Neurology India, Vol. 58, No. 2, March-April, 2010, pp. 298-299
Spinal actinomycosis: A rare disease
Rakesh K Dua, Dhananjaya I Bhat, Devi B Indira
Department of Neurosurgery, NIMHANS, Bangalore, India
Correspondence Address: Rakesh K Dua, Department of Neurosurgery, NIMHANS, Bangalore, India, email@example.com
Date of Acceptance: 01-Feb-2010
Code Number: ni10074
AbstractActinomycosis is an indolent, slowly progressive infection caused by Actinomyces species. Of human actinomycosis, the spinal form is rare and actinomycosis-related spinal neurological deficit is uncommon. We report two cases with cervical and dorsal actinomycosis and one of them with spinal neurological deficit.
Keywords: Actinomycosis, bacteria, misdiagnosed disease, spine
Actinomycosis is an indolent, slowly progressive infection caused by Actinomyces species, which are anaerobic-to-microaerophilic gram-positive bacteria.  It normally colonizes gastrointestinal, bronchial, and female genital tracts. Actinomyces israelii is the most common species of actinomycosis causing human actinomycosis. Human actinomycosis presents with a myriad of clinical manifestations and at one time it was called the "most misdiagnosed disease." Virtually any site of the body can be affected in human actinomycosis.  Spinal actinomycosis is rare and fewer cases have been reported. We report two patients with spinal actinomycosis.
A 26-year-old male, manual laborer by occupation, presented with a complaint of mid-back pain of two months duration, insidious in onset and gradually progressive. On examination, he had mild thoracic scoliosis and tenderness at T3-5 level. Neurological examination and other system examinations were normal. Investigations revealed an elevated erythrocyte sedimentation rate during the first hour. Plain X-ray of the spine revealed destruction of T5 vertebra. Computed tomography (CT) and magnetic resonance imaging (MRI) of the spine showed partial destruction of the T5 body with no spinal cord compression [[Figure - 1]a-d].
He underwent CT-guided biopsy. Histopathology showed fragmented bony lamellae, marrow space containing epitheloid cell granuloma admixed with polymorphs. In addition multiple pale bluish globular masses with filamentous internal structure were seen. Gram stain revealed numerous clusters of gram-positive cocci in chains. Giemsa stain (GMS) revealed thin slender branching hyphal form. Periodic acid-Schiff (PAS) stain showed strongly PAS-positive thin filamentous structure.
Patient was treated with oral tetracycline and bed-rest for three months. He reported significant relief in the pain. A follow-up scan done after 12 weeks did not show any progression of the disease.
A 30-year-old lady, manual laborer by occupation, was investigated for cough and expectoration as a case of pulmonary tuberculosis and was put on antitubercular treatment (ATT). But she did not show much improvement on ATT and over a period of the next four to five months she developed multiple discharging sinuses over the neck and spastic weakness of lower limbs. Cultures from the discharging sinus were negative. Patient was evaluated with X-ray chest, CT brain and myelogram. Myelogram showed cutoff of contrast at level C7 [Figure - 2].
She underwent laminectomy and decompression. Histological features of the biopsy were suggestive of actinomycosis. She was treated with tetracycline for three months. There was healing of the sinus and other systemic symptoms also subsided.
Of the human actinomycoses, oral cervico-facial region actinomycosis accounts for about half of the cases and actinomycosis of the musculoskeletal system and central nervous system is rare. , The risk factors for actinomycosis include: male gender, poor oral hygiene, use of intrauterine devices, and immunocompromised states. ,,
Actinomycosis of the central nervous system is rare and the manifestations include brain abscess, meningitis or meningoencephalitis, subdural empyema, and epidural abscess.  In a review of the literature, Eftekhar et al.  found fewer than 100 cases of spinal actinomycosis and only 13 cases with actinomycosis-related spinal neurological deficit. The actinomycois-related spinal neurological deficits include myelopathy or myeloradiculopathy and are due to compression from epidural mass lesions. ,,,,,,,,, Actinomycosis of the spine is usually due to adjacent spread of soft tissue infection but the infection may occur following trauma or it can be hematogenous spread.  In Case 1 there was no other focus to explain the possible mode of infection. We feel that the vertebral involvement may be primary. In Case 2 the spinal involvement may be from the skin sinuses or lung. As the progression of the disease is slow, both bone destruction and new bone formation can be seen. 
In patients with spinal actinomycosis, treatment strategies depend on the presenting clinical features. Medical treatment may be sufficient for patients without any neurological deficits as in Case 1 and surgery may be considered in patients with neurological deficits as in Case 2. Actinomyces species are susceptible to penicillin, tetracycline, clindamycin, erythromycin (indicated in pregnant women), imipenem, ceftriaxone, and ciprofloxacine.  Failure to isolate any organisms on cultures in the second case may be related to the fact that the patients was on antituberculous drugs. Rifampicin is one of the recommended drugs for the treatment of actinomycosis.
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