AJR 2003; 180:969-971
© American Roentgen Ray Society
Pyothorax-Associated Lymphoma: Diagnosis at Percutaneous Core Biopsy with CT Guidance
Vincent Brun1,
Marie Pierre Revel1,
Claire Danel2,
Laure S. Fournier1,
Reda Souilamas3 and
Guy Frija1
1 Department of Radiology, Georges Pompidou Hospital, 20 rue Leblanc, 75015
Paris, France.
2 Department of Pathology, Georges Pompidou Hospital, 75015 Paris, France.
3 Department of Thoracic Surgery, Georges Pompidou Hospital, 75015 Paris,
France.
Received April 1, 2002;
accepted after revision August 27, 2002.
Address correspondence to M. P. Revel.
Introduction
Pyothorax-associated lymphoma is a rare late complication of tuberculous
pleuritis or induced pneumothorax. Since the first description of
pyothorax-associated lymphoma by Iuchi et al. in 1987
[1], more than 40 cases have
been reported, mainly by Japanese authors. However, radiologists are generally
unfamiliar with pyothorax-associated lymphoma because most chest-wall tumors
seen on CT correspond to soft-tissue or bone metastases
[2]. We describe a case of
pyothorax-associated lymphoma diagnosed at percutaneous core biopsy with CT
guidance in a patient with a history of therapy for lung collapse.
Case Report
An 82-year-old man presented with a painful mass in the right lateral chest
wall. He had noticed the mass 2 months previously, and had developed weight
loss, asthenia, and night sweats. Physical examination showed a palpable hard
mass under the right clavicle and extending laterally.
The patient had a history of tuberculosis, and an artificially induced
pneumothorax had been created 48 years previously. Blood tests disclosed a
slightly elevated C-reactive protein level, an erythrocyte sedimentation rate
of 60 mm in the first hour, and a moderate lactate dehydrogenase
elevation.
Chest radiography (Fig. 1A)
showed a thickening of the upper right pleural space and of the right chest
wall. Thick calcifications of the visceral pleura were present in the right
lung. Contrast-enhanced CT (Fig.
1B) revealed a homogeneous nonenhancing soft-tissue mass of the
right chest wall, without destruction of the contiguous ribs or pleural
effusion. Chest and abdominal CT showed no other abnormalities.
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Fig. 1A. —82-year-old man with painful mass in right lateral chest
wall. Chest radiograph shows right superior thoracic pleural mass associated
with thickening of axillary soft tissue, suggesting axillary extension. Note
superficial pleural calcifications in right hemithorax, and right hilum
retraction.
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Fig. 1B. —82-year-old man with painful mass in right lateral chest
wall. Contrast-enhanced CT scan of chest shows chest wall invasion adjacent to
chronic pleural changes, which are visualized as nonenhancing homogeneous
soft-tissue density mass.
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Percutaneous core biopsy was performed with a semiautomatic 18-gauge
coaxial biopsy needle (Biosphere Medical, Louvres, France). Pathologic
analysis revealed necrotic cells that were diffusely labeled by
anti–CD20 antibodies (Dako SA, Glostrup, Denmark) and were highly
suggestive of high-grade B cell proliferation.
Surgical resection of the mass was unsuccessful. Analysis of an open biopsy
specimen confirmed the previous histologic findings and also showed that the
lymphoid proliferation was Epstein-Barr virus–encoded positive for
latent membrane protein.
Chemotherapy was ineffective, and the disease progressed locally. No
evidence of extrathoracic localization was seen. The patient died 4 months
after the initial symptoms of infectious complications after chemotherapy.
Discussion
Pyothorax-associated lymphoma occurring in patients with chronic
tuberculous empyema is relatively rare, especially in Western countries
[3,
4]. Most cases have been
reported by Japanese authors
[1,
5,
6]. Clinical features include a
general deterioration of health, pain caused by pleural invasion, and a
palpable hard mass in the chest wall. These features were all present in our
patient. There are few reported data on imaging findings in the literature,
and no article dealing specifically with radiographic patterns of
pyothorax-associated lymphoma. In the series of Jardin et al.
[3], a short description of CT
findings is given. In that series, pyothorax-associated lymphoma mainly
presented as an inhomogeneous soft-tissue mass. The degree of contrast
enhancement is not indicated. No bone destruction occurred in four of the six
reported cases, as in our patient, whereas the two remaining patients in that
series had rib destruction. MR imaging findings reported by Kinoshita et al.
[6] in one patient are
prolonged T1 and T2 relaxation times and inhomogeneous contrast enhancement
better delineated by the fat-suppression technique on contrast-enhanced
T1-weighted images. For those authors, contrast-enhanced MR images provided
information on the appropriate site for biopsy in cases in which the malignant
lymphoma is contiguous with a chronic tuberculous empyema, because empyema
does not enhance.
The interval between therapy for collapsed lung and the onset of lymphoma
was 48 years in our patient, which is within the range (22–58 years)
reported elsewhere [5]. A long
history of empyema is a known risk factor for lymphoma and may be observed in
two contexts—namely, tuberculous pleuritis and induced pneumothorax
performed for treatment of tuberculosis. In the series by Aozasa et al.
[5], patients with an induced
pneumothorax created for tuberculous pleuritis had a greater risk of
developing pleural lymphoma than similar patients without an induced
pneumothorax. However, 26% of cases of pyothorax-associated lymphoma occur in
patients with no history of induced pneumothorax.
Soft-tissue masses in the chest wall of patients with chronic pyothorax may
correspond to several disease processes
[2]. Acute reactivation of
tuberculosis is one possibility, together with aspergilloma and infection by
more common microorganisms. Neoplasms other than lymphoma, such as
mesothelioma, squamous-cell carcinoma, and soft-tissue sarcoma, have been
associated with chronic tuberculous empyema.
Because it is difficult to distinguish pyothorax-associated lymphoma from
these other diseases by means of imaging alone, biopsy is needed to confirm
the diagnosis. Percutaneous core biopsy is the simplest way of obtaining
samples of tumors close to the skin surface. There is no risk of pneumothorax
or vascular lesions because the path of the biopsy needle is extrapleural and
there is no vascular interposition. Percutaneous core biopsy obviates surgery,
which is an appreciable advantage for patients with pyothorax-associated
lymphoma, whose health status is usually poor.
Although these lymphomas are localized, local spread at diagnosis usually
rules out surgical resection. The average survival after symptom onset is
about 5 months [3]. Factors of
poor prognosis include old age, performance status, and histology.
Two reasons explain the low frequency of pyothorax-associated lymphoma in
Western countries. Lung collapse therapy, which is a major factor for
pyothorax-associated lymphoma, has been much more widely performed in
Southeast Asia, especially in Japan, than in Western countries
[5]. A second explanation for
the higher prevalence of pyothorax-associated lymphoma in Japan is the link
between the Epstein-Barr virus and pyothorax-associated lymphoma. More than
90% of Japanese are infected with the Epstein-Barr virus in early life
[7]. In situ hybridization
experiments have shown that pyothorax-associated lymphomas have high levels of
Epstein-Barr virus gene expression
[4,
8]. Chronic pleural
inflammation may favor the clonal growth of Epstein-Barr virus–infected
B cells through the production of growth factors or immunosuppressive
cytokines produced by inflammatory cells
[4]. Other Epstein-Barr
virus–associated malignancies, such as Burkitt's lymphoma and
undifferentiated nasopharyngeal carcinoma, also show geographic variations in
frequency. Our patient came from North Africa, an area endemic for the
Epstein-Barr virus.
Although chest wall primary lymphoma is uncommon, it should be considered
as a potential diagnosis in patients with chest wall masses, especially those
who have a history of induced pneumothorax and who are from areas endemic for
the Epstein-Barr virus. Diagnosis can be obtained by means of percutaneous
core biopsy, which obviates exploratory surgery.
References
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Introduction
Case Report
