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| CASE REPORT |
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| Year : 2022 | Volume
: 5
| Issue : 2 | Page : 37-40 |
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Simultaneous coexisting intracranial gliosarcoma and pituitary adenoma: A rare phenomenon
Sunit Mediratta1, Sudheer Kumar Tyagi2, Debnath Dwaipayan2
1 Department of Neurosurgery, Indraprastha Apollo Hospital, New Delhi, India
2 Department of Neurosurgery, Apollo Hospital, New Delhi, India
| Date of Submission | 07-Nov-2022 |
| Date of Acceptance | 12-Mar-2023 |
| Date of Web Publication | 08-Apr-2023 |
Correspondence Address:
Dr. Sunit Mediratta
Department of Neurosurgery, Apollo Hospital, Sarita Vihar, New Delhi 110076
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/IJNO.IJNO_9_22
Simultaneous occurrence of two primary brain tumors with different pathologies is rare. These tumors have occasionally been reported with history of phakomatosis or radiotherapy. De novo occurrence is extremely rare with only 71 cases reported. We present a case report of a simultaneous occurrence of a temporal lobe gliosarcoma with a pituitary adenoma. The patient underwent surgery for both lesions in the same hospitalization and survived 13 months. Keywords: Diverse pathology, multiple brain tumor, pituitary adenoma, sarcoma, simultaneous
How to cite this article:
Mediratta S, Tyagi SK, Dwaipayan D. Simultaneous coexisting intracranial gliosarcoma and pituitary adenoma: A rare phenomenon. Int J Neurooncol 2022;5:37-40 |
| Introduction | |  |
Gliosarcoma (GSM) is a primary central nervous system tumor composed of both malignant glial and sarcomatous elements. According to the World Health Organization 2016 classification of glial tumors, this tumor type is considered grade IV, and it has been suggested that approximately 2% of otherwise conventional glioblastomas generate malignant mesenchymal components.[1] Pituitary adenomas are one of the most common lesions in the sellar area, and they are generally classified into functioning and nonfunctioning neoplasms. Lactotrophic tumors or prolactinomas constitute the most prevalent secreting pituitary adenomas.[2],[3]
The simultaneous occurrence of multiple primary brain tumors with different histology has been reported previously. However, in most of these patients, there was either a history of previous cranial radiotherapy[4] or familial tumor syndromes.[5]De novo occurrence of these simultaneous tumors is extremely rare with only 71 cases reported[6] in English literature. There has been no case report of simultaneous occurrence of GSM and pituitary adenoma to the best of our knowledge. This is the first case report to identify the coexistence of a pituitary adenoma and a GSM. The current literature is reviewed, and a common pathogenetic mechanism was proposed.
| Case report | | |
A 60-year-old male presented with weakness of the left upper limb and decreasing memory. On clinical evaluation, he was found to have occasionally impaired immediate memory with bitemporal hemianopia and 4 ± 5 power in the left upper limb. His magnetic resonance imaging brain revealed a lesion in the right temporal lobe and sellar region. The temporal lobe lesion was hypointense on T1-weighted images [Figure 1] and heterogeneously hyperintense on T2-weighted images with a heterogenous contrast enhancement with perilesional edema [Figure 2]. The sellar-suprasellar lesion was hypointense on T1 [Figure 1] and hyperintense on T2-weighted images and showed homogenous contrast enhancement [Figure 2]. He was diagnosed preoperatively to have a temporal lobe glioma along with a pituitary adenoma after imaging studies. | Figure 1: T1-weighted and postcontrast image showing hypointense lesion in right temporal lobe with a well-defined isointense lesion in sellar region
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 | Figure 2: Postcontrast MR image shows heterogenous enhancement of the temporal lobe lesion with perilesional edema. The sellar lesion shows homogenous enhancement
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Hematological studies along with his hormonal profile were normal. He underwent a frontotemporal craniotomy and total excision of the right temporal lobe lesion. This was followed by a transnasal transsphenoidal decompression of the sellar tumor 2 days later. The histopathological examination of the temporal lesion was reported as GSM [Figure 3], and the sellar tumor was reported as a pituitary adenoma [Figure 4]. The patient was discharged without any new neurological deficit and received postoperative radiotherapy. The patient survived for 13 months after the diagnosis was established. | Figure 3: Haemotoxylin & Eosin (H & E) stain suggestive of pituitary adenoma showing a tumor exhibiting solid, pseudopapillary, pseudoacinar, and nested pattern with prominent sinusoidal vascularity. Interspersed individual cells are small, oval to round and monomorphic
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 | Figure 4: H & E stain showing a highly cellular, high grade tumor with mesenchymal and glial components suggestive of gliosarcoma. The tumor cells have pleomorphic round to oval nuclei in fibrillary stroma. Rich arborizing vessels with large foci of necrosis present
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| Discussion | | |
Simultaneous primary lesions of the brain with different histology have been published in the literature and their occurrence is not well understood.[6] Meningioma is the most common tumor associated with the simultaneous occurrence of another primary intracranial tumor. The most frequently described combination is meningioma associated with glioma followed by schwannoma and pituitary adenoma.[7],[8],[9] The simultaneous coexistence of a primary intracranial GSM and pituitary adenoma has never been reported as per our extensive literature search.
Various hypothesis of an association between the “tumor pairs” has been advocated in the past. Perilesional edema caused by the first tumor has been in the past, proposed to initiate neoplastic transformation in the adjacent area owing to tissue irritation. However, it does not explain occurrence of tumors in anatomically distant areas from the primary site.[6],[10],[11] Platelet-derived growth factor receptors have been proposed as key factors in angiogenesis that can lead to tumor development. Suzuki et al.reported simultaneous occurrence of glioblastoma and meningioma with both tumors strongly positive for PGDF receptors.[12] The alpha subunit of PDGFR maps on chromosome 4 and the gene coding the beta subunit of PDGFR maps on chromosome 5. Coexisting tumors should, therefore, theoretically have abnormalities on chromosome 4 or 5. However, studies by other authors have not been able to demonstrate concordance.[6] Simultaneous tumors of varying histology have also been proposed to develop entirely coincidently or from residual embryonic tissue.[9]
The pathogenesis of GSM has been a topic of controversy and currently remains unknown. Reis et al.[13] discovered identical phosphatase and tensin homolog mutations, p53 nuclear accumulation, p16 deletion, and cyclin-dependent kinase 4 amplifications in GSMs. Cander et al.[14] showed that p16 downregulation is a common event in pituitary adenomas and that its alteration may play a significant role in genesis, growth progression, and biological behavior of pituitary adenomas. The common underlying etiology for the concurrent occurrence of GSM and pituitary adenoma remains undefined.
| Conclusion | | |
Familial syndromes should be ruled out in multiple primary brain tumors with different histology. While no causative factors can be derived from a single case report, potential mechanism of this phenomenon is p16 downregulation associated with both tumors.
Ethical statement
All procedures performed in the study were conducted in accordance with the ethics standards given in the 1964 declaration of Helsinki, as revised in 2013
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4]
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