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PubMed1991

[Adrenocortical scintigraphy to evaluate incidental adrenal mass (incidentaloma) discovered on computed tomography].

To assess the nature of adrenal mass incidentally discovered on computed tomography, adrenocortical scintigraphy with I-131-6 beta-iodomethyl-19-norcholest-5(10)-en-3 beta-ol (I-131 Adosterol) in 29 patients was retrospectively reviewed. All patients had no clinical symptoms and biochemical evidence of adrenal hyperfunction. Six of them had extra-adrenal malignancy. Increased tracer uptake on the side of adrenal mass was observed in 13 patients (concordant group): cortical adenoma in 8, macronodular hyperplasia in 1, no growing tumor in 1, and no further examination in the remaining 3 patients. No cancer patients were included in the concordant group. Symmetric uptake was seen in 9 patients (symmetric group): 1 cortical adenoma and 1 ganglioneuroma, 6 no growing tumor (two of them had malignancy), and no further examination in 1 patient. Decreased uptake were shown in 7 patients (dis-concordant group): metastasis in 4, adrenal cyst in 2, and no growing tumor in 1. Adrenocortical scintigraphy was helpful in distinguishing benign functioning adrenal masses from non-functioning adrenal masses such as metastatic tumor and cyst.

Adenoma↗
PubMed1991

[Multiple calcified adrenal myelolipoma suggestive of association of primary aldosteronism: report of a case].

We report a case of adrenal myelolipoma with multiple calcification and hypertension. A 69-year-old woman visited our hospital with a complaint of right flank pain. Computed tomography demonstrated a right adrenal tumor which was a spherical mass with fat density and multiple calcification. Adrenal scintigraphy of I-131 adosterol demonstrated predominant accumulation of the right adrenal gland. Selective venous sampling disclosed a high aldosterone level (303.7 ng/dl) from the affected side. Right adrenalectomy was performed. Pathological diagnosis revealed adrenal myelolipoma with calcification. After the operation the patient became normotensive. The characteristics of the 12 cases of myelolipoma with calcification we found in the Japanese literature are also briefly reviewed.

Adosterol↗
PubMed1999

Successful autotransplantation of an adrenal gland using a new method of omental wrapping: report of a case.

Bilateral total adrenalectomy results in the need for patients to take lifelong supplements of adrenal steroids, with the risk of possible Addisonian crisis. Few reports of the successful autotransplantation of adrenal tissue in muscular pockets have been documented; however, we describe herein the case of a 22-year-old woman in whom autotransplantation of an adrenal gland was successfully performed employing a new method of omental wrapping. The patient underwent bilateral total adrenalectomy for bilateral pheochromocytoma at which time adrenal tissue was sliced into 1-2-mm thick pieces, half of which were placed in muscular pockets in the abdominal rectus muscles, and the remaining half put onto the omentum and wrapped with it. Laboratory examinations done 6 months after surgery showed recovery of her adrenal function, and 4 months later steroid supplements were able to be discontinued. Scintigraphic studies using [131I]-iodomethyl-norcholesterol ([131]I-Adosterol) demonstrated clear activity in the omentum, weak activity in the rectus muscles, and no activity in the adrenal beds. These findings suggest that the omentum may be more suitable as an implantation site for adrenal tissue than muscular pockets.

Adrenal Gland Neoplasms↗
PubMed2003

High [18F] 2-fluoro-2-deoxy-D-glucose (FDG) uptake of adrenocortical adenoma showing subclinical Cushing's syndrome.

A 48-year-old woman with left adrenal tumor, which showed increased uptake of [18F] 2-fluoro-2-deoxy-D-glucose (FDG) was presented. Her adrenal tumor was incidentally discovered, although she had no remarkable illness, and her blood pressure was normal. Hormonal examination including dexamethason suppression test and diurnal variation in serum cortisol level confirmed preclinical Cushing's syndrome. CT, MRI and 131I-adosterol scintigraphy showed findings consistent with adenoma. FDG-PET revealed that tumor had standardized uptake value of 4.8, which was higher than usual benign tumors. Histological diagnosis of the resected adrenal tumor was adrenocortical adenoma without evidence of malignancy. Although the current literature showed that adenomas in general did not exhibit increased FDG uptake, adenoma in the present case with subclinical Cushing's syndrome showed intense uptake of FDG, suggesting FDG-PET could evaluate hormonal function of an adrenocortical adenoma in a completely asymptomatic normocortisolism patient.

Adrenal Cortex Neoplasms↗
PubMed1998

Hormonal characteristics of primary aldosteronism due to unilateral adrenal hyperplasia.

A case of unilateral adrenocortical hyperplasia is presented. A 46-year-old woman with a 7-year history of hypertension and a 1-year-history of hypokalemia was diagnosed with primary aldosteronism. Computed tomography, magnetic resonance imaging, venous sampling and adosterol scintigraphy exhibited a functioning left adrenal mass. The plasma aldosterone concentration increased markedly when furosemide with upright posture and either captopril or adrenocorticotropin were administered. Plasma renin activity was suppressed below the detectable range. Aldosterone secretion displayed a circadian rhythm and was not suppressed by dexamethasone administration. The resected left adrenal mass was pathologically diagnosed as adrenocortical nodular hyperplasia. Unilateral adrenal hyperplasia involving the zona glomerulosa rarely has been reported, with varying and incompletely characterized hormonal characteristics. This case report and literature review suggest unilateral adrenal hyperplasia as a rare cause of hyperaldosteronism with characteristics intermediate between idiopathic hyperaldosteronism and aldosterone-producing adrenocortical adenoma, resembling the functional features of the adenoma more closely.

Adrenal Cortex↗
PubMed2001

Laparoscopic partial or cortical-sparing adrenalectomy by dividing the adrenal central vein.

BACKGROUND: We perform laparoscopic partial adrenalectomy without sectioning the adrenal central vein has been described because it is important to preserve this vein in the remnant adrenal gland in order to maintain its function. In this article, we describe our technique for laparoscopic partial or cortical-sparing adrenalectomy by dividing the adrenal central vein. METHODS: The procedures were performed in four patients with aldosterone-producing adenomas (APA) and two patients with pheochromocytomas. RESULTS: There were no postoperative complications. At follow-up, adrenal 131I-adosterol scintigrams showed that remnant adrenal function had been preserved in all cases. CONCLUSION: Since the vascular bed adjacent to the remnant adrenal gland is integral to the preservation of its function, it is important to perform procedures that do not separate the remnant adrenal gland from the retroperitonium space. Because the operative field is clearly visualized on the high-magnification video monitor, this delicate procedure can be performed with a high degree of accuracy via the laparoscopic approach. We consider this operative technique to be useful for selected cases.

Adrenal Cortex↗
PubMed1993

Clinical experience of incidentally discovered adrenal tumor with particular reference to cortical function.

We reviewed 14 male and 9 female patients with adrenal tumor discovered incidentally by imaging studies in an attempt to assess adrenal function, mainly cortical function. Pathological diagnosis consisted of cortical adenoma in 12 patients, cortical nodular hyperplasia in 2, medullary hyperplasia in 1, cavernous hemangioma in 1, metastatic tumor in 4 and cyst in 2. Another adrenal cyst was diagnosed by percutaneous puncture. In all patients peripheral levels of plasma cortisol and aldosterone were normal. Plasma catecholamine levels were also normal except for 1 patient with medullary hyperplasia who had equivocal elevation. Among the patients with cortical adenoma and hyperplasia, however, 5 of 10 (50%) had excessive excretion of 24-hour urinary 17-hydroxycorticosteroids, 4 of 13 (31%) had a loss of plasma cortisol circadian rhythm and 7 of 14 (50%) had insufficient suppression on the dexamethasone test. Furthermore all patients had an increased ipsilateral uptake that was not suppressed after dexamethasone on 131iodine-adosterol scintigraphy, while a diminished contralateral uptake was noted in 5 of 15 (33%). Postoperatively, hypertension decreased to normal in 2 of 3 patients and impaired glucose tolerance was improved in 1 of 2. Two patients with cortical adenoma who exhibited a loss of plasma cortisol circadian rhythm experienced postoperative adrenal insufficiency. These data indicate that a considerable number of incidentally discovered adrenal tumors are not nonfunctioning particularly in reference to glucocorticoid secretion, and adrenalectomy seems to be beneficial in some of those patients with hypertension or impaired glucose tolerance.

17-Hydroxycorticosteroids↗
PubMed2002

Laparoscopic partial adrenalectomy.

Since corticosteroids are indispensable hormones, partial or cortical-sparing adrenalectomies may be adopted for the surgical treatment of adrenal diseases. In this article, we describe the technique and results of these procedures. Laparoscopic partial or cortical-sparing adrenalectomy has been performed in 10 patients. Seven cases had an aldosterone-producing adenoma (APA) and three had a pheochromocytoma. Three cases with an APA and a case with a pheochromocytoma had tumors located far from the adrenal central vein, and the vein could be preserved. Four cases with an APA and two with a pheochromocytoma had tumors located close to the adrenal central vein, and it was necessary to section the central vein to resect them. All endoscopic procedures were performed successfully. There were no postoperative complications. At follow-up, adrenal 131I-adosterol scintigrams showed the preservation of remnant adrenal function in all patients. Laparoscopic partial or cortical-sparing adrenal surgery was safely performed, and adrenal function was preserved irrespective of whether the adrenal central vein could be preserved or not. We consider this to be a useful operative technique for selected cases.

Adrenal Gland Neoplasms↗
PubMed1997

Adrenocorticotrophin-independent macronodular adrenal hyperplasia in a patient with lysine vasopressin responsiveness but insensitivity to gastric inhibitory polypeptide.

The aetiology of ACTH-independent macronodular adrenal hyperplasia (AIMAH) is uncertain. We examined a 55 year old man with Cushing's syndrome due to AIMAH, whose cortisol levels increased after stimulation with lysine-8-vasopressin (LVP) in vitro as well as in vivo. Abdominal MRI revealed nodular enlargement of both adrenal glands. No adenoma was evident on pituitary MRI. 131I-adosterol scintigraphy exhibited marked uptake into both adrenal glands. Although baseline plasma cortisol levels were within normal limits, urinary free cortisol excretion was 3-fold higher than the upper limit of the normal range. Plasma ACTH levels were undetectable. Oral dexamethasone failed to suppress plasma cortisol levels irrespective of dose, and administration of corticotrophin releasing hormone failed to increase plasma ACTH and cortisol levels. LVP injection failed to increase plasma ACTH levels, but elicited an increase in plasma cortisol levels. The direct stimulatory effect of LVP on cortisol secretion was confirmed in vitro in cultured adrenocortical cells from macronodules obtained at surgery. Food intake, gastric inhibitory polypeptide (GIP), or octreotide administration, which were reported to regulate cortisol release in patients with AIMAH, failed to affect plasma cortisol levels. In conclusion, plasma cortisol responsiveness to LVP, GIP, and octreotide is heterogeneous in patients with AIMAH.

Adrenal Glands↗
PubMed2001

Autonomy of cortisol secretion in clinically silent adrenal incidentaloma.

Recent progress in non-invasive imaging techniques have resulted in an increasing frequency of adrenal incidentaloma discovery. In addition, even clinically silent adrenal tumor has been suggested to possess a subtle production of adrenal hormones. The aim of the study was to ascertain the autonomy of cortisol production in clinically silent adrenocortical incidentaloma. We investigated the hypothalamic-pituitary-adrenal axis in 38 patients with adrenal incidentaloma. Basal plasma cortisol level was reproducibly within normal range in all the patients with adrenal incidentaloma, but was also normal in half of the Cushing's syndrome cases studied. Eighteen of 38 patients showed plasma cortisol above 3 microg/dl after 1 mg dexamethasone (Dex) and above 1 microg/dl after 8 mg Dex, respectively, and were defined as preclinical Cushing's syndrome. These patients were subjected to further evaluation of the autonomy of cortisol production. The incidence of positive findings indicating autonomy of cortisol secretion was as follows: suppressed basal plasma ACTH level in 44%, loss of normal diurnal rhythm in 79%, lack of ACTH response to CRF in 35%, decreased plasma DHEA-S level in 28%, significant laterality of 131I-adosterol uptake in 75%, atrophy of the contralateral side of the adrenal on CT scan in 6%, and histological atrophy of the adjacent adrenal cortex in 56%, respectively. The endocrine feature relevant to the hypothalamic-pituitary-adrenal axis varied from patient to patient, ranging from the non-functioning adrenal adenoma to Cushing's syndrome. In addition, the results of each test did not coincide with others in each patient. These results clearly demonstrated that the incidence of autonomy of cortisol production in the clinically silent adrenal incidentaloma is not infrequent, showing significant diversity. Systemic evaluation of the hypothalamic-pituitary-adrenal axis before adrenal surgery is warranted for an appropriate glucocorticoid replacement after adrenal surgery.

Adrenal Gland Neoplasms↗
PubMed1996

Adrenocortical adenoma producing 18-hydroxycorticosterone.

A 30-year-old man presented at our hospital with microscopic hematuria. Ultrasonography and computed tomography scanning revealed a right adrenal mass measuring 20 x 20 mm. The tumor was asymptomatic, but there was obvious accumulation on the right side when scintigraphy was performed with radioactive iodine (131I)-labeled adosterol. Endocrinology studies showed elevation of the plasma cortisol and renin concentrations, while the plasma aldosterone level was low. Right laparoscopic adrenalectomy was done on July 4, 1994. Histologic examination showed an adrenocortical adenoma. Serum levels of adrenocortical hormones were measured before and after surgery, and the tissue content for the same hormones was determined in the resected tumor. The hormonal studies showed that the tumor produced 18-hydroxycorticosterone.

18-Hydroxycorticosterone↗
PubMed2006

Gastric diverticulum preoperatively diagnosed as one of two left adrenal adenomas.

A 47-year-old man was diagnosed with primary aldosteronism due to two left adrenal adenomas, suggested on computed tomography (CT) to be located at the upper and lower adrenal portion. However, adosterol scintigraphy revealed negligible uptake at the upper portion of the left adrenal. Laparoscopic left adrenalectomy was performed, but macroscopic examination of the specimen revealed only one adrenal tumor. Continued surgical exploration detected another mass between the spleen and the stomach, which was demonstrated to be continuous with the stomach and was eventually diagnosed as a gastric diverticulum. Postoperatively, aldosteronism resolved and repeat CT revealed staining of the adrenal pseudotumor when oral contrast was administered. Since organs located near the adrenals can simulate adrenal tumors, caution must be exercised in interpreting suprarenal masses on CT. To our knowledge, this is the first reported case of concurrent pseudotumor and true tumor of the ipsilateral adrenal.

Adrenalectomy↗
PubMed1998

Aldosterone-producing adrenocortical adenoma complicated by chronic renal failure. Case report and review of the literature.

A 61-year-old Japanese woman was hospitalized because of general malaise. The patient demonstrated hypertension, hypokalemia and chronic renal failure (CRF). Plasma aldosterone concentration and urinary excretion of aldosterone were elevated. Abdominal computed tomographic scan revealed right adrenal tumor and multiple cysts in both kidneys. Adrenal scintigram using 131I-adosterol disclosed uptake of the isotope in the area corresponding to the adrenal tumor. Plasma aldosterone concentration and renin activity (PRA) in an upright posture and daily variations in adrenocorticotropic hormone, cortisol, aldosterone levels and PRA were compatible with aldosterone-producing adrenocortical adenoma. After administration of spironolactone and manidipine hydrochloride, a calcium antagonist, general malaise disappeared, and blood pressure and serum potassium level returned to the normal range without adrenalectomy. Although adrenalectomy is known to be effective for the treatment of aldosterone-producing adrenocortical adenoma, several papers reporting cases of aldosterone-producing adrenocortical adenoma with CRF indicated that surgical therapy was not always optimal in terms of postoperative conditions. Taken together, the conservative therapy may be one of the choices considering the prognoses of hypertension and renal dysfunction in patients with aldosterone-producing adrenocortical adenoma with CRF.

Adrenal Cortex Neoplasms↗
PubMed2004

Preclinical Cushing's syndrome resulting from black adrenal adenoma. A case report.

A 59-year-old Japanese woman, admitted for the treatment of diabetes mellitus and hypertension, was incidentally discovered to have a solid mass of 1.4 cm in diameter by CT scan with the attenuation value of 38 Hounsfield units, relatively higher for ordinary adrenal adenomas. Magnetic resonance imaging revealed no reduction of signal intensity on opposite-phase image on T1-weighted sequence. Adrenal scintigraphy imaging with 131I-adosterol did not show any uptake of the isotope in the area corresponding to both adrenals. Although she had no characteristic feature of overt Cushing's syndrome, her serum cortisol level was not suppressed after an overnight dexamethasone administration. She was diagnosed as having preclinical Cushing's syndrome. Left adrenalectomy was performed, revealing the well-circumscribed black tumor, mainly consisted of compact cell, in which cytoplasm was filled with numerous granules pigmented with dark to golden brown colors on hematoxylin-eosin staining. These findings suggested that her incidentaloma was a black adrenal adenoma. Production of steroid hormones was confirmed by immunohistochemical analysis of steroidogenic enzymes and by measurement of the tissue contents of hormones, whose levels were comparable with those in adenomas of overt Cushing's syndrome. This is the first case report of preclinical Cushing's syndrome resulting from black adrenal adenoma.

17-Hydroxycorticosteroids↗
PubMed1985

[Image diagnosis of adrenal disorders--II. Cushing's syndrome].

Comparative study of image diagnosis of ultrasonography (US) by linear electronic scanner, computed tomography (CT), and adrenal scintigraphy was performed in 14 patients with Cushing's syndrome. Adrenal imaging by scintigraphy was performed at the 5th and 6th day or further 7th day following the injection of 1 mCi of Adosterol. Cushing's disease (11 cases) US failed to detect the adrenals in 4 cases examined. Measurement of the adrenals on CT film demonstrated the enlargement of adrenals (greater than mean + 2SD) in 6 of 7 cases (85.7%). Scintiscanning showed the increased uptake of bilateral adrenals in 4 of 10 cases (40%). Adrenal scintigraphy with dexamethasone pretreatment (DP) still demonstrated the isotope uptake of bilateral adrenals in all of those 4 cases tested, although the other 6 cases were not studied with DP. From these findings, it was suggested that the measurement of adrenal size by CT was useful for the additional image diagnosis of Cushing's disease, and the adrenal scintigraphy with DP was also available for complementary study of Cushing's disease. Cushing's syndrome due to adrenocortical adenoma (3 cases) In one case examined by US, which had the smallest adenoma (0.6 X 1.0 X 2.0 cm) in this syndrome, the adenoma was not detected. All of 3 adrenal adenomas (2.6 X 2.6 X 2.2 cm to 0.6 X 1.0 X 2.0 cm) were detected by CT. Adrenal scintigraphy demonstrated good uptake by adrenal adenoma but no visualization of the contralateral adrenal in every case.

Adenoma↗
PubMed1985

[Image diagnosis of adrenal disorders--I. CT images of control subjects and image diagnosis of primary aldosteronism].

The shape and size of the adrenals in control subjects without adrenal disorders were studied by computed tomography (CT), and a comparative assessment of diagnostic values of ultrasonography (US) by electronic linear scanner, CT, and adrenal scintigraphy was made on 9 patients with primary aldosteronism. Adrenal imaging by scintigraphy was performed on the 5th and 6th day, or further on the 7th day after the injection of 1 mCi of Adosterol. CT findings of the adrenals in control subjects: Eighty-two % of 100 control right adrenals, and 89% of 100 control left adrenals were detected by CT. Seventy-seven % of the right adrenals were in linear-shape, and the others were in V-shape. The shape of the left adrenals could be classified into triangular-shape (54%), Y-shape (28%) and V-shape (18%). The mean width and thickness of the right adrenals were 28.6 +/- 7.5 mm (M +/- SD) and 3.8 +/- 1.4 mm, respectively. Those of the left ones were 19.4 +/- 5.5 mm and 5.3 +/- 1.8 mm. Image diagnosis of primary aldosteronism: In 2 out of 3 patients examined by US, aldosteronomas were detected. In these 2 patients, one had 2 adenomas 2.8 X 1.7 X 1.2 cm and 1.0 X 1.0 X 2.0 cm in size, and the other had one adenoma 0.8 X 1.0 X 2.0 cm in size. On adrenal scintiscanning under dexamethasone pretreatment (DP), the isotope uptake of aldosteronoma was still seen with the disappearance of the contralateral adrenal in 7 out of 9 cases. In these 7 cases, the laterality of the tumor was confirmed. In one of the remaining 2 cases, the bilateral adrenal images were obtained regardless of DP. In the other case, of which aldosteronoma was the smallest (0.6 X 0.6 X 0.8 cm), the image of the affected adrenal with adenoma as well as the contralateral adrenal disappeared under DP. CT delineated all aldosteronomas in 8 cases examined including 2 cases in which adrenal scintiscanning failed to elucidate the localization of aldosteronoma. These results indicated that the combination of these 3 new image diagnostic methods was available for the detection of aldosteronomas of various sizes.

Adenoma↗
PubMed1985

[Image diagnosis of adrenal disorders. III. "Nonfunctioning" adrenal adenoma, weak mineralocorticoids producing adrenal carcinoma, congenital adrenogenital syndrome due to 21-hydroxylase deficiency--simple virilizing form, and pheochromocytoma].

The image diagnoses of a case of so-called "nonfunctioning" adrenal adenoma, weak mineralocorticoids producing adrenal carcinoma, congenital adrenogenital syndrome due to 21-hydroxylase deficiency--simple virilizing form--, and 5 cases of pheochromocytoma were studied. In a patient with so-called "nonfunctioning" adrenal adenoma (2.3 X 3.0 X 3.3 cm), in which steroids biosynthesis was confirmed, computed tomography (CT) delineated the tumor shadow with extremely low density, and ultrasonography (US) demonstrated the round tumor echo with homogenous and low echogenicity at the superior region of the right renal pole. Adrenal scintigraphy also showed the tumor image. A weak mineralocorticoids-producing left adrenal carcinoma (3.5 X 3.5 X 3.0 cm) was shown as a heterogenous round tumor at the left lateral portion of the vertebra by CT. On adrenal scintigraphy under dexamethasone pretreatment, there was good uptake in the tumor and disappearance of the contralateral. Both bilateral adrenal images on CT in a patient with congenital adrenogenital syndrome were linear-shaped and markedly enlarged. The enlarged right adrenal was clearly demonstrated by US with an electronic sector scanner but not with an electronic linear scanner, although the left one was hardly shown by either US instruments. Three of 4 patients with pheochromocytomas examined by US were correctly detected, while in the remaining one the tumor image was judged to be a retroperitoneal tumor. CT also correctly demonstrated the former 3 pheochromocytomas, but misjudged the latter one as a pancreatic cancer. Good uptake of Adosterol by bilateral adrenals was shown in a case of extra-adrenal pheochromocytoma. Three of 4 cases of adrenal pheochromocytoma showed the isotope uptake of the contralateral normal adrenal alone. In another case of right adrenal pheochromocytoma, isotope accumulation in the colon obscured whether the isotope uptake in the right adrenal was shown or not.

Adenoma↗
PubMed1989

[21-hydroxylase deficiency associated with adrenal tumor: case report of two brothers].

Described herein are two brothers with 21-hydroxylase deficiency (21-OHD) associated with adrenal tumors, and these possible mechanisms are discussed. A 34-year-old male was admitted on Jan. 9, 1984 because of an enlarged and tender left breast. Physical examination revealed short stature (152 cm, 76.5 kg), gynecomastia and shortening of metacarpal bone. His testes were small (2.6 X 1.6 X 1.9 cm). Urinary excretion of 17-OHCS was within normal range (5.9 mg/day), but those of 17-KS, 17-KGS and pregnanetriol were markedly increased (44.4, 110 and 22.6 mg/day, respectively). Plasma concentrations of progesterone and ACTH and urinary excretions of estrone, estradiol and estriol were also increased. Urinary excretions of 17-KS were decreased to 11.7 mg/day and 17-KGS to 22.3 mg/day after the ingestion of 2 mg/day dexamethasone for two days. The computed tomography and a scintigraphy with 131I-Adosterol revealed a tumor in the left adrenal gland, and the adrenal arteriography revealed a neovascularity and a tumor stain in the tumor. These data indicated that the patient was suffering from both 21-OHD and the left adrenal tumor. At this point, adenoma or adenocarcinoma of the adrenal gland was suspected. The left adrenal tumor (85 g) was resected on April 10, 1984, and the pathological diagnosis was adrenal adenoma. The patient's endocrinological abnormalities, however, did not improve after the operation. Urinary excretions of 17-KS and KGS were increased to 57.9 and 108.5 mg/day, respectively, in the patient's elder brother, and 63.3 and 127.9 mg/day, respectively, in his younger brother, indicating that they also had 21-OHD. Interestingly, an adrenal tumor was diagnosed by abdominal computed tomography in the elder brother who had the same HLA typing as the present case. The three brothers had 21-OHD, and two of them had both 21-OHD and adrenal tumor. To our knowledge, this is the first report documenting the co-existence of adrenal tumors in brothers with 21-OHD. This suggests that adenoma can be one of the complications of 21-OHD, probably due to the chronic stimulation by ACTH, and that a possible linkage to HLA may exist in such cases.

Adenoma↗