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PubMed1998

Telomerase activity in adrenal cortical tumors and pheochromocytomas with reference to clinicopathologic features.

Telomeres consist of short repeated sequences that are shortened on continuous cell proliferations and synthesized by telomerase, an RNA-dependent DNA polymerase. Recent molecular studies have reported that telomerase is activated in most human cancers, whereas it is not detected in most somatic cells. These findings indicate that the positive telomerase activity is closely related to the malignant potential of human tumors. In several types of human tumors, including adrenal cortical tumors and pheochromocytomas, it is very difficult to predict the malignant potential using conventional histopathologic examination. To determine whether telomerase activity is useful as a diagnostic marker, we examined telomerase activity in adrenal cortical tumors and pheochromocytomas with special reference to their clinicopathologic features. Using a highly sensitive polymerase chain reaction (PCR)-based detection method, telomerase activity was demonstrated in one of 13 adrenal cortical tumors and two of seven pheochromocytomas, whereas all seven normal portions of adrenal gland failed to showed any telomerase activity. Although none of the tumors examined in this study was associated with metastasis, these three telomerase-positive tumors were accompanied by clinicopathologic features suggesting malignant potential. Telomerase activity might be a potential marker for estimating the biologic characteristics of adrenal cortical tumors and pheochromocytomas.

Adolescent↗
PubMed1998

Coexistence of an adrenocortical carcinoma with an abdominal ganglioneuroma in a child.

We report a 3-year, 5-month-old boy with an adrenocortical carcinoma. These tumours are rare and highly malignant in childhood. In most cases they are functional, secreting adrenocortical hormones. In this case there was a misleading coexistence with a second abdominal neoplasm, which was a ganglioneuroma; this is a rare benign tumour arising from the sympathetic nervous system. The imaging investigations and their findings are discussed and correlated with pathology.

Abdominal Neoplasms↗
PubMed2004

Partial versus total adrenalectomy by the posterior retroperitoneoscopic approach: early and long-term results of 325 consecutive procedures in primary adrenal neoplasias.

The retroperitoneoscopic approach is a standardized operative procedure for primary adrenal gland tumors. It allows direct access with a detailed view of the adrenal gland. Thereby, a clear differentiation between normal and neoplastic adrenal tissue is often possible, which permits a planned partial resection of the gland in selected cases. Between July 1994 and November 2003 325 posterior retroperitoneoscopic adrenalectomies were performed for primary benign adrenal gland tumors (106 Conn's adenomas, 83 pheochromocytomas, 76 Cushing's adenomas, 60 nonfunctioning tumors; size: 2.8 +/- 1.5 cm; site: 160 right, 165 left) in 318 patients (122 M, 196 F, age: 49.0 +/- 14.3 years). In 96 patients 100 tumors were removed by partial adrenalectomy (30 Conn's adenomas, 33 pheochromocytomas, 20 Cushing's adenomas, 17 nonfunctioning tumors; site: 61 right, 59 left) maintaining tumor-free parts of the adrenal gland. Of this group, 15 patients suffered from bilateral adrenal neoplastic diseases. During the same period, 225 total adrenalectomies (76 Conn's adenomas, 50 pheochromocytomas, 56 Cushing's adenomas, 34 nonfunctioning tumors; site: 109 right, 116 left) were performed in 224 patients. There was no mortality. Major complications were seen in 1.8%, minor complications in 14.5%. Three conversions were necessary to an open or a laparoscopic approach (2 patients and 1 patient, respectively). There are no differences between the two groups (total versus partial adrenalectomy) with regard to tumor size (2.8 +/- 1.6 cm versus 2.8 +/- 1.5 cm), operating time (80 +/- 44 minutes versus 79 +/- 42 minutes), and blood loss (33 +/- 71 ml versus 29 +/- 31 ml). In all patients with partial adrenalectomy, biochemical healing was proven. Fourteen of 15 patients with bilateral diseases had preservation of adrenocortical function. After a mean follow up of 51 months (range: 7-120 months) local recurrence or relapse of the initial diseases was noticed in 6 patients after total adrenalectomy: in 4 patients with Conn's syndrome and bilateral hyperplasia, and in 2 patients with malignant pheochromocytoma and adrenocortical carcinoma, respectively. Our data demonstrate that partial adrenalectomy is a safe procedure not only perioperatively but also in the long-term follow-up.

Adrenal Cortex Neoplasms↗
PubMed2006

The role of adrenal venous sampling in the surgical management of primary aldosteronism.

OBJECTIVE: Primary aldosteronism is the most common endocrine form of secondary hypertension, but no single test or imaging method always identifies it. Identification of a unilateral overproduction of aldosterone due to Conn's adenoma or unilateral hyperplasia is of utmost importance to the surgeon. MATERIALS AND METHODS: We reviewed our experience with primary aldosteronism in 46 consecutive patients who had undergone adrenalectomy at the Surgical Pathology Institute, University of Padua since 1993. All the patients underwent a CT scan. Adrenal venous sampling was performed in those patients with negative or equivocal findings on imaging studies. RESULTS: Computed tomography was non-contributory in 12 patients and frankly misleading in 2 patients, demonstrating a probable mass lesion in the contralateral but not in the ipsilateral adrenal. Eighteen patients had selective venous sampling that was successful in altering the management of 14 cases. Eleven patients who biochemically had an adrenal adenoma, had normal/equivocal CT, while the remaining 3 had bilateral or contralateral adrenal masses. Venous sampling localized aldosterone secretion and an adenoma, less than 1 cm in diameter, was removed, curing their hypertension. Eleven patients were treated by open adrenalectomy and 35 by the lateral transperitoneal laparoscopic approach. Histological examination revealed 45 Conn's adenomas, of which 13 had a diameter of less than 1 cm (range 0.3-0.8), and 1 case of nodular hyperplasia. CONCLUSIONS: Patients who have equivocal or unexpected CT findings should proceed to hormonal localization. Adrenal venous sampling is essential in patients with equivocal CT scans to avoid unnecessary and inappropriate adrenalectomy.

Adrenal Cortex Neoplasms↗
PubMed2007

Posterior retroperitoneoscopic adrenalectomy: a comparison between the initial experience in the invention phase and introductory phase of the new surgical technique.

BACKGROUND: Today, the posterior retroperitoneoscopic technique has become a standard procedure in adrenal surgery. The procedure allows direct access to the adrenal glands, but it seems to be difficult because of the uncommon anatomic view. This study compares the learning period of the new procedure of "posterior retroperitoneoscopic adrenalectomy" in the primary invention phase and the secondary introductory phase in a different hospital 10 years later. MATERIALS AND METHODS: The analysis included 100 posterior retroperitoneoscopic adrenalectomies (PRA) and involved 50 procedures in each center. Group A consisted of 44 patients (14 males, 30 females; age: 48.7 +/- 14.5 years) undergoing surgery between 07/1994 and 8/1996 (24 right, 26 left; 8 Cushing adenomas, 14 Conn adenomas, 11 pheochromocytomas, 7 nonfunctioning adrenocortical adenomas, 10 ACTH-dependent adrenal hyperplasias). Group B consisted of 50 patients (12 males, 38 females; mean age 59.3 +/- 10.7 years) operated between 01/2004 and 01/2006 (28 right, 22 left tumors; 5 Cushing adenomas, 12 Conn adenomas, 4 pheochromocytomas, 29 nonfunctioning adrenocortical adenomas). All PRAs were performed with the patient in the prone position with 3-4 trocars placed caudally in the region of the 11th and 12th ribs. In group A, the surgical team developed the technique of PRA themselves. Before their first PRA, the surgical team of group B was introduced to the technique by the group A surgeons and afterwards were supervised continuously. RESULTS: No serious intraoperative or postoperative complication occurred in either group. Group A experienced 7 conversions to open surgery, whereas group B had one conversion and one early reoperation due to bleeding (P = 0.03; chi(2)-test). The mean operative time was 117 +/- 41 minutes versus 83 +/- 35 minutes (group A and B respectively; P < 0.001; t-test). Estimated blood loss was similar in the two groups (47.2 +/- 46.2 ml versus 54 +/- 16.3 ml, group A versus B, respectively; P = 0.36; t-test). CONCLUSIONS: The study demonstrates the feasibility, safety, and reproducibility of the new surgical method of PRA both when it is employed in the early phase of invention, as well as when performed by surgeon-learners. After comprehensive training, the operative time and conversion rate are dramatically reduced, allowing for a short learning period.

Adrenal Cortex Neoplasms↗
PubMedDate not supplied

Adrenal surgery in the elderly: too risky?

Surgical treatment for adrenal disease may be withheld from elderly patients because of concern about prohibitive operative morbidity and mortality. To obtain objective data in our practice, we analyzed the results of adrenalectomy for patients aged 65 years and older. From 1984 to 1993 there were 85 patients (41 men, 44 women) with ages ranging from 65 to 84 years (median 69 years) who underwent adrenalectomy for Cushing syndrome (n = 19), pheochromocytoma (n = 16), adrenocortical carcinoma (n = 7), benign adenoma (n = 26), or primary hyperaldosteronism (n = 17) at our institution. Median follow-up was 26 months (range 1 month to 9.1 years). A retrospective review with respect to preoperative risks and postoperative morbidity and mortality was performed utilizing the American Society of Anesthesiologists (ASA) physical status classification and the modified Goldman multifactorial cardiac risk scheme. Survival was estimated by the Kaplan-Meier methods. Operative mortality was 7% (six patients). No patients with pheochromocytoma or primary hyperaldosteronism died during the postoperative period. Patients undergoing adrenalectomy for adrenocortical carcinoma had a significantly higher operative mortality (43%) (p = 0.006). Postoperative complications developed in 19 patients (22%), and there was a reoperation rate of 6% (5 patients). Nineteen percent of patients required postoperative intensive care admission and had a median stay of 2 days (range, 1-38 days). Median hospital stay was 7 days (range 3-47 days). Seventy-three patients (86%) remained alive at study completion. Two- and five-year survivals were 86% and 84%, respectively. Goldman class II or greater was an excellent predictor of increased morbidity (p = 0.032) and mortality (p = 0.036). With the exception of adrenocortical carcinoma, adrenal surgery for elderly patients can be performed with acceptable morbidity and mortality. The Goldman multifactorial cardiac risk scheme reliably predicts postoperative outcome in this elderly group of patients.

ACTH Syndrome, Ectopic↗
PubMed1996

Adrenalectomy for treatment of Cushing syndrome: results in 122 patients and long-term follow-up studies.

Patients with Cushing syndrome (n = 122) who underwent adrenalectomy from 1957 through 1993 were reviewed for survival and complications. Of the 122 patients, 70 had adrenocortical adenoma, 30 Cushing's disease, 6 primary pigmented nodular adrenocortical disease (PPNAD), 7 other types of primary nodular hyperplasia, 5 adrenocortical carcinoma, and 4 ectopic ACTH syndrome. Sixty-five patients with adrenocortical adenoma are alive, and the survival rate was equal to the age-matched control population, when patients who died of the postoperative complication were excluded. Of the patients with Cushing's disease, 20 are alive; and 10 of 16 patients (63%) who were followed-up and evaluated had skin pigmentation. Four of sixteen patients (25%) developed Nelson's syndrome. Four PPNAD patients and five with other types of nodular hyperplasia are alive. Most of these patients underwent bilateral total adrenalectomy, but some patients remitted after unilateral adrenalectomy. All of five adrenocortical carcinoma patients and four with ectopic ACTH syndrome died within 2 years after operation. The prognosis for patients with adrenocortical adenoma after unilateral adrenalectomy is excellent, though it is important to avoid operative complications. The rapid cure of signs and symptoms of glucocorticoid excess after total adrenalectomy is ensured, and prognosis is satisfactory under careful glucocorticoid replacement, making total adrenalectomy an alternative treatment for Cushing's disease.

ACTH Syndrome, Ectopic↗
PubMed1996

Adrenaloma: a call for more aggressive management.

We review our experience from the surgical management of 57 patients (24 males, 33 females) with a mean age of 48.5 years who underwent adrenalectomy because of the computed tomography (CT) finding of a "nonfunctioning" adrenal tumor (adrenaloma). We found that CT consistently underestimated the real histologic size of the adrenal tumor (p = 0.001). Of the 57 resected tumors, 23 were cortical adenomas, 7 myelolipomas, 8 adrenal cysts, 11 nodular hyperplasias, 2 primary adenocarcinomas, 2 metastatic carcinomas, and 4 pheochromocytomas. The mean diameter was 5.89 cm and the mean weight 114.07 g. The mean diameter of the resected primary adenocarcinomas was 3.0 cm and 4.5 cm, respectively. The operative mortality was zero and the perioperative morbidity minimal. The mean operating time was 137 minutes (range 60-240 minutes). The posterior approach had the shortest operating time and the laparoscopic approach the shortest hospital stay and the least postoperative need for narcotics. During the 6.2 years mean follow-up period, five patients with preoperative hypertension remained normotensive, and both patients with the resected primary adenocarcinomas are alive without recurrence. We suggest a more liberal surgical approach to patients with adrenalomas because: (1) even small tumors can be malignant or potentially lethal (e.g., pheochromocytomas); (2) some tumors that appear to be nonfunctioning may in reality be functioning; and (3) other nonfunctioning tumors may, with time (and without prior notice), function. The low risk of adrenalectomy especially via the laparoscopic approach can provide an early definitive diagnosis and treatment, avoiding the cost of repeated CT scans and other studies as suggested by the currently prevailing conservative management of these tumors.

Adenocarcinoma↗
PubMed2003

[Adrenocortical tumours].

Adrenocortical tumours have to be differentiated from cortical hyperplasias, medullary tumours and extra-adrenal tumours or metastases. These diagnoses are mostly possible using routine paraffin sections. In some cases, immunostaining for differing cortical and medullary tumours and metastases are necessary. Adrenal hyperplasias can be congenital or acquired. They are diffuse, micronodular or macronodular and may be inactive or induce a cortical hyperfunction. Very rarely, macronodular hyperplasias may transform into autonomous adenomas. Primary, pigmented adrenocortical disease is a rare, special type of primary adrenal Cushing's syndrome, showing many small tumours with pleomorphic cells and an atrophic surrounding cortical tissue. Adrenal adenomas are mostly unilateral and solitary tumours weighing less than 50 g. Adrenal cortical carcinomas are mostly larger. Like the adenomas, they may be inactive and may induce a hypercortisolism, a hyperandrogenism or (very rarely in carcinomas) a hyperaldosteronism. Differentiating cortical adenomas and carcinomas is difficult in some cases but is possible using a diagnostic algorithm. Myelolipomas are benign, inactive tumours composed of mature fat cells and bone marrow cells. They have to be differentiated from adenomas with myelolipomatous metaplasia. Further, rare primary cortical tumours are mentioned.

Adenoma↗
PubMed2007

[123I-Metaiodobenzylguanidine- (MIBG-) scintigraphy: paradoxical positivity in an oncocytic adrenocortical carcinoma].

(123)I-metaiodobenzylguanidine (MIBG), a radio-labeled catecholamine analogue, is used for the imaging of pheochromocytoma based on the selective uptake of MIBG by chromaffin tissues. MIBG scintigraphy displays high sensitivity (90%) and specificity (close to 100%). In contrast, the false-positive uptake of MIBG by adrenal cortical carcinoma is rare. Here, we report a metastatic oncocytic adrenal cortical carcinoma with MIBG uptake used for therapeutic purposes.

3-Iodobenzylguanidine↗
PubMed2002

Congenital hemihypertrophy and pheochromocytoma, not a coincidental combination?

UNLABELLED: We describe a 19-year-old female, known to have congenital hemihypertrophy, who presented with bilateral benign pheochromocytoma. This is the second time that this combination has been reported in the literature. We speculate that the combination of congenital hemihypertrophy and pheochromocytoma is not coincidental and could be part of the clinical spectrum of the Beckwith-Wiedemann syndrome. CONCLUSION: in patients with congenital hemihypertrophy, the physician should be aware of the symptoms of pheochromocytoma. Besides screening for abdominal tumours, analysis of plasma and/or urinary catecholamines and/or their metabolites should be considered.

Adrenal Cortex Neoplasms↗
PubMed2003

Virilising adrenocortical tumours in children.

UNLABELLED: Adrenocortical tumours (ACT) are a rare but important cause of virilisation in infancy and childhood. Four cases of virilising ACT are presented. Two girls (age 0.9 years and 3.9 years) and two boys (age 6.2 years and 6.4 years) had symptoms and signs of virilisation before the age of 6 years. Diagnosis of a virilising adrenal tumour was confirmed by laboratory tests, diagnostic imaging and histology. However, one female patient was misdiagnosed and treated for 3 months as atypical congenital adrenal hyperplasia. Ultrasonography of the adrenal region could not visualise the tumour in three out of four cases. The most sensitive method of diagnostic imaging was MRI. In all cases, treatment consisted of complete surgical resection of the adrenal tumour by open abdominal surgery. Immunohistochemistry was performed in all patients and in two patients there was an overexpression of p53, indicating p53 mutation and in three cases the ki67 proliferation index was greater than 5%. The classification of ACT in childhood is extremely difficult. Histology scores adapted from adrenal tumours in adults and molecular markers are under investigation, but there is still not enough clinical experience since ACT are so rare. CONCLUSION: Long-term follow-up is mandatory not only because of the uncertainty in classification of adrenocortical tumours, but also for observation of growth and pubertal development.

17-Hydroxycorticosteroids↗
PubMed2001

Clinical management of malignant adrenal tumors.

Malignant primary adrenal tumors are rare forms of cancer with an estimated incidence of two to ten new cases per one million inhabitants per year. The 5-year survival rate for adrenocortical carcinoma is approximately 35%, whereas the 10-year survival rate of malignant pheochromocytoma reaches 40%. Clinical studies support repeated surgery as the mainstay of treatment, either with curative or palliative intention. For adrenocortical carcinoma, adjunctive treatment with oral mitotane leads to well-documented improvement of survival. Rare malignant pheochromocytomas with distant metastases are preferably treated by 131I-MIBG. Chemotherapy is reserved for unresectable tumors without sufficient response to mitotane or 131I-MIBG, respectively. Cisplatin and etoposide as single therapy, or in combination with doxorubicin or etoposide, appear to be effective in adrenocortical carcinoma. Malignant pheochromocytoma may be treated with vincristine, dacarbazine, and cyclophosphamide. Treatment with octreotide is currently being evaluated. Radiotherapy is indicated if unresectable tumor masses cause local symptoms. If symptoms of endocrine activity are not sufficiently controlled by measures aiming at tumor mass reduction, specific inhibitors of hormone synthesis or action are available. Ketoconazole is widely used for adrenocortical carcinoma, and phenoxybenzamine and metyrosine are available for malignant pheochromocytoma. This review provides guidelines for rational disease management based on still scanty clinical evidence.

Adrenal Cortex Neoplasms↗
PubMed2005

Sub-mesocolic access in laparoscopic left adrenalectomy.

BACKGROUND: This article reports an alternative laparoscopic access to left adrenal gland. METHODS: From January 1994 to August 2004, 209 laparoscopic adrenalectomies were performed in our Department. Indications were Conn adenoma (55 cases), incidentaloma (64), Cushing adenoma (45), pheochromocytoma (32), adreno-genital syndrome (two), mielolipoma (two), and metastatic mass(nine). Of 209, in 12 cases the left adrenalectomy was performed through a submesocolic access (seven pheochromocytoma, two incidentaloma, two Cushing adenoma, one Conn adenoma,). The identification and closure of the adrenal vein with minimal gland manipulation resulted the main benefit of this approach. Moreover, the adrenalectomy was performed with minimal anatomical dissection. RESULTS: No mortality or major complications occurred. During the operation, the blood pressure and cardiac rhythm were significantly more stable, in the group of patients who underwent a left adrenalectomy by the submesocolic approach compared to the anterior or flank lateral transperitoneal group. CONCLUSIONS: Left adrenal lesions, as selected cases of pheochromocytoma, can be safely treated by laparoscopic submesocolic access.

Adrenal Cortex Neoplasms↗
PubMed2005

Endoscopic adrenalectomy: an analysis of the transperitoneal and retroperitoneal approaches and results of a prospective follow-up study.

BACKGROUND: Endoscopic adrenalectomy is currently performed using either a retroperitoneal or transperitoneal approach. The aim of this study was to determine which of these is the optimal surgical technique in a prospectively designed analysis of a large series of patients operated on by a single team over a 10-year period. METHODS: From February 1994 to March 2004, 267 endoscopic adrenalectomies (retroperitoneal in 132 patients and transperitoneal in 135 patients) were performed in 245 consecutive patients. There were 102 right lateral and 121 left lateral procedures (22 patients had a bilateral procedure). The most prevalent indication was incidentaloma (35.9%), followed by pheochromocytoma and Conn's adenoma. RESULTS: The endoscopic procedure was performed in 238 of 245 patients (97.1%). The conversion rate was 1.5% for the transperitoneal approach and 3.8% for the retroperitoneal approach. No statistically significant influence was noted for the parameters of intraoperative blood loss, rate of postoperative complications, and duration of hospital stay with regard to the surgical technique. The operative time and the learning curve proved to be significantly longer for the retroperitoneal adrenalectomy. In addition, a variance analysis identified tumor size (>5 cm) as a significant factor influencing the operative time, whereas body mass index and localization (right/left lateral) did not prove significant. CONCLUSION: Independent of the underlying pathology, endoscopic adrenalectomy using either the trans- or retroperitoneal approach can be performed in 96-98% of all patients. Differences between the two techniques in operative time and learning curves clearly favor the transperitoneal adrenalectomy.

Adrenal Cortex Neoplasms↗
PubMed2006

Diagnostic accuracy of chemical-shift MR imaging to differentiate between adrenal adenomas and non adenoma adrenal lesions.

PURPOSE: The objective of this study was to evaluate the diagnostic accuracy of chemical-shift (CS) magnetic resonance (MR) imaging in the differential diagnosis of adenoma and nonadenoma adrenal masses. MATERIALS AND METHODS: We enrolled 36 patients (9 men, 27 women, mean age 51.3+14.4 years) with unilateral (n=31) or bilateral (n=5) adrenal masses incidentally discovered during imaging examinations [ultrasound (US), computed tomography (CT)] performed for other indications. A total of 41 adrenal lesions were evaluated (mean diameter 3.0+2.2 cm). Histology (n=19), biopsy (n=3) or clinical-imaging follow-up (n=19) demonstrated 29 adenomas, five pheochromocytomas, three cysts and four carcinomas. MR imaging was performed using the following breath-hold sequences: T1-fast field echo (FFE) [repetition time (TR)/echo time (TE)=236/4.6 ms], T2-turbo spin echo-single shot (TSE-SSh) (TR/TE=831/80 ms), T1-DUAL-FFE (TR=236, double TE=4.6/2.3 ms in phase and out of phase) and T1-FFE after gadolinium-DTPA (Gd). Axial and coronal imaging planes were used, with a slice thickness of 3-5 mm. MR images were qualitatively assessed for signal intensity of the adrenal mass relative to the liver on T1, T2, CS and T1-Gd scans; diagnostic criteria for adenomas were isointensity or hypointensity on both T1 and T2 scans, out-of-phase CS signal loss and mild transient enhancement after Gd. RESULTS: Analysis of T1-T2 signal intensity showed diagnostic accuracy, sensitivity, specificity, positive predictive value (PPV) and negative predictive value (NPV) of 80%, 72%, 100%, 100% and 60%, respectively. In contrast, analysis of CS and T1-Gd signal intensity showed diagnostic accuracy, sensitivity, specificity, PPV and NPV for both sequences of 93%, 90% (p<0.05 vs. T1-T2 analysis), 100%, 100% and 80% (p<0.05 vs. T1-T2 analysis), respectively. CONCLUSIONS: CS MR imaging significantly improves characterization of adrenal masses compared with conventional T1-T2-weighted images, providing accuracy similar to that of the T1 sequence after Gd. Therefore, the CS sequence is strongly recommended for MR study of adrenal masses, and its use might obviate the need for Gd administration.

Adrenal Cortex Neoplasms↗
PubMed2002

Aldosteronoma.

Aldosteronoma is a surgically curable cause of hypertension. Recent studies have found aldosteronomas to be a more common cause of hypertension than previously thought. At least 2% of patients with hypertension may have an aldosteronoma. More than 50% of these patients are normokalemic because of earlier diagnosis or milder disease, but still benefit from adrenalectomy. Patients with hypertension should be screened for possible primary hyperaldosteronism regardless of their serum potassium level. When used in conjunction with the appropriate laboratory tests, high-resolution computerized tomography scanning helps the surgeon to differentiate accurately between an adrenal adenoma and bilateral adrenal hyperplasia. Focused approach and laparoscopic resection are the norm for the surgical treatment of aldosteronoma.

Adrenal Cortex Neoplasms↗