S Mekni, H Khiari, S Bouali, R Laamouri, S Essayeh, I Rojbi, I Ben Nacef
{"title":"Therapeutic challenges of giant invasive cystic macroprolactinoma.","authors":"S Mekni, H Khiari, S Bouali, R Laamouri, S Essayeh, I Rojbi, I Ben Nacef","doi":"10.1530/EDM-25-0091","DOIUrl":null,"url":null,"abstract":"<p><strong>Summary: </strong>Giant cystic prolactinomas are extremely rare, with only four cases previously reported in the literature. Herein, we report the case of a 59-year-old male who presented with an acute decline in visual acuity. An urgent brain MRI revealed a large intra- and suprasellar tumor, consisting of a solid (48 × 31 × 23 mm) and a cystic suprasellar (39.9 × 38.7 × 26.3 mm) component, with cavernous sinus invasion and optic chiasm compression. Hormonal workup revealed hyperprolactinemia (1,020 ng/mL) and panhypopituitarism. The patient was initially treated with bromocriptine. After 3 months, his serum prolactin levels increased despite adequate dosing, and no improvement was observed in his visual deficits. Consequently, he underwent transcranial surgery with subtotal tumor resection. Histopathological examination confirmed PRL-secreting pituitary adenoma. Postoperatively, despite sequential treatment with bromocriptine and cabergoline (maximum approved dose of 3 mg/week), prolactin levels failed to normalize. Furthermore, follow-up MRI revealed a persistent 29 × 21 × 33 mm intra- and suprasellar tumor. This report presents a case of a dopamine agonist (DA)-resistant giant cystic prolactinoma, highlighting the therapeutic challenges associated with this rare tumor subtype.</p><p><strong>Learning points: </strong>Giant cystic prolactinomas are extremely rare. Dopamine agonists remain the first-line therapy for both giant and cystic prolactinomas. Early surgical intervention is indicated for vision-threatening conditions or in cases of dopamine agonist resistance.</p>","PeriodicalId":37467,"journal":{"name":"Endocrinology, Diabetes and Metabolism Case Reports","volume":"2025 4","pages":""},"PeriodicalIF":0.9000,"publicationDate":"2025-11-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12666772/pdf/","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Endocrinology, Diabetes and Metabolism Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1530/EDM-25-0091","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/10/1 0:00:00","PubModel":"Print","JCR":"Q4","JCRName":"ENDOCRINOLOGY & METABOLISM","Score":null,"Total":0}
引用次数: 0
Abstract
Summary: Giant cystic prolactinomas are extremely rare, with only four cases previously reported in the literature. Herein, we report the case of a 59-year-old male who presented with an acute decline in visual acuity. An urgent brain MRI revealed a large intra- and suprasellar tumor, consisting of a solid (48 × 31 × 23 mm) and a cystic suprasellar (39.9 × 38.7 × 26.3 mm) component, with cavernous sinus invasion and optic chiasm compression. Hormonal workup revealed hyperprolactinemia (1,020 ng/mL) and panhypopituitarism. The patient was initially treated with bromocriptine. After 3 months, his serum prolactin levels increased despite adequate dosing, and no improvement was observed in his visual deficits. Consequently, he underwent transcranial surgery with subtotal tumor resection. Histopathological examination confirmed PRL-secreting pituitary adenoma. Postoperatively, despite sequential treatment with bromocriptine and cabergoline (maximum approved dose of 3 mg/week), prolactin levels failed to normalize. Furthermore, follow-up MRI revealed a persistent 29 × 21 × 33 mm intra- and suprasellar tumor. This report presents a case of a dopamine agonist (DA)-resistant giant cystic prolactinoma, highlighting the therapeutic challenges associated with this rare tumor subtype.
Learning points: Giant cystic prolactinomas are extremely rare. Dopamine agonists remain the first-line therapy for both giant and cystic prolactinomas. Early surgical intervention is indicated for vision-threatening conditions or in cases of dopamine agonist resistance.
期刊介绍:
Endocrinology, Diabetes & Metabolism Case Reports publishes case reports on common and rare conditions in all areas of clinical endocrinology, diabetes and metabolism. Articles should include clear learning points which readers can use to inform medical education or clinical practice. The types of cases of interest to Endocrinology, Diabetes & Metabolism Case Reports include: -Insight into disease pathogenesis or mechanism of therapy - Novel diagnostic procedure - Novel treatment - Unique/unexpected symptoms or presentations of a disease - New disease or syndrome: presentations/diagnosis/management - Unusual effects of medical treatment - Error in diagnosis/pitfalls and caveats