Postgrad Med J: first published as 10.1136/pgmj.61.715.435 on 1 May 1985. Downloaded from Postgraduate Medical Journal (1985) 61, 435-438

Adrenal insufficiency and bilateral adrenal enlargement: demonstration by computed tomography

T. Wheatley, S. Gallagher', A.K. Dixon' Departments ofMedicine and 'Radiology, Addenbrooke's Hospital, Hills Road, Cambridge CB2 2QQ, UK.

Summary: A patient presented with fever, malaise and a staphylococcal wound infection occurring 3 weeks after severe haemorrhage from a lacerated brachial artery. There were no clinical features to suggest Addison's disease but abdominal computed tomography to exclude abdominal showed bilateral enlargement with preservation ofadrenal shape. This was consistent with resolving adrenal haemorrhage or infarction and endocrinological investigations confirmed primary adrenal failure.

Introduction Primary adrenocortical insufficiency is often 85 mmHg and emergency exploration of the right overlooked because the presenting features, such as ante-cubital fossa was performed. During the opera- malaise, abdominal pain, weight loss and gastro-intes- tion he received 3 units ofwhole blood and his systolic tinal symptoms are non-specific and may be mislead- blood pressure varied between 100 and 140 mmHg.

ing (Bayliss, 1980; Irvine et al., 1979). We report the His post-operative recovery was uncomplicated. copyright. early diagnosis of adrenal failure following the Eleven days later he was readmitted with abdominal observation of adrenal enlargement on computed pain, weight loss, pyrexia and a wound infection from tomography (CT) performed while investigating which Staphylococcus aureus was isolated. His right pyrexia and abdominal pain. brachial blood pressure was 150/75 mmHg and further investigations showed a normal chest radiograph, normal plasma electrolytes, a normochromic nor- Case report mocytic anaemia of 10.6 g/dl, a normal count,

normal clotting studies and sterile blood cultures. He http://pmj.bmj.com/ A previously fit 39 year old English man was admitted was treated with 500 mg flucloxacillin every 6 h but, as with a severe laceration around the right elbow caused his abdominal pain and pyrexia persisted, abdominal by broken glass. There was no past history of renal or CT examination was performed which showed pulmonary tuberculosis, or travel to the tropics or marked bilateral adrenal enlargement (Figure 1). The North America and no family history ofauto-immune CT findings led to endocrinological investigations disease or tuberculosis. On examination he was pale which showed primary adrenocortical insufficiency. A and sweating with substantial arterial from 9 a.m. basal plasma adrenocorticotrophic hormone the right brachial artery, a sinus of 120 (ACTH) concentration was elevated at 270 ng/I (nor- on September 29, 2021 by guest. Protected beats/min and a supine left brachial blood pressure of mal range less than 10-80ng/l) and a low 9a.m. 80/50 mmHg. There was no pigmentation suggestive plasma cortisol concentration of 120 nmol/1 (normal of Addison's disease nor clinical features of other greater than 250 nmol/l) rose only to 130 nmol/l auto-immune diseases or a generalized bleeding disor- 30 min after the intramuscular administration of der. Initial investigations showed a normal chest 250 fig oftetracosactrin (Synacthen). Primary adreno- radiograph, plasma sodium 135 nmol/l, plasma pot- cortical insufficiency was confirmed by a 9 a.m. assium 3.6 nmol/l, plasma bicarbonate 27 mmol/l, plasma cortisol concentration of 160 nmol/l following plasma urea 3 mmol/l and plasma creatinine the intramuscular administration of 1 mg of 90 imol/l. Following treatment with 1.51 of tetracosactrin acetate and zinc complex (Synacthen Haemocell, his systemic blood pressure rose to 130/ depot) for 3 d. Screening for adrenal, thyroid and gastric parietal cell antibodies was negative. A repeat high resolution CT examination of the adrenals 3 Correspondence: T. Wheatley, M.A., M.R.C.P. weeks later showed little overall change in size. Accepted: 15 August 1984 However, after intravenous contrast enhancement © The Fellowship of Postgraduate Medicine, 1985 Postgrad Med J: first published as 10.1136/pgmj.61.715.435 on 1 May 1985. Downloaded from 436 CLINICAL REPORTS

hydrocortisone 20 mg in the morning and 10 mg in the evening and 0.1 mg/d 9-alpha-fludrocortisone, he re- mained asymptomatic with a normal chest radiograph and plasma electrolytes.

Discussion Although it is well known that adrenocortical insuf- ficiency may follow local sepsis, septicaemia and hypotension (Xarli et al., 1978) the diagnosis may be overlooked until an occurs (Portnay et al., 1974; Bayliss, 1980), since ifacute adrenal failure is superimposed upon previously normal adrenal func- tion, the typical features of chronic anorexia, weight loss and pigmentation are absent (Bayliss, 1980). In our case neither the clinical signs nor plasma elec- trolytes suggested the diagnosis of adrenal failure Figure 1 Abdominal CT showing enlarged adrenals while the presenting symptoms initially seemed ad- (arrows) which still retain an adrenal shape. equately explained by concurrent staphylococcal in- fection, so that adrenocortical insufficiency was dis- covered only after an adrenal abnormality had been (50ml of sodium iothalamate, Conray 420), each demonstrated by CT. adrenal gland showed rim enhancement with a fluid The adrenal glands can be visualized by CT in a dense centre (Figure 2). A third scan after 8 weeks majority of patients (Brownlie & Kreel 1978; Mon- showed some diminution in adrenal size but no tagne et al., 1978) and several authors have stressed its of evidence developing calcification. Following treat- value in the diagnosis of primary and secondarycopyright. ment with hydrocortisone and 9-alpha-fludrocort- adrenal tumours and Cushing's syndrome due to isone the patient's abdominal pain, pyrexia and pituitary disease or ectopic adrenocorticotrophic hor- malaise resolved and 3 months later while taking mone secreting (Dunnick et al., 1979; White et al., 1982; Adams et al., 1983). CT has been less useful in the diagnosis of Addison's disease and .... itt although small gland remnants and adrenal calcifica- .:: tion may be seen (Doppman et al., 1982; Huebener &

..o..o.oSSQIS!I111_~~~~~~~~~~~~~~~~~~~...... Treugut, 1984) others have reported normal http://pmj.bmj.com/ appearance of adrenal glands in primary adrenal failure (Korobkin et al., 1979; Eghrari et al., 1980; -.°...... Adams et al., 1983). However, when adrenal failure has been due either to granulomatous infiltration . . caused by tuberculosis (Wilms et al., 1983), histoplas- (arrow).isstillenlarged.Theappearanm..en .n- mosis (Doppman et al., 1982; Wilson et al., 1984) or

*;; .o..°. blastomycosis (Halvorsen et al., 1982) or to acute adrenal haemorrhage and infarction, the CT findings on September 29, 2021 by guest. Protected of bilateral enlargement have contributed to the diagnosis and management. . Korobkin et al. (1979) was the first to describe the association of primary adrenal failure and CT appearances suggestive of adrenal haemorrhage and infarction. Subsequently other reports have shown similar findings in patients with haemorrhagic tenden- cies due either to therapeutic anticoagulation (Swift et Figureven 2ca;Highaoresolutionora post-intravenous contrast CT al., 1981; Liu et al., 1982; Ling, 1983) or throm- scan of the left adrenal gland at 3 weeks. The adrenal bocytopenia in patients with myeloproliferative dis- (arrow) is still enlarged. The appearance ofrim enhan- ease (Albert et al., 1982; Wolverson & Kannegiesser, cement around an avascular centre is consistent with 1984). As the occurrence of adrenal haemorrhage in either infarction or resolving haemorrhage. ivc: inferior this group is well known (Portnay et al., 1974), it vena cava; ao: aorta. should always be considered when such patients CLINICAL REPORTS 437 Postgrad Med J: first published as 10.1136/pgmj.61.715.435 on 1 May 1985. Downloaded from present with abdominal pain, nausea, vomiting, stimulation is important and relevant to our patient in pyrexia or hypotension. whom staphylococcal infection occurred following the However, even when clotting and platelet function stress of previous trauma and substantial blood loss. are normal, bilateral adrenal haemorrhage may follow The administration of bacterial endotoxin to rats an episode of hypotension or infection (Xarli et al., causes an increase in adrenal size and weight, but not 1978). Such patients are at particular risk since haemorrhage. However, ifthese animals are previous- concurrent or infectiu> may provide an ad- ly stressed, or pre-treated with adrencorticotrophic equate explanation of their symptoms, delaying the hormone, the administration ofendotoxin, which may diagnosis of adrenal failure until an Addisonian crisis be concentrated in the (Jones & Carter, occurs. However, these patients may well have CT 1955), causes adrenal haemorrhage (Levin & Cluff, examinations of the abdomen and Wolverson & 1965; Beisel & Rapoport, 1969). Kannegiesser (1984) reported 2 patients in whom Now that CT of the abdomen is becoming more adrenal haemorrhage was noted on CT examination commonplace, bilaterally enlarged adrenal glands will performed to exclude a sub-phrenic abscess complicat- be shown as an incidental finding in numerous ing pneumonia. In our patient the CT appearances patients. This will usually be due to metastatic disease were compatible with either adrenal infarction or which should be recognisable by loss of the normal resolving haemorrhage, as the non-enhancing central adrenal shape, while pituitary and ectopic ACTH portion would be compatible with an area either of secreting tumours should be considered when the resolving haematoma, or oedematous infarcted tissue. adrenal shape is well preserved. However, sometimes Both hypotension and infection with S.aureus have the adrenal glands will be enlarged due to adrenal been associated with adrenal haemorrhage and infarc- infarction and/or resolving haemorrhage which may tion found post-mortem (Xarli et al., 1978) and while lead on to primary adrenal failure. If in these circum- the exact patho-physiology is uncertain, in the absence stances is considered and ex- of a systemic haemorrhagic disorder, the important cluded by performing a short tetracosactrin test, factors are probably the vascular supply to the gland progression to a life threatening Addisonian crisis may and the extent of recent adrenocortical stimulation. be avoided.

The adrenal glands are intensely vascular structures copyright. with extensive small arterial branching but venous drainage often limitw! to one vessel (Anson et al., 1947). This not only predisposes to haemorrhage and Acknowledgements infarction, but facilitates the one leading to the other, We thank Dr D. Rubenstein for permission to report this so that the initial cause may be hard to determine case, Professor R. Y. Calne for the initial referral and Mrs L. (Berte, 1953). The role of recent adrenocortical Stellitano and Mrs M. Watts for typing the manuscript. http://pmj.bmj.com/

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