J Clin Ultrasound 20:217-220, MarchiApril 1992 CCC 0091-2751/92/030217-04 $04.00 0 1992 by John Wiley & Sons, Inc.
Case Report
Aspergillus Osteomyelitis of the Rib: Sonographic Diagnosis Claudio Cartoni, MD,* Alberta Capua, MD,t Costantino Damico, MD,* and Giuseppe Potente, MDt
Aspergillosis is an emerging opportunistic infection observed in immunocompromised patients. Besides the usual ways of presentation such as the pulmonary, gastrointestinal, cerebral, and disseminate forms, bone and joint Aspergillus infections have been infrequently reported.' This paper describes the first case of a child treated for leukemia with rib Aspergillus osteomyelitis detected in its early stages by ultrasonography and diagnosed by needle aspiration puncture. *Hematology, Department of Human Biopathology, and tInstitute of Radiology, University La Sapienza of Rome, Roma, Italy. For reprints contact Claudio Cartoni, MD, Cattedra di Ematologia, via Benevento 6, 00161 Roma, Italy.
Radiographic and computed tomography (CT) findings of the affected rib are also described. CASE REPORT
A 13-year-old boy was referred in December 1988 to our institution with hematologic relapse of acute non-lymphocytic leukemia. The patient had been previously treated elsewhere, and during the first course of chemotherapy he developed a pulmonary infection characterized by bilateral nodular infiltrates as seen in a standard chest radiograph. Some of these infiltrates were cavitated and were interpreted as localizations of fungal infection (mycetomas). Therefore, anti-
FIGURE 1. Oblique sonogram of the anterior part of the seventh right rib (R) shows an anechoic area with scattered internal echoes protruding from a fenestration (arrow) into the subcutaneous space (arrowheads).
217
218
CASE REPORT: CARTON1 ET AL.
FIGURE 2. The chest radiograph shows swelling and osteolysis of the right seventh rib on its anterior extremity (arrows).
FIGURE 3. CT scan shows the enlargement and the bone erosion of the seventh right rib (arrow) and its contiguity with adjacent soft tissue mass (arrowheads).
JOURNAL OF CLINICAL ULTRASOUND
ASPERGILLUS OSTEOMYELITIS
fungal chemotherapy (intravenous amphotericin B) was administered with resolution of the radiologic anomalies. When the patient was admitted in our institution, CT of the brain, chest, and abdomen did not show any evidence of leukemia or infection. He received antileukemic chemotherapy (mitoxantrone, VP-16 etoposide, and cytosine arabinoside), and during the neutropenic phase he began to complain of right thoracic pain with fever. Broad-spectrum antibiotic therapy was instituted, although chest radiographs, thoracic and abdominal CT, and multiple blood cultures had been negative. Ten days later, at the time of peripheral white blood cell count recovery, the patient was still febrile and he experienced a painful edematous swelling on the right anterior hemithorax. Ultrasonography was performed using a highresolution real-time scanner with a 5-MHz electronically focused linear array transducer (Toshiba, Sal-77B). Longitudinal, oblique, and transverse sonograms of the inflamed zone revealed a subcutaneous anechoic area with irregular borders and heterogeneous content consisting of fine internal echoes. This abnormal area, suspected to be composed of fluid because of its echo pattern, was evident above the anterior part of the seventh rib; some sonograms showed continuity with the marrow space of the rib due to a cortical destruction (Figure 1). Chest radiographs and conventional tomography revealed swelling and erosion of the anterior part of the seventh rib, consistent with osteolysis (Figure 2). Thoracic CT showed a low-density subcutaneous area spreading from the medulla of the seventh right rib with destruction of the cortical bone (Figure 3). At the same time, cerebral, thoracic, and abdominal CT scans were normal. Percutaneous needle aspiration of the lesion was performed with a disposable syringe (21gauge needle) after determining the site of puncture on the skin by ultrasonography. A darkreddish fluid was aspirated showing Aspergillus elements, a culture of which yielded Aspergillus fumigutus. The patient did not undergo surgical debridement of the affected rib because of a persistent low platelet count, and he was treated with local and intravenous administration of amphotericin B. He died 7 months later of leukemia. DISCUSSION
Aspergillus osteomyelitis is strongly associated with immunodeficiency,which may be inborn, as VOL. 20,NO.3, MARCHiAPRlL 1992
219
in granulomatous disease, or acquired, as in granulocytopenia or during corticosteroid therapy. Surgical wounds or the presence of prosthetic devices are also considered to be predisposing factor^.',^,^ Bone infection by Aspergillus is rarely reported in the literature except for cranial localizations, which differ clinically and epidemiologically from extracranial o~teomyelitis.~ In 1985 Barnwell collected 26 cases, among which those localized in vertebrae and ribs were the most common sites of Aspergillus fumigutus, which is the predominant infecting s p e ~ i e s .Recently, ~ Flynn et al. have described a child treated for leukemia who had Aspergillus osteomyelitis of the hip diagnosed by surgical b i ~ p s y The . ~ fungus usually spreads to the ribs from contiguous pulmonary infections, as reported by Caligiuri, who observed a case of chest wall invasion from a contiguous pulmonary aspergillosis,6 or else from hematogenous seeding. In our case, the previous pulmonary fungal infection occurring during the induction treatment for leukemia was diagnosed on the basis of the radiographic aspect of the pulmonary lesions (cavitated nodules) and on their complete responsiveness to the antimicotic therapy with amphotericin B. Although no roentgenographically detectable lung disease was evident at the time of osteomyelitis, secondary spread from the pulmonary lesions is possible. In this patient the fungus might have persisted in the rib despite the complete resolution of the previous pneumonia with antimicotic therapy, and it reappeared during the second neutropenic phase with involvement of the bone. In fact, unlike the pulmonary localization, bone involvement by the fungus is less responsive to treatment with amphotericin B as proved by the poor prognosis reported in the literature for patients with Aspergillus osteomyeliti~.~.~ In conclusion, CT and chest radiograph are well-established diagnostic procedures in detecting pulmonary or chest wall anomalies in patients at risk of opportunistic infections. However in the present case, sonography proved helpful in leading to a diagnosis of rib osteomyelitis with subcutaneous abscess by demonstrating the erosion of the rib, the characteristics of the fluid collection adjacent to the bone, and the extension of the abscess into the medullary cavity of bone. Moreover, ultrasonography is useful in guiding the needle aspiration biopsy and in monitoring response to specific treatment.
220
CASE REPORT: CARTON1 ET AL.
REFERENCES Jlidi R, Lagier R, Abele R, Auckenthaler R: Histological study of a chondrocostal aspergillosis case. Ann PathoZ8:49-53, 1988. Tack KJ, Rhame FS, Brown B, Thompson R: Aspergillus osteomyelitis. Report of four cases and review of the literature. Am J Med 73:295-300, 1982. Rothfield I, Eliot G, Begg CF: Aspergilloma of the sinus. N Y State J Med 72:493-495, 1972. Barnwell PA, Jelsma LF, Raff MJ: Aspergillus osteomyelitis. Report of a case and review of the literature. Case report. Diagn Microbiol Infect Dis 3:515-519, 1985.
5. Flynn PM, Magill HL, Jenkins I11 JJ et al: Apergillus osteomyelitis in a child treated for acute lymphoblastic leukemia. Pediatr Infect Dis J 9:733936, 1990. 6. Caligiuri P, MacMahon H, Courtney J , Weiss L: Opportunistic pulmonary aspergillosis with chest wall invasion. Plain film and computed tomographic findings. Arch Intern Med 143:2223-2224, 1983. 7. De Bernardi B, Garventa A, Garre ML, et al.: Two cases of osteomyelitis in acute leukemia in induction phase of treatment. Pediatr Med Chir 5:205209. 1983.
JOURNAL OF CLINICAL ULTRASOUND
Case Report
Aspergillus Osteomyelitis of the Rib: Sonographic Diagnosis Claudio Cartoni, MD,* Alberta Capua, MD,t Costantino Damico, MD,* and Giuseppe Potente, MDt
Aspergillosis is an emerging opportunistic infection observed in immunocompromised patients. Besides the usual ways of presentation such as the pulmonary, gastrointestinal, cerebral, and disseminate forms, bone and joint Aspergillus infections have been infrequently reported.' This paper describes the first case of a child treated for leukemia with rib Aspergillus osteomyelitis detected in its early stages by ultrasonography and diagnosed by needle aspiration puncture. *Hematology, Department of Human Biopathology, and tInstitute of Radiology, University La Sapienza of Rome, Roma, Italy. For reprints contact Claudio Cartoni, MD, Cattedra di Ematologia, via Benevento 6, 00161 Roma, Italy.
Radiographic and computed tomography (CT) findings of the affected rib are also described. CASE REPORT
A 13-year-old boy was referred in December 1988 to our institution with hematologic relapse of acute non-lymphocytic leukemia. The patient had been previously treated elsewhere, and during the first course of chemotherapy he developed a pulmonary infection characterized by bilateral nodular infiltrates as seen in a standard chest radiograph. Some of these infiltrates were cavitated and were interpreted as localizations of fungal infection (mycetomas). Therefore, anti-
FIGURE 1. Oblique sonogram of the anterior part of the seventh right rib (R) shows an anechoic area with scattered internal echoes protruding from a fenestration (arrow) into the subcutaneous space (arrowheads).
217
218
CASE REPORT: CARTON1 ET AL.
FIGURE 2. The chest radiograph shows swelling and osteolysis of the right seventh rib on its anterior extremity (arrows).
FIGURE 3. CT scan shows the enlargement and the bone erosion of the seventh right rib (arrow) and its contiguity with adjacent soft tissue mass (arrowheads).
JOURNAL OF CLINICAL ULTRASOUND
ASPERGILLUS OSTEOMYELITIS
fungal chemotherapy (intravenous amphotericin B) was administered with resolution of the radiologic anomalies. When the patient was admitted in our institution, CT of the brain, chest, and abdomen did not show any evidence of leukemia or infection. He received antileukemic chemotherapy (mitoxantrone, VP-16 etoposide, and cytosine arabinoside), and during the neutropenic phase he began to complain of right thoracic pain with fever. Broad-spectrum antibiotic therapy was instituted, although chest radiographs, thoracic and abdominal CT, and multiple blood cultures had been negative. Ten days later, at the time of peripheral white blood cell count recovery, the patient was still febrile and he experienced a painful edematous swelling on the right anterior hemithorax. Ultrasonography was performed using a highresolution real-time scanner with a 5-MHz electronically focused linear array transducer (Toshiba, Sal-77B). Longitudinal, oblique, and transverse sonograms of the inflamed zone revealed a subcutaneous anechoic area with irregular borders and heterogeneous content consisting of fine internal echoes. This abnormal area, suspected to be composed of fluid because of its echo pattern, was evident above the anterior part of the seventh rib; some sonograms showed continuity with the marrow space of the rib due to a cortical destruction (Figure 1). Chest radiographs and conventional tomography revealed swelling and erosion of the anterior part of the seventh rib, consistent with osteolysis (Figure 2). Thoracic CT showed a low-density subcutaneous area spreading from the medulla of the seventh right rib with destruction of the cortical bone (Figure 3). At the same time, cerebral, thoracic, and abdominal CT scans were normal. Percutaneous needle aspiration of the lesion was performed with a disposable syringe (21gauge needle) after determining the site of puncture on the skin by ultrasonography. A darkreddish fluid was aspirated showing Aspergillus elements, a culture of which yielded Aspergillus fumigutus. The patient did not undergo surgical debridement of the affected rib because of a persistent low platelet count, and he was treated with local and intravenous administration of amphotericin B. He died 7 months later of leukemia. DISCUSSION
Aspergillus osteomyelitis is strongly associated with immunodeficiency,which may be inborn, as VOL. 20,NO.3, MARCHiAPRlL 1992
219
in granulomatous disease, or acquired, as in granulocytopenia or during corticosteroid therapy. Surgical wounds or the presence of prosthetic devices are also considered to be predisposing factor^.',^,^ Bone infection by Aspergillus is rarely reported in the literature except for cranial localizations, which differ clinically and epidemiologically from extracranial o~teomyelitis.~ In 1985 Barnwell collected 26 cases, among which those localized in vertebrae and ribs were the most common sites of Aspergillus fumigutus, which is the predominant infecting s p e ~ i e s .Recently, ~ Flynn et al. have described a child treated for leukemia who had Aspergillus osteomyelitis of the hip diagnosed by surgical b i ~ p s y The . ~ fungus usually spreads to the ribs from contiguous pulmonary infections, as reported by Caligiuri, who observed a case of chest wall invasion from a contiguous pulmonary aspergillosis,6 or else from hematogenous seeding. In our case, the previous pulmonary fungal infection occurring during the induction treatment for leukemia was diagnosed on the basis of the radiographic aspect of the pulmonary lesions (cavitated nodules) and on their complete responsiveness to the antimicotic therapy with amphotericin B. Although no roentgenographically detectable lung disease was evident at the time of osteomyelitis, secondary spread from the pulmonary lesions is possible. In this patient the fungus might have persisted in the rib despite the complete resolution of the previous pneumonia with antimicotic therapy, and it reappeared during the second neutropenic phase with involvement of the bone. In fact, unlike the pulmonary localization, bone involvement by the fungus is less responsive to treatment with amphotericin B as proved by the poor prognosis reported in the literature for patients with Aspergillus osteomyeliti~.~.~ In conclusion, CT and chest radiograph are well-established diagnostic procedures in detecting pulmonary or chest wall anomalies in patients at risk of opportunistic infections. However in the present case, sonography proved helpful in leading to a diagnosis of rib osteomyelitis with subcutaneous abscess by demonstrating the erosion of the rib, the characteristics of the fluid collection adjacent to the bone, and the extension of the abscess into the medullary cavity of bone. Moreover, ultrasonography is useful in guiding the needle aspiration biopsy and in monitoring response to specific treatment.
220
CASE REPORT: CARTON1 ET AL.
REFERENCES Jlidi R, Lagier R, Abele R, Auckenthaler R: Histological study of a chondrocostal aspergillosis case. Ann PathoZ8:49-53, 1988. Tack KJ, Rhame FS, Brown B, Thompson R: Aspergillus osteomyelitis. Report of four cases and review of the literature. Am J Med 73:295-300, 1982. Rothfield I, Eliot G, Begg CF: Aspergilloma of the sinus. N Y State J Med 72:493-495, 1972. Barnwell PA, Jelsma LF, Raff MJ: Aspergillus osteomyelitis. Report of a case and review of the literature. Case report. Diagn Microbiol Infect Dis 3:515-519, 1985.
5. Flynn PM, Magill HL, Jenkins I11 JJ et al: Apergillus osteomyelitis in a child treated for acute lymphoblastic leukemia. Pediatr Infect Dis J 9:733936, 1990. 6. Caligiuri P, MacMahon H, Courtney J , Weiss L: Opportunistic pulmonary aspergillosis with chest wall invasion. Plain film and computed tomographic findings. Arch Intern Med 143:2223-2224, 1983. 7. De Bernardi B, Garventa A, Garre ML, et al.: Two cases of osteomyelitis in acute leukemia in induction phase of treatment. Pediatr Med Chir 5:205209. 1983.
JOURNAL OF CLINICAL ULTRASOUND
