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Unusual splenic hemangioma of a pediatric patient: hypointense on T2-weighted image
Clinical Imaging xxx (2014) xxx–xxx
Contents lists available at ScienceDirect
Clinical Imaging journal homepage: http://www.clinicalimaging.org
Unusual splenic hemangioma of a pediatric patient: hypointense on T2-weighted image Hatice Nursun Ozcan a,⁎, Berna Oguz a, Beril Talim b, Saniye Ekinci c, Mithat Haliloglu a a b c
Division of Pediatric Radiology, Department of Radiology, Hacettepe University Medical School, Ankara, Turkey Department of Pediatric Pathology, Hacettepe University Medical School, Ankara, Turkey Department of Pediatric Surgery, Hacettepe University Medical School, Ankara, Turkey
a r t i c l e
i n f o
Article history: Received 26 October 2013 Received in revised form 19 January 2014 Accepted 10 February 2014 Available online xxxx
a b s t r a c t Splenic hemangioma is a very rare neoplasm in children. On magnetic resonance imaging, splenic hemangioma shows high signal intensity on T2-weighted images. In this report, we present the first pediatric case of a splenic hemangioma, which was significantly hypointense on T2-weighted images. © 2014 Elsevier Inc. All rights reserved.
Keywords: MRI Child Splenic hemangioma
1. Introduction Splenic hemangioma is the most common benign primary neoplasm of the spleen and it is very rare in childhood. It is mostly diagnosed incidentally and clinic presentation is often without symptoms. Imaging features of splenic hemangiomas have been defined with magnetic resonance imaging (MRI) in pediatric patients and adults [1–3]. Most hemangiomas are smaller than 2 cm, welldefined, homogeneous, hypointense to isointense on T1-weighted images, and most commonly hyperintense on T2-weighted images compared with splenic parenchyma. On the other hand, in this paper, we present a pediatric case of splenic hemangioma that was hypointense on the T2-weighted images to the adjacent splenic parenchyma, which is a very unusual finding. 2. Case report An 11-year-old boy was referred to our institution for abdominal pain. His medical history was otherwise unremarkable. Physical examination and hematologic evaluation were normal. Initial abdominal ultrasonography showed a 35×32 mm well-defined hypoechoic lesion in the spleen without any calcification. On color Doppler images, the lesion was slightly vascularized (Fig. 1a and b). On T1weighted images, the lesion was slightly hyperintense, and on T2weighted images, it was significantly hypointense to adjacent splenic
⁎ Corresponding author. Fakülteler Mah, Dirim sok 22/3 Cebeci 06590, Ankara, Turkey. Tel.: +90 532 6967862; fax: +90 312 3112145. E-mail address: [email protected] (H.N. Ozcan).
parenchyma (Fig. 2a and b). A dynamic MRI study was performed after the administration of contrast material. Early arterial dynamic image showed no contrast enhancement and the lesion was mildly enhanced on portal venous phases. On delayed post contrast T1weighted image, the lesion became isointense to the spleen (Fig. 2c and d). Thereafter, splenectomy was performed for definite diagnosis and rule out malignancy. At gross examination, a mass with a diameter of 32 mm was seen in the spleen. Histologic examination of the splenectomy material showed a non-capsulated but limited neoplasm in the spleen. The neoplasm was composed of proliferated vascular channels of capillary size lined with a single layer of endothelium (Fig. 3a and b). There was no sclerosis or thrombosed vessel in the neoplasm. Immunohistochemically, endothelial cells were positive for CD31 and CD34 but negative for CD8. The histologic features were consistent with a cavernous splenic hemangioma. The histologic differential diagnosis of splenic hemangioma includes hamartoma, which is CD8-positive immunohistochemically. 3. Discussion Hemangioma, although rare, is the most common primary neoplasm of the spleen. Their rate of occurrence in large autopsy series has been reported as 0.03–14%, more frequent in male patients [4]. Mostly, they are small lesions that are found incidentally in otherwise asymptomatic patients. Splenic hemangioma may also occur as part of generalized angiomatosis as seen in KlippelTrenaunay syndrome. The radiologic appearance of hemangiomas ranges from solid to cystic, depending on gross morphology; however, predominantly, a hemangioma appears as a solid mass with cystic spaces [5]. On
0899-7071/$ – see front matter © 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.clinimag.2014.02.007
Please cite this article as: Ozcan HN, et al, Unusual splenic hemangioma of a pediatric patient: hypointense on T2-weighted image, Clin Imaging (2014), http://dx.doi.org/10.1016/j.clinimag.2014.02.007
2
H.N. Ozcan et al. / Clinical Imaging xxx (2014) xxx–xxx
Fig. 1. (a) Longitudinal sonogram of the spleen shows a 35×32 mm well-defined hypoechoic lesion in the spleen without any calcification. (b) On color Doppler image, the lesion was slightly vascularized.
ultrasonography, a hemangioma may manifest as a well-defined intrasplenic echogenic solid or complex cystic mass. The MRI appearance of splenic hemangioma is hypointense to isointense on T1-weighted images and hyperintense on T2-weighted images to the adjacent spleen. Ramani et al. [6] in a series of 22 splenic hemangiomas reported that 19 lesions were hyperintense, 2 lesions were isointense, and only 1 lesion was hypointense on T2-weighted images. That particular lesion was histologically found to be sclerotic. However, due to particular complicating features (e.g., hemorrhage, infarction, and thrombosis), larger hemangiomas may show various types of MRI findings. In histopathologic examination of our patient, there was no sclerosis or thrombosed vessel in the neoplasm. Dynamic MRI, after administration of gadolinium, has shown that splenic hemangiomas have three patterns of enhancement: (1) immediate homogeneous enhancement that persists, (2) early peripheral enhancement with uniform delayed enhancement, and (3) peripheral enhancement with centripetal progression but persis-
tent enhancement of a central fibrous scar [7]. In our case, the lesion was significantly hypointense on T2-weighted images, and after administration of contrast material, there was no enhancement on early arterial sequences, mildly enhanced on portal venous phase and isointense to the spleen on delayed T1-weighted images. In conclusion, most hemangiomas are hypointense to the spleen on T1-weighted images and hyperintense on T2-weighted images. The presentation of splenic hemangioma as low signal intensity on T2weighted images is exceedingly rare in adult patients. To our knowledge, our case is the first pediatric patient in the hitherto literature who has splenic hemangioma that is hypointense on T2-weighted images. It should be kept in mind in differential diagnosis of splenic lesions.
References [1] Paterson A, Frush DP, Donnelly LF, Foss JN, O'Hara SM, Bisset GS. A patternoriented approach to splenic imaging in infants and children. Radiographics 1999;19(6):1465–85.
Fig. 2. (a) Axial T1-weighted magnetic resonance (MR) image demonstrates the lesion slightly hyperintense relative to the spleen. (b) Axial T2-weighted MR image shows a hypointense lesion in the central part of the spleen. (c) Early arterial dynamic contrast-enhanced image demonstrates the lesion without contrast enhancement. (d) On delayed T1weighted image, the lesion becomes uniformly isointense.
Please cite this article as: Ozcan HN, et al, Unusual splenic hemangioma of a pediatric patient: hypointense on T2-weighted image, Clin Imaging (2014), http://dx.doi.org/10.1016/j.clinimag.2014.02.007
H.N. Ozcan et al. / Clinical Imaging xxx (2014) xxx–xxx
3
Fig. 3. (a) Non-capsulated, well-demarcated neoplasm (Hematoxylin and Eosin, original magnification ×40). (b) Endothelial cells forming vascular channels (Hematoxylin and Eosin, original magnification ×100).
[2] Abbott RM, Levy AD, Aguilera NS, Gorospe L, Thompson WM. From the archives of the AFIP: primary vascular neoplasms of the spleen: radiologic-pathologic correlation. Radiographics 2004;24(4):1137–63. [3] Panuel M, Ternier F, Michel G, Scheiner C, Bourliere B, Faure F, Guys JM, Devred P. Splenic hemangioma—report of three pediatric cases with pathologic correlation. Pediatr Radiol 1992;22(3):213–6. [4] Pines B, Rabinovitch J. Hemangioma of the spleen. Arch Pathol 1942;33:487–503.
[5] Urrutia M, Mergo PJ, Ros LH, Torres GM, Ros PR. Cystic masses of the spleen: radiologic-pathologic correlation. Radiographics 1996;16(1):107–29. [6] Ramani M, Reinhold C, Semelka RC, Siegelman ES, Liang L, Ascher SM, Brown JJ, Eisen RN, Bret PM. Splenic hemangiomas and hamartomas: MR imaging characteristics of 28 lesions. Radiology 1997;202(1):166–72. [7] Mortele KJ. Imaging of tumoral conditions of the spleen. JBR-BTR 2000;83(4):213–5.
Please cite this article as: Ozcan HN, et al, Unusual splenic hemangioma of a pediatric patient: hypointense on T2-weighted image, Clin Imaging (2014), http://dx.doi.org/10.1016/j.clinimag.2014.02.007
Contents lists available at ScienceDirect
Clinical Imaging journal homepage: http://www.clinicalimaging.org
Unusual splenic hemangioma of a pediatric patient: hypointense on T2-weighted image Hatice Nursun Ozcan a,⁎, Berna Oguz a, Beril Talim b, Saniye Ekinci c, Mithat Haliloglu a a b c
Division of Pediatric Radiology, Department of Radiology, Hacettepe University Medical School, Ankara, Turkey Department of Pediatric Pathology, Hacettepe University Medical School, Ankara, Turkey Department of Pediatric Surgery, Hacettepe University Medical School, Ankara, Turkey
a r t i c l e
i n f o
Article history: Received 26 October 2013 Received in revised form 19 January 2014 Accepted 10 February 2014 Available online xxxx
a b s t r a c t Splenic hemangioma is a very rare neoplasm in children. On magnetic resonance imaging, splenic hemangioma shows high signal intensity on T2-weighted images. In this report, we present the first pediatric case of a splenic hemangioma, which was significantly hypointense on T2-weighted images. © 2014 Elsevier Inc. All rights reserved.
Keywords: MRI Child Splenic hemangioma
1. Introduction Splenic hemangioma is the most common benign primary neoplasm of the spleen and it is very rare in childhood. It is mostly diagnosed incidentally and clinic presentation is often without symptoms. Imaging features of splenic hemangiomas have been defined with magnetic resonance imaging (MRI) in pediatric patients and adults [1–3]. Most hemangiomas are smaller than 2 cm, welldefined, homogeneous, hypointense to isointense on T1-weighted images, and most commonly hyperintense on T2-weighted images compared with splenic parenchyma. On the other hand, in this paper, we present a pediatric case of splenic hemangioma that was hypointense on the T2-weighted images to the adjacent splenic parenchyma, which is a very unusual finding. 2. Case report An 11-year-old boy was referred to our institution for abdominal pain. His medical history was otherwise unremarkable. Physical examination and hematologic evaluation were normal. Initial abdominal ultrasonography showed a 35×32 mm well-defined hypoechoic lesion in the spleen without any calcification. On color Doppler images, the lesion was slightly vascularized (Fig. 1a and b). On T1weighted images, the lesion was slightly hyperintense, and on T2weighted images, it was significantly hypointense to adjacent splenic
⁎ Corresponding author. Fakülteler Mah, Dirim sok 22/3 Cebeci 06590, Ankara, Turkey. Tel.: +90 532 6967862; fax: +90 312 3112145. E-mail address: [email protected] (H.N. Ozcan).
parenchyma (Fig. 2a and b). A dynamic MRI study was performed after the administration of contrast material. Early arterial dynamic image showed no contrast enhancement and the lesion was mildly enhanced on portal venous phases. On delayed post contrast T1weighted image, the lesion became isointense to the spleen (Fig. 2c and d). Thereafter, splenectomy was performed for definite diagnosis and rule out malignancy. At gross examination, a mass with a diameter of 32 mm was seen in the spleen. Histologic examination of the splenectomy material showed a non-capsulated but limited neoplasm in the spleen. The neoplasm was composed of proliferated vascular channels of capillary size lined with a single layer of endothelium (Fig. 3a and b). There was no sclerosis or thrombosed vessel in the neoplasm. Immunohistochemically, endothelial cells were positive for CD31 and CD34 but negative for CD8. The histologic features were consistent with a cavernous splenic hemangioma. The histologic differential diagnosis of splenic hemangioma includes hamartoma, which is CD8-positive immunohistochemically. 3. Discussion Hemangioma, although rare, is the most common primary neoplasm of the spleen. Their rate of occurrence in large autopsy series has been reported as 0.03–14%, more frequent in male patients [4]. Mostly, they are small lesions that are found incidentally in otherwise asymptomatic patients. Splenic hemangioma may also occur as part of generalized angiomatosis as seen in KlippelTrenaunay syndrome. The radiologic appearance of hemangiomas ranges from solid to cystic, depending on gross morphology; however, predominantly, a hemangioma appears as a solid mass with cystic spaces [5]. On
0899-7071/$ – see front matter © 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.clinimag.2014.02.007
Please cite this article as: Ozcan HN, et al, Unusual splenic hemangioma of a pediatric patient: hypointense on T2-weighted image, Clin Imaging (2014), http://dx.doi.org/10.1016/j.clinimag.2014.02.007
2
H.N. Ozcan et al. / Clinical Imaging xxx (2014) xxx–xxx
Fig. 1. (a) Longitudinal sonogram of the spleen shows a 35×32 mm well-defined hypoechoic lesion in the spleen without any calcification. (b) On color Doppler image, the lesion was slightly vascularized.
ultrasonography, a hemangioma may manifest as a well-defined intrasplenic echogenic solid or complex cystic mass. The MRI appearance of splenic hemangioma is hypointense to isointense on T1-weighted images and hyperintense on T2-weighted images to the adjacent spleen. Ramani et al. [6] in a series of 22 splenic hemangiomas reported that 19 lesions were hyperintense, 2 lesions were isointense, and only 1 lesion was hypointense on T2-weighted images. That particular lesion was histologically found to be sclerotic. However, due to particular complicating features (e.g., hemorrhage, infarction, and thrombosis), larger hemangiomas may show various types of MRI findings. In histopathologic examination of our patient, there was no sclerosis or thrombosed vessel in the neoplasm. Dynamic MRI, after administration of gadolinium, has shown that splenic hemangiomas have three patterns of enhancement: (1) immediate homogeneous enhancement that persists, (2) early peripheral enhancement with uniform delayed enhancement, and (3) peripheral enhancement with centripetal progression but persis-
tent enhancement of a central fibrous scar [7]. In our case, the lesion was significantly hypointense on T2-weighted images, and after administration of contrast material, there was no enhancement on early arterial sequences, mildly enhanced on portal venous phase and isointense to the spleen on delayed T1-weighted images. In conclusion, most hemangiomas are hypointense to the spleen on T1-weighted images and hyperintense on T2-weighted images. The presentation of splenic hemangioma as low signal intensity on T2weighted images is exceedingly rare in adult patients. To our knowledge, our case is the first pediatric patient in the hitherto literature who has splenic hemangioma that is hypointense on T2-weighted images. It should be kept in mind in differential diagnosis of splenic lesions.
References [1] Paterson A, Frush DP, Donnelly LF, Foss JN, O'Hara SM, Bisset GS. A patternoriented approach to splenic imaging in infants and children. Radiographics 1999;19(6):1465–85.
Fig. 2. (a) Axial T1-weighted magnetic resonance (MR) image demonstrates the lesion slightly hyperintense relative to the spleen. (b) Axial T2-weighted MR image shows a hypointense lesion in the central part of the spleen. (c) Early arterial dynamic contrast-enhanced image demonstrates the lesion without contrast enhancement. (d) On delayed T1weighted image, the lesion becomes uniformly isointense.
Please cite this article as: Ozcan HN, et al, Unusual splenic hemangioma of a pediatric patient: hypointense on T2-weighted image, Clin Imaging (2014), http://dx.doi.org/10.1016/j.clinimag.2014.02.007
H.N. Ozcan et al. / Clinical Imaging xxx (2014) xxx–xxx
3
Fig. 3. (a) Non-capsulated, well-demarcated neoplasm (Hematoxylin and Eosin, original magnification ×40). (b) Endothelial cells forming vascular channels (Hematoxylin and Eosin, original magnification ×100).
[2] Abbott RM, Levy AD, Aguilera NS, Gorospe L, Thompson WM. From the archives of the AFIP: primary vascular neoplasms of the spleen: radiologic-pathologic correlation. Radiographics 2004;24(4):1137–63. [3] Panuel M, Ternier F, Michel G, Scheiner C, Bourliere B, Faure F, Guys JM, Devred P. Splenic hemangioma—report of three pediatric cases with pathologic correlation. Pediatr Radiol 1992;22(3):213–6. [4] Pines B, Rabinovitch J. Hemangioma of the spleen. Arch Pathol 1942;33:487–503.
[5] Urrutia M, Mergo PJ, Ros LH, Torres GM, Ros PR. Cystic masses of the spleen: radiologic-pathologic correlation. Radiographics 1996;16(1):107–29. [6] Ramani M, Reinhold C, Semelka RC, Siegelman ES, Liang L, Ascher SM, Brown JJ, Eisen RN, Bret PM. Splenic hemangiomas and hamartomas: MR imaging characteristics of 28 lesions. Radiology 1997;202(1):166–72. [7] Mortele KJ. Imaging of tumoral conditions of the spleen. JBR-BTR 2000;83(4):213–5.
Please cite this article as: Ozcan HN, et al, Unusual splenic hemangioma of a pediatric patient: hypointense on T2-weighted image, Clin Imaging (2014), http://dx.doi.org/10.1016/j.clinimag.2014.02.007