https://orcid.org/0000-0001-7900-7560
Abstract
Destructive spondyloarthropathy (DSA) is a rare complication of long-term hemodialysis that often mimics infectious spondylitis, making diagnosis challenging. We report two patients with chronic kidney disease on long-term hemodialysis who presented with progressive neurological deficits and destructive spinal lesions suggestive of infection. In both cases, percutaneous biopsy of the lumbar destructive lesions showed no definitive evidence of infection, although occult infection could not be completely excluded. A 56-year-old woman underwent cervical reconstruction and achieved significant neurological recovery with solid fusion at 24 months. In contrast, a 62-year-old man underwent lumbar reconstruction but developed severe postoperative wound complications and died following a fatal cardiac event. These cases highlight the importance of considering DSA in the differential diagnosis of destructive spinal lesions in hemodialysis patients. Histological confirmation is essential before definitive treatment, and surgical outcomes depend not only on technical success but also on the patient's systemic condition and comorbidities.
Introduction
Destructive spondyloarthropathy (DSA) is a complication of long-term hemodialysis characterized by destructive spinal changes that may mimic infectious spondylitis [1–5]. Poor bone quality and systemic comorbidities complicate surgical management [6, 7]. We report two cases of presumed dialysis-associated DSA with contrasting postoperative courses, illustrating diagnostic uncertainty and the challenges of spinal reconstruction in this population.
Case reports
The Institutional Review Board approved this retrospective study and waived informed consent for participation. Separate written informed consent for the publication of clinical information and images was obtained for both cases.
Case 1
A 56-year-old woman on hemodialysis for over 10 years presented with severe lower back pain and quadriparesis (motor grade III). Lumbar imaging showed L3–4 destruction suggestive of infectious spondylitis. Bilateral Hoffmann’s signs prompted a cervical magnetic resonance imaging (MRI), which revealed severe cervical cord compression (Fig. 1).
(A) Preoperative lumbar spine T1-weighted Gd-enhanced sagittal MRI showing changes suspicious for infectious spondylitis at the L3–4 level; (B) preoperative cervical spine T2-weighted sagittal MRI showing bony destruction and angular kyphosis with cord compression at the C5–6–7 level with high signal change; Gd, gadolinium.
The white blood cell count, C-reactive protein, and erythrocyte sedimentation rate were normal. No antibiotics were administered before the percutaneous biopsy of the lumbar lesion, which showed degenerative fibrocartilaginous tissue without specific evidence of infection; cultures showed no growth. DSA was not initially suspected, and amyloid-specific staining was not performed.
Progressive cervical myelopathy prompted a C6 corpectomy and anterior cervical corpectomy and fusion from C5 to C7. A mesh cage was packed with allograft and demineralized bone matrix because the harvested autologous bone was of poor quality. Attempted anterior plating failed because of repeated screw pullout in severely compromised bone (Fig. 2), necessitating unplanned supplemental posterior fixation. A rigid cervical collar was maintained until pedicle screw fixation at C4, C5, C6, C7, and T1 was performed 3 days later. Motor strength improved to grade IV+, and independent ambulation was regained. At 24 months, computed tomography (CT) showed a maintained reconstruction without cage subsidence or implant failure (Fig. 3d, Table 1). Low back pain persisted, but after discussion of perioperative risks, the patient declined additional lumbar surgery and chose conservative management.
Intraoperative C-arm image showing screw pull-out during attempted anterior cervical plate fixation (arrow).
Changes in cervical sagittal alignment parameters: C2–7 lordosis (white line), C2–7 sagittal vertical axis (white dotted line), segmental Cobb angle (black line), T1 slope (black dotted line); (A) preoperative image; (B) postoperative 24-month image; (C) postoperative T2-weighted MRI showing spinal cord decompression with persistent cord signal change (dotted circle); (D) sagittal CT image obtained at the 24-month follow-up showing the maintained position of the mesh cage without subsidence or implant failure; a residual screw track from the previous anterior fixation attempt (white arrow) is visible in the vertebral body above the mesh cage.
Preoperative and postoperative (24 months) radiographic and clinical parameters
| Parameter | Preoper-ative | Postoper-ative | Change values |
|---|---|---|---|
| C2–7 lordosis (°) | −15.7 | 10.8 | +26.5 |
| C2–7 SVA (mm) | 16.5 | 6.2 | −10.3 |
| Segmental Cobb angle (°) | −18.2 | 8.4 | +26.6 |
| T1 slope (°) | 14.2 | 15.4 | +1.2 |
| T1S–CL (°) | 29.9 | 4.6 | −25.3 |
| mJOA scorea | 5 | 11 | +6 |
| VAS (neck) | 8 | 2 | −6 |
| NDI | 48 | 12 | −36 |
SVA, sagittal vertical axis; T1S–CL, T1 slope minus cervical lordosis; mJOA, modified Japanese Orthopaedic Association; VAS, visual analog scale; NDI, Neck Disability Index.
aThe sphincter function domain was excluded because urinary function could not be evaluated due to anuria associated with long-term hemodialysis; therefore, the maximum adapted mJOA score was 15 points.
Case 2
A 62-year-old man on hemodialysis for over 11 years presented with progressive low back pain and paraparesis (motor grade II). MRI showed L3–4 destruction suggestive of infectious spondylitis (Fig. 4). The white blood cell count was 12 350/μl, C-reactive protein was 58 mg/l, and the erythrocyte sedimentation rate was 17 mm/h. Percutaneous L3–4 biopsy on hospital Day 2 showed degenerated and necrotic bone; Gram stain/culture, acid-fast bacillus and fungal studies, and real-time polymerase chain reaction testing for mycobacterium tuberculosis and nontuberculous mycobacteria were negative. DSA was not initially suspected, and amyloid-specific staining was not performed.
(A) Initial lumbar lateral radiograph; degenerative spondylolisthesis was seen at the L3–4 level; (B) follow-up image after 1 year; bony destruction and angular kyphosis at the L3–4 level; (C) preoperative lumbar spine T1-weighted Gd-enhanced sagittal MRI showing changes suspicious for infectious spondylitis at the L3–4 level.
Pulmonary edema and pneumonia were diagnosed on hospital Day 6, and piperacillin–tazobactam was initiated. Preoperative coronary angiography demonstrated three-vessel coronary artery disease. Despite substantial operative risk, the patient requested surgery for persistent severe pain and progressive neurological deficits. Surgery was performed 2 weeks after antibiotic initiation. During anterior decompression and mesh cage reconstruction, small yellowish abscess-like pockets were encountered, raising concern for occult infection despite negative preoperative studies. Total L3 laminectomy and posterior pedicle screw fixation at L1, L2, L3, L5, and S1 followed (Fig. 5).
Postoperative lateral radiograph; lateral anterior column reconstruction using a mesh cage was followed by posterior decompression and instrumented fusion.
Postoperative wound necrosis with purulent discharge developed. Irrigation and debridement were performed on postoperative Day (POD) 14. Persistent deep wound infection required repeat debridement and removal of all posterior pedicle screws on POD 43. Attempted mesh cage removal was unsuccessful because of firm fixation. No additional internal fixation was performed, and the patient remained bedridden during open-wound management. On POD 46, bradycardia and cyanosis preceded cardiac arrest, and cardiopulmonary resuscitation restored spontaneous circulation. Further debridement and erector spinae local advancement flap coverage were performed on POD 58. On POD 103, ventricular fibrillation and recurrent cardiac arrest occurred, and the patient died despite prolonged resuscitation (Fig. 6).
(A) Postoperative wound necrosis with purulent discharge was observed; (B) clinical photograph obtained on POD 96 after debridement and local advancement flap coverage, showing the residual open wound.
Discussion
Dialysis-associated DSA has classically been linked to β2-microglobulin amyloid deposition and predominantly affects the intervertebral discs and adjacent vertebral endplates [1–3]. It should be distinguished from renal osteodystrophy and osteoporosis, which may coexist and contribute to skeletal fragility [8, 9]. Differentiation from infectious spondylitis is challenging because both conditions can cause endplate destruction, disc-space narrowing, and spinal instability [1, 3]. In these cases, DSA was considered retrospectively based on prolonged hemodialysis and the clinical and radiological findings. Lumbar biopsies were nonspecific, and amyloid-specific staining, including Congo red staining, was not performed. Additional staining of archived specimens was subsequently considered but could not be performed. The pathological findings therefore did not establish amyloid deposition as the cause of the destructive lesions. Likewise, negative microbiological studies did not confirm a noninfectious process. The small yellowish abscess-like pockets encountered in Case 2 further limited the exclusion of a concomitant infection. Accordingly, we describe these cases as presumed dialysis-associated DSA, reflecting the overall clinical context rather than definitive histopathological confirmation.
Poor bone quality can compromise fixation, while systemic comorbidities complicate perioperative management in patients undergoing long-term hemodialysis [6, 7, 9, 10]. Case 1 illustrates the difficulty of securing anterior fixation and the need to modify the operative plan when screw purchase is inadequate. The maintained reconstruction at 24 months documents the absence of cage subsidence or implant failure, but does not alone establish solid fusion. Case 2 illustrates the challenges of a reconstruction complicated by a deep wound infection and implant removal. These observations support the careful assessment of bone quality and systemic disease when planning surgery, but do not establish the superiority of a particular surgical strategy. Although tissue sampling and microbiological evaluation can aid diagnosis when clinically feasible, histological confirmation is not always required before treatment, particularly in patients with progressive neurological compromise.
This report is limited by a retrospective diagnostic assessment without amyloid-specific confirmation; therefore, occult or concomitant infection cannot be completely excluded. Furthermore, differences in lesion locations, procedures, and comorbidities preclude a direct comparison or the attribution of the contrasting outcomes to any single factor. These cases illustrate diagnostic and perioperative challenges rather than establishing determinants of surgical success or failure.
Conclusion
Presumed dialysis-associated DSA should be considered in patients on long-term hemodialysis who present with destructive spinal lesions. Negative biopsy and microbiological findings do not definitively exclude infection. Surgical planning for mechanical instability or progressive neurological deficits should account for poor bone quality and systemic comorbidities, with careful interpretation of the available diagnostic evidence.
Conflicts of interest
None declared.
Funding
None declared.
References
Author notes
Seong Gyu Kim and Yeong Ha Jeong contributed equally to this work and share first authorship.
