ECE2022 Eposter Presentations Adrenal and Cardiovascular Endocrinology (131 abstracts)
Primary adrenal insufficiency due to bilateral adrenal hemorrhage-adrenal infarction in a patient with systemic lupus erythematosus and antiphospholipid syndrome
Katerina Bouki 1 , Vasiliki Venetsanaki 1 , Maria Chrysoulaki 1 , Aikaterini Pateromichelaki 2 , George Bertsias 2 , Vasiliki Daraki 1 , Konstantinos Spanakis 3 , Grigoria Betsi 1 , Nikolaos Sbyrakis 4 , Maria Sfakiotaki 1 , Kalliopi Kontolaimaki 1 , Eleni-Konstantina Syntzanaki 1 , Rodanthi Vamvoukaki 1 , Panagiotis-Nikolaos Tsakalomatis 1 , Prodromos Sidiropoulos 2 & PARASKEVI XEKOUKI 1
1University General Hospital of Heraklion, Endocrinology and Diabetes Clinic, HERAKLION, CRETE, Greece; 2University General Hospital of Heraklion, Rheumatology and Clinical Immunology Clinic, HERAKLION, CRETE, Greece; 3University General Hospital of Heraklion, Radiology Department, Department of Medical Imaging, HERAKLION, CRETE, Greece; 4University General Hospital of Heraklion, Emergency Department, HERAKLION, CRETE, Greece.
Background: Systemic lupus erythematosus (SLE) is an autoimmune disease with multisystem involvement and varied presentation but its association with adrenal insufficiency is rarely reported.
Case presentation: A 30-year-old female presented to the emergency department with fever, lethargy and syncopal episodes during the last 48 h. She complained of general weakness, abdominal pain, nausea, vomiting, weight loss and anorexia for five months. Symptoms deteriorated progressively but were attributed to cholelithiasis for which she underwent a cholecystectomy one month prior, without symptom resolution. She had been hospitalized two years ago due to acute myocarditis and pulmonary embolism and was diagnosed with systematic lupus erythematosus (SLE) and secondary antiphospholipid syndrome (APS). Treatment included hydroxychloroquine, mycophenolic acid (MMF), tinzaparin and periodical corticosteroid treatment. At presentation, physical examination revealed severe hypovolemia, anuria, abdominal tenderness and skin and mucosal hyperpigmentation. Laboratory evaluation revealed anemia, hyperkalemia, hyponatremia, hypercalcemia, troponinemia, prolonged clotting time tests, acute renal insufficiency (serum creatinine:6,5 mg/dl) and pyuria. Primary adrenal insufficiency was suspected and was later confirmed by a low plasma cortisol level (1 μg/dl). She was initially admitted to the Intensive Care Unit (ICU) and treated with fluid resuscitation, continuous hydrocortisone infusion, inotropic agents, antiarrhythmics, anticoagulants, antibiotics and blood transfusions with a good clinical response. Low levels of C3, C4 combined with high titres of SLE-related autoantibodies, lupus anticoagulant and a positive direct Coombs test were suggestive of clinically active SLE. Anti-adrenal antibodies were negative. Adrenal computed tomography performed on the seventh day revealed high density bilateral adrenal enlargement with fluid stranding of the adjacent fat probably due to recent bilateral adrenal haemorrhage. The patient remained in the ICU for three days and was then transferred to the Endocrine and subsequently to Rheumatology Clinic for further treatment. She was discharged 18 days later, on corticosteroids, hydroxychloroquine, MMF, bisoprolol, acenocoumarol and instructions about stress steroid dosing.
Discussion: Bilateral adrenal involvement in our patient was probably a manifestation of a hypercoagulable state due to APS, which can cause adrenal vein thrombosis, followed by adrenal haemorrhagic infarction, although haemorrhage due to tinzaparin therapy cannot be excluded. Addison disease is rare in patients with SLE however, adrenal failure in the presence of elevated antiphospholipid antibodies has been reported. High index of clinical suspicion is required in patients with lupus or APS presenting with unexplained symptoms and adrenal insufficiency should be included in the differential diagnosis.
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Endocrine Abstracts
ISSN 1470-3947 (print) | ISSN 1479-6848 (online)
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