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Pterygopalatine fossa metastasis with severe trigeminal neuralgia from neuroendocrine carcinoma of the breast
G Model
ANL-2128; No. of Pages 3 Auris Nasus Larynx xxx (2016) xxx–xxx Contents lists available at ScienceDirect
Auris Nasus Larynx journal homepage: www.elsevier.com/locate/anl
Pterygopalatine fossa metastasis with severe trigeminal neuralgia from neuroendocrine carcinoma of the breast Taro Takanami a,b,*, Yasushi Ota b, Mitsuya Suzuki b a b
Department of Otolaryngology, Sanraku Hospital, Kandasurugadai 2-5, Chiyoda, Tokyo 101-0622, Japan Department of Otolaryngology, Toho University Sakura Medical Center, Shimoshizu 564-1, Sakura, Chiba 285-8741, Japan
A R T I C L E I N F O
A B S T R A C T
Article history: Received 20 February 2016 Accepted 18 April 2016 Available online xxx
A 57-year-old woman presented to our department with a 2-month history of pain and paresthesia on the left side of the face 12 years after having undergone surgery for breast cancer. We performed an endoscopic biopsy and diagnosed metastatic breast cancer to the pterygopalatine fossa. There has been no recurrence for two years since the metastatic tumor was treated by radiation therapy. A literature search shows only one case of metastatic breast cancer with severe trigeminal neuralgia located in the V2 division of the trigeminal nerve area. Metastatic disease should be considered part of the differential diagnosis in patients presenting with trigeminal neuropathy. ß 2016 Elsevier Ireland Ltd. All rights reserved.
Keywords: Pterygopalatine fossa Neuroendocrine carcinoma Trigeminal neuralgia
1. Introduction Breast carcinoma is the most common cancer encountered in women, with increasing incidence and mortality rates in Japan over the past 20–30 years. Breast carcinoma metastases to the head and neck region are not infrequent. The larynx, nasopharynx, parotid gland, nasal and paranasal sinuses, and temporal bone may harbor metastatic deposits from breast carcinoma. However, pterygopalatine fossa (PPF) metastasis is not common, and there is only one report in the literature of breast cancer metastasis involving the PPF [1]. Here we report a case of metastatic breast cancer to the PPF manifesting as severe trigeminal neuralgia located in the V2 division of the trigeminal nerve area 12 years after surgery for breast cancer. 2. Case report A 57-year-old woman presented to our department with a 2month history of pain and paresthesia on the left side of the * Corresponding author at: Department of Otolaryngology, Sanraku Hospital, Kandasurugadai 2-5, Chiyoda, Tokyo 101-0622, Japan. Tel.: +81 08051973133. E-mail address: [email protected] (T. Takanami).
face. The pain was unresponsive to pharmacological treatment, increasing in frequency and intensity, and characteristic of trigeminal neuralgia, which presents as episodes of intense, stabbing, electric shock-like pain in areas of the face supplied by the V2 branch of the trigeminal nerve. The patient had been diagnosed with carcinoma of the left breast (neuroendocrine carcinoma, invasive type: stage T2N0M0) 12 years earlier. She had undergone a left modified radical mastectomy with axillary lymph node dissection. There were no findings of recurrence at the original site during the follow-up period. Computed tomography (CT) revealed bony lysis of the posterior wall of the left maxillary sinus. Magnetic resonance imaging (MRI) showed an enhancing soft tissue mass within the left PPF and posterior portion of the maxillary sinus (Fig. 1). The lesion was suspected to be either a metastasis from her original breast cancer or a primary PPF tumor. On the basis of her previous history, the lesion in the PPF was considered more likely to be a solitary metastasis from her initial breast carcinoma, so we performed an endoscopic sinus biopsy. Hematoxylin and eosin staining showed tumor cells that ranged in shape from oval to spindle and plasmacytoid. The cells contained mildly pleomorphic nuclei with fine pepper chromatin and were separated by delicate fibrous stroma (Fig. 2). Immunohistochemistry revealed positive staining for the chromogranin
http://dx.doi.org/10.1016/j.anl.2016.04.011 0385-8146/ß 2016 Elsevier Ireland Ltd. All rights reserved.
Please cite this article in press as: Takanami T, et al. Pterygopalatine fossa metastasis with severe trigeminal neuralgia from neuroendocrine carcinoma of the breast. Auris Nasus Larynx (2016), http://dx.doi.org/10.1016/j.anl.2016.04.011
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ANL-2128; No. of Pages 3 T. Takanami et al. / Auris Nasus Larynx xxx (2016) xxx–xxx
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Fig. 1. Magnetic resonance imaging showing an enhancing soft tissue mass within the right pterygopalatine fossa and posterior portion of the maxillary sinus (axial T1 – weighted MRI).
receptor, consistent withthe receptor status of the patient’s original breast carcinoma. The pathology report showed metastatic neuroendocrine carcinoma; the morphology was consistent with breast carcinoma. We concluded that the cause of the trigeminal neuralgia was direct metastasis of breast cancer to the cisternal portion of the trigeminal nerve or ganglion. There is no other metastasis than pterygopalatine fossa. She received radiation therapy of total 40 Gy in 20 fractions delivered to the distant metastasis and her disease has remained stable. The follow up period is 3 years, and the new growth of tumor does not appear for this period. There is no other metastasis. 3. Discussion Metastasis of breast carcinoma to the PPF is not common. The only report of breast cancer metastasis involving the PPF was published by Albayram et al. [1]. The PPF is the pyramidal space located inferior to the orbital apex and posterior to the maxillary sinus. It contains the maxillary nerve (the second
division of the trigeminal nerve and its branches), the pterygopalatine ganglion, and the terminal branches of the internal maxillary artery. The patient’s symptom was caused by the metastatic carcinoma compressing the nerve and causing neuralgia unresponsive to pharmacological treatment. The differential diagnosis of a PPF mass typically includes perineural tumor extension along the second division of the trigeminal nerve, a nerve sheath tumor (schwannoma, neurofibroma), angiofibroma, lymphoma, hemangioma, and an ectopic lesion of the minor salivary glands [2,3]. Therefore, diagnosis of a PPF mass requires radiologic assessment and histologic confirmation of the lesion, which will guide further treatment. However, metastatic carcinoma to the head and neck region is generally associated with a poor prognosis because the primary disease is usually advanced and widespread by the time secondary deposits in this area become clinically manifest. A case reported by Albayram et al. had no more histological assessment or medication in terms of pulmonary metastasis. In our case, we suspected a solitary PPF metastasis from the original breast cancer because metastatic lesions had not been observed on the head, chest, and abdominal CT scans. Isolated PPF lesions can be treated with radiation therapy. Therefore, we carried out an endoscopic sinus biopsy for histologic assessment. In cases such as this, we recommend that the tumor be diagnosed by histological assessment, unless the patient clearly has advanced disease. To the best of our knowledge, this is the first report of severe trigeminal neuralgia that was caused by a solitary PPF metastasis from breast cancer that was confirmed by endoscopic sinus biopsy. Another unusual feature of our patient was the history of neuroendocrine carcinoma of the breast. Neuroendocrine tumors of the breast are rare, accounting for less than 0.1% of all breast cancers and less than 1% of all neuroendocrine tumors [4]. Little is known about their evolution, biologic behavior, or optimal treatment. The malignant potential of neuroendocrine tumors has been alluded to in a few reports; metastases at diverse sites have been found years after treatment of the primary site in the breast [5]. In our case, we hypothesized that it was possible that a distant metastasis from a low grade and slow-growing neuroendocrine carcinoma could appear 12 years after surgical resection of the primary tumor. In the future, a better knowledge of the biology of these tumors will hopefully provide new therapeutic targets for personalized treatment. There has been no report to date regarding metastasis affecting the PPF and causing direct compression of the trigeminal ganglion. In this case, the initial radiographic examination of the face was an important factor in the diagnosis. In summary, we report a case of metastatic breast cancer that clinically manifested as left facial paresthesia.
4. Conclusion Fig. 2. Hematoxylin and eosin stains showing that the shape of the tumor cells ranged from oval to spindle and plasmacytoid; the cells were separated by delicate fibrous stroma (hematoxylin and eosin; original magnification 400).
We have encountered a rare case of metastasis from neuroendocrine carcinoma of the breast to the PPF presenting as severe trigeminal neuralgia. We would like to highlight that
Please cite this article in press as: Takanami T, et al. Pterygopalatine fossa metastasis with severe trigeminal neuralgia from neuroendocrine carcinoma of the breast. Auris Nasus Larynx (2016), http://dx.doi.org/10.1016/j.anl.2016.04.011
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ANL-2128; No. of Pages 3 T. Takanami et al. / Auris Nasus Larynx xxx (2016) xxx–xxx
metastasis to PPF should be kept in mind when a patient with history of breast cancer presents with trigeminal neuralgia. Conflict of interest There is no conflict of interests. References [1] Albayram S, Adaletli I, Selcuk H, Gulsen F, Islak C, Kocer N. Breast cancer metastasis involving pterygopalatine fossa. Headache 2004;44:927–8.
3
[2] Chan LL, Chong J, Gillenwater AM, Ginsberg LE. The pterygopalatine fossa: postoperative MR imaging appearance. AJNR Am J Neuroradiol 2000;21:1315–9. [3] Daniels DL, Mark LP, Ulmer JL, Mafee MF, McDaniel J, Shah NC. Osseous anatomy of the pterygopalatine fossa. AJNR Am J Neuroradiol 1998;19:1423–32. [4] Ogawa H, Nishio A, Satake H, Naganawa S, Imai T, Sawaki M. Neuroendocrine tumor in the breast. Radiat Med 2008;26:28–32. [5] Konstantions M, Spyridon D, Nikolaos P, Loanna G, Chris D. Neuroendocrine breast carcinoma metastatic to the liver: report of a case and review of the literature. Int J Surg Case Rep 2014;5:540–3.
Please cite this article in press as: Takanami T, et al. Pterygopalatine fossa metastasis with severe trigeminal neuralgia from neuroendocrine carcinoma of the breast. Auris Nasus Larynx (2016), http://dx.doi.org/10.1016/j.anl.2016.04.011
ANL-2128; No. of Pages 3 Auris Nasus Larynx xxx (2016) xxx–xxx Contents lists available at ScienceDirect
Auris Nasus Larynx journal homepage: www.elsevier.com/locate/anl
Pterygopalatine fossa metastasis with severe trigeminal neuralgia from neuroendocrine carcinoma of the breast Taro Takanami a,b,*, Yasushi Ota b, Mitsuya Suzuki b a b
Department of Otolaryngology, Sanraku Hospital, Kandasurugadai 2-5, Chiyoda, Tokyo 101-0622, Japan Department of Otolaryngology, Toho University Sakura Medical Center, Shimoshizu 564-1, Sakura, Chiba 285-8741, Japan
A R T I C L E I N F O
A B S T R A C T
Article history: Received 20 February 2016 Accepted 18 April 2016 Available online xxx
A 57-year-old woman presented to our department with a 2-month history of pain and paresthesia on the left side of the face 12 years after having undergone surgery for breast cancer. We performed an endoscopic biopsy and diagnosed metastatic breast cancer to the pterygopalatine fossa. There has been no recurrence for two years since the metastatic tumor was treated by radiation therapy. A literature search shows only one case of metastatic breast cancer with severe trigeminal neuralgia located in the V2 division of the trigeminal nerve area. Metastatic disease should be considered part of the differential diagnosis in patients presenting with trigeminal neuropathy. ß 2016 Elsevier Ireland Ltd. All rights reserved.
Keywords: Pterygopalatine fossa Neuroendocrine carcinoma Trigeminal neuralgia
1. Introduction Breast carcinoma is the most common cancer encountered in women, with increasing incidence and mortality rates in Japan over the past 20–30 years. Breast carcinoma metastases to the head and neck region are not infrequent. The larynx, nasopharynx, parotid gland, nasal and paranasal sinuses, and temporal bone may harbor metastatic deposits from breast carcinoma. However, pterygopalatine fossa (PPF) metastasis is not common, and there is only one report in the literature of breast cancer metastasis involving the PPF [1]. Here we report a case of metastatic breast cancer to the PPF manifesting as severe trigeminal neuralgia located in the V2 division of the trigeminal nerve area 12 years after surgery for breast cancer. 2. Case report A 57-year-old woman presented to our department with a 2month history of pain and paresthesia on the left side of the * Corresponding author at: Department of Otolaryngology, Sanraku Hospital, Kandasurugadai 2-5, Chiyoda, Tokyo 101-0622, Japan. Tel.: +81 08051973133. E-mail address: [email protected] (T. Takanami).
face. The pain was unresponsive to pharmacological treatment, increasing in frequency and intensity, and characteristic of trigeminal neuralgia, which presents as episodes of intense, stabbing, electric shock-like pain in areas of the face supplied by the V2 branch of the trigeminal nerve. The patient had been diagnosed with carcinoma of the left breast (neuroendocrine carcinoma, invasive type: stage T2N0M0) 12 years earlier. She had undergone a left modified radical mastectomy with axillary lymph node dissection. There were no findings of recurrence at the original site during the follow-up period. Computed tomography (CT) revealed bony lysis of the posterior wall of the left maxillary sinus. Magnetic resonance imaging (MRI) showed an enhancing soft tissue mass within the left PPF and posterior portion of the maxillary sinus (Fig. 1). The lesion was suspected to be either a metastasis from her original breast cancer or a primary PPF tumor. On the basis of her previous history, the lesion in the PPF was considered more likely to be a solitary metastasis from her initial breast carcinoma, so we performed an endoscopic sinus biopsy. Hematoxylin and eosin staining showed tumor cells that ranged in shape from oval to spindle and plasmacytoid. The cells contained mildly pleomorphic nuclei with fine pepper chromatin and were separated by delicate fibrous stroma (Fig. 2). Immunohistochemistry revealed positive staining for the chromogranin
http://dx.doi.org/10.1016/j.anl.2016.04.011 0385-8146/ß 2016 Elsevier Ireland Ltd. All rights reserved.
Please cite this article in press as: Takanami T, et al. Pterygopalatine fossa metastasis with severe trigeminal neuralgia from neuroendocrine carcinoma of the breast. Auris Nasus Larynx (2016), http://dx.doi.org/10.1016/j.anl.2016.04.011
G Model
ANL-2128; No. of Pages 3 T. Takanami et al. / Auris Nasus Larynx xxx (2016) xxx–xxx
2
Fig. 1. Magnetic resonance imaging showing an enhancing soft tissue mass within the right pterygopalatine fossa and posterior portion of the maxillary sinus (axial T1 – weighted MRI).
receptor, consistent withthe receptor status of the patient’s original breast carcinoma. The pathology report showed metastatic neuroendocrine carcinoma; the morphology was consistent with breast carcinoma. We concluded that the cause of the trigeminal neuralgia was direct metastasis of breast cancer to the cisternal portion of the trigeminal nerve or ganglion. There is no other metastasis than pterygopalatine fossa. She received radiation therapy of total 40 Gy in 20 fractions delivered to the distant metastasis and her disease has remained stable. The follow up period is 3 years, and the new growth of tumor does not appear for this period. There is no other metastasis. 3. Discussion Metastasis of breast carcinoma to the PPF is not common. The only report of breast cancer metastasis involving the PPF was published by Albayram et al. [1]. The PPF is the pyramidal space located inferior to the orbital apex and posterior to the maxillary sinus. It contains the maxillary nerve (the second
division of the trigeminal nerve and its branches), the pterygopalatine ganglion, and the terminal branches of the internal maxillary artery. The patient’s symptom was caused by the metastatic carcinoma compressing the nerve and causing neuralgia unresponsive to pharmacological treatment. The differential diagnosis of a PPF mass typically includes perineural tumor extension along the second division of the trigeminal nerve, a nerve sheath tumor (schwannoma, neurofibroma), angiofibroma, lymphoma, hemangioma, and an ectopic lesion of the minor salivary glands [2,3]. Therefore, diagnosis of a PPF mass requires radiologic assessment and histologic confirmation of the lesion, which will guide further treatment. However, metastatic carcinoma to the head and neck region is generally associated with a poor prognosis because the primary disease is usually advanced and widespread by the time secondary deposits in this area become clinically manifest. A case reported by Albayram et al. had no more histological assessment or medication in terms of pulmonary metastasis. In our case, we suspected a solitary PPF metastasis from the original breast cancer because metastatic lesions had not been observed on the head, chest, and abdominal CT scans. Isolated PPF lesions can be treated with radiation therapy. Therefore, we carried out an endoscopic sinus biopsy for histologic assessment. In cases such as this, we recommend that the tumor be diagnosed by histological assessment, unless the patient clearly has advanced disease. To the best of our knowledge, this is the first report of severe trigeminal neuralgia that was caused by a solitary PPF metastasis from breast cancer that was confirmed by endoscopic sinus biopsy. Another unusual feature of our patient was the history of neuroendocrine carcinoma of the breast. Neuroendocrine tumors of the breast are rare, accounting for less than 0.1% of all breast cancers and less than 1% of all neuroendocrine tumors [4]. Little is known about their evolution, biologic behavior, or optimal treatment. The malignant potential of neuroendocrine tumors has been alluded to in a few reports; metastases at diverse sites have been found years after treatment of the primary site in the breast [5]. In our case, we hypothesized that it was possible that a distant metastasis from a low grade and slow-growing neuroendocrine carcinoma could appear 12 years after surgical resection of the primary tumor. In the future, a better knowledge of the biology of these tumors will hopefully provide new therapeutic targets for personalized treatment. There has been no report to date regarding metastasis affecting the PPF and causing direct compression of the trigeminal ganglion. In this case, the initial radiographic examination of the face was an important factor in the diagnosis. In summary, we report a case of metastatic breast cancer that clinically manifested as left facial paresthesia.
4. Conclusion Fig. 2. Hematoxylin and eosin stains showing that the shape of the tumor cells ranged from oval to spindle and plasmacytoid; the cells were separated by delicate fibrous stroma (hematoxylin and eosin; original magnification 400).
We have encountered a rare case of metastasis from neuroendocrine carcinoma of the breast to the PPF presenting as severe trigeminal neuralgia. We would like to highlight that
Please cite this article in press as: Takanami T, et al. Pterygopalatine fossa metastasis with severe trigeminal neuralgia from neuroendocrine carcinoma of the breast. Auris Nasus Larynx (2016), http://dx.doi.org/10.1016/j.anl.2016.04.011
G Model
ANL-2128; No. of Pages 3 T. Takanami et al. / Auris Nasus Larynx xxx (2016) xxx–xxx
metastasis to PPF should be kept in mind when a patient with history of breast cancer presents with trigeminal neuralgia. Conflict of interest There is no conflict of interests. References [1] Albayram S, Adaletli I, Selcuk H, Gulsen F, Islak C, Kocer N. Breast cancer metastasis involving pterygopalatine fossa. Headache 2004;44:927–8.
3
[2] Chan LL, Chong J, Gillenwater AM, Ginsberg LE. The pterygopalatine fossa: postoperative MR imaging appearance. AJNR Am J Neuroradiol 2000;21:1315–9. [3] Daniels DL, Mark LP, Ulmer JL, Mafee MF, McDaniel J, Shah NC. Osseous anatomy of the pterygopalatine fossa. AJNR Am J Neuroradiol 1998;19:1423–32. [4] Ogawa H, Nishio A, Satake H, Naganawa S, Imai T, Sawaki M. Neuroendocrine tumor in the breast. Radiat Med 2008;26:28–32. [5] Konstantions M, Spyridon D, Nikolaos P, Loanna G, Chris D. Neuroendocrine breast carcinoma metastatic to the liver: report of a case and review of the literature. Int J Surg Case Rep 2014;5:540–3.
Please cite this article in press as: Takanami T, et al. Pterygopalatine fossa metastasis with severe trigeminal neuralgia from neuroendocrine carcinoma of the breast. Auris Nasus Larynx (2016), http://dx.doi.org/10.1016/j.anl.2016.04.011