(Radiology. 1999;210:515-517.)
© RSNA, 1999
Fatty Infiltration of Osseous Structures: A Long-term Complication of Oleothorax—Case Report
Benjamin J. Freedman, MD1,
Denise M. McCarthy, MD1,
Frieda Feldman, MD1 and
Nikki Feirt, MD1
1 Department of Radiology, Columbia-Presbyterian Medical Center, 622 W 168th St, New York, NY 10032-3784.
 |
Abstract
|
|---|
Thoracic imaging of a patient treated for pulmonary tuberculosis with oleothorax therapy before the antibiotic era demonstrated a rare complication. Gross invasion by lipid with subsequent pathologic fracture of the adjacent thoracic vertebra may give rise to symptomatic spinal cord compression. Magnetic resonance imaging is a useful modality for help in diagnosing treatment complications of oleothorax.
Index terms: Oleothorax, 60.459 • Pleura, 66.459 • Ribs, 471.23, 471.249 • Spine, infection, 32.232 • Spine, MR, 32.121411, 32.121415 • Spine, radionuclide studies, 32.1216 • Thorax, CT, 471.12111 • Tuberculosis, 32.232
|
Case Report
|
|---|
The patient was a 72-year-old woman who contracted pulmonary tuberculosis in 1944 while an inmate at the Auschwitz concentration camp in Poland. She was treated in 1948 in Heidelberg, Germany, with left pleural paraffin (oleothorax therapy), which helped relieve her symptoms. She did not receive antibiotics, nor was the oleothorax drained. The patient had been in excellent health before coming to medical attention in 1997.
In June 1997, she presented at our institution with a 3-month history of middle-thoracic back pain, unsteady gait, and progressive mild four-extremity weakness (lower greater than upper). She reported a 10-lb (4.54-kg) weight loss over the 3 months but had no respiratory complaints, fevers, chills, or night sweats. At the onset of symptoms in March 1997, computed tomography (CT) of the chest, performed elsewhere, demonstrated a large left oleothorax. At that time, specimens obtained at CT-guided thoracentesis and needle biopsy of the left pleura yielded negative cultures.
The patient subsequently developed a large, firm, tender, progressively expanding left axillary mass. The patient was afebrile at the time of admission and had a normal white blood cell count. Neurologic findings included brisk lower extremity deep tendon reflexes, unsteady gait, and a positive Romberg sign. Laboratory values disclosed an elevated erythrocyte sedimentation rate (110 mm/h). All other laboratory values were unremarkable.
A chest CT scan obtained without contrast material enhancement revealed a 10-cm-diameter heterogeneous left axillary mass of predominantly fluid and soft-tissue attenuation, with focal regions of fat attenuation in contiguity with the known oleothorax at the level of the anterolateral left first intercostal space. The large pleural fat-containing collection abutted the osseous elements, eroded the posterior left third rib, and directly infiltrated its marrow. CT numbers obtained over the latter bone measured -31 HU, which indicated fat content (Figs 1–3).
View larger version (124K):
[in this window]
[in a new window]
|
Figure 1. Axial CT image obtained at the T3 level. Rounded areas of hyperlucency (straight arrow), which represent focal fatty collections, infiltrate the vertebral body and adjoining left rib. Note neighboring intrathoracic collection with fat-fluid level (curved arrow).
|
|
View larger version (153K):
[in this window]
[in a new window]
|
Figure 3. Axial CT image. Rounded fat collections (-31 HU) can be seen in the left third rib (cursor, 1). Note the left axillary phlegmon (arrow).
|
|
A whole-body bone scan demonstrated abnormally increased radiotracer uptake in the third thoracic vertebra and the left third and fourth posterior ribs. The osseous structures were otherwise normal (Fig 4).
View larger version (75K):
[in this window]
[in a new window]
|
Figure 4. Whole body bone scan. Abnormal radiotracer uptake is visible in T3 vertebra and in the left third and fourth posterior ribs (arrows), with no evidence of a distant osseous abnormality.
|
|
The presumptive diagnosis was left axillary tubercular phlegmon. Cultures of percutaneous 14-F catheter drainage were positive for Pseudomonas species, with no acid-fast bacilli identified. The patient developed a persistent low-grade fever despite appropriate antibacterial and antimycobacterial chemotherapy. Her neurologic symptoms progressed.
Thoracic spine magnetic resonance (MR) imaging was performed by using a 1.5-T magnet (Signa; GE Medical Systems, Milwaukee, Wis) equipped with a dedicated spine coil. Axial and sagittal spin-echo T1-weighted (repetition time msec/echo time msec, 450/14) and T2-weighted (4,000/140) images were obtained both with and without fat suppression.
A complex upper-thoracic fat-containing collection was identified abutting the third thoracic vertebral body. Areas of markedly increased T1 and heterogeneous T2 signal intensities were noted in the third thoracic vertebral body, as well as in the posterior arch of the fourth thoracic vertebra and the posterior left third rib. The areas of abnormally increased signal intensity were uniformly suppressed on the fat-saturated images (Fig 5). Intervertebral disk spaces were preserved. Spinal canal stenosis with mild cord compression and edema were noted at the T3–T4 level (Fig 6). The findings were compatible with fatty infiltration of the osseous structures secondary to direct invasion by lipid elements of the adjacent oleothorax rather than primary tuberculous osteomyelitis. Such a fatty infiltrative process may theoretically stimulate a secondary inflammatory response with subsequent mycobacterial superinfection.
View larger version (98K):
[in this window]
[in a new window]
|
Figure 5a. Sagittal T1-weighted spin-echo MR images. (a) Image obtained without fat suppression (450/14) shows a thick-walled collection with central high-signal-intensity fat (curved arrow) in the posterosuperior thorax, with abnormal marrow signal intensity (straight arrow) in the immediately adjacent T3 vertebra. (b) Image obtained with fat suppression (600/14) shows uniform intracavitary (curved arrow) and intravertebral (arrow) decreased signal intensity.
|
|
View larger version (112K):
[in this window]
[in a new window]
|
Figure 5b. Sagittal T1-weighted spin-echo MR images. (a) Image obtained without fat suppression (450/14) shows a thick-walled collection with central high-signal-intensity fat (curved arrow) in the posterosuperior thorax, with abnormal marrow signal intensity (straight arrow) in the immediately adjacent T3 vertebra. (b) Image obtained with fat suppression (600/14) shows uniform intracavitary (curved arrow) and intravertebral (arrow) decreased signal intensity.
|
|
View larger version (163K):
[in this window]
[in a new window]
|
Figure 6. Axial T1-weighted spin-echo MR image (450/14). Heterogeneously increased fat signal intensity within the expanded third thoracic vertebra extends into posterior elements and ribs. Note the stenosis of the spinal canal (arrowhead).
|
|
The left axillary mass was resected en bloc with portions of the third and fourth rib. An Eloesser flap was created. Defervescence occurred promptly after surgery. Neurologic symptoms abated, and the patient was prescribed antibiotics and discharged.
Histopathologic analysis of the resected mass demonstrated caseating granulomas lining a large cavity filled with necrotic debris and a "chicken wire" pattern of inflammatory tissue alternating with fat. Gram and acid-fast bacilli stains were negative, but cultures subsequently revealed Mycobacterium tuberculosis. Histopathologic evaluation of the ribs demonstrated foci of fibrosis in the marrow cavity, which contained large empty spaces rimmed by histiocytes (Fig 7). These findings were compatible with infiltration of oil into the bone marrow and secondary foreign-body granuloma formation.
View larger version (133K):
[in this window]
[in a new window]
|
Figure 7. Photomicrograph of excisional biopsy specimen of the left third rib. Fibromuscular tissue and bone (long straight arrows) are infiltrated by lipid (short straight arrow), with an associated granulomatous response (curved arrow). (Hematoxylin-eosin stain; original magnification, x4.)
|
|
|
Discussion
|
|---|
Collapse therapy was a well-established treatment for pulmonary tuberculosis before the advent of antibiotics. Artificial pneumothorax was a technique advocated by James Carson as early as 1821 as an alternative to surgical thoracoplasty (1). Intra- and extrapleural "filling" pneumolysis was introduced as an alternate method for pulmonary compression. Various materials were used to occupy the potential pleural spaces and to displace lung parenchyma. These included autologous omental, muscle, or bone fragment grafts; inflated rubber gloves; gauze packing; fibroid tumors; liquefied human fat ("humanol"); and various lipid-based substances such as mineral oil, cottonseed oil, olive oil, and nut oils. The use of solid, moldable paraffin wax was described in 1913 (6). Paraffin had the advantages of being readily available, poorly absorbed, and relatively elastic.
The intrapleural administration of oil-based substances is termed oleothorax. In addition to exerting a mass effect on the adjacent lung, such substances were caustic and produced an obliterative pleuritis, which justified their use in tuberculous empyemas. Other caustic agents, such as extract of the myrtle plant ("gomonol"), were occasionally added to the mixture. Volumes ranged from 100 to 2,000 mL. The recommended length of therapy was up to 2 years, after which the material was to be removed. However, asymptomatic patients were often lost to follow-up, so no pleural evacuation was performed (2).
Previously described long-term complications of oleothorax include expansion of the oleothorax (3), lipoid pneumonia secondary to bronchopleural fistula formation (4,5), pleurocutaneous fistula (6), recurrent tuberculous empyema (7), mediastinal compression with superior vena cava syndrome, secondarily infected pleurolith (8), chest wall abscess, and oil embolism. Paraffinomas—that is, extruded collections of lipoid material—have been described (3) in the chest wall and mediastinum. Extrapleural oleothorax produced fewer complications than did the pleural variety (4).
The use of oleothorax was abandoned with the advent of antituberculous chemotherapy in the 1950s. In retrospect, most authors (9) concluded that the results of oleothorax therapy were generally unsatisfactory.
Long-term complications of oleothorax may manifest as osseous involvement. The authors of previous reports (8) have described nocardial infections complicating a postoleothorax pleurolith, with fungal erosion of adjacent bone. In contrast, the findings in the present case suggest direct invasion of adjacent bony structures by lipid molecules. Infiltration of the thoracic vertebra results in sufficient compromise of the osseous architecture so as to cause partial cortical collapse, with concomitant mass effect on contiguous central nervous system structures. The infiltrating lipid molecules may subsequently stimulate a granulomatous inflammatory response that mimics recurrent mycobacterial infection radiographically, as well as histopathologically. The clinical scenario must be carefully reviewed, and mycobacterial cultures must be obtained, to conclusively exclude superinfection.
Review of the current medical literature did not reveal previous use of MR imaging in the evaluation of complicated oleothorax. Although conventional radiography, CT (10,11), and nuclear scintigraphy (12) are valuable for the assessment of pathologic pulmonary manifestations, MR imaging is superior in that it can be used to simultaneously demonstrate bone as well as soft-tissue involvement. With its multiplanar capability, MR imaging is useful for assessing the extent of disease, including the degree of spinal cord compression, and fat-saturation techniques can help confirm the etiology of the fatty infiltrative process.
View larger version (125K):
[in this window]
[in a new window]
|
Figure 2. Axial CT image. Multiple large, rounded lipid globules replace the osseous elements of the posterior left fourth rib (arrow). Note the calcified left pleura (arrowhead).
|
|
|
Footnotes
|
|---|
Address reprint requests to B.J.F.
Author contributions: Guarantor of integrity of entire study, B.J.F.; study concepts, D.M.M.; literature research, B.J.F.; clinical studies, F.F., N.F.; manuscript preparation, B.J.F.; manuscript editing and review, F.F.
Received October 2, 1997;
revision requested December 15, 1997; revision received July 22, 1998;
accepted September 28, 1998.
|
References
|
|---|
-
Riviere C. The pneumothorax treatment of pulmonary tuberculosis London, England: Oxford Medical, 1917.
-
Alexander J. Extrapleural pneumolysis In: The surgery of pulmonary tuberculosis. Philadelphia, Pa: Lea & Febiger, 1925; 202-224.
-
Hutton L. Oleothorax: expanding pleural lesion. AJR 1984; 142:1107-1110.[Abstract/Free Full Text]
-
Twersky J, Himmelfarb E, Kim C. Post traumatic bronchopleural fistula: a complication of long-standing extrapleural oleothorax. Br J Radiol 1972; 45:693-694.[Abstract/Free Full Text]
-
Tsou E, Yeager H, Jr, Katz S. Late bronchopleural fistula and Hemophilus influenzae empyema complicating longstanding oleothorax: report of two cases. Chest 1978; 74:573-575.[Abstract/Free Full Text]
-
Kirshenbaum KJ, Burke RC, Kirshenbaum MD, Cavallino RP. Pleurocutaneous fistula as a complication of oleothorax: CT findings in three patients. Clin Imaging 1995; 19:125-128.[Medline]
-
Forouhi NG, Walker W, Leitch AG. Tuberculous empyema presenting 62 years after oleothorax: a case report. Respir Med 1994; 88:391-392.[Medline]
-
Taylor CE, Schwartz R, Madoff IM. Nocardiosis associated with a giant pleurolith secondary to a long-standing oleothorax. Ann Intern Med 1967; 66:960-965.
-
Kayne GG, Pagel W, O'Shaughnessy L. Oleothorax In: Pulmonary tuberculosis. 2nd ed. London, England: Oxford University Press, 1948; 609.
-
Deboisblanc BP, Burch WC, Jr, Buechner HA, Haponik EF. Computed tomographic appearance of an oleothorax. Thorax 1988; 43:572-573.[Abstract/Free Full Text]
-
Mullin DM, Rodan BA, Bean WJ, Gocke TM, Feng TS. Computed tomography of oleothorax. J Comput Tomogr 1986; 10:197-199.[Medline]
-
Groshar D, Mahmud S, Zeligsohn E. Bone and gallium scintigraphy of an oleothorax. Eur J Nucl Med 1987; 13:478-479.[Medline]
TOP
Abstract
Case Report
