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17-Year-Delayed Fistula Formation After Elective Spinal Instrumentation: A Case Report


1 Department of Neurological Surgery, Ghaem Hospital, Mashhad University of Medical Sciences (MUMS), Mashhad, IR Iran
*Corresponding Author: Babak Ganjeifar, Department of Neurological Surgery, Ghaem Hospital, Mashhad University of Medical Sciences (MUMS), Mashhad, IR Iran. Tel: + 98-5138012613, Fax: + 98-5138413493, E-mail: b_ganjeifar@yahoo.com.
Iranian Red Crescent Medical Journal. 17(5): e28090 , DOI: 10.5812/ircmj.17(5)2015.28090 | PMID: 26082855 | PMCID: PMC4464381
Article Type: Case Report; Received: Feb 20, 2015; Revised: Mar 19, 2015; Accepted: Apr 18, 2015; epub: May 25, 2015; collection: May 2015

Abstract


Introduction: A late-developing infection after an uneventful initial spinal instrumentation procedure is rare. Delayed infection and new fistula formation have been reported from a few months to 13 years. Here we report an unusual 17-year-delayed fistula formation after primary spinal instrumentation. The patient underwent hardware removal surgery with antibiotic therapy as a definitive treatment.

Case Presentation: Here we report an unusual 17-year delayed fistula formation after primary spinal instrumentation due to spinal trauma. He was admitted to Ghaem General Hospital, a chief referral center, Mashhad, North-East of Iran in August 2014. The patient underwent hardware removal surgery with antibiotic therapy as a definitive treatment.

Conclusions: Late inflammation may occur around spinal instruments and results in cutaneous fistula formation. After oral or intravenous antibiotic treatment, total device extraction is the cornerstone of treatment.

Keywords: Delayed; Fistula; Instrumentation

1. Introduction


Postoperative wound infection following spinal surgery poses high morbidity and mortality, and in this regard, posterior instrumentation has been reported the most frequent procedure (1). Total instrument removal and subsequent primary wound closure with systemic antibiotics is the treatment of choice (2, 3). However, the situation becomes problematic where insufficient bony fusion or pseudarthrosis requires spinal stabilization. Primary implant removal with subsequent reinstrumentation is susceptible to complications of the potentially infectious wound.

2. Case Presentation


A 53-year-old man complaining of exudative fluid discharge of his right flank since one month ago, was admitted to Ghaem General Hospital, a chief referral center, in Mashhad City, North-East of Iran in August 2014. On the physical examination, he was not pyretic; a draining fistula was located in his right flank, associated with pain, erythema, and local edema. His past history was positive for a falling accident 17 years ago with burst fracture of the third lumbar vertebra, and subsequent posterolateral fusion and instrumentation. The surgical and postsurgical periods were both uneventful with a 10-year follow up. Fistula tract was demonstrated on the fistulography and CT-fistulography, opening to the first lumbar pedicle screw (Figure 1A, 1B). There was no abnormality in his laboratory tests except for elevated erythrocyte sedimentation rate. Blood and smear samples were collected, which they were both negative for any microorganism.


Our patient underwent hardware removal surgery with perioperative empirical antibiotics (cephalexin and ciprofloxacin). During the procedure exudative fluid was discovered at the site of the right first lumbar pedicle screw. Adequate smear and culture were obtained with a specimen withdrawn for pathological studies. All the samples were negative for bacteria. The pathological study was consistent with fibro-muscular tissue and non-specific chronic inflammation. Fistula site healed spontaneously and our patient was discharged 10 days after the operation without any complication (Figure 2A, 2B).


Figure 1.
Fistulography

Figure 2.
A, Fistula Drainage preoperatively; B, Resolved Fistula Drainage 6 Months Follow-up

3. Discussion


Spinal fusion and instrumentation infection rate varies from 0.7% to 8.5% in the early postoperative course (4). However, the late onset (longer than one month postoperatively) infection is uncommon, as we demonstrated a 17 years gap between the incident and the fistula formation. Delayed infection rate was reported after instrumented spine surgery from 0.2% to 6.9% (5, 6).


Table 1 summarizes the cases with late infection reported in the literature since 1993. The period between the initial surgical procedure and the inflammatory symptoms varies from a few months to many years, as Mhaidli et al. reported a case with spontaneous drainage 13 years after the first spinal instrumentation (7). Here we report the longest interval, about 17 years.


Staphylococcus species was the most common organism found in the surgical site (8-11), with the next most detected bacteria being Propionibacterium acnes (2, 5, 12). The inflammatory reaction against the metallic device may have a role in these cases, so the removal of the instrument is necessary whether the organism is detected or not. Very late inflammation reaction may occur around spinal instruments and results in cutaneous fistula formation. After oral or intravenous antibiotic treatment, total device extraction is the cornerstone of treatment.


Table 1.
Summary of Late Infections Reported After Instrumented Spine Surgery a

Footnotes

Authors’ Contributions: Hamid Etemadrezaei developed the original idea, revised the manuscript, supervised the treatment and was guarantor. Samira Zabihyan prepared the manuscript and finally revised the manuscript. Aidin Shakeri prepared the manuscript, helped in follow up of the patient and acquisition of data. Babak Ganjeifar helped in follow up of the patient, acquisition of data, abstracted the findings, and revised the manuscript.

References


  • 1. Muschik M, Luck W, Schlenzka D. Implant removal for late-developing infection after instrumented posterior spinal fusion for scoliosis: reinstrumentation reduces loss of correction. A retrospective analysis of 45 cases. Eur Spine J. 2004;13(7):645-51. [DOI] [PubMed]
  • 2. Clark CE, Shufflebarger HL. Late-developing infection in instrumented idiopathic scoliosis. Spine (Phila Pa 1976). 1999;24(18):1909-12. [PubMed]
  • 3. Wimmer C, Gluch H. Aseptic loosening after CD instrumentation in the treatment of scoliosis: a report about eight cases. J Spinal Disord. 1998;11(5):440-3. [PubMed]
  • 4. Robertson PA, Taylor TK. Late presentation of infection as a complication of Dwyer anterior spinal instrumentation. J Spinal Disord. 1993;6(3):256-9. [PubMed]
  • 5. Viola RW, King HA, Adler SM, Wilson CB. Delayed infection after elective spinal instrumentation and fusion. A retrospective analysis of eight cases. Spine (Phila Pa 1976). 1997;22(20):2444-50. [PubMed]
  • 6. Hahn F, Zbinden R, Min K. Late implant infections caused by Propionibacterium acnes in scoliosis surgery. Eur Spine J. 2005;14(8):783-8. [DOI] [PubMed]
  • 7. Mhaidli HH, Der-Boghossian AH, Haidar RK. Propionibacterium acnes delayed infection following spinal surgery with instrumentation. Musculoskelet Surg. 2013;97(1):85-7. [DOI] [PubMed]
  • 8. Bose B. Delayed infection after instrumented spine surgery: case reports and review of the literature. Spine J. 2003;3(5):394-9. [PubMed]
  • 9. Kowalski TJ, Berbari EF, Huddleston PM, Steckelberg JM, Mandrekar JN, Osmon DR. The management and outcome of spinal implant infections: contemporary retrospective cohort study. Clin Infect Dis. 2007;44(7):913-20. [DOI] [PubMed]
  • 10. Mok JM, Guillaume TJ, Talu U, Berven SH, Deviren V, Kroeber M, et al. Clinical outcome of deep wound infection after instrumented posterior spinal fusion: a matched cohort analysis. Spine (Phila Pa 1976). 2009;34(6):578-83. [DOI] [PubMed]
  • 11. Sierra-Hoffman M, Jinadatha C, Carpenter JL, Rahm M. Postoperative instrumented spine infections: a retrospective review. South Med J. 2010;103(1):25-30. [DOI] [PubMed]
  • 12. Heggeness MH, Esses SI, Errico T, Yuan HA. Late infection of spinal instrumentation by hematogenous seeding. Spine (Phila Pa 1976). 1993;18(4):492-6. [PubMed]
  • 13. Dubousset J, Shufflebarger H, Wenger D. Late “infection” with CD instrumentation. Orthop Trans. 1994;18(3):121-6.
  • 14. Antuna SA, Mendez JG, Lopez-Fanjul JC, Paz Jimenez J. Cotrel-Dubousset instrumentation in idiopathic scoliosis a 5-year follow-up. Acta Orthop Belg. 1997;63(2):74-81. [PubMed]
  • 15. Hatch RS, Sturm PF, Wellborn CC. Late complication after single-rod instrumentation. Spine (Phila Pa 1976). 1998;23(13):1503-5. [PubMed]
  • 16. Weinstein MA, McCabe JP, Cammisa FJ. Postoperative spinal wound infection: a review of 2,391 consecutive index procedures. J Spinal Disord. 2000;13(5):422-6. [PubMed]
  • 17. Richards BR, Emara KM. Delayed infections after posterior TSRH spinal instrumentation for idiopathic scoliosis: revisited. Spine (Phila Pa 1976). 2001;26(18):1990-6. [PubMed]
  • 18. Soultanis K, Mantelos G, Pagiatakis A, Soucacos PN. Late infection in patients with scoliosis treated with spinal instrumentation. Clin Orthop Relat Res. 2003;(411):116-23. [DOI] [PubMed]
  • 19. Emel E, Karagoz Guzey F, Guzey D, Seyithanoglu H, Sel B, Alatas I. Delayed infection 6 years after spinal instrumentation: a case report. Turk Neurosurg. 2007;17(2):116-20. [PubMed]
  • 20. Farshad M, Sdzuy C, Min K. Late Implant Removal After Posterior Correction of AIS With Pedicle Screw Instrumentation—A Matched Case Control Study With 10-Year Follow-up. Spin Deformity. 2013;1(1):68-71. [DOI]
  • 21. Messina AF, Berman DM, Ghazarian SR, Patel R, Neustadt J, Hahn G, et al. The management and outcome of spinal implant-related infections in pediatric patients: a retrospective review. Pediatr Infect Dis J. 2014;33(7):720-3. [DOI] [PubMed]

Table 1.

Summary of Late Infections Reported After Instrumented Spine Surgery a

Reference Number of Patients Clinical Symptoms Risk Factors Risk Factors Findings From Culture Treatment Suspected Cause Year of Publication
Heggeness et al. (12) 6 Back pain (3), fever (3), spontaneous drainage of painful swelling (4), retroperitoneal abscess (1), fluctuation mass (1) Paraplegia with neurogenic bladder (2), Pyelonephritis/ renal calculi (1), intravenous drug abuse (2) Paraplegia with neurogenic bladder (2), Pyelonephritis/ renal calculi (1), intravenous drug abuse (2) Propionibacterium acnes (1), S.aureus (3), Streptococcus morvillorium (1), S.epidermides (1) Instrument removed (3), abscess drainage under CT guidance (1), needle aspiration (1) Hematogenous seeding 1993
Robertson and Taylor (4) 3 Elevated ESR (2), feeling of malaise (3), abscess in groin (2) Not stated Not stated Proteus mirabilis (1), none found (1) not stated (1) Instrumentation removed Intraoperative inoculation 1993
Dubousset et al. (13) 18 Incisional swelling and pain, spontaneous drainage Not stated Not stated S.epidermis (2), negative (16) Instrumentation removed Micromotion; metal fretting causing s sterile inflammation 1994
Viola et al. (5) 8 Wound drainage (2), abscess (4) back pain (8), Elevated ESR (7) Smoker (3), Malnutrition (1), Neurogenic bladder (2) Smoker (3), Malnutrition (1), Neurogenic bladder (2) S.epidermidis (6), Proprionibacterium acnes (1), negative (13) Instrumentation removed Intraoperative inoculation 1997
Antuna et al. (14) 1 Pain, spontaneous drainage None None Not reported Instrumentation removed Not stated 1997
Hatch et al. (15) 1 Back pain, tenderness over instrumentation, Westergren sedimentation rate 38% None None Negative Instrumentation removed Metal fretting, Chronic inflammation or low virulent bacteria 1998
Wimmer and Gluch (3) 8 Aseptic loosening of hardware, radiolucency around pedicle screws, pain and swelling in 6 patients who had discharging sinus None None Negative (6), no culture taken (2) Instrumentation removed (6), none (2) Metal fretting, micromotion 1998
Clark and Shufflebarger (2) 22 Fluctuant mass or drainage, pain rarely a factor, no fever none none S.epidermides (6), Enterococcus (2), S.aureus (1), Propionibacterium acnes (3) Instrumentation removed Intraoperative inoculation 1999
Weinstein et al. (16) 3 (of a series of 46 infections) Not specifically stated for 3 patients Not specifically stated Not specifically stated Not specifically stated; S. aureus found in 34 of 46 Instrumentation removed Not stated 2000
Richards and Emara (17) 23 Spontaneous drainage (15) fluctuance (6), pain (9), fever (3) Reoperation for dislodgement of hook (1), intravenous drug abuse (1) Reoperation for dislodgement of hook (1), intravenous drug abuse (1) Propionibacterium acnes (12), S epidermides (4), Micrococcus varians (13), S. aureus (1), negative (5) Instrumentation removed Intraoperative inoculation 2001
Soultanis et al. (18) 5 local subcutaneous abscess, whereas the remaining patients had a local drainage instrumentation failure and loosening instrumentation failure and loosening coagulase-negative Staphylococci (3), Acinetobacter baumani (1), Peptostreptococcus (1) Instrumentation removed Not stated 2003
Bose (8) 4 Back pain (1), incisional swelling (2), spontaneous drainage (1), abscess (1), hip pain (1), mental confusion (1) Dental infection (1), neurogenic bladder (1), kidney infection (1), Knee replacement surgery (1) Dental infection (1), neurogenic bladder (1), kidney infection (1), Knee replacement surgery (1) S. aureus (2), not tested (1) S. aureus suspected but no organisms found probably due to long-term course of antibiotics Irrigation/debridement (2), instrumentation removed (2), antibiotic therapy alone (1) Hematogenous seeding (4) 2003
Muschik et al. (1) 45 Wound sinus and spontaneous drainage of fluid (40), local pain (38), swelling (34), redness (28), Fever > 38.0°C (7) History of allergic predisposition, protracted postoperative fever, and nonunion of the fusion History of allergic predisposition, protracted postoperative fever, and nonunion of the fusion Staphylococcus aureus (6) Staphylococcus epidermidis (2) Instrumentation removed Implant bulk, metallurgic reactions, contamination with low-virulence microorganisms 2004
Hahn et al. (6) 7 Sudden Onset Local pain and swelling (7), Sinus Drainage (2) None None Propionibacterium acnes (6), Not found (1) Instrumentation removed Intraoperativeinoculation 2005
Kowalski et al. (9) 51 Back Pain (33), Wound drainage (16), Sinus tract present (13), Neurologic deficits (8) Diabetes mellitus (1), Systemic malignancy (6), Hepatic failure (1), Immunosuppressive medication use (8), End-stage renal disease (1) Diabetes mellitus (1), Systemic malignancy (6), Hepatic failure (1), Immunosuppressive medication use (8), End-stage renal disease (1) coagulase-negative staphylococci (9), Propionibacterium acnes (6), Staphylococcus aureus (11), Gram-negative bacilli (1), Streptococci (4), Polymicrobial infection (12), negative (8) Antibiotic therapy (6), Instrumentation removed (45) Intraoperative inoculation of low-virulence organisms 2007
Emel et al. (19) 1 Purulent flow developed in the posterior skin scar L3-Giant cell tumor L3-Giant cell tumor Staphylococcus aureus Instrumentation removed Intraoperative inoculation 2007
Mok et al. (10) 4 Drainage (2), Operative site pain (2) None None S. epidermidis (4 of 4), Propionibacterium acnes (3 of 4) 2009
Sierra-Hoffman et al. (11) 7 Drainage (4), Fever (3), Erythema (1) Not specified, For late onset infection Not specified, For late onset infection Methicillin-sensitive Staphylococcus aureus (3), Pseudomonas aeruginosa (2), Enterococcus faecalis (2) Antibiotic therapy (1), Instrumentation removed (6) Intraoperativeinoculation 2010
Mhaidli et al. (7) 1 Spontaneous drainage Non Stated Non Stated Propionibacterium acnes Instrumentation removed Intraoperative inoculation 2012
Farshad et al. (20) 7 Sudden Pain, Swelling and Flactuation Not Stated Not Stated Propionibacterium acnes (6) Instrumentation removed Intraoperative inoculation, metal fretting 2012
Messina et al. (21) 7 (of a series of 23 infections) Not specifically, stated, wound drainage (19), pain (8), fever (7) Not Stated Not Stated Not specified, for late onset infection Instrumentation removed Intraoperative inoculations 2014
Our Case 1 Elevated ESR, Fistula formation none none negative Instrumentation removed Not stated
a Abbreviation: ESR: erythrocyte sedimentation rate.

Figure 1.

Fistulography
A, CT Fistulography; B, Coronal view.

Figure 2.

A, Fistula Drainage preoperatively; B, Resolved Fistula Drainage 6 Months Follow-up