British Journal of Radiology (2005) 78, 639-641
© 2005 British Institute of Radiology
doi: 10.1259/bjr/33935946
Pulmonary benign metastasising leiomyomas presenting as fluid-containing masses on CT in a patient with two unrelated malignancies
A Osadchy, MD1,
T Zehavi, MD, PhD2 and
R Zissin, MD1
Departments of 1 Diagnostic Imaging and 2 Pathology Meir Hospital, Sapir Medical Center, Kfar-Saba, 44281, Israel, affiliated to the Sackler school of Medicine, Tel-Aviv University, Tel-Aviv, Israel
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Introduction
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Benign metastasising leiomyoma (BML) is a rare entity with less than 100 documented cases published in the medical literature usually as scattered case reports [1, 2]. BML originates from an antecedent leiomyoma of the uterus in virtually all cases. The affected women are usually asymptomatic and present with multiple lesions incidentally discovered on routine chest radiographs [1–3]. We have recently encountered a case of BML discovered in a young woman with two known different malignancies and we wish to present the CT findings and to review the literature.
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Case report
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A 49-year-old woman was admitted for the evaluation of a right anterior neck swelling of 1-week duration. Her medical history included hysterectomy 20 years previously for a uterine leiomyoma. Physical examination revealed enlargement of the right lobe of thyroid gland. Laboratory tests were unremarkable. Chest radiograph showed multiple, bilateral, well-circumscribed pulmonary masses ranging in size from several centimetres up to 13 cm (Figure 1a
). Ultrasound examination of the cervical lesion revealed a hypoechoic solid mass within the right lobe of the thyroid. Subsequent fine needle aspiration (FNA) under ultrasound guidance disclosed papillary adenocarcinoma. Chest and abdominal CT, following both oral and intravenous contrast medium, were than performed. Multiple pulmonary masses of varying size were seen, scattered throughout both lungs (Figure 1b, c). All lesions were smoothly marginated and had well-defined borders. On the soft tissue (mediastinal) window settings the lesions were apparently multiloculated, fluid containing cystic lesions, with multiple septations, several of which were slightly calcified (Figure 1b). A non-homogeneous soft tissue mass, of approximately 5 cm in diameter, was incidentally found within the left kidney. Ultrasound-guided FNAs from the left renal mass and from two pulmonary lesions were performed. Renal cell carcinoma was diagnosed while clear fluid was aspirated from the lung lesions without evidence of malignancy. Nephrectomy and thoracotomy with pulmonary wedge resection were initially performed while subsequent thyroidectomy was planned. At gross pathology the pulmonary mass was composed of numerous cystic lesions, 3–30 mm in size. Microscopic examination revealed benign spindle cells, stained for smooth muscle markers (HHF 35, desmin and actin), entrapping bronchiolar structures, consistent with BML.
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Figure 1. A 49-year-old woman presented with neck mass. (a) A chest radiograph reveals numerous well-defined pulmonary masses ranging in size up to 13 cm. Enhanced helical CT with (b) soft-tissue and (c) lung window settings the lung bases show large, multiloculated fluid filled masses with internal septation (black arrow), with calcified septum in one (white arrow).
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The post-operative course was uneventful.
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Discussion
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Multiple pulmonary BMLs are rare lesions that have been sporadically reported in the worldwide medical literature [1–4]. They are often incidentally discovered on chest radiographs of asymptomatic mature women, performed for unrelated reasons. Typically, these women had undergone hysterectomy for benign leiomyomas several months to many years earlier. Despite the high incidence of uterine leiomyomas in the general population, BML is a rare condition. The origin of the tumour remains controversial; initially regarded as malformative tumours, they were later interpreted as haematogenous metastases of uterine leiomyomas via pelvic venous channels to extrauterine organs, the lung being the most common site of involvement [1–4]. These tumours typically respond to hormonal therapy and have a benign course, showing regression on progesterone treatment and progression on oestrogen regimen [1, 2]. Biopsy, however, is needed to confirm what is likely to be a benign diagnosis, and regular follow up is required [4].
BML is histologically similar to pulmonary fibroleiomyomatous hamartoma that appear in men and children from an extrauterine source hence have a different clinical setting and response to treatment [4, 5]. Other multifocal leiomyomatous lung lesions that should be differentiated from BML include metastatic leiomyoma and lymphangioleiomyomatosis [5]. The latter is, however, characterized by different imaging features including reticulonodular and honeycomb pattern on chest radiographs and numerous thin-walled lung cysts on HRCT, as well as distinct pathological findings.
Regarding the imaging findings, BML characteristically present as numerous well-defined pulmonary nodules of various size ranging from few millimetres to several centimetres that may rarely cavitate [1–7]. To our knowledge tiny calcification, as was seen in several lesions in our patient, has not been previously described. Based on the published data, the nodules typically neither calcify nor enhance following intravenous contrast injection [1]. The enormous pulmonary masses were surprisingly fluid filled. We, however, found no information regarding the density of most of the nodules detected on CT, and furthermore, most of the published figures are shown on lung window settings only that do not express their density (Figure 1c
). We did find a case of pulmonary leiomyomatosis with giant cyst formation published in 1988, when CT was not available. In that case, followed for 20 years with gradual increasing of the lesions, some solid round shadows had changed into cystic lesions on chest radiographs due to their endobronchial extension [8]. Cavitation of the lesions with complicating pneumothorax has also been reported recently, and it may not necessarily imply sinister pathology [7].
Recently, we have examined another asymptomatic, healthy woman after hysterectomy for leiomyoma with multiple nodules disclosed incidentally on a chest radiograph. On CT, the nodules were sharply marginated and composed of fluid density (Figure 2). A presumed pre-operative diagnosis of BML was made which was further confirmed on surgery.
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Figure 2. A 64-year-old asymptomatic woman, after hysterectomy for leiomyoma, was referred for chest CT for incidental lung nodules detected on a routine chest radiograph. Contrast enhanced CT at the lung bases shows two well-circumscribed lung nodules (arrows). The larger nodule is composed of fluid density, measuring 14 Hounsfield units (thicker arrow).
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The explanation for the fluid content of the masses is obscure and requires further investigation.
To conclude, our case is unique not only for the rarity of pulmonary BML but also for its coincidence with two different malignancies. Radiologists should be aware of the possibility of this unusual entity and include it in the differential diagnosis in the appropriate clinical setting of an asymptomatic woman after hysterectomy for leiomyoma, when multiple, fluid-containing pulmonary nodules are seen on CT.
Received for publication November 18, 2004.
Revision received February 3, 2005.
Accepted for publication February 7, 2005.
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References
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- Camenzuli A, Twaite E, Huda B, Haqqani M, Warburton CJ, Curtis J. Cavitation in lung masses from benign metastizing leiomyomatosis. Clin Radiol Extra 2004;59:83–5.[CrossRef]
- Uyama T, Monden Y, Harada K, Sumitomo M, Kimura S. Pulmonary leiomyomatosis showing endobronchial extension and giant cyst formation. Chest 1988;94:644–6.[Abstract/Free Full Text]
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Introduction
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