|Year : 2022 | Volume
| Issue : 2 | Page : 204-211
Aspergillus spinal epidural abscess: A case report and review of the literature
Mohammad Humayun Rashid1, Mohammad Nazrul Hossain1, Nazmin Ahmed1, Raad Kazi1, Gianluca Ferini2, Paolo Palmisciano3, Gianluca Scalia4, Giuseppe Emmanuele Umana3, Samer S Hoz5, Bipin Chaurasia6
1 Department of Neurosurgery, H Ibrahim Cardiac Hospital and Research Institute, Dhaka, Bangladesh
2 Department of Radiation Oncology, REM Radioterapia Srl, Viagrande, Italy
3 Department of Neurosurgery, Trauma Center, Gamma Knife Center, Cannizzaro Hospital, Catania, Italy
4 Department of Neurosurgery, Highly Specialized Hospital and of National Importance “Garibaldi”, Catania, Italy
5 Department of Neurosurgery, Neurosurgery Teaching Hospital, Baghdad, Iraq
6 Department of Neurosurgery, Bangabandhu Sheikh Mujib Medical University, Dhaka, Bangladesh
|Date of Submission||22-Feb-2022|
|Date of Acceptance||02-Apr-2022|
|Date of Web Publication||13-Jun-2022|
Giuseppe Emmanuele Umana
Department of Neurosurgery, Cannizzaro Hospital, Trauma Center, Gamma Knife Center, Via Messina, 829, 95126 Catania
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Aspergillus spinal epidural abscess (ASEA) is a rare entity that may mimic Pott's paraplegia as it commonly affects immunocompromised patients. We present one institutional case of ASEA with concomitant review of the literature. A 58-year-old female presented with intermittent low back pain for 10 years recently aggravated and with concurrent spastic paraparesis, fever, and weight loss. Emergent magnetic resonance imaging (MRI) showed T11–T12 epidural abscess with discitis and osteomyelitis. After empirical treatment with antibiotics, computed tomography-guided, percutaneous biopsy with drainage was performed, showing granulomatous tubercular-like collection. Antitubercular therapy was initiated, but after 1 month, the patient's condition deteriorated. Repeat MRI showed growth of the spinal epidural abscess with significant cord compression and vertebral osteomyelitis. T11–T12 laminectomy and tissue removal were performed with a posterior midline approach. Tissue histopathology showed necrotic debris colonies of Aspergillus spp. Antifungal therapy was started, and the patient rapidly improved. ASEA may mimic Pott's disease at imaging, leading to immediate start of antitubercular treatment without prior biopsy, leading to severe worsening of patients' clinical status. Cases of ASEA should be considered at pretreatment planning, opting for biopsy confirmation before treatment initiation so to prevent the occurrence of fatal infection-related complications.
Keywords: Aspergillus infection, spinal epidural abscess, spine infection, spine osteomyelitis
|How to cite this article:|
Rashid MH, Hossain MN, Ahmed N, Kazi R, Ferini G, Palmisciano P, Scalia G, Umana GE, Hoz SS, Chaurasia B. Aspergillus spinal epidural abscess: A case report and review of the literature. J Craniovert Jun Spine 2022;13:204-11
|How to cite this URL:|
Rashid MH, Hossain MN, Ahmed N, Kazi R, Ferini G, Palmisciano P, Scalia G, Umana GE, Hoz SS, Chaurasia B. Aspergillus spinal epidural abscess: A case report and review of the literature. J Craniovert Jun Spine [serial online] 2022 [cited 2023 Feb 9];13:204-11. Available from: https://www.jcvjs.com/text.asp?2022/13/2/204/347324
| Introduction|| |
Aspergillus species inhabit the soil and plants worldwide and may become pathogenic only in immunocompromised hosts., Inhaled by humans, Aspergillus infections may result in severe pulmonary diseases with human-to-human spread via blood contact. Invasive aspergillosis rarely affects bone structures, with the spine being the most common site. In immunocompromised patients, Aspergillus spinal epidural abscesses (ASEAs) may occur via hematogenous route or contiguous spread following any surgery or procedure in the respiratory system, gastrointestinal tract. ASEAs are typically found in the thoracic or lumbar spine but have been reported also in the cervical spine. In 1.9% of patients, they may also involve the skeletal muscle system. Due to their delayed onset and nonspecific clinical manifestations, ASEAs are often misdiagnosed at initial presentation as tuberculous spondylitis, especially due to the imaging similarities between the two entities. However, accurate differential diagnosis is mandatory as their management strategy differs, and severe risks of fatal complications may follow the initiation of inappropriate therapeutic plans. Owing to the rarity of this condition, only a few patients with ASEA have been reported. We present one institutional case of ASEA successfully treated with a combination of antifungal and surgical management and further review the current literature.
| Case Illustration|| |
A 58-year-old female normotensive, nonasthmatic, and diabetic presented intermittent low back pain for 10 years recently aggravated. The pain gradually increased in the last 4 months, radiating down to the anterior left leg and associated with decrease in walking distance. Intermittent fever and prolonged weight loss were also referred. Physical examination revealed spastic paraparesis with normal sensation and reflexes. She had normal rectal tone. Complete blood count was negative for leukocytosis or anemia, and erythrocyte sedimentation rate was elevated at 75.
Emergent magnetic resonance imaging (MRI) showed a T11–T12 epidural abscess with discitis and osteomyelitis [Figure 1]. The patient was admitted and started an empirical antibiotic treatment with meropenem, linezolid, and metronidazole for a presumed bacterial spinal epidural abscess. Percutaneous computed tomography (CT)-guided abscess aspiration and fluid cytopathology were positive for granulomatous tubercular-like infection [Figure 2]. Smear showed adequate cellular material containing plenty of degenerative polymorphs, lymphocytes, and histiocytes, and few epithelioid cell granulomas of tubercular origin. Decompressive surgery was not indicated because the patient lacked any neurological deficits or signs of cauda equina syndrome. Antitubercular therapy was initiated with rifampicin, pyrazinamide, ethambutol, isoniazid, and pyridoxine.
|Figure 1: Sagittal (a), coronal (b), and axial (c) magnetic resonance imaging T2WI scans showing T11–T12 epidural abscess with concurrent discitis|
Click here to view
|Figure 2: Computed tomography-guided fine-needle aspiration cytology of the T11 lesion suggestive of a granulomatous tubercular-like inflammation|
Click here to view
After 1 month of antitubercular therapy, the patient's condition worsened, with aggravating pain and increased weakness in the left lower limb. A new MRI study showed T11–T12 paravertebral soft-tissue intensity with central liquefaction consistent with spinal epidural abscess, and concurrent significant cord compression with vertebral osteomyelitis. The patient underwent partial T11–T12 laminectomy and subtotal removal of the granulation tissue via a posterior midline approach [Figure 3]. Tissue was sent for histopathology, showing negative bacteriological findings but revealing necrotic debris of Aspergillus spp. [Figure 4]. Long-term oral voriconazole was administered together with the antitubercular therapy. The patient was discharged to a skilled nursing facility to complete the planned 3-month course of antifungal therapy. Physical and occupational therapy noted the patient to progress well.
|Figure 3: Perioperative findings showing granulation tissue with purulent collection (a) evacuated via a posterior midline approach (b)|
Click here to view
|Figure 4: Histological specimen stained with H and E showing necrotic debris and infection from Aspergillus spp|
Click here to view
A literature search was performed on PubMed and returned 21 articles reporting patients with ASEA [Table 1]. Most patients were males in their third to sixth decade of life. The most common comorbidities were diabetes mellitus, tuberculosis, immunodeficiency, cancer, and kidney failure. The thoracic spine and the lumbar spine were the most affected. Common presenting symptoms were lower back pain, neurological deficits, low-grade fever, and weight loss. Diagnostic management mostly consisted of MRI imaging and/or CT-guided fine-needle aspiration. The most common pathogen was the Aspergillus fumigatus (60%) [Table 2]. Treatment commonly comprised a combination of antibiotics and surgical intervention. Only a few cases were treated with antibiotics alone. Most patients recovered completely or partially at a mean follow-up of 14.4 months. Some patients developed drug-related complications and died at a mean of 1.8 months.
|Table 1: Overview of all studies reporting patients with Aspergillus spinal epidural abscess|
Click here to view
|Table 2: Aspergillus species found in the 30 patients with Aspergillus spinal epidural abscess|
Click here to view
| Discussion|| |
ASEA is a rare entity with a recent increase in incidence owing to raising prevalence of systemic diseases responsible for immunosuppressive states (e.g., diabetes, kidney failure, and cancer). Corticosteroid therapy and intravenous drug abuse also play a major role in the development of these opportunistic infections. In immunocompromised patients, Aspergillus may spread to the spine from continuous lung foci, exposure to contaminated blood, or direct inoculation from the surrounding air during trauma or surgery and involve vertebral bodies with intervertebral discs causing osteomyelitis. Clinical features are nonspecific, mostly characterized by lower back pain with or without fever. Symptoms of spinal cord compression may occur after time. Men are most frequently affected, with involvement of their thoracic and lumbar spine regions.
For diagnostic confirmation, routine fungal culture, microbiological testing, imaging examinations, and histopathology need to be combined to exclude differential diagnoses. White blood cell counts have poor sensitivity and specificity. Specific genetic Aspergillus examinations often fail to rule out Aspergillus infection because immunocompromised patients cannot produce a significant inflammatory response, but inflammatory markers monitoring may help to assess response to treatment.,,, The differential diagnosis between spinal aspergillosis and tuberculosis is challenging only based on clinico-radiological examinations, but delay in diagnosis and treatment may be responsible for the development of invasive aspergillosis with high morbidity and mortality burden. From a radiological perspective, spinal tuberculosis frequently begins in the anteroinferior portion of the vertebral body and then spreads beneath the anterior longitudinal ligament to involve the adjacent vertebral body with secondary narrowing of the disc space., In invasive aspergillosis, the lesions often expand circumferentially destroying the surrounding spinal structures (vertebral bodies and discs) and the contiguous structures (ribs, thoracic wall, and lungs), as seen in our case. This imaging feature may support in the correct differential diagnosis. If the diagnosis is missed or delayed until there is extensive paravertebral, para-aortic, chest wall, or skull base invasion, complete eradication of disease by antifungal agents or surgery is not possible.,, For this reason, expedite diagnosis and distinction from the more common tubercular spondylitis are mandatory. The most reliable diagnostic methods for ASEA are histopathological examination and bacterial culture. When hematological and imaging examinations are doubtful, CT-guided fine-needle biopsy should be promptly performed to confirm the suspicion and start the appropriate therapy. Methods of molecular biology including enzyme-based and polymerase chain reaction-based assays can aid in the earlier diagnosis, but these are limited by lower sensitivity and higher false positives. In our case, the granulomatous inflammation detected at CT-guided biopsy posed some challenges in the differential diagnosis. Indeed, CT-guided biopsy is less invasive than intraoperative biopsy, but provides an insufficient number of specimens to make a proper diagnosis.
A review of the available literature shows that combined operative and medical treatment in early cases offers the best chance for the patient.,, Surgery is usually indicated for spinal cord compression, microbiological diagnosis, and stabilization of the spine. Biopsy and decompression by posterior approach followed by antifungal therapy may be successful in most cases. In our case, this strategy was pursed as the disease was limited in only two spine segments with no spine instability. However, extension of the pulmonary aspergillosis infection to the chest wall and mediastinum carries higher risks of morbidity and mortality burden.,
As regards antifungal therapy, amphotericin B was used in earlier cases of ASEA but later disregarded because of its high nephrotoxicity and its ineffectiveness in invasive aspergillosis, owing to the lack of entry into bones. As shown in more recent cases, second-generation broad-spectrum triazoles are often tolerated well and favor survival improvement in ASEA patients.,, The aspergillosis treatment guidelines proposed by the Infectious Diseases Society of America recommend voriconazole as primary therapy for extrapulmonary central nervous system aspergillosis including Aspergillus osteomyelitis, while itraconazole can be used in patients who cannot tolerate voriconazole. Voriconazole is associated with higher survival rate and higher remission rate than amphotericin B. In our patient, we started voriconazole obtaining optimal clinical improvement with no neurological deficit in postoperative period.
| Conclusion|| |
ASEA represents a rare cause of compressive myelopathy, which shows clinico-radiological similarities with tubercular spine disease. Early definitive diagnosis is challenging, but surgery with antifungal drugs is often indicated to establish the definite diagnosis and decompression of the spine in severe cases.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Ur-Rahman N, Jamjoom ZA, Jamjoom A. Spinal aspergillosis in nonimmunocompromised host mimicking Pott's paraplegia. Neurosurg Rev 2000;23:107-11.
Shweikeh F, Zyck S, Sweiss F, Sangtani A, Shweikeh M, Issa H, et al. Aspergillus
spinal epidural abscess: Case presentation and review of the literature. Spinal Cord Ser Cases 2018;4:19.
Dai G, Wang T, Yin C, Sun Y, Xu D, Wang Z, et al. Aspergillus
spondylitis: Case series and literature review. BMC Musculoskelet Disord 2020;21:572.
Chi CY, Fung CP, Liu CY. Aspergillus flavus
epidural abscess and osteomyelitis in a diabetic patient. J Microbiol Immunol Infect 2003;36:145-8.
Takagi Y, Yamada H, Ebara H, Hayashi H, Kidani S, Okamoto S, et al
. Aspergillus terreus spondylodiscitis following an abdominal stab wound: a case report. J Med Case Rep 2019;13:172. doi: 10.1186/s13256-019-2109-5.
Holmes PF, Osterman DW, Tullos HS. Aspergillus
discitis. Report of two cases and review of the literature. Clin Orthop Relat Res 1988;240-6.
van Ooij A, Beckers JM, Herpers MJ, Walenkamp GH. Surgical treatment of Aspergillus
spondylodiscitis. Eur Spine J 2000;9:75-9.
Gupta PK, Mahapatra AK, Gaind R, Bhandari S, Musa MM, Lad SD. Aspergillus
spinal epidural abscess. Pediatr Neurosurg 2001;35:18-23.
Auletta JJ, John CC. Spinal epidural abscesses in children: A 15-year experience and review of the literature. Clin Infect Dis 2001;32:9-16.
Saigal G, Donovan Post MJ, Kozic D. Thoracic intradural Aspergillus
abscess formation following epidural steroid injection. AJNR Am J Neuroradiol 2004;25:642-4.
Vaishya S, Sharma MS. Spinal Aspergillus
vertebral osteomyelitis with extradural abscess: Case report and review of literature. Surg Neurol 2004;61:551-5.
Son JM, Jee WH, Jung CK, Kim SI, Ha KY. Aspergillus
spondylitis involving the cervico-thoraco-lumbar spine in an immunocompromised patient: A case report. Korean J Radiol 2007;8:448-51.
Tew CW, Han FC, Jureen R, Tey BH. Aspergillus
vertebral osteomyelitis and epidural abscess. Singapore Med J 2009;50:e151-4.
Batra S, Arora S, Meshram H, Khanna G, Grover SB, Sharma VK. A rare etiology of cauda equina syndrome. J Infect Dev Ctries 2011;5:79-82.
Chang HM, Yu HH, Yang YH, Lee WI, Lee JH, Wang LC, et al.
Successful treatment of Aspergillus flavus
spondylodiscitis with epidural abscess in a patient with chronic granulomatous disease. Pediatr Infect Dis J 2012;31:100-1.
Sethi S, Siraj F, Kalra K, Chopra P. Aspergillus
vertebral osteomyelitis in immunocompetent patients. Indian J Orthop 2012;46:246-50.
] [Full text]
Jiang Z, Wang Y, Jiang Y, Xu Y, Meng B. Vertebral osteomyelitis and epidural abscess due to Aspergillus nidulans
resulting in spinal cord compression: Case report and literature review. J Int Med Res 2013;41:502-10.
Raj KA, Srinivasamurthy BC, Nagarajan K, Sinduja MG. A rare case of spontaneous Aspergillus
spondylodiscitis with epidural abscess in a 45-year-old immunocompetent female. J Craniovertebr Junction Spine 2013;4:82-4.
Yoon KW, Kim YJ. Lumbar Aspergillus
osteomyelitis mimicking pyogenic osteomyelitis in an immunocompetent adult. Br J Neurosurg 2015;29:277-9.
McCaslin AF, Lall RR, Wong AP, Lall RR, Sugrue PA, Koski TR. Thoracic spinal cord intramedullary Aspergillus
invasion and abscess. J Clin Neurosci 2015;22:404-6.
Sathyapalan D, Balachandran S, Kumar A, Mangalath Rajamma B, Pillai A, Menon VP. Long term outcome of medical and surgical co-management of craniospinal aspergillosis in an immunocompromised patient. Med Mycol Case Rep 2016;14:33-7.
Yang H, Shah AA, Nelson SB, Schwab JH. Fungal spinal epidural abscess: A case series of nine patients. Spine J 2019;19:516-22.
Tavakoli M, Hedayati MT, Mirhendi H, Nouripour-Sisakht S, Hedayati N, Saghafi F, et al.
The first rare and fatal case of invasive aspergillosis of spinal cord due to Aspergillus nidulans
in an Iranian child with chronic granulomatosis disease: Review of literature. Curr Med Mycol 2020;6:55-60.
Takagi Y, Yamada H, Ebara H, Hayashi H, Kidani S, Okamoto S, et al. Aspergillus terreus
spondylodiscitis following an abdominal stab wound: A case report. J Med Case Rep 2019;13:172.
Smith RM, Schaefer MK, Kainer MA, Wise M, Finks J, Duwve J, et al.
Fungal infections associated with contaminated methylprednisolone injections. N Engl J Med 2013;369:1598-609.
Gamaletsou MN, Rammaert B, Bueno MA, Moriyama B, Sipsas NV, Kontoyiannis DP, et al
osteomyelitis: epidemiology, clinical manifestations, management, and outcome. J Infect 2014;68:478-93. doi: 10.1016/j.jinf.2013.12.008.
Naim-Ur-Rahman, Jamjoom A, al-Hedaithy SS, Jamjoom ZA, al-Sohaibani MO, Aziz SA. Cranial and intracranial aspergillosis of sino-nasal origin. Report of nine cases. Acta Neurochir (Wien) 1996;138:944-50. doi: 10.1007/BF01411283. PMID: 8890991.
Patterson TF, Thompson GR 3rd
, Denning DW, Fishman JA, Hadley S, Herbrecht R, et al.
Practice guidelines for the diagnosis and management of aspergillosis: 2016 Update by the Infectious Diseases Society of America. Clin Infect Dis 2016;63:e1-60.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
[Table 1], [Table 2]