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      Spindle cell oncocytoma of the adenohypophysis in a woman: a case report and review of the literature

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          Abstract

          Introduction

          Spindle cell oncocytoma of the adenohypophysis is a rare tumour recently reported by Roncaroli et al. in 2002. This tumour is considered a grade I tumour by the World Health Organization.

          Case presentation

          We describe what is, to the best of our knowledge, the 14th case of its kind in the literature. A 45-year-old African woman presented clinical and radiological findings related to a nonfunctioning pituitary adenoma. The diagnosis was made on the basis of histological and immunohistochemical findings.

          Conclusion

          The purpose of this work is to report a rare pituitary tumour and to describe its histological and immunohistochemical features, which were characterized by the expression of thyroid transcription factor 1 antigen by tumour cells. This fact could support the theory of a possible common origin of these tumours in pituicytomas. In fact, thyroid transcription factor 1 is considered to be a specific marker of pituicytes.

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          Most cited references8

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          Thyroid transcription factor 1 expression in sellar tumors: a histogenetic marker?

          Pituicytomas are rare low-grade gliomas of the neurohypophysis. Their morphology and variable immunophenotype have led to speculation that they arise from pituicytes. Given the role of thyroid transcription factor 1 (TTF-1) in the developing rodent infundibulum and its expression in the adult rat neurohypophysis, we speculated that TTF-1 would be a marker of human pituicytes. Using immunohistochemistry, we found that TTF-1 is strongly expressed in fetal and adult human pituicytes. A survey of sellar masses demonstrated specific TTF-1 expression in pituicytomas (n = 3), atypical pituicytomas (n = 2), and granular cell tumors (n = 4), indicating a common pituicyte lineage. TTF-1 expression in spindle cell oncocytomas (n = 8) is less easily explained but invites speculation. Our observations may have implications for the classification of these rare sellar neoplasms, all the while acknowledging the morphological diversity of pituicyte-related neoplasms.
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            'Spindle cell oncocytoma' of the adenohypophysis: a tumor of folliculostellate cells?

            We describe five primary tumors of the adenohypophysis featuring mitochondrion-rich spindle cells. The patient ages ranged from 53 to 71 years (mean 61.6 years); two were female. All presented with panhypopituitarism. Two also had visual field defect. On neuroimaging all tumors showed suprasellar extension and were indistinguishable from pituitary adenoma. None showed imaging or operative evidence of dural involvement. All were gross totally removed: four by transsphenoidal surgery and one by frontal craniotomy. Follow-up ranged from 2 to 68 months (mean 35.4 months). No recurrences were noted. The clinical workup was noncontributory in all but two patients: one (case no. 4) with an oncocytic thyroid adenoma and another (case no. 5) with squamous carcinoma of both the uterine cervix and of vocal cord. Histologically, the five tumors were composed mainly of fascicles of spindle cells with eosinophilic, granular cytoplasm. Mitoses were rare and necrosis was absent. Neoplastic cells were immunoreactive for vimentin, epithelial membrane antigen, S-100 protein, and galectin-3. Stains for pituitary hormones, synaptophysin, chromogranin, glial fibrillary acidic protein, cytokeratin CAM5.2, smooth muscle actin, CD34, and CD68 were negative. No thyroglobulin immunoreactivity was noted in the tumor of case no. 4. Ultrastructurally, the neoplastic cells contained numerous mitochondria with lamellar cristae. The neoplastic cells were linked by intermediate junctions and desmosomes. No secretory granules were noted. The histologic, immunohistochemical, and fine structural features of these tumors were unlike those of pituitary adenoma or any other primary sellar tumor. A derivation from adenohypophyseal folliculostellate cells is suggested.
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              Spindle cell oncocytoma of the adenohypophysis: report of two recurrent cases.

              The recently described "spindle cell oncocytoma of the adenohypophysis" is a very rare and often misdiagnosed entity. A benign biologic behavior has been suggested based on the absence of recurrences with a median follow-up of 3 years. Herein, we present 2 cases of recurrent spindle cell oncocytomas. One patient is a 71-year-old woman (case no. 1) and the other a 76-year-old man (case no. 2). Recently, both underwent transsphenoidal reexploration for recurrent "pituitary adenoma." Patient no. 1 had initial surgery 11 years ago with a recurrence after 3 years that was initially stable. Ultimately, a partial resection was performed after compression of optic pathways by the tumor, and approximately 1 year later, re-resection was carried out. Patient no. 2 had initial surgery 10 years ago with recurrence and resection after 3 years. He recently presented with a large mass that involved the pituitary fossa and base of the skull, with extension into the nasopharynx and nasal cavity. The primary and recurrent lesions of both cases showed similar architecture with interlacing fascicles of spindle cells that alternated with areas of epithelioid-like cells that exhibited eosinophilic, granular cytoplasm. Neoplastic cells were positive for vimentin, S-100 protein, and epithelial membrane antigen, and negative for glial fibrillary acidic protein, chromogranin, and pituitary hormones. Increased mitotic activity was noted in 1 lesion (case no. 2), although both cases had high Ki-67 indices (18% and 20%, respectively). The ultrastructural features of both cases were characteristic with intracytoplasmic accumulations of large mitochondria. The histopathologic features of these lesions are consistent with spindle cell oncocytoma of the adenohypophysis. In summary, we are reporting 2 cases of recurrent spindle cell oncocytoma of adenohypophysis with longer follow-up than previously published cases, suggesting the possibility of a more aggressive behavior than has been initially considered.
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                Author and article information

                Journal
                J Med Case Reports
                Journal of Medical Case Reports
                BioMed Central
                1752-1947
                2011
                14 February 2011
                : 5
                : 64
                Affiliations
                [1 ]Department of Pathology, La Rabta Hospital, Bab Saadoun, Tunis 2037, Tunisia
                [2 ]Department of Neurosurgery, La Rabta Hospital, Bab Saadoun, Tunis 2037, Tunisia
                Article
                1752-1947-5-64
                10.1186/1752-1947-5-64
                3047430
                21320334
                5fa8b242-deca-4d25-b6d3-ded6b1fc75ab
                Copyright ©2011 Mlika et al; licensee BioMed Central Ltd.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 4 March 2010
                : 14 February 2011
                Categories
                Case Report

                Medicine
                Medicine

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