ORL 2014;76:105–109 DOI: 10.1159/000362247 Received: February 4, 2014 Accepted: March 14, 2014 Published online: May 1, 2014
© 2014 S. Karger AG, Basel 0301–1569/14/0762–0105$39.50/0 www.karger.com/orl
Case Report
Salivary Neoplasms Presenting with Radiologic Venous Invasion: An Imaging Pearl to Diagnosing Metastatic Renal Cell Carcinoma Pejman Maralani a Suyash Mohan b Laurie A. Loevner b, c
Christopher H. Rassekh c
a Department of Radiology, Neuroradiology Division, University of Toronto, Sunnybrook Health Sciences Center, Toronto, Ont., Canada; b Department of Radiology, Neuroradiology Division, and c Department of Otolaryngology and Head and Neck Surgery, Perelman School of Medicine at University of Pennsylvania, Philadelphia, Pa., USA
Key Words Venous invasion · Salivary glands · Metastases · Renal cell carcinoma Abstract We report the case of a 64-year-old otherwise healthy woman who presented with left facial swelling. Imaging of the neck revealed multiple masses in the salivary and thyroid glands. The mass in the left parotid gland was associated with an intravenous extension into the retromandibular, facial and internal jugular veins in the left neck. Based on multiplicity of these masses and the presence of radiologic venous invasion, the diagnosis of metastatic renal cell carcinoma (RCC) was suggested on imaging, which was subsequently confirmed on systemic workup and pathology findings. Although RCC metastasizes to the salivary glands, the primary presentation of RCC with both salivary and thyroid gland masses is extremely rare, with only a few reports. The above feature and its imaging diagnosis based on local venous inva© 2014 S. Karger AG, Basel sion are the highlights of this report.
Introduction
In this report, we will discuss a patient who presented with multiple masses involving the bilateral parotid, submandibular gland and the thyroid glands. The patient was otherwise not known to have any medical conditions. Based on multiplicity of masses and the imaging Laurie A. Loevner, MD Department of Radiology, Division of Neuroradiology Perelman School of Medicine at University of Pennsylvania 219 Dulles Building, 3400 Spruce Street, Philadelphia, PA 19104 (USA) E-Mail laurie.loevner @ uphs.upenn.edu
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ORL 2014;76:105–109 © 2014 S. Karger AG, Basel www.karger.com/orl
DOI: 10.1159/000362247
Maralani et al.: Salivary Neoplasms Presenting with Radiologic Venous Invasion: An Imaging Pearl to Diagnosing Metastatic Renal Cell Carcinoma
a
b
c
d
e
f
Fig. 1. a Axial T1-weighted image demonstrates a lobulated solid mass involving the superficial and deep lobes of the left parotid gland (arrows). b Axial post-contrast fat-saturated T1-weighted image demonstrates enhancing thrombus in the left retromandibular vein (curved arrow). Axial contrast-enhanced CT image demonstrates thrombus in the expanded left retromandibular vein (notched arrow, c), and in the left IJV (arrow heads) on a coronal reformatted image (d). e Axial T2-weighted image in the lower neck demonstrates a heterogeneous mass in the right lobe of the thyroid lobe (arrows). f Axial post-contrast fat-saturated T1weighted image demonstrates enhancing mass in the left submandibular gland (open arrows).
finding of venous invasion, the imaging diagnosis of metastatic renal cell carcinoma (RCC) was suggested, which was then proved with systemic workup and surgical pathology. Case Report A 64-year-old woman with no significant past medical history presented to her family physician with a few months of slowly progressive left facial swelling. The patient’s physical examination was otherwise unremarkable. A contrast-enhanced neck CT scan was ordered for further evaluation and was reported to show masses in the bilateral parotid glands, the left submandibular gland, and in the right lobe of the thyroid gland. The patient was referred to our institution for further evaluation. As part of her workup, MRI of the neck was obtained. This study revealed a 2.8-cm mass involving the superficial and deep lobes of the left parotid gland, as well as smaller focal masses in the superficial right parotid gland, left submandibular gland, and in the right lobe of the thyroid gland (fig. 1a–f). On close evaluation, the left parotid mass was noted to invade the left retromandibular and facial veins, with inferior extension into the left internal jugular vein (IJV) (fig. 1b–d). The finding of multiple masses was most consistent with metastatic disease, and based on the presence of direct intravenous extension of the neoplasm the diagnosis of metastatic RCC was proposed. An ultrasound-guided fine needle aspiration of the left parotid mass demonstrated an oncocytic neoplasm with multiple epithelioid cells. Immunohistochemical staining was negative for CK5/6 and p63 consistent with adenocarcinoma as opposed to squamous cell carcinoma. Mucicarmine stain was negative for mucin.
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ORL 2014;76:105–109 DOI: 10.1159/000362247
© 2014 S. Karger AG, Basel www.karger.com/orl
Maralani et al.: Salivary Neoplasms Presenting with Radiologic Venous Invasion: An Imaging Pearl to Diagnosing Metastatic Renal Cell Carcinoma
Fig. 2. Coronal reformatted image of a contrast-enhanced CT of the abdomen and pelvis demonstrating a heterogeneously enhancing 9-cm mass in the mid-lower pole of the right kidney (arrows), with no extension into the right renal vein (asterisk).
The patient subsequently underwent contrast-enhanced CT of the abdomen and pelvis, which demonstrated a 9-cm solid and cystic right renal mass, with no extension into the right renal vein (fig. 2). The next day, the patient had a partial left parotidectomy for biopsy. The tumor was found to be hypervascular and extensive. Frozen section was consistent with RCC. Final surgical pathology analysis of the specimen showed the tumor with venous invasion, and histology revealed cells with clear cytoplasm and irregular hyperchromatic nuclei. In light of the findings on the abdomen CT, immunohistochemical staining was performed to differentiate primary salivary adenocarcinoma from metastatic adenocarcinoma from a systemic site. The tumor cells were positive for AE1/3, CAM5.2, CD10, and focally for EMA and negative for chromogranin, synaptophysin and S100 consistent with metastatic clear-cell RCC. The patient subsequently underwent radical right nephrectomy and surgical pathology revealed grade II clear-cell RCC. The neoplasm was entirely confined within the renal capsule. The patient was put on oral sunitinib on which the metastatic nodules decreased in size. One-year follow-up scans demonstrated stable disease.
Discussion
The most common cause of metastatic disease to the head and neck is squamous cell carcinoma of the upper aerodigestive tract, presenting as metastatic adenopathy [1]. Metastatic disease to the head and neck from thyroid cancer, melanoma, and primary salivary neoplasms is relatively common. Metastases to the neck from systemic malignancies such as breast and lung carcinoma are much less common and also usually involve the lymph nodes [1]. Metastases to the salivary glands are most commonly seen in the parotid glands and are related to intraglandular nodal metastasis from face and scalp skin cancers, and less commonly from squamous cell carcinomas of the upper aerodigestive tract [2]. The parotid gland is the last of the salivary glands to be encapsulated by the superficial layer of the deep cervical fascia, which occurs after the development of its lymphatic system in the embryonic period. As such, the parotid gland is the only salivary gland to have intraglandular lymph nodes. Metastatic disease to the major salivary glands related to systemic cancers is rare and most often occurs to the parotid gland [3], with lung, breast and renal cell carcinomas. Metas-
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ORL 2014;76:105–109 DOI: 10.1159/000362247
© 2014 S. Karger AG, Basel www.karger.com/orl
Maralani et al.: Salivary Neoplasms Presenting with Radiologic Venous Invasion: An Imaging Pearl to Diagnosing Metastatic Renal Cell Carcinoma
tases to the submandibular glands are exceedingly rare [3]. Metastatic disease to the thyroid is also quite rare representing 1.4–3% of thyroid tumors [4]. A recent review shows [4] that metastases to the thyroid gland in decreasing order of frequency are RCC, colorectal and lung carcinomas, with RCC accounting for almost half of these, and colorectal cancer for about 10% [5] of cases [6, 7]. The most common sites for RCC to metastasize are the lungs, bones, and the liver. Metastatic disease to the head and neck occurs in 8–14% of cases. Within the neck, the thyroid is the most common site of metastatic involvement. Invasion of the IJV by neck neoplasms has been described with thyroid cancers [6, 7], paragangliomas [8] and skull base neoplasms such as meningiomas [9]. RCC is known for its predilection to invade the renal vein and other veins in the retroperitoneum by contiguous extension [10]. There are reports of superior vena cava syndrome in RCC that metastasized to the thyroid gland via direct extension to and invasion of the IJV [11], or from metastatic lymphadenopathy that invades the brachiocephalic vein [12]. To the best of our knowledge, metastatic lesion in the parotid gland demonstrating frank radiologic venous invasion into the adjacent retromandibular and facial veins with contiguous spread into the IJV has not been reported previously. Though there are a few reported cases of metastatic RCC to the thyroid and submandibular glands 7–10 years after radical nephrectomy [4, 13], multiple neck masses involving the major salivary and thyroid glands as the initial clinical presentation of RCC is exceedingly rare, and illustrated by our case. The involvement of multiple glands should suggest a systemic process such as metastatic disease in an otherwise healthy patient. Involvement of the thyroid gland and the presence of neoplastic venous invasion are highly suggestive of RCC and should prompt appropriate workup. Conclusion
Metastatic disease to the thyroid and salivary glands is very rare. RCC is the most common cause of metastatic disease to the thyroid gland, and is also known for its propensity for venous invasion. The presence of multiple masses involving the thyroid and salivary glands with local venous invasion should suggest the diagnosis of metastatic RCC.
References 1 2 3 4 5 6 7 8
Yanlan C, Liping S, Shaomin C, et al: Metastasis to the parotid region as an initial presentation of renal cell carcinoma: a case report. Oncol Lett 2013;5:997–999. Rodriguez MS, Reija MF, Rodilla IG: Primary clear cell carcinoma of parotid gland: case report and review of literature. J Oral Maxillofac Pathol 2013;17:101–105. Serouya SM, Dultz LA, Concors SJ, et al: Late solitary metastasis of renal cell carcinoma to the submandibular gland. J Oral Maxillofac Surg 2012;70:2356–2359. Miah MS, White SJ, Oommen G, et al: Late simultaneous metastasis of renal cell carcinoma to the submandibular and thyroid glands seven years after radical nephrectomy. Int J Otolaryngol 2010;2010:698014. Chung AY, Tran TB, Brumund KT, et al: Metastases to the thyroid: a review of the literature from the last decade. Thyroid 2012;22:258–268. Nakashima T, Nakashima A, Murakami D, et al: Follicular carcinoma of the thyroid with massive invasion into the cervical and mediastinum great veins: our own experience and literature review. Laryngoscope 2012;122: 2855–2857. Chao TC, Lin JD, Chen MF: Insular carcinoma: infrequent subtype of thyroid cancer with aggressive clinical course. World J Surg 2004;28:393–396. Arsene D, Ardeleanu C, Danaila L: An intrajugular paraganglioma. Unusual presentation of a classical tumor. Rom J Morphol Embryol 2007;48:189–193.
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ORL 2014;76:105–109 DOI: 10.1159/000362247
© 2014 S. Karger AG, Basel www.karger.com/orl
Maralani et al.: Salivary Neoplasms Presenting with Radiologic Venous Invasion: An Imaging Pearl to Diagnosing Metastatic Renal Cell Carcinoma
9 10 11 12 13
Vachhrajani S, Jea A, Rutka JA, et al: Meningioma with dural venous sinus invasion and jugular vein extension. J Neurosurg Pediatr 2008;2:391–396. Davits RJ, Blom JH, Schroder FH: Surgical management of renal carcinoma with extensive involvement of the vena cava and right atrium. Br J Urol 1992;70:591–593. Davison BD, Mammen L, Ritchie J, et al: Descending tumor thrombus from renal cell carcinoma metastases with SVC syndrome. Cardiovasc Intervent Radiol 2006;29:687–690. Sakura M, Tsujii T, Yamauchi A, et al: Superior vena cava syndrome caused by supraclavicular lymph node metastasis of renal cell carcinoma. Int J Clin Oncol 2007;12:382–384. Moudouni SM, Tligui M, Doublet JD, et al: Late metastasis of renal cell carcinoma to the submaxillary gland 10 years after radical nephrectomy. Int J Urol 2006;13:431–432.
Copyright: S. Karger AG, Basel 2014. Reproduced with the permission of S. Karger AG, Basel. Further reproduction or distribution (electronic or otherwise) is prohibited without permission from the copyright holder.
© 2014 S. Karger AG, Basel 0301–1569/14/0762–0105$39.50/0 www.karger.com/orl
Case Report
Salivary Neoplasms Presenting with Radiologic Venous Invasion: An Imaging Pearl to Diagnosing Metastatic Renal Cell Carcinoma Pejman Maralani a Suyash Mohan b Laurie A. Loevner b, c
Christopher H. Rassekh c
a Department of Radiology, Neuroradiology Division, University of Toronto, Sunnybrook Health Sciences Center, Toronto, Ont., Canada; b Department of Radiology, Neuroradiology Division, and c Department of Otolaryngology and Head and Neck Surgery, Perelman School of Medicine at University of Pennsylvania, Philadelphia, Pa., USA
Key Words Venous invasion · Salivary glands · Metastases · Renal cell carcinoma Abstract We report the case of a 64-year-old otherwise healthy woman who presented with left facial swelling. Imaging of the neck revealed multiple masses in the salivary and thyroid glands. The mass in the left parotid gland was associated with an intravenous extension into the retromandibular, facial and internal jugular veins in the left neck. Based on multiplicity of these masses and the presence of radiologic venous invasion, the diagnosis of metastatic renal cell carcinoma (RCC) was suggested on imaging, which was subsequently confirmed on systemic workup and pathology findings. Although RCC metastasizes to the salivary glands, the primary presentation of RCC with both salivary and thyroid gland masses is extremely rare, with only a few reports. The above feature and its imaging diagnosis based on local venous inva© 2014 S. Karger AG, Basel sion are the highlights of this report.
Introduction
In this report, we will discuss a patient who presented with multiple masses involving the bilateral parotid, submandibular gland and the thyroid glands. The patient was otherwise not known to have any medical conditions. Based on multiplicity of masses and the imaging Laurie A. Loevner, MD Department of Radiology, Division of Neuroradiology Perelman School of Medicine at University of Pennsylvania 219 Dulles Building, 3400 Spruce Street, Philadelphia, PA 19104 (USA) E-Mail laurie.loevner @ uphs.upenn.edu
106
ORL 2014;76:105–109 © 2014 S. Karger AG, Basel www.karger.com/orl
DOI: 10.1159/000362247
Maralani et al.: Salivary Neoplasms Presenting with Radiologic Venous Invasion: An Imaging Pearl to Diagnosing Metastatic Renal Cell Carcinoma
a
b
c
d
e
f
Fig. 1. a Axial T1-weighted image demonstrates a lobulated solid mass involving the superficial and deep lobes of the left parotid gland (arrows). b Axial post-contrast fat-saturated T1-weighted image demonstrates enhancing thrombus in the left retromandibular vein (curved arrow). Axial contrast-enhanced CT image demonstrates thrombus in the expanded left retromandibular vein (notched arrow, c), and in the left IJV (arrow heads) on a coronal reformatted image (d). e Axial T2-weighted image in the lower neck demonstrates a heterogeneous mass in the right lobe of the thyroid lobe (arrows). f Axial post-contrast fat-saturated T1weighted image demonstrates enhancing mass in the left submandibular gland (open arrows).
finding of venous invasion, the imaging diagnosis of metastatic renal cell carcinoma (RCC) was suggested, which was then proved with systemic workup and surgical pathology. Case Report A 64-year-old woman with no significant past medical history presented to her family physician with a few months of slowly progressive left facial swelling. The patient’s physical examination was otherwise unremarkable. A contrast-enhanced neck CT scan was ordered for further evaluation and was reported to show masses in the bilateral parotid glands, the left submandibular gland, and in the right lobe of the thyroid gland. The patient was referred to our institution for further evaluation. As part of her workup, MRI of the neck was obtained. This study revealed a 2.8-cm mass involving the superficial and deep lobes of the left parotid gland, as well as smaller focal masses in the superficial right parotid gland, left submandibular gland, and in the right lobe of the thyroid gland (fig. 1a–f). On close evaluation, the left parotid mass was noted to invade the left retromandibular and facial veins, with inferior extension into the left internal jugular vein (IJV) (fig. 1b–d). The finding of multiple masses was most consistent with metastatic disease, and based on the presence of direct intravenous extension of the neoplasm the diagnosis of metastatic RCC was proposed. An ultrasound-guided fine needle aspiration of the left parotid mass demonstrated an oncocytic neoplasm with multiple epithelioid cells. Immunohistochemical staining was negative for CK5/6 and p63 consistent with adenocarcinoma as opposed to squamous cell carcinoma. Mucicarmine stain was negative for mucin.
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ORL 2014;76:105–109 DOI: 10.1159/000362247
© 2014 S. Karger AG, Basel www.karger.com/orl
Maralani et al.: Salivary Neoplasms Presenting with Radiologic Venous Invasion: An Imaging Pearl to Diagnosing Metastatic Renal Cell Carcinoma
Fig. 2. Coronal reformatted image of a contrast-enhanced CT of the abdomen and pelvis demonstrating a heterogeneously enhancing 9-cm mass in the mid-lower pole of the right kidney (arrows), with no extension into the right renal vein (asterisk).
The patient subsequently underwent contrast-enhanced CT of the abdomen and pelvis, which demonstrated a 9-cm solid and cystic right renal mass, with no extension into the right renal vein (fig. 2). The next day, the patient had a partial left parotidectomy for biopsy. The tumor was found to be hypervascular and extensive. Frozen section was consistent with RCC. Final surgical pathology analysis of the specimen showed the tumor with venous invasion, and histology revealed cells with clear cytoplasm and irregular hyperchromatic nuclei. In light of the findings on the abdomen CT, immunohistochemical staining was performed to differentiate primary salivary adenocarcinoma from metastatic adenocarcinoma from a systemic site. The tumor cells were positive for AE1/3, CAM5.2, CD10, and focally for EMA and negative for chromogranin, synaptophysin and S100 consistent with metastatic clear-cell RCC. The patient subsequently underwent radical right nephrectomy and surgical pathology revealed grade II clear-cell RCC. The neoplasm was entirely confined within the renal capsule. The patient was put on oral sunitinib on which the metastatic nodules decreased in size. One-year follow-up scans demonstrated stable disease.
Discussion
The most common cause of metastatic disease to the head and neck is squamous cell carcinoma of the upper aerodigestive tract, presenting as metastatic adenopathy [1]. Metastatic disease to the head and neck from thyroid cancer, melanoma, and primary salivary neoplasms is relatively common. Metastases to the neck from systemic malignancies such as breast and lung carcinoma are much less common and also usually involve the lymph nodes [1]. Metastases to the salivary glands are most commonly seen in the parotid glands and are related to intraglandular nodal metastasis from face and scalp skin cancers, and less commonly from squamous cell carcinomas of the upper aerodigestive tract [2]. The parotid gland is the last of the salivary glands to be encapsulated by the superficial layer of the deep cervical fascia, which occurs after the development of its lymphatic system in the embryonic period. As such, the parotid gland is the only salivary gland to have intraglandular lymph nodes. Metastatic disease to the major salivary glands related to systemic cancers is rare and most often occurs to the parotid gland [3], with lung, breast and renal cell carcinomas. Metas-
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ORL 2014;76:105–109 DOI: 10.1159/000362247
© 2014 S. Karger AG, Basel www.karger.com/orl
Maralani et al.: Salivary Neoplasms Presenting with Radiologic Venous Invasion: An Imaging Pearl to Diagnosing Metastatic Renal Cell Carcinoma
tases to the submandibular glands are exceedingly rare [3]. Metastatic disease to the thyroid is also quite rare representing 1.4–3% of thyroid tumors [4]. A recent review shows [4] that metastases to the thyroid gland in decreasing order of frequency are RCC, colorectal and lung carcinomas, with RCC accounting for almost half of these, and colorectal cancer for about 10% [5] of cases [6, 7]. The most common sites for RCC to metastasize are the lungs, bones, and the liver. Metastatic disease to the head and neck occurs in 8–14% of cases. Within the neck, the thyroid is the most common site of metastatic involvement. Invasion of the IJV by neck neoplasms has been described with thyroid cancers [6, 7], paragangliomas [8] and skull base neoplasms such as meningiomas [9]. RCC is known for its predilection to invade the renal vein and other veins in the retroperitoneum by contiguous extension [10]. There are reports of superior vena cava syndrome in RCC that metastasized to the thyroid gland via direct extension to and invasion of the IJV [11], or from metastatic lymphadenopathy that invades the brachiocephalic vein [12]. To the best of our knowledge, metastatic lesion in the parotid gland demonstrating frank radiologic venous invasion into the adjacent retromandibular and facial veins with contiguous spread into the IJV has not been reported previously. Though there are a few reported cases of metastatic RCC to the thyroid and submandibular glands 7–10 years after radical nephrectomy [4, 13], multiple neck masses involving the major salivary and thyroid glands as the initial clinical presentation of RCC is exceedingly rare, and illustrated by our case. The involvement of multiple glands should suggest a systemic process such as metastatic disease in an otherwise healthy patient. Involvement of the thyroid gland and the presence of neoplastic venous invasion are highly suggestive of RCC and should prompt appropriate workup. Conclusion
Metastatic disease to the thyroid and salivary glands is very rare. RCC is the most common cause of metastatic disease to the thyroid gland, and is also known for its propensity for venous invasion. The presence of multiple masses involving the thyroid and salivary glands with local venous invasion should suggest the diagnosis of metastatic RCC.
References 1 2 3 4 5 6 7 8
Yanlan C, Liping S, Shaomin C, et al: Metastasis to the parotid region as an initial presentation of renal cell carcinoma: a case report. Oncol Lett 2013;5:997–999. Rodriguez MS, Reija MF, Rodilla IG: Primary clear cell carcinoma of parotid gland: case report and review of literature. J Oral Maxillofac Pathol 2013;17:101–105. Serouya SM, Dultz LA, Concors SJ, et al: Late solitary metastasis of renal cell carcinoma to the submandibular gland. J Oral Maxillofac Surg 2012;70:2356–2359. Miah MS, White SJ, Oommen G, et al: Late simultaneous metastasis of renal cell carcinoma to the submandibular and thyroid glands seven years after radical nephrectomy. Int J Otolaryngol 2010;2010:698014. Chung AY, Tran TB, Brumund KT, et al: Metastases to the thyroid: a review of the literature from the last decade. Thyroid 2012;22:258–268. Nakashima T, Nakashima A, Murakami D, et al: Follicular carcinoma of the thyroid with massive invasion into the cervical and mediastinum great veins: our own experience and literature review. Laryngoscope 2012;122: 2855–2857. Chao TC, Lin JD, Chen MF: Insular carcinoma: infrequent subtype of thyroid cancer with aggressive clinical course. World J Surg 2004;28:393–396. Arsene D, Ardeleanu C, Danaila L: An intrajugular paraganglioma. Unusual presentation of a classical tumor. Rom J Morphol Embryol 2007;48:189–193.
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ORL 2014;76:105–109 DOI: 10.1159/000362247
© 2014 S. Karger AG, Basel www.karger.com/orl
Maralani et al.: Salivary Neoplasms Presenting with Radiologic Venous Invasion: An Imaging Pearl to Diagnosing Metastatic Renal Cell Carcinoma
9 10 11 12 13
Vachhrajani S, Jea A, Rutka JA, et al: Meningioma with dural venous sinus invasion and jugular vein extension. J Neurosurg Pediatr 2008;2:391–396. Davits RJ, Blom JH, Schroder FH: Surgical management of renal carcinoma with extensive involvement of the vena cava and right atrium. Br J Urol 1992;70:591–593. Davison BD, Mammen L, Ritchie J, et al: Descending tumor thrombus from renal cell carcinoma metastases with SVC syndrome. Cardiovasc Intervent Radiol 2006;29:687–690. Sakura M, Tsujii T, Yamauchi A, et al: Superior vena cava syndrome caused by supraclavicular lymph node metastasis of renal cell carcinoma. Int J Clin Oncol 2007;12:382–384. Moudouni SM, Tligui M, Doublet JD, et al: Late metastasis of renal cell carcinoma to the submaxillary gland 10 years after radical nephrectomy. Int J Urol 2006;13:431–432.
Copyright: S. Karger AG, Basel 2014. Reproduced with the permission of S. Karger AG, Basel. Further reproduction or distribution (electronic or otherwise) is prohibited without permission from the copyright holder.
