Case report: schwannoma arising from the unilateral adrenal area with bilateral hyperaldosteronism.
Case report: schwannoma arising from the unilateral adrenal area with bilateral hyperaldosteronism.
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DOI：
10.1186/s12902-017-0225-z
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发表时间：
2017-12-06
影响因子：
2.7
通讯作者：
Ikegami H
中科院分区：
文献类型：
--
作者：
Babaya N;Makutani Y;Noso S;Hiromine Y;Ito H;Taketomo Y;Ueda K;Ushijima H;Komoike Y;Yamazaki Y;Sasano H;Kawabata Y;Ikegami H
我们报告一例罕见的肾上腺旁神经鞘瘤，手术切除前无法与肾上腺肿瘤区分，并发双侧醛固酮增多症。据我们所知，这是第一个病例中，肾上腺旁神经鞘瘤和醛固酮增多症并存。一名69岁男性，因高血压接受治疗，腹部计算机断层扫描发现左肾上肿块（5.8 × 5.2 cm）。他的实验室数据显示，他的血浆醛固酮浓度（PAC）在正常范围内，但他的血浆肾素活性（PRA）降低，导致醛固酮/肾素比值（ARR）升高。在站立位的负荷试验中，卡托普利或速尿表现出自发的醛固酮分泌和肾素抑制。肾上腺静脉取样（AVS）促肾上腺皮质激素刺激表明双侧醛固酮分泌过多。左肾上肿瘤切除，因为恶性的可能性，并被发现是一个良性神经鞘瘤产生的邻肾上腺区连同肾上腺。在球状体中，解剖的左侧肾上腺形态增生，但CYP 11B 2（醛固酮合成酶）化学染色呈阴性。在肾上腺中检测到多个CYP 11B 2阳性肾上腺皮质微结节，表明微结节增生。尽管术前AVS显示双侧醛固酮增多症，但切除单侧肿瘤后PRA、PAC和ARR值均在各自的参考范围内，表明切除后无法检测到剩余右侧病变激素分泌的轻微增加。肾上腺旁神经鞘瘤的临床和形态学诊断很困难，特别是在醛固酮增多症的情况下，如本病例所示。这些资料提示肾上腺偶发瘤诊断的复杂性和困难性。
We report a rare case of a juxta-adrenal schwannoma that could not be discriminated from an adrenal tumor before surgical resection and was complicated by bilateral hyperaldosteronism. To the best of our knowledge, this is first case in which both a juxta-adrenal schwannoma and hyperaldosteronism co-existed. A 69-year-old male treated for hypertension was found to have a left supra-renal mass (5.8 × 5.2 cm) by abdominal computed tomography. His laboratory data showed that his plasma aldosterone concentration (PAC) was within the normal range, but his plasma renin activity (PRA) was reduced, resulting in an increased aldosterone/renin ratio (ARR). Load tests of captopril or furosemide in the standing position demonstrated autonomous aldosterone secretion and renin suppression. Adrenal venous sampling (AVS) with ACTH stimulation indicated bilateral hypersecretion of aldosterone. A left supra-renal tumor was resected because of the possibility of malignancy and was found to be a benign schwannoma arising from the juxta-adrenal region together with an adrenal gland. The dissected left adrenal gland was morphologically hyperplastic in the zona glomerulosa, but was immunohistochemically negative for CYP11B2 (aldosterone synthase). Multiple CYP11B2-positive adrenocortical micronodules were detected in the adrenal gland, indicating micronodular hyperplasia. Although bilateral aldosteronism was indicated by AVS before the operation, the PRA, PAC and ARR values were within their respective reference ranges after resection of the unilateral tumor, suggesting that the slight increase in hormone secretion from the remaining right-sided lesion could not be detected after resection. A clinical and morphologic diagnosis of juxta-adrenal schwannoma is difficult, particularly in a case of hyperaldosteronism, as shown in this case. These data suggest the complexity and difficulty diagnosing adrenal incidentaloma.
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DOI：
10.1196/annals.1353.004
发表时间：
2006-01-01
期刊：
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影响因子：
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作者：
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