Case Presentation: A 37-year-old male with a history of intravenous drug use (IVDU), recurrent skin infections, and treated hepatitis C presented to the ED with 4 days of loose bloody stools, nausea, chest pain with radiation to the back, and tender nodular lesions on his bilateral forearms. He denied recent IVDU.Initial exam, vitals, and labs (CBC, CMP, lactate) were unremarkable. His EKG, head CT without contrast, and chest CTA showed no acute pathology. Blood cultures were obtained, and he was discharged with cephalexin for presumed bilateral upper extremity cellulitis. He was advised to return to the emergency department two days later due to positive blood cultures: one set showed Staphylococcus epidermidis, and both sets grew gram-negative rods. He was admitted for further evaluation.He was afebrile with normal vital signs. Exam revealed erythematous nodules (1-2 cm in diameter) on the bilateral forearms and dorsal hands and an area of induration and erythema on the right forearm (Figure 1). He was cultured and started on cefepime and vancomycin. Burkholderia gladioli grew in both initial sets of blood cultures; Microbacterium was isolated in one set. He was switched to meropenem and vancomycin. His HIV serologies, RPR, and hepatitis C viral RNA were negative. Biopsy of the cutaneous lesions revealed no abscess formation and nonspecific lymphocytic and neutrophilic infiltrate (Figure 2); the culture grew B. gladioli consistent with hematogenous seeding. His surveillance blood cultures remained negative.He was diagnosed with B. gladioli bacteremia, the source of which remained unclear. He improved on meropenem and vancomycin, then was discharged to complete a course of oral levofloxacin, based on susceptibility testing.
Discussion: Burkholderia gladioli is a gram-negative environmental organism known for causing flower rot and first recognized as a human pathogen in the early 1990s.1 It primarily infects individuals with cystic fibrosis, chronic granulomatous disease, and other immunocompromised states. B. gladioli is highly susceptible to lysis in human serum, suggesting that most healthy individuals are protected.2-3 Even among the immunocompromised, it remains an extremely rare pathogen often linked to poor prognosis.1Due to its rarity, information on B. gladioli’s clinical manifestations is limited. It has been associated with various conditions, including respiratory infections, septicemia, abscesses, osteomyelitis, keratitis, adenitis, and neonatal sepsis.3-4 Notably, there is only one case of B. gladioli-related abscesses in an immunocompetent patient after an intradermal filler injection, and abscesses are typically localized, with only one case involving multiple skin sites.2Treatment is challenging due to resistance mechanisms, including beta-lactamase production and biofilm formation.5 The organism is generally susceptible to ciprofloxacin, imipenem, and ticarcillin-clavulanic acid, but it shows resistance to aminoglycosides and is fully resistant to colistin.3,4 This case is unique, presenting a rare cause of bacteremia, without typical risk factors, and unusual skin manifestations not previously reported.
Conclusions: This case highlights the rarity of Burkholderia gladioli as a cause of bacteremia in an immunocompetent host, along with its unusual skin manifestations, underscoring the importance for hospitalists to recognize such atypical presentations for timely diagnosis and treatment.
