- Division of Neurosurgery, Northwestern University, Feinberg, School of Medicine, Chicago, Illinois, US
- Division of Pathology, Northwestern University, Feinberg, School of Medicine, Chicago, Illinois, US
- Division of Neurology, Northwestern University, Feinberg, School of Medicine, Chicago, Illinois, USA
James P. Chandler
Division of Neurosurgery, Northwestern University, Feinberg, School of Medicine, Chicago, Illinois, US
DOI:10.4103/2152-7806.83932Copyright: © 2011 Ogiwara H. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
How to cite this article: Ogiwara H, Dubner S, Shafizadeh S, Raizer J, Chandler JP. Spindle cell oncocytoma of the pituitary and pituicytoma: Two tumors mimicking pituitary adenoma. Surg Neurol Int 17-Aug-2011;2:116
How to cite this URL: Ogiwara H, Dubner S, Shafizadeh S, Raizer J, Chandler JP. Spindle cell oncocytoma of the pituitary and pituicytoma: Two tumors mimicking pituitary adenoma. Surg Neurol Int 17-Aug-2011;2:116. Available from: http://sni.wpengine.com/surgicalint_articles/spindle-cell-oncocytoma-of-the-pituitary-and-pituicytoma-two-tumors-mimicking-pituitary-adenoma/
Background:Spindle cell oncocytoma (SCO) and pituicytoma are rare nonfunctioning tumors of the pituitary. Both tumors are low grade and macroscopically indistinguishable from a nonfunctioning pituitary adenoma.We report one case of SCO and one case of pituicytoma and review the previous literature.
Case Description:One patient was a 39-year-old man who presented with progressive headache, visual blurring, and polyuria of 3-year duration. He underwent partial resection (30% of the tumor) and postoperative adjuvant radiation therapy. Histopathology revealed SCO. However, after 9 months, the residual tumor grew and partial resection (70% of the tumor) was performed again. Four months after the second surgery, the tumor recurred again and he underwent transsphenoidal resection of the tumor with stable residual tumor to date. The other patient was a 59-year-old man who presented with a 3-month history of visual decline, fatigue, difficulty in writing, and polyuria. He underwent transsphenoidal resection (total) of the tumor. Histopathology revealed pituicytoma. He has been stable without evidence of recurrence for 1 year and 4 months.
Conclusion:To date, there are 15 reported cases of SCO and 45 reported cases of pituicytoma including our cases. An incomplete resection of the tumor was a significant risk factor for recurrence in both SCO and pituicytoma (P = 0.0014 and P = 0.019, respectively). These tumors have a tendency to be hypervascular, which may hamper total resection. Epithelial membrane antigen (EMA) and mitochondria positivity is characteristic to SCO and they are considered to be important immunomarkers to distinguish these tumors.
Keywords: Pituicytoma, recurrence, spindle cell oncocytoma, total resection
Spindle cell oncocytoma (SCO) of the pituitary and pituicytoma are rare nonfunctional tumors, which have recently been codified as distinct entities by the 2007 WHO classification of central nervous system (CNS) tumors. SCO is defined as a spindled-to-epithelioid, oncocytic, nonendocrine neoplasm of the anterior hypophysis that manifests in adults and follows a benign clinical course.[
To date, there are 14 reported cases of SCO[
Surgical specimens were formalin fixed and routinely processed. Five-micron-thick tissue sections were stained by hematoxylin and eosin, and immunohistochemistry. The following antibodies were applied: anti-glial fibrillary acidic protein (GFAP; polyclonal, 1:2800; DAKO), anti-S-100 protein (polyclonal, 1:2000; DAKO), antithyroid transcription factor 1 (TTF-1; monoclonal, prediluted; Cell Marque), anti-EMA (epithelial membrane antigen; monoclonal, 1:500; DAKO), anti-calretinin (monoclonal, prediluted; Ventana), anti-Ki-67 (MIB-1; monoclonal, 1:200; DAKO), anti-galectin-3 (monoclonal, 1:25; Invitrogen), anti-CD34 (monoclonal, 1:25; DAKO), anti-CD57 (monoclonal, 1:50; Becton Dickenson), anti-CD68 (monoclonal, 1:400; DAKO), anti-mitochondria (monoclonal, 1:200, Leinco Technologies), anti-chromogranin (monoclonal, prediluted; Ventana), and anti-synaptophysin (polyclonal, 1:50; DAKO).
Case 1 (spindle cell oncocytoma)
A 39-year-old man presented with progressive headache, loss of stamina and libido, visual blurring, and polyuria of 3-year duration to an outside hospital. Visual field assessment revealed a bitemporal hemianopia. Endocrinologic evaluation revealed low testosterone and high prolactin levels. He was started on bromocriptine and testosterone replacement. The MRI scan revealed a suprasellar lesion with the compression of the optic nerves [Figure
Histopathologic findings for patient 1 (original magnification, ×400). These images are from the patient's third resection. Microscopic evaluation reveals interlacing fascicles of spindled to epithelioid cells with eosinophilic, variably oncocytic cytoplasm. Mild to moderate nuclear atypia is present. (a) Mitotic figures are rare. Immunostains for (b) transcription factor 1, (c) epithelial membrane antigen, (d) and mitochondria are positive
Case 2 (pituicytoma)
A 59-year-old man presented with a 3-month history of visual decline, fatigue, difficulty in writing, and polyuria. Visual field assessment revealed bitemporal hemianopia. The serum pituitary hormonal profile was grossly normal. The serum sodium level was 136 mEq/l. An MRI scan revealed a giant suprasellar clival lesion with the distortion of the chiasm and invasion of the clivus [Figure
Histopathologic findings for patient 2 (original magnification of both, ×400). Microscopic examination reveals a solid neoplasm composed of elongate, bipolar spindle cells arranged in interlacing fascicles or in a storiform pattern. Individual tumor cells contain abundant eosinophilic cytoplasm and cell shapes range from short and plump to elongate and angulated. There is no significant cytoplasmic granularity or vacuolization. (a) Mitotic figures are rare. (b) Immunostain for transcription factor 1 is positive
SCO was first described by Roncaroli et al. as a tumor containing fascicles of spindle cells with eosinophilic, granular cytoplasm, and a distinct immunophenotype which includes positive immunoreaction to vimentin, EMA, S-100, and galectin-3 with negative immunoreactions to pituitary hormones, synaptophysin, chromogranin, and cytokeratins. They suggested a benign nature based on the absence of cellular anaplasia, mitoses, and necrosis along with a low Ki-67 proliferation labeling index, and that no recurrence was observed in all five cases of their series with a mean follow-up of 3 years.[
However, more recently a further nine additional cases have been described, six of which recurred.[
The previous report described SCOs as firm, fibrous, and adherent to surrounding structures making dissection difficult especially in recurrent cases.[
Pituicytoma is a rare, solid, glial, and low-grade tumor of adult pituitary. Histologically, pituicytoma consists of elongate, bipolar spindle cells arranged in interlacing fascicles or assuming a storiform pattern.[
Of 29 cases of pituicytoma with a detailed follow-up, 6 cases (20.7%) recurred. The Ki-67 labeling index was low and mitosis or necrosis was not observed in any recurrent case. The mean time between the original surgery and recurrence was 1.1 years (ranging from 5 months to 2 years). All the recurrent cases underwent an incomplete resection at the original surgery. Similar to SCO, an incomplete resection of the tumor was a significant risk factor for recurrence (P = 0.019;
The immunostain by S-100 was positive in all the reported cases of SCOs and pituicytomas. GFAP positivity varied in pituicytomas. The immunostain by GFAP was available in 38 cases of pituicytoma, of which 9 cases (23.7%) were negative for GFAP. The immunostain by galectin-3 was positive in our cases of SCO and pituicytoma. Rodríguez et al. showed that galectin-3 is expressed not only in SCO but also in pituicytoma and granular cell tumors. And they suggested that this marker is of little use to differentiate those diagnoses.[
In all 15 reported cases of SCO, the immunostain by EMA was available and showed positivity in every case. The immunostain by EMA was available in 32 cases of pituicytoma. EMA was negative in 28 (87.5%) cases and only focally positive in 4 cases. Therefore, EMA is considered to be important to distinguish these tumors.
There is little evidence for the sensitivity of SCOs to radiotherapy. Four of five SCO cases which underwent radiation therapy recurred.[
SCO of pituitary and pituicytoma are rare entities with 15 and 45 cases reported to date, respectively, including our cases. Clinical behaviors of these tumors are very similar. Both tumors have a tendency to recur locally if not completely resected. The incomplete resection of the tumor was a significant risk factor for recurrence in both SCO and pituicytoma (P = 0.0014 and P = 0.019, respectively). Both tumors have a tendency to be hypervascular, which may hamper total resection. EMA and mitochondria positivity is characteristic of SCO and they are considered to be important immunomarkers to distinguish these tumors.
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