Bilateral adrenal uptake of 123I MIBG scintigraphy with mild catecholamine elevation, the diagnostic dilemma, and its characteristics

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作者
Yuiko Inaba
Masaaki Yamamoto
Shin Urai
Masaki Suzuki
Seiji Nishikage
Maki Kanzawa
Yayoi Aoyama
Tomonori Kanda
Katsumi Shigemura
Hironori Bando
Genzo Iguchi
Yasuhiro Nakamura
Masato Fujisawa
Akihisa Imagawa
Hidenori Fukuoka
Wataru Ogawa
机构
[1] Kobe University Hospital,Division of Diabetes and Endocrinology, Department of Internal Medicine
[2] Osaka Medical and Pharmaceutical University,Department of Internal Medicine(I)
[3] Kobe University Graduate School of Medicine,Division of Diabetes and Endocrinology
[4] Kobe University Hospital,Department of Diagnostic Pathology
[5] Tohoku University Hospital,Department of Pathology
[6] Kobe University Graduate School of Medicine,Department of Radiology
[7] Kobe University Graduate School of Medicine,Division of Urology, Department of Organ Therapeutics, Faculty of Medicine
[8] Kobe University Graduate School of Health Science,Department of Public Health
[9] Kobe University Graduate School of Medicine,Division of Development of Advanced Therapy for Metabolic Disease
[10] Kobe University,Medical Center for Student Health
[11] Kobe University Graduate School of Medicine,Department of Biosignal Pathophysiology
[12] Tohoku Medical and Pharmaceutical University,Division of Pathology
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Scientific Reports | / 12卷
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摘要
Cases in which bilateral adrenal 123I-Metaiodobenzylguanidine (123I-MIBG) scintigraphy accumulation is sometimes shown, with mildly elevated catecholamine (CA) or metanephrine (MN) levels (within 3 times the upper reference limit) are diagnostic dilemmas. We experienced 3 cases of adrenal incidentalomas with this dilemma in the differential diagnosis. The clinical diagnosis was subclinical Cushing's syndrome in 2 cases, and primary aldosteronism in 1. Despite suspected CA excess in clinical symptoms and imaging findings, the pathological findings of all these tumors were revealed to be cytochrome P450 family 11 subfamily B member 1 (CYP11B1) positive adrenocortical adenomas. Interestingly, adrenal medullary hyperplasia (AMH) was detected in the adrenal parenchyma of all those backgrounds. To clarify the clinical features of such cases, a cross-sectional study was conducted at the Kobe University Hospital from 2014 to 2020. One-hundred sixty-four patients who had undergone 123I-MIBG scintigraphy were recruited. Among them, 10 patients (6.1%) met the above criteria, including the presented 3 cases. Plasma adrenaline, noradrenaline, urinary metanephrine, and normetanephrine had values of 0.05 ± 0.05 ng/mL, 0.63 ± 0.32 ng/mL, 0.22 ± 0.05 mg/day, and 0.35 ± 0.16 mg/day, respectively. Nine cases were complicated with hypertension, and symptoms related to CA excess were observed. Half of them (5 cases) including presented 3 cases had unilateral adrenal tumors. These suggest that in cases of bilateral adrenal uptake on 123I-MIBG, AMH needs to be considered. Adrenocortical adenomas may be associated with AMH and further larger investigation is needed for this pathology.
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