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A 57-year-old woman was admitted to the hospital for the further evaluation of a left adrenal incidentaloma measuring 45 mm x 33 mm. She had no signs of the clinical manifestation of hypercortisolism.... An endocrine evaluation revealed that her ACTH level was normal and cortisol values were almost normal pattern excluding the value at 9 PM slightly rising, however, the cortisol was not completely suppressed by the overnight administration of 1 mg dexamethasone. These findings indicated that subtle abnormalities of the hypothalamo-pituitary-adrenal axis were present in this case. After 3 months, surprisingly, the ACTH was suppressed to low levels. Further hormonal investigations revealed that the cortisol level was normal but had an abnormal diurnal rhythm and was not suppressed completely by a 1 mg or an 8 mg overnight dexamethasone dose. Adrenal scintigraphy revealed positive uptake in the left adrenal tumor with no uptake in the right adrenal gland. The patient underwent a left laparoscopic adrenalectomy. Microscopically, the tumor displayed histopathological features in common with ACTH-independent macronodular adrenocortical hyperplasia , including clear cell predominance, a pattern of small compact nests in clear cell areas, and a cord-like arrangement of small compact cells. An in situ hybridization study demonstrated the hybridization signals for P-450scc, 3β-HSD, P-450c21, P-45011β, and P-45017αwhich were observed in the clear cells as well as compact cells, the compact cells being more intensely stained. This case indicates the ability of autonomous cortisol production to become clear during a very short term and a more detailed and careful short-time follow-up should be recommended in patients with adrenal incidentalomas.
症例は57 歳，女性．45 ｍｍｘ 33 ｍｍの左副腎偶発腫瘍の精査目的で当科に入院となった．高コルチゾール血症による身体所見は認めず，内分泌学的検査では，コルチゾールの夜間９時の値が軽度上昇している以外は，ACTH，コルチゾール値ともほぼ正常で日内リズムも保たれていた．しかしデキサメサゾン1mg 抑制試験ではコルチゾールは完全には抑制されなかった．よってわずかな間脳下垂体副腎系の異常が示唆され，腫瘍径も4cm を超えており摘出も検討したが，患者さんは保存的経過観察を希望され手術は施行しなかった．ところが３ケ月後の血液検査でACTH 値は感度以下の低値で，コルチゾール基礎値は正常であるが，デキサメサゾン1mgまたは8mg 抑制試験でコルチゾールは完全に抑制されなかった．アドステロール副腎シンチグラフィー検査では左副腎腫瘍に高集積を認め，右副腎には集積を認めなかった．そこで今回患者さんの同意を得て腹腔鏡下で左副腎摘出術を施行した．摘出副腎腫瘍は光顕像ではACTH-independent macronodularadrenocortical hyperplasia (AIMAH)の組織像であり，in situ hybridization studyではcompact cellにより強いP-450scc, 3β-HSD，P-450c21，P-45011βそしてP-45017αのシグナルが認めた．本症例は短期間のうちにコルチゾールの自律的分泌能が明確となった副腎偶発腫瘍の一例であり，より注意深い，詳細な，短期間のうちの内分泌学的経過観察が副腎偶発腫瘍に対して必要であることが示唆された．続きを見る
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Mendeley出力
fam99-7_p150