Nonfunctioning Pituitary Adenoma That Changed to a Functional Gonadotropinoma

Hindawi Case Reports in Endocrinology Volume 2018, Article ID 5027859, 4 pages https://doi.org/10.1155/2018/5027859 Case Report Nonfunctioning Pituitary Adenoma That Changed to a Functional Gonadotropinoma Gerson Geovany Andino-Ríos ,1 Lesly Portocarrero-Ortiz,1 Carlos Rojas-Guerrero,1 Alejandro Terrones-Lozano ,1 Alma Ortiz-Plata,2 and Alfredo Adolfo Reza-Albarrán 3 1 Neuroendocrinology Department, Instituto Nacional de Neurologı́a y Neurocirugı́a Manuel Velasco Suárez, Ciudad de México, Mexico 2 Experimental Neuropathology Laboratory, Instituto Nacional de Neurologı́a y Neurocirugı́a Manuel Velasco Suárez, Ciudad de México, Mexico 3 Endocrinology and Metabolism Department, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán, Ciudad de México, Mexico Correspondence should be addressed to Gerson Geovany Andino-Rı́os; Received 3 November 2017; Accepted 8 March 2018; Published 29 April 2018 Academic Editor: Takeshi Usui Copyright © 2018 Gerson Geovany Andino-Rı́os et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Objective. Pituitary adenomas can be classified as clinically functional or silent. Depending on the reviewed literature, these are the first or second place in frequency of the total pituitary adenomas. Even rarer is the presence of a functional gonadotropinoma since only very few case reports exist to date. The conversion of a clinically silent to functional pituitary adenoma is extraordinarily rare; the mechanisms that explain these phenomena are unknown or not fully understood. Methods. We report the case of a woman who initially had a nonfunctional gonadotropinoma and in the course of her medical condition showed biochemical changes in her hormonal pituitary profile compatible with a functional gonadotropinoma. Results. We considered that the patient had a functional gonadotropinoma due to the hyperestrogenemia in the context of secondary amenorrhea, resolving the hyperestrogenemia after almost complete resection of the tumor. Conclusion. It is necessary to point out from a clinical and/or biochemical point of view the change in functionality that a nonfunctional pituitary adenoma may have. In the case of our patient, the suspicion of this change in functionality became evident when we found an increase in the FSH/LH ratio and a progressive increase in serum estradiol concentrations when the patient had amenorrhea. 1. Introduction 2. Case Report Nonfunctioning gonadotropinomas are the second most common type of pituitary adenoma. Its clinical diagnosis is based on the presence of symptoms associated with compression by mass effect; the true prevalence of functioning gonadotropinomas is unknown since the vast majority of reports mentioning this pituitary entity are case reports. To date, there are very few cases reported in the medical literature discussing the conversion of a nonfunctional to functional pituitary adenoma. We describe the case of a patient who initially was diagnosed with a nonfunctional gonadotropinoma that, at a later clinical follow-up, diagnosis was changed to a functional gonadotropinoma, where the main diagnostic key was the elevation of estradiol in the context of amenorrhea. A 41-year-old woman came in for consultation in July 2013. Her menarche was at age of 13, no pregnancies occurred, and her menstrual cycles were regular, every 28–30 days. At age of 36 she had noticed oligomenorrhea and subsequently amenorrhea. She also mentioned headache and blurred vision, so she was submitted to magnetic resonance imaging (MRI), which showed a pituitary adenoma of 43 × 40 × 29 mm. Her visual field test showed bilateral temporal hemianopia and she was then sent to a reference center. The first hormonal pituitary profile showed the following results: TSH: 1.9 𝜇IU/mL (0.34–5.60), FT4 : 7.2 pmol/L (8.11– 17.25), TT3 : 1.3 nmol/L (0.98–2.78), LH: 2.6 mIU/mL (2.4– 12.6), FSH: 7.3 mIU/mL (3.5–12.5), estradiol 14.4 pg/mL 2 Case Reports in Endocrinology Figure 1: Coronal MRI T1 sequence with gadolinium. Intra- and suprasellar tumor with extension to the floor of third ventricle, after first surgery. (a) (b) Figure 2: Coronal and sagittal MRI T1 sequence with gadolinium. Intra- and suprasellar tumor with tumor growth related to previous study. (25.0–195.0), prolactin: 20.0 ng/mL (3.3–26.7), ACTH: 15.0 pg/mL (4.7–48.8), cortisol: 12.4 𝜇g/dL (8.7–22.4), and IGF-1: 53.8 ng/mL (56–194). The patient was considered to have hypopituitarism; however, only 50 mcg oral levothyroxine treatment was started every day due to central hypothyroidism. Dynamic test was not performed to rule out secondary adrenal insufficiency due to the risk of causing pituitary apoplexy. The patient underwent transsphenoidal resection in August 2013 (Figure 1). Despite the important tumor remnant, it was not specified whether the macroscopic appearance of the tumor had any special feature that made resection difficult during surgery. Immunostaining was positive for FSH and LH. During clinical follow-up, she presented some improvement in her visual fields but amenorrhea persisted. In 2015 there was deterioration in the visual fields; new hormonal determinations showed FSH of 6.31 mIU/mL and LH of 2.20 mIU/mL, as well as increase in estradiol levels at 135 pg/mL; it was considered to perform a new tumor resection to protect the vision but a short-term surgical date could not be obtained. In March 2016, a new MRI with a focus on the sellar region was performed, finding tumor remnant growth (Figures 2(a) and 2(b)). No visual worsening was reported. A new gonadal profile was requested which showed estradiol of 394.5 pg/mL and dissociation between FSH and LH (6.19 mIU/mL and 1.98 mIU/mL, resp.); amenorrhea persisted. After the annual biochemical monitoring of the gonadal hormones, it was concluded that the gonadotropinoma became functional, so it was decided to surgically intervene by transcranial approach that resulted in a significant reduction of the tumor lesion (Figures 3(a) and 3(b)). The decision to perform a transcranial approach was taken by the neurosurgery team due to their experience with this type of approach and the objective of resecting as much tumor tissue as possible. Ten days after the second surgery, a new gonadal profile measurement was performed: FSH 2.81 mIU/mL, LH 0.57 mIU/mL, and estradiol 20.3 pg/ mL. Immunostaining index (percentage of immune-positive cells) was slightly higher for FSH (38.9%; range 31–48%) than LH (35.1%; range 29–39%). MIB-1 (Ki-67) labeling index was 1.7%. Histologically tumors cells arranged in papillary pattern were found (Figures 4(a) and 4(b)). It is noteworthy highlighting that hormonal therapy with estrogen and/or progestagens was never initiated during the entire management of the patient. Unfortunately, pelvic ultrasound could (...truncated)