TY - JOUR
T1 - Case report
T2 - schwannoma arising from the unilateral adrenal area with bilateral hyperaldosteronism
AU - Babaya, Naru
AU - Makutani, Yukako
AU - Noso, Shinsuke
AU - Hiromine, Yoshihisa
AU - Ito, Hiroyuki
AU - Taketomo, Yasunori
AU - Ueda, Kazuki
AU - Ushijima, Hokuto
AU - Komoike, Yoshifumi
AU - Yamazaki, Yuto
AU - Sasano, Hironobu
AU - Kawabata, Yumiko
AU - Ikegami, Hiroshi
PY - 2017/12/6
Y1 - 2017/12/6
N2 - BACKGROUND: 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.CASE PRESENTATION: 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.CONCLUSION: 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.
AB - BACKGROUND: 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.CASE PRESENTATION: 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.CONCLUSION: 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.
KW - Aldosterone-producing cell clusters
KW - Hyperaldosteronism
KW - Incidentaloma
KW - Micronodular hyperplasia
KW - Paradoxical hyperplasia
UR - http://www.scopus.com/inward/record.url?scp=85050879095&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85050879095&partnerID=8YFLogxK
U2 - 10.1186/s12902-017-0225-z
DO - 10.1186/s12902-017-0225-z
M3 - Article
C2 - 29212491
AN - SCOPUS:85050879095
SN - 1472-6823
VL - 17
SP - 74
JO - BMC Endocrine Disorders
JF - BMC Endocrine Disorders
IS - 1
M1 - 74
ER -
