adhesions between it and the duodenum. The gall- stone was analyzed and found to be a pure choles- terol stone; the wire was essentially pure cop- per. The wire, presumably, caused irritation of the gallbladder epithelium and served as the nidus for the formation of the gallstone.
We are indebted to Fred D’Altroy, Ph.D., Bell Telephone Laboratories, Allentown, Pennsylvania, for analysis of the wire and to John L. Madden, M.D., director of the Depart- ment of Surgery, St. Clare’s Hospital and Health Center, New York City, for analysis of the gallstone.
REFERENCES
1. Rains AJH: Gallstones: Causes and treatment. Springfield, Illinois. Charles C Thomas, 1964
2. Bockus HL: Gastroenterology. Second edition. Vol 3. Philadelphia, WB Saunders Company. 1965
3. Small DM, Rapo S: Source of abnormal bile in patients with cho- lesterol gallstones. New Eng J Med 283:53-57, 1970
4. Bouchier IAD, Freston JW: The aetiology of gallstones. Lancet 1: 340-344. 1968
ACTH-Responsive, Dexamethasone- Suppressible Adrenocortical Carcinoma*
ELLIOT J. RAYFIELD, M.D., LESLIE I. ROSE, M.D., JOHN P. CAIN, M.D., ROBERT G. DLUHY, M.D., AND GORDON H. WILLIAMS, M.D.
A DRENOCORTICAL neoplasms are not thought to be adrenocorticotrophin (ACTH) dependent since they fail to show suppression of 17-hydroxy- corticosteroids (17-OHCS) with the administration of 8 mg of dexamethasone per 24 hours.1 Recently,
Kendall and Sloop2 described a patient with an ad- renocortical adenoma in whom 2 mg of dexametha- sone per 24 hours suppressed urinary 17-OHCS excretion from 18 to 3.5 mg per 24 hours. The fol- lowing report describes a patient with a metastatic adrenocortical carcinoma that physiologically mim- icked adrenocortical hyperplasia.
CASE REPORT
Eight months after right nephroadrenalectomy for adreno- cortical “adenoma” with Cushing’s syndrome a 70-year-old woman was admitted to the Clinical Center of the Peter Bent Brigham Hospital with complaints of facial and leg edema. On admission, the blood pressure was 140/70. There was increased facial hair, red cheeks, liver enlargement to 16 cm in the mid-clavicular line and 4+ pitting edema of the lower extremities. Laboratory data disclosed a normal com- plete blood count, urinalysis, serum electrolytes, calcium and phosphorus. The serum creatinine was 1.2 mg per 100 ml, alkaline phosphatase 200 IU per milliliter (normal, 9 to 41 IU per milliliter) and serum glutamic oxalacetic transaminase 55 U per milliliter (normal, 11 to 33 U per milliliter). X-ray study revealed osteoporosis and an osteolytic lesion of the left 2d rib, and a liver scan was consistent with metastatic disease. After completion of the tests outlined in Table 1, the patient was treated with ortho para ‘DDD 4 g per day, and dexamethasone, 0.75 mg per day. She died 3 months later.
Pathological examination of the surgical specimen demon- strated an adrenocortical tumor, 11 by 12 by 8 cm, adherent to the right renal capsule but without microscopical evidence of renal invasion. Histologic examination showed a well differentiated tumor characteristic of an adrenocortical ade- noma. Post-mortem examination disclosed tumor invasion of the liver, both lungs and the inferior vena cava. The pitui- tary gland weighed 0.720 g and showed Crooke’s degenera- tion. The left adrenal gland was atrophic, weighing 2.8 g. The metastatic lesions and tumor histologically showed a cellular pattern similar to that of the surgical specimen, with the addition of numerous areas of anaplasia and pleomor- phism.
| TIME OF COLLECTION | CREATININE | 17- KETOSTEROIDS | 17-HYDROXYCORTI- COSTEROIDS | DRUG |
|---|---|---|---|---|
| mg/total volume | ||||
| 7 a.m .- 7 p.m. (12 hr) | 324 | 3.3 | 9.9 | - |
| 7 p.m .- 7 a.m. (12 hr) | 372 | 4.8 | 13.3 | - |
| 7 a.m .- 7 a.m. (24 hr) | 714 | 31.0 | 111.2 | 40 U of ACTH intravenously over 8 hr |
| 7 a.m .- 7 a.m. (24 hr) | 702 | 42.6 | 110.4 | 40 U of ACTH intravenously over 8 hr |
| - | - | - | - | - |
| - | - | - | - | - |
| 7 a.m .- 7 a.m. (24 hr) | 648 | 10.9 | 27.9 | - |
| 7 a.m .- 7 a.m. (24 hr) | 708 | 11.6 | 27.8 | Dexamethasone, 2.0 mg by mouth/6 hr |
| 7 a.m .- 7 a.m. (24 hr) | 676 | 6.8 | 9.8 | Dexamethasone, 2.0 mg by mouth/6 hr |
*From the Endocrine-Metabolic Unit and the Department of Medi- cine, Peter Bent Brigham Hospital and Harvard Medical School (address reprint requests to Dr. Williams at the Peter Bent Brigham Hospital, Boston, Mass. 02115).
Supported in part by a grant (9893) from the John A. Hartford Foundation, and in part by a research grant (AM-5100-14) from the National Institute of Arthritis and Metabolic Diseases, U. S. Public Health Service; the clinical studies were carried out in the Clinical Research Center of the Peter Bent Brigham Hospital with the support of a grant (8-M01-FR-31-06) (Dr. Rayfield is a U. S. Public Health Postdoctoral Research Fellow [Grant 1-F03-HE-46,714-01], Dr. Rose a U. S. Public Health Postdoctoral Research Fellow [Grant 1-F2-AM- 36,726], and Dr. Williams an investigator of the Howard Hughes Medical Institute).
DISCUSSION
The present case is of interest because although the histology of the surgical specimen resembled an adrenocortical adenoma, there were widespread metastases at autopsy. The tumor responded to in- travenous ACTH infusions with an increase of al- most five times in 17-OHCS and 17-ketosteroids, whereas 85 to 95 per cent of adrenocortical carcino- mas are not responsive to ACTH.1,3 It is unlikely that the urinary 17-OHCS originated from the markedly
atrophic left adrenal gland found at autopsy. Furthermore, dexamethasone, 8 mg per 24 hours over a two-day period, resulted in a greater than 50 per cent decrease in urinary 17-OHCS from control levels - a response characteristic of bilateral adreno- cortical hyperplasia.4 It is possible that pituitary radiation or hypophysectomy is indicated in the management of some adrenocortical carcinomas and that a larger dose of dexamethasone than 0.75 mg per day would have been physiologically sounder in suppressing tumor growth. This report emphasizes the fact that the diagnosis of adrenocortical carci- noma cannot be excluded on the basis of respon- siveness to ACTH or dexamethasone suppression.
REFERENCES
1. Liddle GW: Cushing’s syndrome, The Adrenal Cortex. Edited by AB Eisenstein. Boston, Little, Brown and Company, 1967, pp 523-551
2. Kendall JW, Sloop PR Jr: Dexamethasone-suppressible adreno- cortical tumor. New Eng J Med 279:532-535, 1968
3. Lipsett MB, Hertz R, Ross GT: Clinical and pathophysiologic aspects of adrenocortical carcinoma. Amer J Med 35:374-383, 1963
4. Liddle GW: Tests of pituitary-adrenal suppressibility in the diagno- sis of Cushing’s syndrome. J Clin Endocr 20:1539-1560, 1960
Acute Renal Failure and Nephrotic Syndrome after Angiocardiography with Meglumine Diatrizoate*
SONIA BORRA, M.D., DAVID HAWKINS, M.D., F.R.C.P. (C), WILLIAM DUGUID, M.B., B.CH., M.R.C. (PATH.), AND MICHAEL. KAYE, M.B., F.R.C.P. (C)
R ENAL complications of angiography are rare.1-5 In the following case oliguria was followed by the development of a nephrotic syndrome and sub- sequent recovery.
CASE REPORT
L.L., a 49-year-old man, was admitted to the hospital with acute pulmonary edema on January 12, 1970. Aortic insufficiency was diagnosed, and he responded to treatment with digitalis and diuretics. The blood urea nitrogen (BUN) was 11 mg, and the serum creatinine 1 mg per 100 ml.
Urinalysis gave normal results. On January 27, he under- went uneventful cardiac catheterization, receiving 157 ml of meglumine diatrizoate (Renografin 70) for angiocardiog- raphy. After the procedure he was anuric for 72 hours, and a maculopapular rash appeared on the skin. On Janu- ary 30, the total white-cell count was 5600, with 15 per cent eosinophils. The BUN was 76 mg, and the serum creatinine 10 mg per 100 ml. A specimen of 15 ml of urine gave a 4+ test for protein, with numerous red cells and granular casts in the sediment. The urinary creatinine was 14 mg per 100 ml, the sodium 111 mEq per liter, and the osmolarity 313
*From the divisions of Nephrology and Immunology, Department of Medicine, and the Department of Pathology, Montreal General Hospi- tal (address reprint requests to Dr. Kaye at the Montreal General Hospital, 1650 Cedar Ave., Montreal 109, Quebec, Canada).
mOsm per liter. X-ray study of the abdomen demonstrated a bilateral nephrogram, with gallbladder opacification.6 The patient remained oliguric for 5 weeks and was maintained on 4 peritoneal dialyses and then 4 hemodialyses.
The urinary sodium, which was initially high, fell to 7 mEq per liter, and the creatinine rose to over 600 mg per 100 ml. Renal biopsy on February 23 showed proliferative glomerulonephritis (Fig. 1),7 and on electron microscopy, fusion of epithelial-cell foot processes was seen. Immuno- fluorescent studies failed to demonstrate immune deposits in the kidney tissue, and no antibody binding was detected on radioimmunoelectrophoresis, with the use of the patient’s serum and radioiodinated meglumine diatrizoate as antigen.
Dialysis was discontinued on March 3, when diuresis started. Gross proteinuria, with excretion of 13 g per day, was observed. Edema developed, and the serum albumin fell to 1.6 g per 100 ml. By March 18 proteinuria ceased, and the BUN and creatinine were normal. One month later the se- rum albumin was also normal, and aortic-valve replacement was performed. The patient was discharged 3 months after admission.
DISCUSSION
Serious nephrotoxicity of uncertain pathogenesis was observed in this patient, and the sequence of