Case: A 35-year-old woman was allergic to iodine contrast medium and was diagnosed with primary aldosteronism (PA) based on functional confirmatory tests. She was suspected to have unilateral PA because of marked hypertension, spontaneous hypokalemia, high plasma aldosterone, reduced plasma renin activity, and a right hypodense adrenal tumor. She wanted to become pregnant and requested adrenalectomy instead of medical treatment with mineralocorticoid receptor antagonists. Localization of PA by adrenal vein sampling (AVS) was necessary, but angiography with iodine contrast medium was not possible because of her allergy. AVS was performed using gadolinium contrast agent (gadoterate meglumine) instead of iodine, in combination with computed tomography angiography (CTA) because the American College of Radiology manual does not recommend prior steroid administration for high-risk patients who have already exhibited adverse reactions to iodine contrast medium. In AVS, before and after adrenocorticotropic hormone (ACTH) loading, 12 blood samples were drawn from the right adrenal vein, left adrenal central vein, left adrenal common duct, left and right renal veins, and the lower inferior vena cava with only 5 mL of gadolinium medium. There were no complications during AVS. Examination revealed an elevated aldosterone/cortisol ratio on the right side, lateralized ratio of 7.4, and contralateral ratio of 0.76; the patient was diagnosed with right unilateral PA. She underwent right adrenalectomy and showed improvements in aldosterone level from 312.4 pg/mL to 83.0 pg/mL, potassium from 3.0 mEq/L to 3.9 mEq/L, and systolic blood pressure from 138 mmHg to 117 mmHg. She is currently off her medications. Conclusion: In PA patients with iodine allergy, AVS can be performed safely and precisely using gadolinium contrast combined with CTA.
Abstract Background Laparoscopic single-site surgery has recently emerged in the field of urology and this minimally-invasive surgery has resulted in a further reduction in morbidity compared with traditional laparoscopy. We present our initial experience with laparoendoscopic single-site surgery of partial adrenalectomy (LESS-PA) to treat aldosterone-producing adenomas. Case presentation A 60-year-old woman was diagnosed with aldosterone-producing macroadenomas in the left adrenal and aldosterone-producing microadenomas in the right adrenal. A two-step operation was planned. The first step involved transumbilical LESS-PA for the left adrenal tumors. A multichannel port was inserted through the center of the umbilicus and the left adrenal gland was approached using bent instruments according to standard traditional laparoscopic procedures. The tumors were resected using an ultrasonic scalpel, and the resected site was coagulated using a vessel sealing instrument and then sealed with fibrin glue. Operative time was 123 minutes and blood loss was minimal. The patient was discharged from hospital within 72 hours. Her right adrenal microadenomas will be treated in the next several months. Conclusions Although our experience is limited, LESS-PA appears to be safe and feasible for treating aldosterone-producing adenomas. More cases and comparisons with the multiport technique are needed before drawing any definite conclusions concerning the technique.
Recent increased use of ultrasonography and computed tomography (CT) has detected a substantial number of incidentally discovered adrenal tumors (incidentaloma). In our institute the discovery rate of adrenal incidentaloma per number of abdominal CT examinations was 0.43%. Those incidentalomas were incidentally found during examination for abdominal discomfort and lumbar pains or evaluation of the cause of hypertension. In the group study by the research committee on“Disorders of Adrenal Steroid Hormones”under the sponsorship of the Ministry of Health and Welfare of Japan, in 53 of 149 patients with incidentaloma, surgical resection was done during 5 years between 1983 and 1989. The pathological examination of those tumors revealed 67.9% of non-hyperfunctioning adrenocortical adenoma, 9.4% of adrenal cancer, 9.4% of ganglioneuroma, 5.7% of pheochromocytoma, 5.7% of adrenal cyst and 1.9% of myelolipoma.The criteria for resection of adrenal incidentaloma have not been established. However, tumors having a diameter of over 5cm are generally excised in most institutes. Tumors having a diameter of between 3cm and 5cm are usually resected if the shape of the tumor is ambiguous and the margin of the tumor is irregular. Tumors having a diameter of less than 3cm should be carefully followed up by abdominal CT examination every 6 months.
To the Editor: We have read the comments by Goulielmos, et al 1 regarding the structural significance of the signal transducer and activator of transcription 1 ( STAT1 ) gain-of-function (GOF) c.970T>C (p.C324R) mutation, in the DNA binding domain (DBD) of STAT1 protein. Based on the results of an in silico analysis, Goulielmos, et al have proposed that the replacement of this position by a positively charged arginine residue triggers a heavily modified interaction of STAT1 with the DNA residues through the change of loop 325–332 structure. First, we are very grateful for the comments by Goulielmos, et al because their structural data1 strengthen our understanding of the pathogenesis of several clinical phenotypes of our case. To date, 12 patients carrying the C324R mutation in STAT1 have been identified2,3, … Address correspondence to Dr. K. Maeshima, Department of Endocrinology, Metabolism, Rheumatology and Nephrology, Faculty of Medicine, Oita University, 1-1 Idaigaoka, Hasama-machi, Yufu, Oita, 879-5593, Japan. E-mail: maeshima{at}oita-u.ac.jp
