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Bilateral acute tibial osteomyelitis in a patient without an underlying disease: a case report
© Sipahioglu et al.; licensee BioMed Central Ltd. 2014
Received: 27 March 2014
Accepted: 17 September 2014
Published: 26 November 2014
The simultaneous presentation of osteomyelitis in more than one bone is rare and is commonly accompanied by a chronic disease. Even in such cases, other conditions that arise commonly in the long bones of children—such as chronic recurrent multifocal osteomyelitis and Ewing’s sarcoma—must be ruled out.
We present the case of a 5-year-old boy with bilateral acute tibial osteomyelitis without an underlying chronic disease who was treated with surgical debridement. We also review the pertinent literature.
Early diagnosis, appropriate antibiotic therapy, and timely surgical intervention—including proactive efforts to prevent fractures—all increase the chance of a successful outcome for these patients.
Acute hematogenous osteomyelitis is the most common type of bone infection. The annual incidence of this infection is 1:5000 children younger than 13 years of age, and most of them are younger than 5 years of age. It arises characteristically in the metaphysis of long bones, such as the femur, tibia, and humerus . Surgery followed by antibiotic therapy is a suitable approach for patients who do not respond to parenteral antibiotics . The recommended duration of antibiotic therapy is 4 to 8 weeks, although successful outcomes have been reported in uncomplicated cases with a mean of only 23 days of therapy . The simultaneous involvement of two or more bones has rarely been reported in the literature. When it does occur, it is usually accompanied by an underlying chronic disease, such as sickle cell anemia . In this report, we present a case of osteomyelitis with bilateral tibial involvement that developed in a child without a chronic disease or preceding trauma. Our report is highlighted by a discussion of the differential diagnosis.
After diagnosing his condition, we carried out open irrigation and surgical drainage in his legs. After opening cortical fenestrations in the metaphysis of each tibia, we performed curettage of the medullary canal, carefully avoiding the growth plate. Approximately 150cc of purulent fluid was drained from the left tibia and approximately 200cc from the right tibia. Microbiological and pathological samples were taken during surgery for Gram staining, blood and bone cultures, staining for acid-resistant bacilli, and a TB culture, all of which were negative. The results of a pathological examination were consistent with acute osteomyelitis. In our search for an underlying chronic disease, we carried out the following tests, all of which produced no indications of any abnormalities: urinalysis; a routine biochemical analysis; a peripheral blood smear; hemoglobin electrophoresis (to rule out sickle cell anemia); an abdominal ultrasound; immunoglobulin and complement factor analysis; tests for rheumatoid factor, antinuclear antibody, anti-Smith antibody, and anti-ribonucleoprotein antibody; intermittent Salmonella agglutination tests; VDRL; and urine and blood cultures.
Osteomyelitis is rarely seen in more than one bone at the same time. When it does appear simultaneously in two or more bones, it is usually accompanied by a chronic illness, such as sickle cell anemia. For example, Bachmeyer and colleagues reported bilateral tibial osteomyelitis caused by Pantoea agglomerans (a rare gram-negative bacillus) in a patient with sickle cell anemia . Other disorders seen with bilateral tibial osteomyelitis include Guillain–Barré syndrome, retroperitoneal fibrosis, and systemic lupus erythematosus. Javaloyas and colleagues treated two patients who developed bilateral symmetric osteomyelitis caused by a Gram-negative bacillus after a latent period following an episode of sepsis . Picillo and colleagues reported the case of a patient with systemic lupus erythematosus and arthritis in the knee, accompanying bilateral femoral septic osteomyelitis caused by Salmonella enteritidis. In some cases, an underlying condition is not found. Crook, for example, carried out numerous tests to search for an underlying condition in a patient with bilateral tibial osteomyelitis caused by Staphylococcus aureus, but every test was negative . Similarly, we failed to identify any underlying chronic disorders in our patient, other than anemia, and his hemoglobin level (the basis of the diagnosis) returned to normal after treatment. At his 18-month follow-up visit, we found no new signs or symptoms and concluded that he did not have any other disease.
Chronic recurrent multifocal osteomyelitis is another disorder that needs to be ruled out in the differential diagnosis of bilateral osteomyelitis. King and colleagues proposed the following diagnostic criteria: two or more bone lesions identified radiographically, pain and swelling lasting at least six months (including periods of remission), and failure to respond to antibiotic therapy given for at least one month . Chronic recurrent multifocal osteomyelitis is usually accompanied by other inflammatory conditions, such as palmoplantar pustulosis, psoriasis, inflammatory bowel disease, and acne. A biopsy often reveals acute and chronic inflammation with lymphocyte dominance, but cultures are almost always negative. The pathogenesis of this condition remains unknown, although an autoinflammatory process or an infection by an unrecognized causative agent has been suggested . Because our patient responded to antibiotic therapy and purulent material was drained from both tibias, we were able to exclude this diagnosis.
Ewing’s sarcoma—the second most common primary malignant tumor of bone in children—should also be ruled out. Ewing’s sarcoma has a predilection for individuals ages 10 to 20 years. Its initial symptoms may include pain and swelling. X-rays reveal radiolucent areas suggestive of a periosteal reaction that sometimes can be distinguished from osteomyelitis only by histopathological examination . Mycobacterium tuberculosis, which can have signs of extrapulmonary involvement, may also cause an osteoarticular infection, although this is not common . Tuberculous osteomyelitis can be diagnosed by isolation of Mycobacterium tuberculosis from cultures of bone biopsy material. A positive tuberculin skin test, Ziehl–Neelsen staining, polymerase chain reaction assays, radiological findings, and histopathology of the bone lesions are also helpful in confirming the diagnosis. We were able to exclude both TB and Ewing’s sarcoma on the basis of laboratory and histopathological findings.
The simultaneous presentation of osteomyelitis in more than one bone is rare and is commonly accompanied by a chronic disease. As we observed in our patient, it can also develop in the absence of an underlying disease. Even in such cases, other conditions that arise commonly in the long bones of children must be ruled out. Early diagnosis, appropriate antibiotic therapy, and timely surgical intervention all increase the chance of a successful outcome for these patients.
Written informed consent was obtained from the patient’s father for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
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