British Journal of Radiology (2007) 80, e4-e6
© 2007 British Institute of Radiology
doi: 10.1259/bjr/93530580
Solitary lymphangioma of the spleen: dynamic MR findings with pathological correlation
W-C Chang, MD
C-H LIOU, MD
H-W KAO, MD
C-C HSU, MD
C-Y CHEN, MD
and
C-Y Yu, MD
Department of Radiology, Tri-Service General Hospital, National Defense Medical Center, 325, Cheng-Kung Road, Sec. 2, Neihu 114, Taipei, Taiwan, Republic of China
Correspondence: Chih-Yung Yu, MD, Department of Radiology, Tri-Service General Hospital, 325, Cheng-Kung Road, Sec. 2, Neihu 114, Taipei, Taiwan, Republic of China. E-mail: chougo2002{at}yahoo.com.tw
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Abstract
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Solitary lymphangioma of the spleen is an extremely rare, benign vasoformative tumour that typically shows no notable enhancement on imaging studies. Few descriptions of the MRI features have been reported and, to our knowledge, the findings on dynamic gadolinium-enhanced MR images have never been described. We report a case in which MR images showed diffuse and prolonged enhancement of a splenic mass. On histological correlation, splenic lymphangiomas with abundant fibrous stroma may account for this specific enhancing pattern.
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Introduction
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Splenic lymphangioma is composed of a single cyst or multiple cysts of various sizes that are lined by endothelium and filled with proteinaceous fluid. Previous case reports described non-specific findings on imaging studies, which were invariably performed without contrast enhancement [1–4]. We present a histologically proven case in which a solitary nodular mass of the spleen appeared isointense on T1 weighted MRI and inhomogeneously hyperintense on T2 weighted images. Dynamic MRI revealed progressive, peripheral enhancement during the arterial phase and prolonged, diffuse enhancement on subsequent images. To our knowledge, this unique MR appearance has not previously been described.
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Case report
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A 46-year-old woman reported mild dullness in the left upper quadrant of the abdomen that was not associated with trauma or any other systemic manifestations. Physical examination, performed when she was admitted to our institution, revealed no other signs or notable findings. The patient had no history of parasitic infection or foreign travel. Her laboratory results were normal except for a lactate dehydrogenase concentration of 1500 IU l–1 (normal range 115–245 IU l–1).
Routine abdominal ultrasound showed a 4.0 cm hypoechoic mass in the posterior aspect of the spleen, which was not enlarged. CT scans further confirmed a splenic mass, showing delayed and mildly diffuse enhancement after the administration of contrast agent. No internal soft-tissue nodules or spotty calcifications were seen in the splenic mass.
Because malignant neoplasm of the spleen could not be completely excluded, MRI of the abdomen was performed. The mass was isointense on T1 weighted images (Figure 1a
) and inhomogeneously hyperintense on T2 weighted images (Figure 1b). Dynamic MRI revealed peripheral enhancement of the tumour during the arterial phase, which become a diffuse and prolonged enhancement pattern on subsequent images (Figure 2). Although these findings suggested a benign splenic lesion, such as a haemangioma or hamartoma, the patient underwent splenectomy because of a clinical concern about neoplasm.
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Figure 1. The splenic mass showed isointensity on(a) T1 weighted images and slight inhomogeneous hyperintensity on (b) T2 weighted images.
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Figure 2. Dynamic MRI showed isointense on(a) pre-contrast fat-saturated T1 weighted images, slight peripheral enhancement of the tumour during (b) arterial phase images, and progressive peripheral enhancement with central fill-in pattern during (c) portal venous phase images and (d) delayed 10 min images.
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Final histopathological results revealed numerous dilated lymphatic spaces with abundant collagenous stroma (Figure 3). Each cystic lumen contained various amounts of watery, pink eosinophilic material. The pathological diagnosis was cavernous lymphangioma of the spleen. During the one year follow-up period, the patient recovered uneventfully.
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Figure 3. Histopathological study showed multiple thin-walled lymph spaces (white arrows) embedded in the abundant fibrous stroma (black arrows) (haematoxylin and eosin x100).
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Discussion
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Lymphangiomas are benign congenital malformations of the lymphatic system. These tumours are commonly found in the neck and axillary regions and, less frequently, in the mediastinum, retroperitoneum, extremities and viscera [1]. Solitary lymphangiomas of the spleen are extremely rare and usually found incidentally during ultrasound or CT examinations. Final pathological diagnosis is required.
As reflected in the histopathological finding of congenital dilated lymphatic channels, splenic lymphangiomas are classified as capillary, cavernous, or cystic [2]. In most solitary splenic lymphangiomas, the cystic components predominate, and CT or MR studies typically show a thin-walled, cystic mass without substantial enhancement. We presented a histologically confirmed case in which the stromal tissue was dominant and in which contrast-enhanced MR images depicted unique delayed and diffuse enhancement.
Splenic lymphangiomas can be visualized as thin-walled cystic lesions with low signal intensity on T1 weighted imaging and high signal intensity on T2 weighted imaging without substantial internal enhancement [1, 5–7]. MRI generally reflects the histological characteristics of the splenic lesion more precisely than CT [7]. Internal bleeding or a high proteinaceous content of the lesion usually accounts for the high signal intensity on T1 weighted images. T2 weighted images show multiloculated, hyperintense areas that correspond to the dilated lymphatic channels. The intervening hypointense septa always reflect the presence of fibrous connective tissue [5–7].
Dynamic MRI with gadolinium enhancement is frequently performed to characterize abdominal lesions, but it is of limited value in assessing splenic lymphangioma because this tumour is almost always non-enhancing [1, 5]. To our knowledge, diffuse and prolonged enhancement on contrast-enhanced CT and MRI has not been previously described in the literature. Our patient received a pre-operative diagnosis of splenic haemangioma or hamartoma. However, the diffuse and prolonged enhancement on MR images may have been a useful finding in differentiating benign splenic tumours from malignant lesions of the spleen, and especially in distinguishing them from nodular lesions of malignant lymphoma. On the basis of the histological correlation, we propose that the unique enhancing pattern of the splenic lymphangioma was probably due to stagnant contrast material in the abundant stromal tissue.
In conclusion, dynamic gadolinium-enhanced MRI is an important diagnostic tool for evaluating splenic lesions and should be perform in cases with atypical findings. We suggest that lymphangiomas might be considered in the differential diagnosis of a splenic mass with diffuse and prolonged enhancement on MR images. In the present case, splenic lymphangiomas with abundant fibrous stroma might have accounted for the specific enhancement pattern observed on dynamic MR images.
Received for publication October 17, 2005.
Revision received January 3, 2006. Revision received December 6, 2005.
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References
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Abstract
Introduction
