European Spine Journal A Case of spinal cord transection for an intramedullary abscess containing gas --Manuscript Draft-Manuscript Number:
ESJO-D-21-00772
Full Title:
A Case of spinal cord transection for an intramedullary abscess containing gas
Article Type:
Case Report
Keywords:
intramedullary abscess, spinal cord transection, infection, gas
Abstract:
Abstract Purpose The purpose of this paper is to report a case in which four debridements and spinal cord transection were performed for a gas-containing intramedullary abscess in addition to a gas-containing abscess found epidurally. To the best of our knowledge, there are no reports concerning gas-containing intramedullary abscesses. Methods The patient, a 72-year old man, was admitted to our hospital. His physical characteristics on admission were: a temperature of 37.6 ° C, blood pressure of 77/49 mmHg, and a heart rate of 120 beats/min. The muscle strength of all lower limbs was manual muscle testing (MMT) 0, and complete sensory loss below T5 was observed. Gas-containing abscesses were found epidurally at T4-S1 and in the dorsal muscle. Debridement was attempted three times. Inflammation began to decrease, but on the 45th day after admission, fever recurred, and an intramedullary abscess containing gas at T5-8 was found. Subsequently, debridement and spinal cord transection were performed. Results Debridement and spinal cord transection were successfully performed in a case involving gas-containing intramedullary abscesses. The dura mater was found to be thickened, and pus emanated from the spinal cord when the dura mater and the spinal cord were incised. A spinal cord amputation was performed to remove the infected spinal cord. The dura mater was sutured, and stump formation was performed. Conclusion The serious condition of this patient was life-threatening in nature. Four debridements and spinal cord transection were successfully performed and effectively saved the life of the patient.
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A Case of spinal cord transection for an intramedullary abscess containing gas
Abstract
Purpose The purpose of this paper is to report a case in which four debridements and spinal cord transection were
performed for a gas-containing intramedullary abscess in addition to a gas-containing abscess found epidurally.
To the best of our knowledge, there are no reports concerning gas-containing intramedullary abscesses.
Methods The patient, a 72-year old man, was admitted to our hospital. His physical characteristics on admission
were: a temperature of 37.6 ° C, blood pressure of 77/49 mmHg, and a heart rate of 120 beats/min. The muscle
strength of all lower limbs was manual muscle testing (MMT) 0, and complete sensory loss below T5 was observed.
Gas-containing abscesses were found epidurally at T4-S1 and in the dorsal muscle. Debridement was attempted
three times. Inflammation began to decrease, but on the 45th day after admission, fever recurred, and an
intramedullary abscess containing gas at T5-8 was found. Subsequently, debridement and spinal cord transection
were performed.
Results Debridement and spinal cord transection were successfully performed in a case involving gas-containing
intramedullary abscesses. The dura mater was found to be thickened, and pus emanated from the spinal cord when
the dura mater and the spinal cord were incised. A spinal cord amputation was performed to remove the infected
spinal cord. The dura mater was sutured, and stump formation was performed.
Conclusion The serious condition of this patient was life-threatening in nature. Four debridements and spinal cord
transection were successfully performed and effectively saved the life of the patient.
1
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Key words: intramedullary abscess, spinal cord transection, infection, gas
Introduction Cases of abscesses containing gas in the spinal canal are extremely rare, but the previously reported cases indicate a high mortality rate. Furthermore, to the best of our knowledge, there have been no reports on intramedullary abscesses containing gas. We report a case in which both epidural and intramedullary abscesses containing gas were found and treated with four debridements and spinal cord transection.
Case report The patient, a 72-year old man, sought treatment a different hospital after suddenly losing the ability to walk. The patient had experienced the onset of low back pain five days prior to seeking treatment. The following day, he was transfered to our hospital. His physical characteristics on admission were: a temperature of 37.6 ° C, blood pressure of 77/49 mmHg, and a heart rate of 120 beats/min. The muscle strength of all lower limbs was MMT 0, and complete sensory loss below T5 was observed. Swelling and a sensation of heat were observed in the lumbar region. The patellar tendon reflex and the Achilles tendon reflex had disappeared. Bladder and rectal disorders were observed. Blood test findings were a white blood cell (WBC) count of 36000 cells/mm3, a C-reactive protein (CRP) level of 30.14, blood urea
2
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nitrogen (BUN) of 53.1 mg/dl, creatinine (Cr) of 1.70 mg/dl, a glomerular filtration rate (GFR) of 31.6 ml/min/1.73m2, and hemoglobin A1c (HbA1c) of 8.8%. The patient's medical history included diabetes mellitus and recurrent liver abscess. Computed tomography (CT) showed extradural gas in the T4-S1 region and gas in the dorsal muscle (Fig.1). Abdominal CT showed a low density area in the right lobe of the liver. On the day of admission to our hospital, after percutaneously draining the liver abscess, the abscess in the lumbar muscle layer was drained under local anesthesia. Extensive resection of the back muscles and Vacuum Assisted Closure (VAC) was performed. Highly invasive surgery targeting the extradural gas was considered to be a life-threatening risk due to the patient's shock vital status, and subsequently abandoned. Intravenous administration of Tazobactam / Piperacillin 4.5g 4 days a day was started. Klebsiella Pneumoniae was detected in blood culture, liver abscess culture, and back abscess culture. We concluded that this was a liver abscess associated with the extradural gas in the T4-S1 region and the likely cause of the lower limb paralysis. Magnetic Resonance Imaging (MRI) revealed an extradural space-occupying lesion at T3-7 and intramedullary hyperintensity of less than T6. In addition, a space-occupying lesion in the epidural space below T12, and scattered lesions were observed (Fig.2). We concluded that these spaceoccupying lesions constituted an epidural abscess. Since the patient's general condition had stabilized by the 11th day following admission, thoracolumbar laminectomy (T3-7, T12-L5) was performed under general anesthesia. The epidural abscess was found and debridement was 3
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performed. VAC was applied to the back muscle defect. On the 25th day after admission, granulation had subsided, and debridement and wound closure were performed. At the L3-5 level, the dura mater had melted and cauda equina had escaped. Inflammation began to decrease, but on the 45th day after admission, a fever was detected. WBC count had increased to 12900 cells/mm3, and CRP level had increased to 13.04. CT and MRI of the thoracolumbar spine were performed. The CT showed intramedullary gas at T5-8, and the MRI showed a T24 intramedullary low signal and a spinal cord high signal below T5 (Fig.3). Subsequently, the lumbar region was incised according to the previous incision, and a large amount of pus was washed away. Following this, inflammation and fever recurred. CT showed a decrease in intramedullary gas, but gas remained at the T9 level. On Day 125 after admission thoracolumbar laminectomy (T7-12) + spinal cord transection (T8) was performed. The dura mater was found to be thickened, and pus emanated from the spinal cord when the dura mater and the spinal cord were incised. A spinal cord amputation was performed at the T8 level to remove the infected spinal cord (Fig.4). The dura mater was sutured, and stump formation was performed. The pathological findings showed scattered abscess tissue in the spinal cord with necrosis and degeneration. Administration of antibiotics was continued, and inflammation gradually decreased. WBC count decreased to 7100 cells/mm3, and CRP level decreased to 0.43. The patient was transferred 253 days after admission. At 352 days after onset, the patient's WBC count was 9680 cells/mm3 and his CRP level was 0.30. Complete 4
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sensory loss below T4 was noted, and both lower limbs were completely paralyzed, but wheelchair usage was possible.
Discussion Gas-containing infections progress very rapidly and are known to cause life-threatening conditions [1] [2]. The occurrence of gas in the spinal canal is very rare, with only 17 cases having been previously reported. The summary of case reports is found in Table 1. Of these, 16 involved extradural gas and 1 involved intradural extramedullary gas [3-19]. To the best of our knowledge, ours is the first report of an intramedullary gas-containing infection.
Localization of gas evolution has been reported to range from the level of 1 vertebral body[4] [9] to widespread occurrence up to C4-S [16]. There are various cases in which gas is contained only in the spinal canal [3,4,6,9-11,13,15-17], cases in which gas is also present in the surrounding soft tissues [5] [7] [8], and cases in which gas is also present in the vertebral body [12] [18]. The major risk factor for gas-containing infectious diseases is diabetes [1] [2]. Thirteen of the 17 previously reported cases involved diabetes mellitus (76.5%). Fever and back pain were reported to be the most common symptoms [2]. Among the 17 previously reported cases, fever was reported in 11 cases (64.7%), and back pain in 14 (82.4%). Conversely, neurological symptoms were relatively infrequent, reported in just 7 of the 17 cases (41.2%). Early diagnosis 5
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and appropriate treatment are important for infectious diseases including gas-containing infection. CT is considered to be the most effective method for early diagnosis. X-ray imaging can miss early changes, and MRI can delay diagnosis. Confirming the presence of gas by CT and recognizing fever and back pain are the keys to early diagnosis of gas-containing infections [2]. In the 17 previously reported cases, staphylococcus aureus was the most common pathogen [3] [4] [6] [8] [9], followed by Escherichia coli [8] [9] [12] [16]. Various routes of infection were reported, including epidural catheters [4], spinal surgeries [10] [11], urinary tract [1] [15] [16], foot ulcers [8] [9], and decubitus ulcers of the buttock [7]. Appropriate antibiotics and aggressive surgical treatment are recommended for the treatment of gas-containing infections [1] [2]. Surgery was performed in 15 of 17 previously reported cases (88.2%) [3-13, 15, 17, 18]. Conversely, among the cases in which gas was localized in the dura, there were cases in which improvement was achieved by administration of antibiotics alone. Among these [14-16], a case of epidural abscess containing gas in an extremely wide area from C4 to S5 is also included [16]. These findings indicate that treatment tailored to each individual case is required. However, even with early diagnosis and appropriate management, infection may be difficult to control, and death may result from multiple organ failure. Six of the 17 cases (35.2%) in which gas was found in the spinal canal resulted in death [5,810,15,18,19]. This is a very rare case involving an intramedullary gas abscess. To our knowledge, this is 6
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the first case of a gas-containing abscess occurring in the spinal cord. Fever, back pain, and paralysis of both lower limbs were observed. CT showed a wide range of gas images inside and outside the spinal canal, so the original diagnosis of epidural abscess was relatively easy. The patient's medical history included diabetes mellitus and recurrent liver abscess. Because the patient had recurrent liver abscesses, and Klebsiella Pneumoniae was detected in the liver abscess culture, blood culture, and back abscess culture, it was thought that sepsis developed from the liver abscess, which led to the development of the lumbar and epidural abscesses. MRI at the time of admission showed that the spinal cord below T6 had a high signal (Fig.2), and it is possible that the infection had already spread into the spinal cord. However, no gas or abscess images were observed in the spinal cord at this time. Since there was a risk that the infection would spread into the dura by surgically opening the dura, this course was abandoned at this early stage. After three debridement attempts, gas was also found in the spinal cord, and spinal cord transection was performed after inflammation showed no decrease. Intraoperative
findings
revealed
intramedullary
pus
emanation,
confirming
the
intramedullary abscess. At 352 days after onset, the patient's WBC count was 9680 cells/mm3 and his CRP level was 0.30. Complete sensory loss below T4 was noted, and both lower limbs were completely paralyzed, but wheelchair usage was possible. Conclusion The serious condition of this patient was life-threatening in nature. Four debridements and 7
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spinal cord transection were successfully performed and effectively saved the life of the patient.
Declarations
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StatPearls. Treasure Island (FL).
2. Ner EB, Chechik Y, Lambert LA, Anekstein Y, Mirovsky Y, Smorgick Y (2020) Gas forming
infection of the spine: a systematic and narrative review. Eur Spine J. https://doi.org/10.1007/s00586-
020-06646-7
3. Kirzner H, Oh YK, Lee SH (1988) Intraspinal air: a CT finding of epidural abscess. AJR Am J
Roentgenol 151:1217-1218. https://doi.org/10.2214/ajr.151.6.1217
4. Shintani S, Tanaka H, Irifune A, Mitoh Y, Udono H, Kaneda A, Shiigai T (1992) Iatrogenic acute
spinal epidural abscess with septic meningitis: MR findings. Clin Neurol Neurosurg 94:253-255.
https://doi.org/10.1016/0303-8467(92)90099-o
5. Kokes F, Iplikcioglu AC, Camurdanoglu M, Bayar MA, Gokcek C (1993) Epidural spinal abscess
containing gas: MRI demonstration. Neuroradiology 35:497-498.
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6. Fujisawa H, Hasegawa M, Tsukada T, Kita D, Tachibana O, Yamashita J (1998) Intraspinal air: an
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anaerobic bacteria, producing a mass of gas]. Rinsho Shinkeigaku 38:224-227
8. Spock CR, Miki RA, Shah RV, Grauer JN (2006) Necrotizing infection of the spine. Spine (Phila Pa
1976) 31:E342-344. https://doi.org/10.1097/01.brs.0000217631.73632.37
9. Nadkarni T, Shah A, Kansal R, Goel A (2010) An intradural-extramedullary gas-forming spinal
abscess in a patient with diabetes mellitus. J Clin Neurosci 17:263-265.
https://doi.org/10.1016/j.jocn.2009.05.019
10. Hur JW, Lee JB, Kim JH, Kim SH, Cho TH, Suh JK, Park YK (2012) Unusual fatal infections after
anterior cervical spine surgeries. Korean J Spine 9:304-308. https://doi.org/10.14245/kjs.2012.9.3.304
11. Lee JS, Choi SM, Kim KW (2013) Triparesis caused by gas-containing extensive epidural abscess
secondary to Aeromonas hydrophila infection of a thoracic vertebroplasty: a case report. Spine J 13:e9-
e14. https://doi.org/10.1016/j.spinee.2013.03.045
12. Akagawa M, Kobayashi T, Miyakoshi N, Abe E, Abe T, Kikuchi K, Shimada Y (2015) Vertebral
osteomyelitis and epidural abscess caused by gas gangrene presenting with complete paraplegia: a case
report. J Med Case Rep 9:81. https://doi.org/10.1186/s13256-015-0567-y
13. Bang JH, Cho KT (2015) Rapidly Progressive Gas-containing Lumbar Spinal Epidural Abscess.
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Meningitis in an Adult. Korean J Spine 14:17-19. https://doi.org/10.14245/kjs.2017.14.1.17
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Thoracic Spinal Epidural abscess secondary to Klebsiella Pneumonia infection. Neurology 90
16. Matsuo T, Tanji A, Tateyama K, Yoda Y, Kamata Y, Urabe T (2019) Pneumorachis from the
cervical to the sacral spinal canal with spinal epidural abscess by gas gangrene. J Orthop Surg (Hong
Kong) 27:2309499019860072. https://doi.org/10.1177/2309499019860072
17. Money AJ, Molloy S, Grabowski G (2019) Epidural Abscess Caused by Gas-Producing Clostridium
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Medical Emergency for Spine Surgeon. World Neurosurg 121:124-126.
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Figure Captions Fig. 1
11
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Sagittal computerized tomography (CT) (Thoracic spine - sacral spine) CT shows extradural and intramuscular gas in T4-S1
Fig. 2
Sagittal T2-weighted magnetic resonance imaging (MRI) (cervical spine - sacral spine) An epidural occupying lesion is visible in T3-7, and intramedullary hyperintensity of T6 or less is apparent. In addition, occupying lesions epidural below T12 are visible
Fig. 3
Sagittal computerized tomography (CT) (Thoracic spine) (A), Sagittal (B) and axial (C) T2weighted magnetic resonance imaging (MRI) (Thoracic spine) CT shows an intramedullary gas image in T5-8, and MRI shows an intramedullary low signal in T2-4 and a high signal below T5
Fig. 4
Intraoperative photo The dura mater is thickened. When the dura mater and the spinal cord are incised, pus emanates from the spinal cord
12
Figure
Fig.1
Figure
Fig.2
Figure
Fig.3
C
A
B
Figure
Fig.4
Died + Staphylococcus aureus Urethra Epidural(T8-S1)
-
+
+
+
68
M M
F F
F M M
Bang (2015)
Kim (2017)
Hsieh (2018)
Teles (2019)
Matsuo (2019)
Money (2019)
Smorgick (2020)
70
61
73
56
72
78
+
+
+
-
+
+
+
+
+
+
+
+
+
+
-
+
N/A
+
+
-
N/A
-
-
-
-
+
-
+
+
+
Vertebral body, Soft tissue
Epidural (C4-S5)
Vertebral body, Soft tissue
Epidural(L3)
Epidural (C5-T5)
Soft tissue
Epidural (C4-5)
Epidural (L4-5)
Vertebral body, Soft tissue
Epidural (T11-12)
Epidural (C4-T8)
Epidural (C4-5)
Table.1 Summary of gas-containing infections in the spinal canal
N/A: not available
Recovered
+
Clostridium septicum
N/A
Epidural (T11-L4)
-
+
+
+
69
F
+
-
intradural–extramedullar(C2)
Urethra
N/A
Urethra
N/A
N/A
N/A
Spine surgery
Spine surgery
Foot ulcers
Escherichia coli
Klebsiella pneumoniae
Klebsiella pneumoniae
Steptococcus constellatus
Steptococcus anginosus
Enterococcus faecalis
Escherichia coli
Clostridium perfringens
Aeromonas hydrophilia
Steptococcus anginosus
Escherichia coli
Staphylococcus aureus
Escherichia coli
Staphylococcus aureus
-
+
+
-
+
+
+
+
+
+
Recovered
Died
Died
Recovered
Recovered
Recovered
Recovered
Died
Died
Died
Recovered
Recovered
Died
Recovered
Recovered
Akagawa (2015)
72
68
+
Foot ulcers
+
+
+
+
+
M
+
Soft tissue
Epidural (L3-5)
Peptostreptococcus
Bacteroides fragilis
Staphylococcus aureus
Bacterioides
Peptostreptococcus
Streptococcus
Staphylococcus aureus
Staphylococcus aureus
M
+
-
Decubitus of sacrum
Unknown
Decubitus of sacrum
Epidural catheter
Urethra
Lee (2013)
+
+
Soft tissue
Epidura(T9, Sacral)
Epidural (C2-6)
Soft tissue
Epidural (T12-L5),
Epidural (L3)
Epidural (T12-L5)
Hur (2013)
61
+
-
+
-
-
+
symptoms
M
+
N/A
+
+
+
+
Nadkarni (2010)
65
-
+
-
-
+
pain
F
-
+
+
-
+
Spock (2006)
54
55
52
71
66
F
Outcomes
Nakatani (1998)
Surgery
F
Bacteria
Fujisawa (1998)
Infectious source
M
Gas location
Kökeş (1993)
Neurological
F
Fever
Shintani (1992)
Back
F
Diabetes
Kirzner (1988)
Age
Sex
Author
Table.1
Table