scholarly journals Acute adrenal failure as the heralding symptom of primary antiphospholipid syndrome: report of a case and review of the literature

2005 ◽  
Vol 153 (4) ◽  
pp. 507-514 ◽  
Author(s):  
Fabio Presotto ◽  
Francesca Fornasini ◽  
Corrado Betterle ◽  
Giovanni Federspil ◽  
Marco Rossato

Acute adrenal failure is a potentially fatal condition if overlooked. Occasionally, acute adrenal insufficiency may ensue from bilateral adrenal haemorrhage in patients with known antiphospholipid syndrome (APS). APS is characterized by recurrent arterial and venous thrombosis, pregnancy complications and detection of autoantibodies to phospholipids. This syndrome may be associated with non-organ specific diseases (e.g. connective tissue disorders) or with malignancies, but it may also appear in isolated form (primary APS). In a very few cases the heralding manifestation is given by adrenal failure. We report here a 63-year-old man presenting with acute adrenal insufficiency as the opening clinical manifestation of an APS. We also carried out a computer-aided search of the literature to identify all cases of primary adrenal failure as the first-recognized expression of a primary APS, a condition that not so infrequently may be tackled by endocrinologists. 20 patients fulfilled the inclusion criteria. The great majority of them were males (75%) with a mean age of 42 years. Abdominal pain was present in 14 patients, followed by fever (13 patients) and hypotension (12 patients). The main morphological findings by computed tomography or magnetic resonance were consistent with bilateral adrenal haemorrhage in 11 patients. Lupus anticoagulant was present in all of the 19 tested patients. Our observations emphasize the importance in the assessment of clotting times, and possibly of antiphospholipid antibodies, in all patients with diagnosis of rapidly progressive adrenal failure and concurrent abdominal pain.

2011 ◽  
Vol 50 (23) ◽  
pp. 2923-2926 ◽  
Author(s):  
Anastasios Mavridis ◽  
Marina Skopeliti ◽  
Panagiota Galani ◽  
Sofia Lafoyianni ◽  
Constantinos Christopoulos

2012 ◽  
Vol 38 (1) ◽  
pp. 49
Author(s):  
Nicola Improda ◽  
Maria Alessio ◽  
Donatella Capalbo ◽  
Giustina Russo ◽  
Ida D'Acunzo ◽  
...  

2011 ◽  
Vol 9 (S1) ◽  
Author(s):  
G Russo ◽  
R Carlomagno ◽  
C Forni ◽  
L De Martino ◽  
D Capalbo ◽  
...  

Author(s):  
Iza F R Machado ◽  
Isabel Q Menezes ◽  
Sabrina R Figueiredo ◽  
Fernando Morbeck Almeida Coelho ◽  
Debora R B Terrabuio ◽  
...  

Abstract Context Coronavirus disease 2019 (COVID-19) is a proinflammatory and prothrombotic condition, but its impact on adrenal function has not been adequately evaluated. Case report A 46-year-old woman presented with abdominal pain, hypotension, skin hyperpigmentation after COVID-19 infection. The patient had hyponatremia, serum cortisol <1.0 ug/dL, ACTH of 807 pg/mL and aldosterone <3 ng/dL. Computed tomography (CT) findings of adrenal enlargement with no parenchymal and minimal peripheral capsular enhancement after contrast were consistent with bilateral adrenal infarction. The patient had autoimmune hepatitis and positive antiphospholipid antibodies, but no previous thrombotic events. The patient was treated with intravenous hydrocortisone, followed by oral hydrocortisone and fludrocortisone. Discussion Among 115 articles, we identified nine articles, including case reports, of new-onset adrenal insufficiency and/or adrenal hemorrhage/infarction on CT in COVID-19. Adrenal insufficiency was hormonally diagnosed in five cases, but ACTH levels were measured in only three cases (high in one case and normal/low in other two cases). Bilateral adrenal non- or hemorrhagic infarction was identified in five reports (two had adrenal insufficiency, two had normal cortisol levels and one case had no data). Interestingly, the only case with well-characterized new-onset acute primary adrenal insufficiency after COVID-19 had a previous diagnosis of antiphospholipid syndrome. In our case, antiphospholipid syndrome diagnosis was established only after the adrenal infarction triggered by COVID-19. Conclusions Our findings support the association between bilateral adrenal infarction and antiphospholipid syndrome triggered by COVID-19. Therefore, patients with positive antiphospholipid antibodies should be closely monitored for symptoms or signs of acute adrenal insufficiency during COVID-19.


2018 ◽  
Vol 2018 ◽  
pp. 1-3
Author(s):  
Petros Ioannou ◽  
George Alexakis

Retroperitoneal bleeding is a rare and difficult to diagnose condition, defined as bleeding in the retroperitoneal space without associated trauma or iatrogenic manipulation. It has been associated with hematologic diseases and malignancies and is more common in patients receiving systemic anticoagulation. A 50-year-old man on aspirin presented with abdominal pain. Physical examination revealed abdominal tenderness and a palpable mass at the left abdominal area. An abdominal CT revealed a spontaneous retroperitoneal hematoma due to bleeding of an intraparenchymal branch of the left renal artery. The patient underwent left nephrectomy in order to control the bleeding. Pathology of the kidney showed evidence of acute and chronic microangiopathy, renal artery stenosis, and renal vein thrombosis. Further work-up led to diagnosis of primary antiphospholipid syndrome. Treatment of spontaneous retroperitoneal bleeding varies from conservative in hemodynamically stable patients to invasive or even surgery in hemodynamically unstable patients. In our case, open surgery was performed due to the rapidly deteriorating patient’s condition and the inability to embolize the bleeding vessel by interventional radiology. Physicians should always think of retroperitoneal bleeding in patients presenting with abdominal pain and signs of hypovolemia, especially if they have a bleeding disorder or receive anticoagulants or antiplatelets.


2020 ◽  
Vol 48 (7) ◽  
pp. 030006052090365
Author(s):  
Katarina Mlekuš Kozamernik ◽  
Mojca Jensterle ◽  
Aleš Ambrožič ◽  
Marija Pfeifer

We herein report two cases of primary adrenal insufficiency (AI) associated with antiphospholipid syndrome (APS). In both patients, the main finding that led to the diagnosis was hyponatraemia. The major difference between the two cases was the time at which AI evolved during the course of APS. In the first patient, AI developed acutely along with other presenting features of APS. In the second patient, the AI was unmasked during a stressful situation induced by severe inflammation that occurred 7 years after the first APS manifestation and had probably evolved slowly during the previous few years. These cases emphasise the importance of considering AI in patients with either suspected or newly diagnosed APS as well as in patients who have long been known to have APS. The symptoms and signs alerting the clinician to possible AI are general abdominal complaints, fever, hypotension, and hyponatraemia. Conversely, patients with primary AI should be questioned about the signs and symptoms of APS.


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