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Temozolomide-Induced Shrinkage of Invasive Pituitary Adenoma in Patient with Nelson's Syndrome: A Case Report and Review of the Literature.

Kurowska M, Nowakowski A, Zieliński G, Malicka J, Tarach JS, Maksymowicz M, Denew P - Case Rep Endocrinol (2015)

Bottom Line: Due to a rapid regrowth of the tumour, the patient did not receive gamma-knife therapy and was treated with cabergoline and somatostatin analogue for some time.Only TMZ therapy resulted in marked clinical, biochemical, and radiological improvement.Conclusion.

View Article: PubMed Central - PubMed

Affiliation: Department of Endocrinology, Medical University of Lublin, Lublin, Poland.

ABSTRACT
Introduction. Invasive tumours in Nelson's syndrome need aggressive therapy. Recent reports have documented the efficacy of temozolomide (TMZ) in the treatment of adenomas resistant to conventional management. Objective. The review of the literature concerning TMZ treatment of atypical corticotroph adenomas and a case study of 56-year-old woman who developed Nelson's syndrome. Treatment Proceeding. The patient with Cushing's disease underwent transsphenoidal adenomectomy followed by a 27-month-long period of remission. Due to a regrowth of the tumor, she underwent two reoperations followed by stereotactic radiotherapy. Because of treatment failures, bilateral adrenalectomy was performed. Then she developed Nelson's syndrome. A fourth transsphenoidal adenomectomy was performed, but there was a rapid recurrence. Five months later, she underwent a right frontotemporal craniotomy. Due to a rapid regrowth of the tumour, the patient did not receive gamma-knife therapy and was treated with cabergoline and somatostatin analogue for some time. Only TMZ therapy resulted in marked clinical, biochemical, and radiological improvement. To date, this is the first case of invasive corticotroph adenoma in Nelson's syndrome treated with temozolomide in Poland. Conclusion. In our opinion, temozolomide can be an effective treatment option of invasive adenomas in Nelson's syndrome.

No MeSH data available.


Related in: MedlinePlus

Ultrastructural features of sparsely granulated corticotroph adenoma (original magnification, 9700x). The cell has well developed Golgi complexes, dilated endoplasmic reticulum, and sparse, variable in shape and electron density secretory granules that measure 200 to 250 nm in diameter. Perinuclear bundles of cytokeratin filaments are absent.
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fig2: Ultrastructural features of sparsely granulated corticotroph adenoma (original magnification, 9700x). The cell has well developed Golgi complexes, dilated endoplasmic reticulum, and sparse, variable in shape and electron density secretory granules that measure 200 to 250 nm in diameter. Perinuclear bundles of cytokeratin filaments are absent.

Mentions: A consecutive pathologic electron microscopy showed an atypical sparsely granulated corticotroph tumour, with an MIB-1 index >5% (Figures 1 and 2), very low activity of O6-methylguanine DNA methyltransferase (MGMT) in tumour cells (Figure 3), and positive receptor SSRT 2A (weak cytoplasmic) reaction. In order to help bridge the time gap until gamma-knife qualification, another desperate attempt of medical treatment was undertaken. Considering the fact that current literature proves the expression of somatostatin and dopamine receptors in corticotroph adenoma tissues [2, 18, 19] and taking into account the result of the somatostatin receptor scintigraphy and postoperative tumour receptor SSRT 2A examination, the patient was administered 2 injections of long-acting release somatostatin analog (lanreotide 120 mg) at a 4-week interval and cabergoline (2 mg/week) for 8 weeks. No positive effect was observed.


Temozolomide-Induced Shrinkage of Invasive Pituitary Adenoma in Patient with Nelson's Syndrome: A Case Report and Review of the Literature.

Kurowska M, Nowakowski A, Zieliński G, Malicka J, Tarach JS, Maksymowicz M, Denew P - Case Rep Endocrinol (2015)

Ultrastructural features of sparsely granulated corticotroph adenoma (original magnification, 9700x). The cell has well developed Golgi complexes, dilated endoplasmic reticulum, and sparse, variable in shape and electron density secretory granules that measure 200 to 250 nm in diameter. Perinuclear bundles of cytokeratin filaments are absent.
© Copyright Policy - open-access
Related In: Results  -  Collection

Show All Figures
getmorefigures.php?uid=PMC4499620&req=5

fig2: Ultrastructural features of sparsely granulated corticotroph adenoma (original magnification, 9700x). The cell has well developed Golgi complexes, dilated endoplasmic reticulum, and sparse, variable in shape and electron density secretory granules that measure 200 to 250 nm in diameter. Perinuclear bundles of cytokeratin filaments are absent.
Mentions: A consecutive pathologic electron microscopy showed an atypical sparsely granulated corticotroph tumour, with an MIB-1 index >5% (Figures 1 and 2), very low activity of O6-methylguanine DNA methyltransferase (MGMT) in tumour cells (Figure 3), and positive receptor SSRT 2A (weak cytoplasmic) reaction. In order to help bridge the time gap until gamma-knife qualification, another desperate attempt of medical treatment was undertaken. Considering the fact that current literature proves the expression of somatostatin and dopamine receptors in corticotroph adenoma tissues [2, 18, 19] and taking into account the result of the somatostatin receptor scintigraphy and postoperative tumour receptor SSRT 2A examination, the patient was administered 2 injections of long-acting release somatostatin analog (lanreotide 120 mg) at a 4-week interval and cabergoline (2 mg/week) for 8 weeks. No positive effect was observed.

Bottom Line: Due to a rapid regrowth of the tumour, the patient did not receive gamma-knife therapy and was treated with cabergoline and somatostatin analogue for some time.Only TMZ therapy resulted in marked clinical, biochemical, and radiological improvement.Conclusion.

View Article: PubMed Central - PubMed

Affiliation: Department of Endocrinology, Medical University of Lublin, Lublin, Poland.

ABSTRACT
Introduction. Invasive tumours in Nelson's syndrome need aggressive therapy. Recent reports have documented the efficacy of temozolomide (TMZ) in the treatment of adenomas resistant to conventional management. Objective. The review of the literature concerning TMZ treatment of atypical corticotroph adenomas and a case study of 56-year-old woman who developed Nelson's syndrome. Treatment Proceeding. The patient with Cushing's disease underwent transsphenoidal adenomectomy followed by a 27-month-long period of remission. Due to a regrowth of the tumor, she underwent two reoperations followed by stereotactic radiotherapy. Because of treatment failures, bilateral adrenalectomy was performed. Then she developed Nelson's syndrome. A fourth transsphenoidal adenomectomy was performed, but there was a rapid recurrence. Five months later, she underwent a right frontotemporal craniotomy. Due to a rapid regrowth of the tumour, the patient did not receive gamma-knife therapy and was treated with cabergoline and somatostatin analogue for some time. Only TMZ therapy resulted in marked clinical, biochemical, and radiological improvement. To date, this is the first case of invasive corticotroph adenoma in Nelson's syndrome treated with temozolomide in Poland. Conclusion. In our opinion, temozolomide can be an effective treatment option of invasive adenomas in Nelson's syndrome.

No MeSH data available.


Related in: MedlinePlus