Cerebrospinal fluid rhinorrhoea in young women after 6.5 years of therapy giant prolactinoma with Cabergolin

Cerebrospinal fluid (CSF) leak is a rare complication in of the prolactinomas treatment with dopamine agonists. In most cases CSF leak develops within the first three months of treatment starting. The article presents a rare clinical case – later development of CSF leak after pharmacological treatment of giant prolactinoma. Women 29 years with giant endo-supra-infra-laterasellar pituitary adenoma (8,7 cm in diameter), visual impairment, cachexia, secondary amenorrhea and prolactin level more than 2 million 200 thousand mU/l treated with cabergoline. On this background, complete regression of the tumor, recovery of visual function, body mass and normalization of prolactin level was marked. Nevertheless, after 6.5 years after beginning of cabergoline therapy CSF leak occured, which required endoscopic endonasal surgery with the plastic of a CSF fistula. All patients with large and giant prolactinomas which invade into skull base structures, receiving cabergoline even for a long time, should be aware of the possibility of such complications as CSF leak and, if necessary, should urgent appeals to the otolaryngologist and the neurosurgeon. In this case closure of skull base defect is a main tactics of treatment.

1. Corsello SM, Ubertini G, Altomare M, et al. Giant prolactinomas in men: efficacy of cabergoline treatment. Clin Endocrinol (Oxf). 2003;58(5):662-670. doi: 10.1046/j.1365-2265.2003.01770.x.

2. Shrivastava RK, Arginteanu MS, King WA, Post KD. Giant prolactinomas: clinical management and long-term follow up. J Neurosurg. 2002;97(2):299-306. doi: 10.3171/jns.2002.97.2.0299.

3. Астафьева Л.И., Кадашев Б.А., Калинин П.Л., и др. Клиническая картина, диагностика и результаты первичной медикаментозной терапии больших и гигантских пролактинсекретирующих аденом гипофиза. // Вопросы нейрохирургии им. Н.Н. Бурденко. – 2008. – №4. – С. 36–39. [Astaf'eva LI, Kadashev BA, Kalinin PL, et al. Clinical presentation, diagnostics and results of primary conservative treatment of large and giant prolactin-secreting pituitary adenomas. Zh Vopr Neirokhir Im N N Burdenko. 2008;(4):36-39. (In Russ.)]

4. Maiter D, Delgrange E. Therapy of endocrine disease: the challenges in managing giant prolactinomas. Eur J Endocrinol. 2014;170(6):R213-227. doi: 10.1530/EJE-14-0013.

5. Shimon I, Sosa E, Mendoza V, et al. Giant prolactinomas larger than 60 mm in size: a cohort of massive and aggressive prolactin-secreting pituitary adenomas. Pituitary. 2016;19(4):429-436. doi: 10.1007/s11102-016-0723-4.

6. Астафьева Л.И., Кадашев Б.А., Калинин П.Л., и др. Выбор тактики лечения гигантских пролактинсекретирущих аденом гипофиза. // Вопросы нейрохирургии им. Н.Н. Бурденко. – 2009. – №2. – С. 23–28. [Astaf'eva LI, Kadashev BA, Kalinin PL, et al. Selection of management tactics in treatment of giant prolactin-secreting pituitary adenomas. Zh Vopr Neirokhir Im N N Burdenko. 2009;(2):23-28. (In Russ.)]

7. Moraes AB, Silva CM, Vieira Neto L, Gadelha MR. Giant prolactinomas: the therapeutic approach. Clin Endocrinol (Oxf). 2013;79(4):447-456. doi: 10.1111/cen.12242.

8. Suliman SG, Gurlek A, Byrne JV, et al. Nonsurgical cerebrospinal fluid rhinorrhea in invasive macroprolactinoma: incidence, radiological, and clinicopathological features. J Clin Endocrinol Metab. 2007;92(10):3829-3835. doi: 10.1210/jc.2007-0373.

9. Lam G, Mehta V, Zada G. Spontaneous and medically induced cerebrospinal fluid leakage in the setting of pituitary adenomas: review of the literature. Neurosurg Focus. 2012;32(6):E2. doi: 10.3171/2012.4.FOCUS1268.

10. Калинин П.Л., Шкарубо А.Н., Астафьева Л.И., и др. Назальная ликворея при первичном лечении больших и гигантских пролактином агонистами дофамина. // Вопросы нейрохирургии им. Н.Н. Бурденко. – 2017. – Т. 81. – №6. – С. 32–39. [Kalinin PL, Shkarubo AN, Astafieva LI, et al. [Cerebrospinal fluid rhinorrhea in primary treatment of large and giant prolactinomas with dopamine agonists]. Zh Vopr Neirokhir Im N N Burdenko. 2017;81(6):32-39. (In Russ.)] doi: 10.17116/neiro201781632-39.

11. Aslan K, Bekci T, Incesu L, Ozdemir M. Giant invasive basal skull prolactinoma with CSF rhinorrhoea and meningitis. Clin Neurol Neurosurg. 2014;120:145-146. doi: 10.1016/j.clineuro.2014.02.028.

12. Bronstein MD, Musolino NR, Benabou S, Marino R. Cerebrospinal fluid rhinorrhea occurring in long-term bromocriptine treatment for macroprolactinomas. Surg Neurol. 1989;32(5):346-349. doi: 10.1016/0090-3019(89)90137-7.

13. Prague JK, Ward CL, Mustafa OG, et al. Delayed presentation of late-onset cerebrospinal fluid rhinorrhoea following dopamine agonist therapy for giant prolactinoma. Endocrinol Diabetes Metab Case Rep. 2014;2014:140020. doi: 10.1530/EDM-14-0020.

14. Greenman Y, Tordjman K, Stern N. Increased body weight associated with prolactin secreting pituitary adenomas: weight loss with normalization of prolactin levels. Clin Endocrinol (Oxf). 1998;48(5):547-553. doi: 10.1046/j.1365-2265.1998.00403.x.

15. Breyer Freitas B, Rothen RE, Zeni D, et al. Nutritional and metabolic assessment in overweight patients with and without hyperprolactinemia caused by prolactinoma. Nutr Hosp. 2015;32(5):2030-2037. doi: 10.3305/nh.2015.32.5.9673.

16. Deepak D, Daousi C, Javadpour M, MacFarlane IA. Macroprolactinomas and epilepsy. Clin Endocrinol (Oxf). 2007; 66(4):503-507. doi: 10.1111/j.1365-2265.2007.02759.x.