Nontyphoidal Salmonella Multiple Osteomyelitis in a Child With Sickle Cell Anemia

To the Editors: Here, we report a case of multiple Nontyphoidal Salmonella (NTS) osteomyelitis with good prognosis. A 2-year-old black child with a diagnosis of sickle cell anemia at birth was given long-term oral folic acid and penicillin and repeatedly suffered abdominal pain and pneumonia. The child developed a high fever and abdominal pain with a body temperature of 39 °C. No cough, expectoration, diarrhea, convulsions or other associated symptoms were observed. Blood culture, urine culture, cerebrospinal fluid examination and chest radiograph were taken. Neither abscess nor vegetation was observed in the abdominal contrast CT and the cardiac ultrasound, respectively. Ceftriaxone was carried out empirically. The blood culture showed the growth of Salmonella group C, which produced extended-spectrum β-lactamase (ESBL). The drug susceptibility testing was as follows: ciprofloxacin was intermediate; ceftriaxone, trimethoprim-sulfamethoxazole and ampicillin were all resistant; imipenem-cilastatin and azithromycin were susceptible. Accordingly, ceftriaxone was switched to meropenem combined with azithromycin. Occasionally, the child was found that his crying behaviors were bound up with the limbs’ passive movements. The CT scan of bilateral limbs suggested left humerus pathologic fracture. The contrast magnetic resonance imaging of the entire spine and limbs revealed left humerus osteomyelitis and suspicious osteomyelitis in the spine, pelvis and bilateral femur, which needed to be differentiated with abnormal bone marrow transformation due to hematologic diseases. The operation of left humerus fenestration and K-wire fixation under general anesthesia was performed. There was a mass of necrotic tissue washed out from the medullary cavity. Bone tissue culture showed the growth of ESBL-producing Salmonella group C. Nevertheless, the fever did not vanish, and the peak declined to about 38.5 °C. Then, the antibiotics were switched to imipenem-cilastatin combined with azithromycin and fosfomycin. The positron emission tomography/computed tomography was performed for screening occult foci. The result showed diffused hypermetabolism in the marrow cavity, including limbs, multiple ribs, skull, maxillofacial bone, bilateral clavicle, sternum, pelvis and several vertebral bodies of the cervicothoracic and lumbar spine, which was overall osteomyelitis caused by NTS sepsis (Fig. 1).FIGURE 1.: The PET/CT showed multiple osteomyelitis.Fortunately, the fever subsided after 5-week treatment. The child recovered and was discharged in good condition. Fosfomycin combined with azithromycin was administrated orally as a long-term medication. The child received the removal of internal fixation 1 year later and was followed up without recurrence until now. It is reported that sickle cell anemia patients have predisposition to NTS infection and usually have bad outcome.1–3 The ESBL-producing NTS is relatively rare and intractable to deal with.4 Considering the high bone concentration of imipenem-cilastatin and fosfomycin, we attempted to combine these 2 with azithromycin. No drug-related adverse effects were observed during the treatment. Usually, the antibiotic course of osteomyelitis is at least 6 weeks after the last debridement.5 The internal fixation was performed when infection was not controlled, and the multiple osteomyelitis could be hardly cleaned up. Therefore, we advised oral antibiotics till the internal fixation was removed.