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      Effective long-term temozolomide rechallenge in a macroprolactinoma

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          Summary

          We describe the 20-year course of a 63-year-old male with a macroprolactinoma that acquired resistance to treatment and aggressive behavior after a 4-year successful treatment with cabergoline. He was submitted to multiple surgical resections by a skilled surgeon, fractionated radiotherapy and was eventually treated with temozolomide. After a first 6-month standard cycle, a relapse occurred and he was treated again successfully.

          Learning points:
          • Prolactinomas are the most frequent type of pituitary adenoma.

          • They usually have a benign course.

          • In most cases dopamine-agonist drugs, mainly cabergoline, are first-line (and usually only) treatment.

          • Occasionally prolactinomas can have or acquire resistance to treatment and/or aggressive behavior.

          • Temozolomide (TMZ), an oral alkylating drug, can be effective in such aggressive tumors.

          • Multimodal treatment (surgery, radiation, cabergoline and TMZ) is warranted in aggressive pituitary tumors.

          • We describe here successful rechallenge with TMZ after relapse occurring 18 months after a first TMZ cycle.

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          Most cited references8

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          Advances in the treatment of prolactinomas.

          Prolactinomas account for approximately 40% of all pituitary adenomas and are an important cause of hypogonadism and infertility. The ultimate goal of therapy for prolactinomas is restoration or achievement of eugonadism through the normalization of hyperprolactinemia and control of tumor mass. Medical therapy with dopamine agonists is highly effective in the majority of cases and represents the mainstay of therapy. Recent data indicating successful withdrawal of these agents in a subset of patients challenge the previously held concept that medical therapy is a lifelong requirement. Complicated situations, such as those encountered in resistance to dopamine agonists, pregnancy, and giant or malignant prolactinomas, may require multimodal therapy involving surgery, radiotherapy, or both. Progress in elucidating the mechanisms underlying the pathogenesis of prolactinomas may enable future development of novel molecular therapies for treatment-resistant cases. This review provides a critical analysis of the efficacy and safety of the various modes of therapy available for the treatment of patients with prolactinomas with an emphasis on challenging situations, a discussion of the data regarding withdrawal of medical therapy, and a foreshadowing of novel approaches to therapy that may become available in the future.
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            Guidelines of the Pituitary Society for the diagnosis and management of prolactinomas.

            In June 2005, an ad hoc Expert Committee formed by the Pituitary Society convened during the 9th International Pituitary Congress in San Diego, California. Members of this committee consisted of invited international experts in the field, and included endocrinologists and neurosurgeons with recognized expertise in the management of prolactinomas. Discussions were held that included all interested participants to the Congress and resulted in formulation of these guidelines, which represent the current recommendations on the diagnosis and management of prolactinomas based upon comprehensive analysis and synthesis of all available data.
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              Temozolomide treatment can improve overall survival in aggressive pituitary tumors and pituitary carcinomas.

              Only few retrospective studies have reported an efficacy rate of temozolomide (TMZ) in pituitary tumors (PT), all around 50%. However, the long-term survival of treated patients is rarely evaluated. We therefore aimed to describe the use of TMZ on PT in clinical practice and evaluate the long-term survival.
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                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                EDM
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                01 October 2018
                2018
                : 2018
                : 18-0092
                Affiliations
                [1 ]Endocrinology Unit , Niguarda Hospital, Milan, Italy
                [2 ]Neurooncology Unit , Fondazione IRCCS Neurological Institute Carlo Besta, Milan, Italy
                [3 ]Endocrinology Service , Galeazzi Institute IRCCS, Milan, Italy
                Author notes
                Correspondence should be addressed to B Zampetti; Email: benedettazampetti@ 123456yahoo.it
                Article
                EDM180092
                10.1530/EDM-18-0092
                6169538
                d0426146-9f0e-4293-b61f-999dff1bf6ce
                © 2018 The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivs 3.0 Unported License.

                History
                : 21 August 2018
                : 10 September 2018
                Categories
                Novel Treatment

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