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Neurological Surgery No Shinkei Geka Volume 48, Issue 3 （March 2020）

Neurological Surgery 脳神経外科 48巻3号（2020年3月発行）

Japanese English

症例

腫瘍の局在診断に苦慮した症候性ACTHおよびGH産生性重複下垂体腺腫の1例 Coexisting Symptomatic Adrenocorticotropic Hormone- and Growth Hormone-producing Double Pituitary Adenomas with Difficulty in Tumor Localization:A Case Report 伊藤裕平 ¹ , 神宮字伸哉 ¹ , 佐藤祐介 ¹ , 佐藤拓 ¹ , 村上友太 ¹ , 藤井正純 ¹ , 佐久間潤 ¹ , 岩崎麻里子 ² , 工藤明宏 ² , 島袋充生 ² , 喜古雄一郎 ³ , 岡田正康 ⁴ , 齋藤清 ¹ Yuhei ITO ¹ , Shinya JINGUJI ¹ , Yusuke SATO ¹ , Taku SATO ¹ , Yuta MURAKAMI ¹ , Masazumi FUJII ¹ , Jun SAKUMA ¹ , Mariko IWASAKI ² , Akihiro KUDO ² , Michio SHIMABUKURO ² , Yuichiro KIKO ³ , Masayasu OKADA ⁴ , Kiyoshi SAITO ¹ ¹福島県立医科大学脳神経外科学講座 ²福島県立医科大学糖尿病内分泌代謝内科学講座 ³福島県立医科大学病理病態診断学講座 ⁴新潟大学脳研究所脳神経外科 ¹Department of Neurosurgery, Fukushima Medical University ²Department of Diabetes, Endocrinology and Metabolism School of Medicine, Fukushima Medical University ³Department of Diagnostic Pathology, Fukushima Medical University ⁴Department of Neurosurgery, Brain Research Institute, Niigata University キーワード： pituitary adenoma , endonasal endoscopic surgery , Cushing's disease , acromegaly Keyword: pituitary adenoma , endonasal endoscopic surgery , Cushing's disease , acromegaly pp.253-260

発行日 2020年3月10日 Published Date 2020/3/10

DOI https://doi.org/10.11477/mf.1436204171

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I．はじめに

　下垂体内に免疫組織化学的に分離することのできる2つ以上の下垂体腺腫が重複する病態は稀ではあるものの，剖検例や手術症例の検討で一定の頻度が存在することが報告されている^3,9,13,14)．産生するホルモンの組み合わせにより頻度が異なり，特にadrenocorticotropic hormone（ACTH)-growth hormone（GH）産生性の重複下垂体腺腫の頻度は低い^2,5)．中でも，先端巨大症とCushing病の臨床症状を合併した症例の報告は数例にとどまる^1,18)．今回われわれは，臨床的に先端巨大症とCushing病を合併したACTHおよびGH産生性重複下垂体腺腫の1例を経験したため，ここに報告する．

　Double functional pituitary adenomas are rare, and only a few cases of excessive clinical symptoms of both adrenocorticotropic hormone（ACTH）and growth hormone（GH）have been reported. We herein report a case of symptomatic ACTH-and GH-producing double pituitary adenomas, which were discretely located within the same pituitary gland.

　A 38-year-old woman presented with general malaise, facial and lower limb edema, unexplained weight gain, facial redness, acne, and nasal enlargement. Endocrinological findings matched with the diagnostic criteria for both acromegaly and Cushing's disease. Preoperative magnetic resonance imaging showed a 15-mm cyst-like lesion on the right side of the sellae surrounded by what was thought to be the normal contrast-enhancing pituitary gland. We assumed that the cyst-like lesion was an adenoma and performed endoscopic endonasal transsphenoidal surgery. However, the cyst-like lesion was a parenchymal tumor. Furthermore, the region we considered to be a normal pituitary gland was also found to be an adenoma. Both adenomas were completely resected. The postoperative blood analysis showed ACTH＜1.0pg/dL, cortisol 1.8μg/dL, and insulin-like growth factor-1 60ng/mL, all of which were below reference levels. The histopathological examination confirmed the coexistence of two adenomas, a GH-producing adenoma and an ACTH-producing adenoma. We concluded that these adenomas were endocrinologically active within the pituitary gland. Thus, a diagnosis of double pituitary adenomas was made.

　When treating a patient with symptoms caused by hypersecretion of multiple anterior pituitary hormones, the possibility of coexisting multiple pituitary adenomas should be considered.