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LETTER TO EDITOR
Year : 2012  |  Volume : 60  |  Issue : 5  |  Page : 560-562

Lumbosacral actinomycosis with direct involvement and compression of conus medullaris and cauda equina nerve roots: An extremely rare case


1 Department of Pathology, Gujarat Cancer and Research Institute, Gujarat, India
2 Department of Radiology, Gujarat Imaging Centre, Gujarat, India
3 Department of Neurosurgery, Gujarat Cancer and Research Centre Ahmedabad, Gujarat, India

Date of Web Publication3-Nov-2012

Correspondence Address:
Aditi Dewan
Department of Pathology, Gujarat Cancer and Research Institute, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.103228

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How to cite this article:
Dewan A, Gupta A, Trivedi P, Agrawal G, Patel DD, Shah M. Lumbosacral actinomycosis with direct involvement and compression of conus medullaris and cauda equina nerve roots: An extremely rare case. Neurol India 2012;60:560-2

How to cite this URL:
Dewan A, Gupta A, Trivedi P, Agrawal G, Patel DD, Shah M. Lumbosacral actinomycosis with direct involvement and compression of conus medullaris and cauda equina nerve roots: An extremely rare case. Neurol India [serial online] 2012 [cited 2019 Aug 23];60:560-2. Available from: http://www.neurologyindia.com/text.asp?2012/60/5/560/103228


Sir,

Actinomyces are slow growing, non-spore forming gram-positive, branching bacilli that thrive in anaerobic and microaerophilic conditions and common sites of infections are oral and cervico-facial regions. Spinal actinomycosis is unusual and spread occurs by direct invasion or hematological dissemination. [1] Direct spinal cord involvement has not yet been described in the literature and hence this report.

A 22-year-old male presented with severe lower back pain and urinary incontinence of 1 month duration and paraparesis of 18 months duration. The patient gave a history of non-healing ulcer over right foot for the past 4 years. Subsequently, the patient developed gradual onset weakness followed by tingling and numbness of both lower limbs. Neurological examination revealed grade 4/5 motor power in lower limbs, bilateral foot drop, and a sensory level at L5. Bilateral ankle and knee reflexes were absent. Magnetic resonance imaging (MRI) of spine revealed a T2-weighted hypointense intraspinal lesion from L1 to S3 levels filling nearly entire lumbosacral spinal canal with posterior scalloping of lumbar vertebral bodies, infiltration of lumbosacral vertebral bodies, and their posterior elements with associated thin prevertebral soft tissue component, infiltration of posterior paraspinal muscles and overlying subcutaneous fat. Intervertebral disc were spared. Extraspinal extension of the lesion through neural foramina at multiple levels on either side was seen. The lesion was involving and compressing conus medullaris with associated lower dorsal cord edema [Figure 1]a. Changes of neurogenic bladder were seen with bilateral hydronephrosis and hydroureter [Figure 1]b and c. At surgery a lesion was identified involving L1-S1 spinal canal which was hard in consistency, engulfing the lower spinal cord and cauda equina nerve roots adherent to the dura. The surface of the lesion showed some yellow grey granules. On cutting through the lesion, purulent foul smelling material came out. The lesion was extending up to S3-S4 level with entrapment of nerve roots. A frozen section revealed dense clumps of fibrillary organisms in a background of inflammatory cells, suggesting a possibility of actinomycosis [Figure 2]a. Subsequently, L1-S1 laminectomy was done, dura was kept open, and a Romodrain was kept. Histopathological examination revealed a tissue reaction composed of acute inflammatory cells penetrating adjacent structures. Multiple microabscess formation was observed and each one contained dense bacterial colonies consisting of radiating filaments capped by eosinophilic hyaline material, thus confirming the diagnosis of actinomycosis [Figure 2]b. Patient received benzyl penicillin 2 MU i.v. every 4 h for 6 weeks followed by oral phenoxymethyl penicillin 1.0 g qid and probenacid 0.5 g bd for a total duration of 12 months. The neurological symptoms steadily improved and the patient was ambulatory with-out any support by the end of treatment.
Figure 1: (a) Sagittal T2W images reveal hypointense intraspinal lumbar lesion involving conus medullaris and cauda equina nerve roots (white arrow showing its extent) with lower dorsal spinal cord edema (grey arrow) and patchy infiltrates involving vertebral bodies and their posterior elements, posterior paraspinal soft tissue and overlying subcutaneous fat with thin pre-vertebral component and sparing of intervertebral discs. (b and c) Axial T2W images reveal similar findings with dorsal cord edema scalloping of the posterior margin of vertebral bodies with dilated ureters on either side.

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Figure 2: (a) Frozen section revealed clumps of fibrillary organisms along with inflammatory cells in the background (H and E). (b) Paraffin section showing typical sulfur granules surrounded by acute inflammatory cells (H and E, ×400)

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More than 100 cases of spinal actinomycosis have been reported in the literature with only 13 cases showing spinal cord compression usually due to epidural abscess. [2],[3] However, in none of the cases infiltration of conus medullaris was seen. Our patient had an extremely rare presentation of spinal actinomycosis showing several radiological features not described earlier in the literature. Bony tissue reacts to actinomycosis in a very characteristic, immediately around the focus of infection there is absorption of the bone; beyond that the cells are stimulated to form new bone resulting in sclerosis. The disease may be limited to one vertebra but more usually affects several vertebrae. Unlike tuberculosis, it involves the adjacent pedicles, transverse processes, also heads of neighboring ribs. The intervertebral discs are not affected. Rarefaction and sclerosis are found in varying proportions. When the disease involves the whole vertebra, the appearance is characteristic. The vertebra is riddled by network of suppurating channels which are bound by bones of increasing density giving it a honeycomb appearance. Due to the formation of dense trabeculae, it is uncommon for the vertebral body to collapse although it may occur in very severe cases. [4]

Actinomycosis of the spine is always secondary to infection of the contiguous tissue. In our patient, a non-healing ulcer was present over the foot for several years and MRI revealed the presence of prevertebral soft tissue involvement along with infiltration of associated posterior paraspinal muscle and overlying subcutaneous fat. The MRI findings reflected the histologic features of abundant granulation tissue in the sites infected with actinomycosis. Direct involvement of the conus medullaris as seen in our patient has not yet been described in the literature. Also, scalloping of the vertebral body instead of the saw-toothed borders has also not been described. These features led to the misdiagnosis of a malignant tumor.

Radiological differential diagnosis includes tuberculosis, septic osteomyelitis of spine, and malignancy. In tuberculosis, the intervertebral discs are more commonly affected and vertebral collapse is very common. Septic osteomyelitis gives rise to formation of sequestra, an occurrence almost unknown in actinomycosis. [4],[5] The only certain method of diagnosis is demonstration of the organism in the pus from the affected part. Close histological differential diagnosis is nocardiosis. On hematoxylin and eosin stain, actinomyces filaments become more basophilic and terminate in eosinophilic clubs but the nocordia species do not. Culture still remains the gold standard for the diagnosis. However, culturing is difficult as the bacteria are slow growing and proliferate in culture in less than 50% cases. Their growth is often precluded by prior administration of antibiotics. [4] Treatment involves long-term antibiotic therapy, surgical drainage, and decompression of the spinal canal. Drug of choice is intravenous high dose penicillin G for 6-8 weeks followed by long-term oral antibiotics.


 » Acknowledgments Top


The authors wish to express their thanks to Dr. S. N. Shukla Honrary Director of The Gujarat Cancer and Research institute to allow us to publish this case report.

 
 » References Top

1.Crank RN, Sundaram M, Shields JB. Case report 197. Cervical actinomycosis with spinalinvolvement. Skeletal Radiol 1982;8:164-7.  Back to cited text no. 1
    
2.Eftekhar B, Ketabachi E, Ghodsi M, Ahmadi A. Cervical epidural actinomycosis. J Neurosurgery 2001;95(1 Suppl):132-4.  Back to cited text no. 2
    
3.Vernon V, Pranav G, Palande D. Actinomycosis of the neck causing cervical epidural cord compression.'A case report and review of literature'. Spinal Cord 2007;45:787-9.  Back to cited text no. 3
    
4.Cope VZ. Actinomycosis of bone with special reference to infection of vertebral column. J Bone Joint Surg Br 1951;33:205-14.  Back to cited text no. 4
    
5.Briggs S, Seel M, Pegler RE. Actinomycotic vertebral osteomyelitis with spinal cord compression: A case report and review of the literature. InfectDisClinPract 2002;11:141-45.  Back to cited text no. 5
    


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