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      Late presentation of acromegaly in medically controlled prolactinoma patients

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          Summary

          Co-secretion of growth hormone (GH) and prolactin (PRL) from a single pituitary adenoma is common. In fact, up to 25% of patients with acromegaly may have PRL co-secretion. The prevalence of acromegaly among patients with a newly diagnosed prolactinoma is unknown. Given the possibility of mixed GH and PRL co-secretion, the current recommendation is to obtain an insulin-like growth factor-1 (IGF-1) in patients with prolactinoma at the initial diagnosis. Long-term follow-up of IGF-1 is not routinely done. Here, we report two cases of well-controlled prolactinoma on dopamine agonists with the development of acromegaly 10–20 years after the initial diagnoses. In both patients, a mixed PRL/GH-cosecreting adenoma was confirmed on the pathology examination after transsphenoidal surgery (TSS). Therefore, periodic routine measurements of IGF-1 should be considered regardless of the duration and biochemical control of prolactinoma.

          Learning points:
          • Acromegaly can develop in patients with well-controlled prolactinoma on dopamine agonists.

          • The interval between prolactinoma and acromegaly diagnoses can be several decades.

          • Periodic screening of patients with prolactinoma for growth hormone excess should be considered and can 
lead to an early diagnosis of acromegaly before the development of complications.

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          Most cited references7

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          Medical progress: Acromegaly.

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            Galactorrhea: a study of 235 cases, including 48 with pituitary tumors.

            An analysis of 235 patients with galactorrhea (5.5 per cent males) showed that 20 per cent of all patients, and 34 per cent of women with associated amenorrhea, had radiologically evident pituitary tumors; these patients had the highest serum prolactin concentrations. The largest single group (32 per cent) consisted of women with idiopathic galactorrhea without amenorrhea; prolactin was normal in 86 per cent of these cases. Five patients had the empty-sella syndrome. Prolactin response was tested in selected patients by thyrotropin-releasing hormone, chlorpromazine, L-dopa, 24-hour sampling and other means. Tests with thyrotropin-releasing hormone were most useful in identifying patients with pituitary tumors. Surgery and radiotherapy lowered prolactin to a similar degree in patients with tumor, but galactorrhea, and amenorrhea often persisted after treatment. The ergot derivatives, bromergocryptine and lergotrile mesylate, lowered prolactin in all 18 patients with idiopathic hyperprolactinemia or pituitary tumor, stopped galactorrhea in over 50 per cent, restored menses in over 70 per cent, and allowed pregnancy in three.
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              Acidophil stem cell adenoma of the human pituitary: clinicopathologic analysis of 15 cases.

              In material of 347 surgically removed pituitary adenomas, 15 tumors (4.3%) were diagnosed as acidophil stem cell adenomas. These are immature neoplasms, assumed to derive from the common progenitor of growth hormone and prolactin cells, and usually containing both hormones by the immunoperoxidase technique. Clinically, they are regularly associated with hyperprolactinemia. Some patients may exhibit physical stigmata of acromegaly without biochemical evidence of the disease ("fugitive acromegaly"). The entity is also characterized by (1) relatively short clinical history; (2) large (grade III--IV), locally invasive adenoma, and (3) relatively low hormonal activity. By electron microscopy, these tumors are unicellular with immature cytoplasm, exhibiting some features of adenomatous growth hormone and prolactin with immature cytoplasm, exhibiting some features of adenomatous growth hormone and prolactin cells and frequently mitochondrial abnormalities as well. They are more aggressive than the well-differentiated adenomas of the "acidophil" cell line--a fact to be considered in postoperative management.
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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
                17 October 2016
                2016
                : 16-0069
                Affiliations
                [1 ]Department of Endocrinology , Diabetes and Metabolism
                [2 ]Department of Neurosurgery
                [3 ]Department of Pathology , University of Rochester, Rochester, New York, USA
                Author notes
                Correspondence should be addressed to Ismat Shafiq; Email: ismat_shafiq@ 123456urmc.rochester.edu
                Article
                EDM160069
                10.1530/EDM-16-0069
                5093383
                ed270f78-8dd3-40b8-8451-42adf88b2c7a
                This is an Open Access article distributed under a Creative Commons Attribution-NonCommercial-NoDerivs 3.0 Unported License.

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

                History
                : 14 September 2016
                : 20 September 2016
                Categories
                Unique/Unexpected Symptoms or Presentations of a Disease

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