- Email: [email protected]
Oncocytic meningioma presenting with intratumoral hemorrhage
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Case Reports / Journal of Clinical Neuroscience 20 (2013) 1622–1624
4. Conclusions
References
We present, to our knowledge, the first report of a primary germinoma of the fourth ventricle in Europe. A previous patient reported by Yen et al. concluded that despite its rarity and nonspecific imaging findings, primary germinoma should be included in the differential diagnosis of medullary masses with extension into the fourth ventricle occurring in young Asian women.5 Our patient’s case supports the inclusion of primary germinoma in the differential of such masses, and highlights that it should be considered in patients of all ethnicities and location.
1. Sano KL. Pathogenesis of intracranial germ cell tumours reconsidered. J Neurosurg 1999;90:258–64. 2. Shuto T, Ohtake M, Matsunga S, et al. Primary medulla oblongata germinoma in a male patient. J Clin Neurosci 2012;19:769–71. 3. Yasuhara T, Ichikawa T, Miyoshi Y, et al. Primary Germinoma in the medulla oblongata. Neurol Med Chir (Tokyo) 2011;51:326–9. 4. Kyritsis A. Management of primary intracranial germ cell tumours. J Neurooncol 2010;96:143–9. 5. Yen P, Chou A, Chen C, et al. Primary medulla oblongata germinoma: a case report and review of the literature. J Neurooncol 2003;62:339–42.
doi:http://dx.doi.org/10.1016/j.jocn.2012.11.025
Oncocytic meningioma presenting with intratumoral hemorrhage Yasuo Sasagawa ⇑, Osamu Tachibana, Takaaki Iida, Hideaki Iizuka Department of Neurosurgery, Kanazawa Medical University, 1-1 Daigaku, Uchinada 920-0293, Ishikawa, Japan
a r t i c l e
i n f o
Article history: Received 25 September 2012 Accepted 17 November 2012
Keywords: Intratumoral hemorrhage Mitochondria Oncocytic meningioma
a b s t r a c t Oncocytic meningiomas have been recently reported as a rare variant of meningiomas. Immunohistochemical analysis shows that neoplastic cells are positive for antimitochondrial antibodies. We report our first patient with oncocytic meningioma, presenting with intratumoral bleeding. A 72-year-old woman suffered from a disturbance of consciousness. A CT scan showed a tumor with intratumoral hemorrhage. An emergency craniotomy was performed and the tumor and hematoma were removed. Examination of the tumor revealed meningothelial cells with oncocytic change. We discuss the clinicopathological considerations of this uncommon variant and review the pertinent literature. Ó 2013 Elsevier Ltd. All rights reserved.
1. Introduction The presence of numerous oncocytic cells with cytoplasm rich in large mitochondria allows us to define some tumors as ‘‘oncocytoma’’. Oncocytic transformation is an infrequent event within the central nervous system. It is limited to neoplasms of the pituitary gland and choroid plexus.1 Roncaroli et al.2 first described the morphological features of this meningioma type as ‘‘oncocytic meningioma’’ in 1997. The tumors were composed mostly of large polygonal cells with finely granular eosinophilic cytoplasm. Results of immunohistochemical analysis showed that neoplastic cells were immunoreactive for antimitochondrial antibodies. We describe our first patient with oncocytic meningioma, who presented with intratumoral hemorrhage. We discuss the clinicopathological considerations of this uncommon variant based on a review of the pertinent literature. 2. Case report A 72-year-old woman was admitted to hospital due to bacterial enteritis in July 2011. She had fallen and sustained blunt forehead trauma. MRI was performed to rule out traumatic intracranial hemorrhage and revealed a 40 35 30 mm tumor located in the left frontal region. The tumor showed isointensity on ⇑ Corresponding author. Tel.: +81 762 86 2211; fax: +81 762 86 1702. E-mail address: [email protected] (Y. Sasagawa).
T1-weighted images and hyperintensity on T2-weighted images. It was revealed as a moderately and homogeneously enhancing, well-circumscribed tumor after gadolinium administration (Fig. 1a). However, no distinct intracranial hemorrhage was seen. One week later, she had a sudden disturbance of consciousness and was transferred to our hospital. A CT scan demonstrated a massive intratumoral hemorrhage and brain shift (Fig. 1b). An emergency craniotomy was performed. The tumor attached to the dura was elastic, but partially hard. The boundary between the tumor and cerebral parenchyma was clear. A total resection of the hematoma, tumor and attached dura was achieved. Microscopically, the lesion included classical meningothelial cells and the presence of fibroblastic proliferation. Foci of recent hemorrhage into the tumor were seen. In addition, many areas were composed of sheets and nests of neoplastic elements in which cells were polygonal with distinct cytoplasmic borders and finely granular eosinophilic cytoplasm (Fig. 2a and b). These cells constituted a large proportion of the total neoplastic population. Immunohistochemically, the tumor cells stained positive for epithelial membrane antigen. There were two mitotic figures per 10 high power fields and the proliferation index was 5% based on the Ki-67 labeling index. Neither necrosis nor cerebral invasion was present. The eosinophilic cells were strongly positive for antimitochondrial antibodies (Fig. 2c). Electron microscopy from a paraffin-embedded block showed cytoplasmic accumulations of a large number of swollen mitochondria (Fig. 2d). The microscopic and ultrastructural findings determined oncocytic meningioma as the final diagnosis. Postoperative MRI showed complete resection with no
Case Reports / Journal of Clinical Neuroscience 20 (2013) 1622–1624
1623
Fig. 1. (a) T1-weighted coronal MRI with gadolinium showing a lesion with dural attachment in the left frontal lobe. (b) Coronal CT scan showing intratumoral hemorrhage with midline shift and ventricular rupture.
Fig. 2. (a) Histopathological examination of the surgical sample showing the lesion composed of sheets of neoplastic elements delimited by fibrous connective tissue rich in vessels (haematoxylin and eosin [H&E] stain, original magnification 100). (b) Sample of the oncocytic meningioma cells showing polygonal cells with eosinophilic granular cytoplasm (H&E, original magnification 400). (c) Sample of the oncocytic meningioma cells showing immunoreactivity for antimitochondrial antibodies (original magnification 400). (d) Electron microscopy from a paraffin-embedded block showing numerous swollen mitochondria in the cytoplasm of granular cells (original magnification 8000).
evidence of residual tumor. Unfortunately, the patient made an unsatisfactory postoperative recovery with motor aphasia and hemiparesis, and was transferred to another hospital for rehabilitation.
3. Discussion Twenty-one patients, including the one reported here, with oncocytic meningiomas have been reported2–6 (Table 1). The first
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Case Reports / Journal of Clinical Neuroscience 20 (2013) 1622–1624
Table 1 Summary of reported cases of oncocytic meningioma Case 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21
Author (year) 2
Roncaroli (1997)
Caldarella3 (2002) Gallina4 (2006)
Marucci5 (2007) Zunarelli6 (2010)
present patient
Age/sex
Location
Necrosis
Brain invasion
Mitoses (HPF)
MIB-1 LI (%)
Follow-up (months)
54/F 61/F 68/F 73/M 55/F 66/F 30/M 51/F 64/M 52/F 54/F 49/F 35/F 26/M 41/M 59/M 48/M 51/M 45/M 49/M 72/F
left sphenoid falx left occipital C frontal C right parietotemporal C right sphenoid right parietal C left sphenoid olfactory groove olfactory groove left frontal C frontal base right optic nerve right frontal C right parietotemporal C right frontal C right frontal C right frontal C right parietal C right frontal C left frontal C
(+) (+) (+) ( ) (+) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( )
( ) ( ) ( ) (+) (+) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( )
12/20 8/20 21/20 6/20 18/20 3/20 2/20 absent absent absent 1/10 2/10 1/10 2/10 1/10 6/10 2/10 2/10 1/10 1/10 2/10
20 2 20 3 30 4 10 3 1 1 7 10 2 5 3 20 5 5 2 4 5
82 rec rec 8 7 rec 8 46 45 11 6 rec 9 29 54 rec 61 46 36 48 24
(18, 27) (30)
(24)
(12, 48)
(18, 34)
C = convexity, HPF = per high power field, LI = labeling index, rec = recurrence.
six patients with oncocytic meningiomas reported by Roncaroli et al.2 accounted for 1% of all 589 patients with meningioma. Based on histopathological and clinical findings, Roncaroli et al. argued that oncocytic meningiomas have a worse prognosis than benign meningiomas and advocated the use of postsurgical radiation therapy. Five patients recurred after surgery, so the recurrence rate of 5/21 (23.8%) patients was higher than that of meningiomas in general. However, necrosis and brain invasion were not significant in any patients, except for those in the study of Roncaroli et al. Thus, the absence of other established histopathological findings realted to likelihood of recurrence and/or aggressive behavior led other authors to conclude that oncocytic morphology should not automatically be assigned a higher grade, and that the diagnosis of oncocytic meningioma should not indicate a need for adjuvant radiotherapy in itself.4,6 In fact, some authors reported a favorable outcome in most oncocytic tumors, including salivary gland neoplasms and pituitary adenomas.1 Additionally, all patients, except the one reported here, were Italian. These tumors may therefore be influenced by genetic and/or environmental factors. Interestingly, our patient had an intratumoral hemorrhage, which is unusual for meningioma, occurring in only 1.3–2.4% of all meningiomas. Bošnjak et al.7 reviewed 145 literature-derived cases and found three main factors associated with an increased propensity for hemorrhage in meningomas: intraventricular and convexity locations; fibrous histopathology; and age <30 or >70 years. Indeed, our patient exhibited all of these proposed features. The possible mechanisms of intratumoral bleeding in meningiomas include endothelial proliferation with subsequent vascular doi:http://dx.doi.org/10.1016/j.jocn.2012.11.023
occlusion and necrosis and thinned and friable vascular walls. Other factors that may promote bleeding in meningiomas include hypertension, traumatic head injury and the use of anticoagulant medications, but the exact pathogenesis is still unclear. In summary, oncocytic meningiomas are a distinct variant of intracranial meningothelial neoplasms though the tumor is not included in the 2007 World Health Organization classification of central nervous system tumors. Given the extreme rarity of this meningioma variant, the clinical and pathological characterization is incomplete. Additional studies are necessary to better understand the features of oncocytic meningiomas.
References 1. Niveiro M, Aranda FI, Payá A, et al. Oncocytic transformation in pituitary adenomas: immunohistochemical analyses of 65 cases. Arch Pathol Lab Med 2004;128:776–80. 2. Roncaroli F, Riccioni L, Cerati M, et al. Oncocytic meningioma. Am J Surg Pathol 1997;21:375–82. 3. Caldarella A, Buccoliero AM, Marini M, et al. Oncocytic meningioma: a case report. Pathol Res Pract 2002;198:109–13. 4. Gallina P, Buccoliero AM, Mariotti F, et al. Oncocytic meningiomas: cases with benign histopathological features and a favorable clinical course. J Neurosurg 2006;105:736–8. 5. Marucci G, Betts CM, Frank G, et al. Oncocytic meningioma: report of a case with progression after radiosurgery. Int J Surg Pathol 2007;15:77–81. 6. Zunarelli E, Tallarico E, Valentini A, et al. Oncocytic meningioma: study of eight new cases and analysis of 13 reported cases. Pathology 2010;42:587–9. 7. Bošnjak R, Derham C, Popovic´ M, et al. Spontaneous intracranial meningioma bleeding: clinicopathological features and outcome. J Neurosurg 2005;103: 473–84.
Case Reports / Journal of Clinical Neuroscience 20 (2013) 1622–1624
4. Conclusions
References
We present, to our knowledge, the first report of a primary germinoma of the fourth ventricle in Europe. A previous patient reported by Yen et al. concluded that despite its rarity and nonspecific imaging findings, primary germinoma should be included in the differential diagnosis of medullary masses with extension into the fourth ventricle occurring in young Asian women.5 Our patient’s case supports the inclusion of primary germinoma in the differential of such masses, and highlights that it should be considered in patients of all ethnicities and location.
1. Sano KL. Pathogenesis of intracranial germ cell tumours reconsidered. J Neurosurg 1999;90:258–64. 2. Shuto T, Ohtake M, Matsunga S, et al. Primary medulla oblongata germinoma in a male patient. J Clin Neurosci 2012;19:769–71. 3. Yasuhara T, Ichikawa T, Miyoshi Y, et al. Primary Germinoma in the medulla oblongata. Neurol Med Chir (Tokyo) 2011;51:326–9. 4. Kyritsis A. Management of primary intracranial germ cell tumours. J Neurooncol 2010;96:143–9. 5. Yen P, Chou A, Chen C, et al. Primary medulla oblongata germinoma: a case report and review of the literature. J Neurooncol 2003;62:339–42.
doi:http://dx.doi.org/10.1016/j.jocn.2012.11.025
Oncocytic meningioma presenting with intratumoral hemorrhage Yasuo Sasagawa ⇑, Osamu Tachibana, Takaaki Iida, Hideaki Iizuka Department of Neurosurgery, Kanazawa Medical University, 1-1 Daigaku, Uchinada 920-0293, Ishikawa, Japan
a r t i c l e
i n f o
Article history: Received 25 September 2012 Accepted 17 November 2012
Keywords: Intratumoral hemorrhage Mitochondria Oncocytic meningioma
a b s t r a c t Oncocytic meningiomas have been recently reported as a rare variant of meningiomas. Immunohistochemical analysis shows that neoplastic cells are positive for antimitochondrial antibodies. We report our first patient with oncocytic meningioma, presenting with intratumoral bleeding. A 72-year-old woman suffered from a disturbance of consciousness. A CT scan showed a tumor with intratumoral hemorrhage. An emergency craniotomy was performed and the tumor and hematoma were removed. Examination of the tumor revealed meningothelial cells with oncocytic change. We discuss the clinicopathological considerations of this uncommon variant and review the pertinent literature. Ó 2013 Elsevier Ltd. All rights reserved.
1. Introduction The presence of numerous oncocytic cells with cytoplasm rich in large mitochondria allows us to define some tumors as ‘‘oncocytoma’’. Oncocytic transformation is an infrequent event within the central nervous system. It is limited to neoplasms of the pituitary gland and choroid plexus.1 Roncaroli et al.2 first described the morphological features of this meningioma type as ‘‘oncocytic meningioma’’ in 1997. The tumors were composed mostly of large polygonal cells with finely granular eosinophilic cytoplasm. Results of immunohistochemical analysis showed that neoplastic cells were immunoreactive for antimitochondrial antibodies. We describe our first patient with oncocytic meningioma, who presented with intratumoral hemorrhage. We discuss the clinicopathological considerations of this uncommon variant based on a review of the pertinent literature. 2. Case report A 72-year-old woman was admitted to hospital due to bacterial enteritis in July 2011. She had fallen and sustained blunt forehead trauma. MRI was performed to rule out traumatic intracranial hemorrhage and revealed a 40 35 30 mm tumor located in the left frontal region. The tumor showed isointensity on ⇑ Corresponding author. Tel.: +81 762 86 2211; fax: +81 762 86 1702. E-mail address: [email protected] (Y. Sasagawa).
T1-weighted images and hyperintensity on T2-weighted images. It was revealed as a moderately and homogeneously enhancing, well-circumscribed tumor after gadolinium administration (Fig. 1a). However, no distinct intracranial hemorrhage was seen. One week later, she had a sudden disturbance of consciousness and was transferred to our hospital. A CT scan demonstrated a massive intratumoral hemorrhage and brain shift (Fig. 1b). An emergency craniotomy was performed. The tumor attached to the dura was elastic, but partially hard. The boundary between the tumor and cerebral parenchyma was clear. A total resection of the hematoma, tumor and attached dura was achieved. Microscopically, the lesion included classical meningothelial cells and the presence of fibroblastic proliferation. Foci of recent hemorrhage into the tumor were seen. In addition, many areas were composed of sheets and nests of neoplastic elements in which cells were polygonal with distinct cytoplasmic borders and finely granular eosinophilic cytoplasm (Fig. 2a and b). These cells constituted a large proportion of the total neoplastic population. Immunohistochemically, the tumor cells stained positive for epithelial membrane antigen. There were two mitotic figures per 10 high power fields and the proliferation index was 5% based on the Ki-67 labeling index. Neither necrosis nor cerebral invasion was present. The eosinophilic cells were strongly positive for antimitochondrial antibodies (Fig. 2c). Electron microscopy from a paraffin-embedded block showed cytoplasmic accumulations of a large number of swollen mitochondria (Fig. 2d). The microscopic and ultrastructural findings determined oncocytic meningioma as the final diagnosis. Postoperative MRI showed complete resection with no
Case Reports / Journal of Clinical Neuroscience 20 (2013) 1622–1624
1623
Fig. 1. (a) T1-weighted coronal MRI with gadolinium showing a lesion with dural attachment in the left frontal lobe. (b) Coronal CT scan showing intratumoral hemorrhage with midline shift and ventricular rupture.
Fig. 2. (a) Histopathological examination of the surgical sample showing the lesion composed of sheets of neoplastic elements delimited by fibrous connective tissue rich in vessels (haematoxylin and eosin [H&E] stain, original magnification 100). (b) Sample of the oncocytic meningioma cells showing polygonal cells with eosinophilic granular cytoplasm (H&E, original magnification 400). (c) Sample of the oncocytic meningioma cells showing immunoreactivity for antimitochondrial antibodies (original magnification 400). (d) Electron microscopy from a paraffin-embedded block showing numerous swollen mitochondria in the cytoplasm of granular cells (original magnification 8000).
evidence of residual tumor. Unfortunately, the patient made an unsatisfactory postoperative recovery with motor aphasia and hemiparesis, and was transferred to another hospital for rehabilitation.
3. Discussion Twenty-one patients, including the one reported here, with oncocytic meningiomas have been reported2–6 (Table 1). The first
1624
Case Reports / Journal of Clinical Neuroscience 20 (2013) 1622–1624
Table 1 Summary of reported cases of oncocytic meningioma Case 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21
Author (year) 2
Roncaroli (1997)
Caldarella3 (2002) Gallina4 (2006)
Marucci5 (2007) Zunarelli6 (2010)
present patient
Age/sex
Location
Necrosis
Brain invasion
Mitoses (HPF)
MIB-1 LI (%)
Follow-up (months)
54/F 61/F 68/F 73/M 55/F 66/F 30/M 51/F 64/M 52/F 54/F 49/F 35/F 26/M 41/M 59/M 48/M 51/M 45/M 49/M 72/F
left sphenoid falx left occipital C frontal C right parietotemporal C right sphenoid right parietal C left sphenoid olfactory groove olfactory groove left frontal C frontal base right optic nerve right frontal C right parietotemporal C right frontal C right frontal C right frontal C right parietal C right frontal C left frontal C
(+) (+) (+) ( ) (+) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( )
( ) ( ) ( ) (+) (+) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( ) ( )
12/20 8/20 21/20 6/20 18/20 3/20 2/20 absent absent absent 1/10 2/10 1/10 2/10 1/10 6/10 2/10 2/10 1/10 1/10 2/10
20 2 20 3 30 4 10 3 1 1 7 10 2 5 3 20 5 5 2 4 5
82 rec rec 8 7 rec 8 46 45 11 6 rec 9 29 54 rec 61 46 36 48 24
(18, 27) (30)
(24)
(12, 48)
(18, 34)
C = convexity, HPF = per high power field, LI = labeling index, rec = recurrence.
six patients with oncocytic meningiomas reported by Roncaroli et al.2 accounted for 1% of all 589 patients with meningioma. Based on histopathological and clinical findings, Roncaroli et al. argued that oncocytic meningiomas have a worse prognosis than benign meningiomas and advocated the use of postsurgical radiation therapy. Five patients recurred after surgery, so the recurrence rate of 5/21 (23.8%) patients was higher than that of meningiomas in general. However, necrosis and brain invasion were not significant in any patients, except for those in the study of Roncaroli et al. Thus, the absence of other established histopathological findings realted to likelihood of recurrence and/or aggressive behavior led other authors to conclude that oncocytic morphology should not automatically be assigned a higher grade, and that the diagnosis of oncocytic meningioma should not indicate a need for adjuvant radiotherapy in itself.4,6 In fact, some authors reported a favorable outcome in most oncocytic tumors, including salivary gland neoplasms and pituitary adenomas.1 Additionally, all patients, except the one reported here, were Italian. These tumors may therefore be influenced by genetic and/or environmental factors. Interestingly, our patient had an intratumoral hemorrhage, which is unusual for meningioma, occurring in only 1.3–2.4% of all meningiomas. Bošnjak et al.7 reviewed 145 literature-derived cases and found three main factors associated with an increased propensity for hemorrhage in meningomas: intraventricular and convexity locations; fibrous histopathology; and age <30 or >70 years. Indeed, our patient exhibited all of these proposed features. The possible mechanisms of intratumoral bleeding in meningiomas include endothelial proliferation with subsequent vascular doi:http://dx.doi.org/10.1016/j.jocn.2012.11.023
occlusion and necrosis and thinned and friable vascular walls. Other factors that may promote bleeding in meningiomas include hypertension, traumatic head injury and the use of anticoagulant medications, but the exact pathogenesis is still unclear. In summary, oncocytic meningiomas are a distinct variant of intracranial meningothelial neoplasms though the tumor is not included in the 2007 World Health Organization classification of central nervous system tumors. Given the extreme rarity of this meningioma variant, the clinical and pathological characterization is incomplete. Additional studies are necessary to better understand the features of oncocytic meningiomas.
References 1. Niveiro M, Aranda FI, Payá A, et al. Oncocytic transformation in pituitary adenomas: immunohistochemical analyses of 65 cases. Arch Pathol Lab Med 2004;128:776–80. 2. Roncaroli F, Riccioni L, Cerati M, et al. Oncocytic meningioma. Am J Surg Pathol 1997;21:375–82. 3. Caldarella A, Buccoliero AM, Marini M, et al. Oncocytic meningioma: a case report. Pathol Res Pract 2002;198:109–13. 4. Gallina P, Buccoliero AM, Mariotti F, et al. Oncocytic meningiomas: cases with benign histopathological features and a favorable clinical course. J Neurosurg 2006;105:736–8. 5. Marucci G, Betts CM, Frank G, et al. Oncocytic meningioma: report of a case with progression after radiosurgery. Int J Surg Pathol 2007;15:77–81. 6. Zunarelli E, Tallarico E, Valentini A, et al. Oncocytic meningioma: study of eight new cases and analysis of 13 reported cases. Pathology 2010;42:587–9. 7. Bošnjak R, Derham C, Popovic´ M, et al. Spontaneous intracranial meningioma bleeding: clinicopathological features and outcome. J Neurosurg 2005;103: 473–84.