Bacille Calmette-Guérin (BCG) spondylitis with adjacent mycotic aortic aneurysm after intravesical BCG therapy: a case report and literature review

Abstract

BackgroundAlthough intravesical bacille Calmette-Guérin (BCG) therapy is accepted as an effective treatment for bladder cancer, serious complications may occur in rare cases. To date, only 4 cases have been reported in which the patient developed a combination of mycotic aortic aneurysm and BCG spondylitis. Accurate diagnosis of BCG spondylitis is important because it is an iatrogenic disease, and its treatment is different from usual tuberculous spondylitis. However, distinguishing BCG spondylitis from usual tuberculous spondylitis is very difficult and takes a long time. In this study, we were able to suspect BCG spondylitis at an early stage from the result of the interferon-gamma release assay (IGRA).Case presentationWe encountered a case of BCG spondylitis with adjacent mycotic aortic aneurysm after intravesical BCG therapy in a 76-year-old man. We performed a 2-stage operation to obtain spine stabilization and replace the aneurysm with a synthetic graft. We started multidrug therapy with antituberculosis medication, excluding pyrazinamide, because the patient’s history of BCG therapy, negative IGRA, and positive of tuberculosis-polymerase chain reaction (Tb-PCR) suggested that the pathogenic bacteria of the spondylitis was BCG. Eventually the bacterial strain was identified as BCG by PCR-based genomic deletion analysis.ConclusionsBCG infection should be considered in patients who have been treated with BCG therapy, even if the treatment was performed several months to several years previously. In the case of a patient with a history of BCG therapy, a positive Tb-PCR result and negative IGRA result probably suggest BCG infections, if the possibility of false-negative IGRA result can be excluded.

Clinical Spectrum of Complications Induced by Intravesical Immunotherapy of Bacillus Calmette-Guérin for Bladder Cancer

“…Osteitis is rare secondary to BCG immunization, occurring in less than 37 per 100,000 cases [ 56 ]. Updated to 2018, only 23 cases of tuberculous spondylitis or vertebral osteomyelitis following intravesical BCG were reported in the English literature [ 57 ]. All these patients were male, with a mean age of 73.2 (35-94) years and with a median interval of 1.3 years (0.5 months-12 years) between BCG instillation and the onset of spondylitis.…”

Complications of intravesical BCG therapy in non-muscle invasive bladder cancer: our tertiary care centre experience

“…A psoas muscle abscess has been stated in the absence of vertebral involvement. In the literature up to 2018, only 23 cases of tuberculous spondylitis/vertebral osteomyelitis following intravesical BCG were reported [41]. Aljada et al [37] reported a case of Pott's disease after BCG instillation.…”

Tuberculous Spondylitis after Intrarenal Bacille Calmette-Guerin (BCG) Therapy

“…Intrarenal BCG is effective in the treatment of upper urinary tract CIS in a long-term follow-up 1. Although BCG therapy is the gold standard of care for high-risk patients intolerant to resection, it can cause rare but serious complications, including sepsis, miliary tuberculosis and spondylitis 2…”

“…The timeframe for BCG spondylitis onset varies considerably (0.5 months–12 years) 2. Leukocyte counts are usually normal, and CRP levels are lower than those seen in pyogenic vertebral infections.…”

“…Leukocyte counts are usually normal, and CRP levels are lower than those seen in pyogenic vertebral infections. In addition, T-SPOT.TB negative results are likely to indicate BCG infections with a deletion of region of difference 1 (RD1), which is present in the DNA of M. tuberculosis complex except with BCG 2. BCG spondylitis should be considered in patients with back pain who have received BCG therapy, even when the treatment was performed several months to several years previously 2…”