolawole TM. Atypical forms of spinal tuberculosis: case report and review of literature. Surg Neurol 1999;51:602–7. [5] Rahman NU. Atypical forms of spinal tuberculosis. J Bone Joint Surg Br 1980;62:162–5. [6] Weaver P, Lifeso RM. The radiological diagnosis of tuberculosis of the adult spine. Skeletal Radiol 1984;12:178–86. [7] Chakravorty S, Sen MK, Tyagi JS. Diagnosis of extrapulmonary tuberculosis by smear, culture, and PCR using universal sample processing technology. J Clin Microbiol 2005;43:4357–62. [8] Tubach F, Salmon-Céron D, Ravaud P. Mariette X and for the RATIO Study Group. The RATIO observatory: French registry of opportunistic infections, severe bacterial infections, and lymphomas complicating antiTnFα therapy. Joint Bone Spine 2005;72:456–60.