open access

Vol 69, No 3 (2018)
Case report
Published online: 2018-01-09
Submitted: 2017-09-22
Accepted: 2017-12-11
Get Citation

Temozolomide therapy for aggressive pituitary Crooke’s cell corticotropinoma causing Cushing’s Disease — a case report with literature review

Aleksandra Gilis-Januszewska, Małgorzata Wilusz, Jacek Pantofliński, Renata Turek-Jabrocka, Grzegorz Sokołowski, Anna Sowa-Staszczak, Łukasz Kluczyński, Dorota Pach, Grzegorz Zieliński, Alicja Hubalewska-Dydejczyk
DOI: 10.5603/EP.a2018.0011
·
Pubmed: 29319131
·
Endokrynologia Polska 2018;69(3):306-312.

open access

Vol 69, No 3 (2018)
Case report
Published online: 2018-01-09
Submitted: 2017-09-22
Accepted: 2017-12-11

Abstract

Abstract Context: Aggressive pituitary tumours causing Cushing’s Disease are very rare, difficult to treat, and usually resistant to conventional therapy. There is growing evidence for the use of temozolomide (TZM), an alkylating chemotherapeutic agent, as first line chemotherapy in tumours resistant to repeated neurosurgery, radiotherapy and adrenalectomy. Objective: To present the response to TMZ in a rare case of an aggressive pituitary tumour in the course of Cushing’s Disease and to review the literature referring to similar cases. Patient: In this report, we present the case of a 61 year old male patient who was diagnosed with Cushing’s Disease in the course of a pituitary invasive macroadenoma in 2011. The patient underwent 4 transphenoidal non-radical neurosurgeries (2012,2013) with rapid tumour progression, repeated non-radical bilateral adrenalectomy (2012, 2013) and stereotactic radiotherapy, and gamma knife surgery (2013, 2015). Histopathological examination revealed macroadenoma with high cell polymorphism and the presence of Crooke's cells, Ki- < 2%. Since 2015 the patient has been treated with 6 cycles of TMZ (320 mg per day for 5 consecutive days, 28-day cycle) with clinical and biochemical improvement and stabilized tumour size and no side effects. TMZ was continued for up to 9 cycles with a stable serum level of cortisol and ACTH being observed. However, clinical symptoms like headaches, visual field impairment, and finally hearing loss started to progress from the eighth cycle. After the ninth cycle of TMZ, there was a sudden increase in the size of the tumour, impairment of the cortisol and ACTH level, marked deterioration of the clinical status with the recurrence of severe headaches, narrowing of the visual field and hearing loss. At the beginning of 2016, a sudden clinical status and sight deterioration, strong headaches, drop of the right eyelid with widening of the pupil were observed. The patient died in February 2016. Lessons: The case of our patient suggests that the response to the TMZ treatment monotherapy in aggressive pituitary tumour causing Cushing’s Disease could be partial and restricted to 7-8 cycles followed by rapid progression of the tumor mass. Therefore, further research should be carried out with regard to new methods to extend the responsiveness and duration of TMZ treatment and to investigate predictors of responsiveness.

< p > < /p >

Abstract

Abstract Context: Aggressive pituitary tumours causing Cushing’s Disease are very rare, difficult to treat, and usually resistant to conventional therapy. There is growing evidence for the use of temozolomide (TZM), an alkylating chemotherapeutic agent, as first line chemotherapy in tumours resistant to repeated neurosurgery, radiotherapy and adrenalectomy. Objective: To present the response to TMZ in a rare case of an aggressive pituitary tumour in the course of Cushing’s Disease and to review the literature referring to similar cases. Patient: In this report, we present the case of a 61 year old male patient who was diagnosed with Cushing’s Disease in the course of a pituitary invasive macroadenoma in 2011. The patient underwent 4 transphenoidal non-radical neurosurgeries (2012,2013) with rapid tumour progression, repeated non-radical bilateral adrenalectomy (2012, 2013) and stereotactic radiotherapy, and gamma knife surgery (2013, 2015). Histopathological examination revealed macroadenoma with high cell polymorphism and the presence of Crooke's cells, Ki- < 2%. Since 2015 the patient has been treated with 6 cycles of TMZ (320 mg per day for 5 consecutive days, 28-day cycle) with clinical and biochemical improvement and stabilized tumour size and no side effects. TMZ was continued for up to 9 cycles with a stable serum level of cortisol and ACTH being observed. However, clinical symptoms like headaches, visual field impairment, and finally hearing loss started to progress from the eighth cycle. After the ninth cycle of TMZ, there was a sudden increase in the size of the tumour, impairment of the cortisol and ACTH level, marked deterioration of the clinical status with the recurrence of severe headaches, narrowing of the visual field and hearing loss. At the beginning of 2016, a sudden clinical status and sight deterioration, strong headaches, drop of the right eyelid with widening of the pupil were observed. The patient died in February 2016. Lessons: The case of our patient suggests that the response to the TMZ treatment monotherapy in aggressive pituitary tumour causing Cushing’s Disease could be partial and restricted to 7-8 cycles followed by rapid progression of the tumor mass. Therefore, further research should be carried out with regard to new methods to extend the responsiveness and duration of TMZ treatment and to investigate predictors of responsiveness.

< p > < /p >
Get Citation

Keywords

aggressive pituitary tumours, Cushing’s Disease, treatment, temozolomide

About this article
Title

Temozolomide therapy for aggressive pituitary Crooke’s cell corticotropinoma causing Cushing’s Disease — a case report with literature review

Journal

Endokrynologia Polska

Issue

Vol 69, No 3 (2018)

Pages

306-312

Published online

2018-01-09

DOI

10.5603/EP.a2018.0011

Pubmed

29319131

Bibliographic record

Endokrynologia Polska 2018;69(3):306-312.

Keywords

aggressive pituitary tumours
Cushing’s Disease
treatment
temozolomide

Authors

Aleksandra Gilis-Januszewska
Małgorzata Wilusz
Jacek Pantofliński
Renata Turek-Jabrocka
Grzegorz Sokołowski
Anna Sowa-Staszczak
Łukasz Kluczyński
Dorota Pach
Grzegorz Zieliński
Alicja Hubalewska-Dydejczyk

References (36)
  1. Daly AF, Rixhon M, Adam C, et al. High prevalence of pituitary adenomas: a cross-sectional study in the province of Liege, Belgium. J Clin Endocrinol Metab. 2006; 91(12): 4769–4775.
  2. Fernandez A, Karavitaki N, Wass JAH. Prevalence of pituitary adenomas: a community-based, cross-sectional study in Banbury (Oxfordshire, UK). Clin Endocrinol (Oxf). 2010; 72(3): 377–382.
  3. Raappana A, Koivukangas J, Ebeling T, et al. Incidence of pituitary adenomas in Northern Finland in 1992-2007. J Clin Endocrinol Metab. 2010; 95(9): 4268–4275.
  4. Halevy C, Whitelaw BC. How effective is temozolomide for treating pituitary tumours and when should it be used? Pituitary. 2017; 20(2): 261–266.
  5. Fadul CE, Kominsky AL, Meyer LP, et al. Long-term response of pituitary carcinoma to temozolomide. Report of two cases. J Neurosurg. 2006; 105(4): 621–626.
  6. Hagen C, Schroeder HD, Hansen S, et al. Temozolomide treatment of a pituitary carcinoma and two pituitary macroadenomas resistant to conventional therapy. Eur J Endocrinol. 2009; 161(4): 631–637.
  7. Raverot G, Sturm N, de Fraipont F, et al. Temozolomide treatment in aggressive pituitary tumors and pituitary carcinomas: a French multicenter experience. J Clin Endocrinol Metab. 2010; 95(10): 4592–4599.
  8. Syro LV, Ortiz LD, Scheithauer BW, et al. Treatment of pituitary neoplasms with temozolomide: a review. Cancer. 2011; 117(3): 454–462.
  9. Di Ieva A, Davidson JM, Syro LV, et al. Crooke's cell tumors of the pituitary. Neurosurgery. 2015; 76(5): 616–622.
  10. Burman P, van Beek AP, Biller BMK, et al. Radiotherapy, Especially at Young Age, Increases the Risk for De Novo Brain Tumors in Patients Treated for Pituitary/Sellar Lesions. J Clin Endocrinol Metab. 2017; 102(3): 1051–1058.
  11. Tampourlou M, Ntali G, Ahmed S, et al. Outcome of Nonfunctioning Pituitary Adenomas That Regrow After Primary Treatment: A Study From Two Large UK Centers. J Clin Endocrinol Metab. 2017; 102(6): 1889–1897.
  12. Ding D, Starke RM, Sheehan JP. Treatment paradigms for pituitary adenomas: defining the roles of radiosurgery and radiation therapy. J Neurooncol. 2014; 117(3): 445–457.
  13. Fadul CE, Kominsky AL, Meyer LP, et al. Long-term response of pituitary carcinoma to temozolomide. Report of two cases. J Neurosurg. 2006; 105(4): 621–626.
  14. Lim S, Shahinian H, Maya MM, et al. Temozolomide: a novel treatment for pituitary carcinoma. Lancet Oncol. 2006; 7(6): 518–520.
  15. Zacharia BE, Gulati AP, Bruce JN, et al. High response rates and prolonged survival in patients with corticotroph pituitary tumors and refractory Cushing disease from capecitabine and temozolomide (CAPTEM): a case series. Neurosurgery. 2014; 74(4): E447–55; discussion E455.
  16. Bruno OD, Juárez-Allen L, Christiansen SB, et al. Temozolomide Therapy for Aggressive Pituitary Tumors: Results in a Small Series of Patients from Argentina. Int J Endocrinol. 2015; 2015: 587893.
  17. Hirohata T, Asano K, Ogawa Y, et al. DNA mismatch repair protein (MSH6) correlated with the responses of atypical pituitary adenomas and pituitary carcinomas to temozolomide: the national cooperative study by the Japan Society for Hypothalamic and Pituitary Tumors. J Clin Endocrinol Metab. 2013; 98(3): 1130–1136.
  18. Kurowska M, Tarach JS, Malicka J, et al. Long-term complete remission of Crooke's corticotropinoma after temozolomide treatment. Endokrynol Pol. 2016; 67(5): 526–533.
  19. Kurowska M, Nowakowski A, Zieliński G, et al. Temozolomide-Induced Shrinkage of Invasive Pituitary Adenoma in Patient with Nelson's Syndrome: A Case Report and Review of the Literature. Case Rep Endocrinol. 2015; 2015: 623092.
  20. Moyes VJ, Alusi G, Sabin HI, et al. Treatment of Nelson's syndrome with temozolomide. Eur J Endocrinol. 2009; 160(1): 115–119.
  21. Curtò L, Torre ML, Ferraù F, et al. Temozolomide-induced shrinkage of a pituitary carcinoma causing Cushing's disease--report of a case and literature review. ScientificWorldJournal. 2010; 10: 2132–2138.
  22. Takeshita A, Inoshita N, Taguchi M, et al. High incidence of low O(6)-methylguanine DNA methyltransferase expression in invasive macroadenomas of Cushing's disease. Eur J Endocrinol. 2009; 161(4): 553–559.
  23. Lau Q, Scheithauer B, Kovacs K, et al. MGMT immunoexpression in aggressive pituitary adenoma and carcinoma. Pituitary. 2010; 13(4): 367–379.
  24. Hirohata T, Asano K, Ogawa Y, et al. DNA mismatch repair protein (MSH6) correlated with the responses of atypical pituitary adenomas and pituitary carcinomas to temozolomide: the national cooperative study by the Japan Society for Hypothalamic and Pituitary Tumors. J Clin Endocrinol Metab. 2013; 98(3): 1130–1136.
  25. Bengtsson D, Schrøder HD, Andersen M, et al. Long-term outcome and MGMT as a predictive marker in 24 patients with atypical pituitary adenomas and pituitary carcinomas given treatment with temozolomide. J Clin Endocrinol Metab. 2015; 100(4): 1689–1698.
  26. McCormack AI, Wass JAH, Grossman AB. Aggressive pituitary tumours: the role of temozolomide and the assessment of MGMT status. Eur J Clin Invest. 2011; 41(10): 1133–1148.
  27. Danson SJ, Middleton MR. Temozolomide: a novel oral alkylating agent. Expert Rev Anticancer Ther. 2001; 1(1): 13–19.
  28. Bush ZM, Longtine JA, Cunningham T, et al. Temozolomide treatment for aggressive pituitary tumors: correlation of clinical outcome with O(6)-methylguanine methyltransferase (MGMT) promoter methylation and expression. J Clin Endocrinol Metab. 2010; 95(11): E280–E290.
  29. Losa M, Bogazzi F, Cannavo S, et al. Temozolomide therapy in patients with aggressive pituitary adenomas or carcinomas. J Neurooncol. 2016; 126(3): 519–525.
  30. Trouillas J, Roy P, Sturm N, et al. members of HYPOPRONOS. A new prognostic clinicopathological classification of pituitary adenomas: a multicentric case-control study of 410 patients with 8 years post-operative follow-up. Acta Neuropathol. 2013; 126(1): 123–135.
  31. Righi A, Agati P, Sisto A, et al. A classification tree approach for pituitary adenomas. Hum Pathol. 2012; 43(10): 1627–1637.
  32. Katznelson L. Sustained improvements in plasma ACTH and clinical status in a patient with Nelson's syndrome treated with pasireotide LAR, a multireceptor somatostatin analog. J Clin Endocrinol Metab. 2013; 98(5): 1803–1807.
  33. Raverot G, Sturm N, de Fraipont F, et al. Temozolomide treatment in aggressive pituitary tumors and pituitary carcinomas: a French multicenter experience. J Clin Endocrinol Metab. 2010; 95(10): 4592–4599.
  34. Losa M, Bogazzi F, Cannavo S, et al. Temozolomide therapy in patients with aggressive pituitary adenomas or carcinomas. J Neurooncol. 2016; 126(3): 519–525.
  35. Kuyumcu S, Özkan ZG, Sanli Y, et al. Physiological and tumoral uptake of (68)Ga-DOTATATE: standardized uptake values and challenges in interpretation. Ann Nucl Med. 2013; 27(6): 538–545.
  36. Xiao J, Zhu Z, Zhong D, et al. Improvement in diagnosis of metastatic pituitary carcinoma by 68Ga DOTATATE PET/CT. Clin Nucl Med. 2015; 40(2): e129–e131.

Important: This website uses cookies. More >>

The cookies allow us to identify your computer and find out details about your last visit. They remembering whether you've visited the site before, so that you remain logged in - or to help us work out how many new website visitors we get each month. Most internet browsers accept cookies automatically, but you can change the settings of your browser to erase cookies or prevent automatic acceptance if you prefer.

Via MedicaWydawcą serwisu jest  "Via Medica sp. z o.o." sp.k., ul. Świętokrzyska 73, 80–180 Gdańsk

tel.:+48 58 320 94 94, faks:+48 58 320 94 60, e-mail:  viamedica@viamedica.pl