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Emphysematous osteomyelitis of the spine mimicking esophageal rupture

      Case Presentation

      An 89-year-old Japanese woman with diabetes mellitus was transferred to the emergency department because of fever and altered mental status. The patient lived alone, and her medical history was unclear because of loss of consciousness. The serum glucose level was 24.1 mmol/L. Gross hematuria was seen. The blood sample could not be tested because of hemolysis. Sepsis was suspected, and the patient was administrated intravenous infusion and antibiotics and was intubated because of unstable breathing.
      A computed tomography scan revealed emphysema around the 8th to 11th thoracic vertebrae and intraosseous air of the 8th and 9th thoracic vertebrae (Fig. 1A, B). Dilatation of the gallbladder with choledocholithiasis was also seen. We at first suspected of esophageal rupture because emphysema was seen adjacent to the esophagus, and examined upper gastrointestinal endoscopy, which revealed no sign of rupture. We then suspected acute cholangitis as a cause of sepsis. Endoscopic retrograde cholangiopancreatography revealed obstructed gallstone and dilated common bile duct, and endoscopic sphincterotomy drained suppurative bile away. Bacterial culture of venous blood and bile both turned out to be positive of Clostridium perfrigens and Escherichia coli. The diagnosis of acute cholangitis and secondary emphysematous osteomyelitis was made. Intravascular hemolysis was thought to be due to C. perfrigens infection. The patient undertook intensive care, surgical intervention, and long-term antibiotics therapy, and her condition was improved.
      Emphysematous osteomyelitis is a rare but fatal disease. The infection is mostly developed hematogenously, and the commonly affected sites are the vertebrae (most common), the pelvis, and the femur.
      • Sung S
      • Lee BH
      • Kim JH
      • et al.
      Emphysematous osteomyelitis of the spine: A rare case report.
      Causative organisms are gas-forming bacteria, including E. coli, Klebsiella pneumoniae, Bacteroides species, and Fusobacterium.
      • Sung S
      • Lee BH
      • Kim JH
      • et al.
      Emphysematous osteomyelitis of the spine: A rare case report.
      In this case, E. coli and C. perfrigens were both detected. Polymicrobial infection is rare as a hematogenous spread.
      • Sung S
      • Lee BH
      • Kim JH
      • et al.
      Emphysematous osteomyelitis of the spine: A rare case report.
      The infection of C. perfringens can cause intravascular hemolysis,
      • Simon TG
      • Bradley J
      • Jones A
      • et al.
      Massive intravascular hemolysis from Clostridium perfringens septicemia: a review.
      as seen in this case. Diabetes mellitus is the most important predisposing factor.
      • Sung S
      • Lee BH
      • Kim JH
      • et al.
      Emphysematous osteomyelitis of the spine: A rare case report.
      Although its mortality is extremely high, early diagnosis and appropriate treatment may improve the prognosis.
      • Simon TG
      • Bradley J
      • Jones A
      • et al.
      Massive intravascular hemolysis from Clostridium perfringens septicemia: a review.
      Emphysematous osteomyelitis developed air density in the bone. Intraosseous gas is also seen in more common conditions, including degenerative changes, osteonecrosis, and neoplasm.
      • Sung S
      • Lee BH
      • Kim JH
      • et al.
      Emphysematous osteomyelitis of the spine: A rare case report.
      Producing gas can be seen also in adjacent soft tissues.
      • Sung S
      • Lee BH
      • Kim JH
      • et al.
      Emphysematous osteomyelitis of the spine: A rare case report.
      In this case, effluent gas into the spine was detected as emphysema around the esophagus, which mimicked esophageal rupture. The imaging features of esophageal rupture include periesophageal fluid, extraluminal air, and pleural effusion.
      • White CS
      • Templeton PA
      • Attar S.
      Esophageal perforation: CT findings.
      These findings may also be detected in patients with emphysematous osteomyelitis of the spine. This case conveys a message: physicians should suspect emphysematous osteomyelitis of the spine when seeing emphysema around the esophagus and thoracic vertebrae in patients with sepsis. The mortality of emphysematous osteomyelitis of the spine is higher than that in other sites,
      • Sung S
      • Lee BH
      • Kim JH
      • et al.
      Emphysematous osteomyelitis of the spine: A rare case report.
      and we cannot emphasize too much on the importance of early diagnosis and treatment.
      In summary, emphysematous osteomyelitis of the spine may present as emphysema around the lower thoracic vertebrae and esophagus. This condition is rare but fatal, and physicians should perform early diagnosis and treatment.

      Funding source

      None.

      Declaration of Competing Interest

      The author has no conflicts of interest to declare.

      References

        • Sung S
        • Lee BH
        • Kim JH
        • et al.
        Emphysematous osteomyelitis of the spine: A rare case report.
        Medicine (Baltimore). 2020; 99: e21113
        • Simon TG
        • Bradley J
        • Jones A
        • et al.
        Massive intravascular hemolysis from Clostridium perfringens septicemia: a review.
        J Intensive Care Med. 2014; 29: 327-333
        • White CS
        • Templeton PA
        • Attar S.
        Esophageal perforation: CT findings.
        AJR Am J Roentgenol. 1993; 160: 767-770