Unilateral adrenal infarction in pregnancy with associated acute hypoadrenalism and subsequent spontaneous biochemical and radiological resolution

Authors describe a rare case of acute adrenal infarction in pregnancy resulting in hypoadrenalism requiring hydrocortisone replacement therapy and anticoagulation with subsequent resolution of the radiological changes and complete recovery of the glucocorticoid reserve during clinical follow-up.

Non-Hemorrhagic Adrenal Infarction during Pregnancy: The Diagnostic Imaging Keys

Background: non-hemorrhagic adrenal infarction (NHAI) is a rare cause of acute abdominal/flank pain during pregnancy; in order to ensure prompt and appropriate treatment, this diagnosis should not be overlooked. This case series highlights pertinent imaging findings, including ultrasounds (USs), computed tomography (CT), and magnetic resonance imaging (MRI) of recent NHAI cases. Methods: we compiled all consecutive NHAI cases from two university hospitals over a two-year period and checked the relevant clinical, laboratory, and imaging findings. Relevant articles on NHAI published from January 2010 to March 2021 were analyzed. Results: six cases were found in our database. CT-scans typically showed enlarged, hypodense, and non-enhanced adrenal glands. Unenhanced MRIs allowed for diagnoses and showed enlarged adrenal glands in the signal hyperintensity on T2 and diffusion-weighted imaging, without any signal hyperintensity on T1. In two of our six cases, USs showed swollen adrenal glands with fluid collection. Conclusion: NHAI and its differential diagnosis—in cases of acute pain during pregnancy—highlight the crucial roles of integrated radiological examination and cooperation between obstetricians and radiologists, both of whom should consider the location of the pain, the accessibility and tolerance of MRI, and the radiation exposure of CT. Despite its supposed poor sensitivity, an US performed because the patient reports pain should also be used to examine the adrenal gland regions. Non-enhanced MRI is clearly of value and access to it in emergencies is important to avoid radiation exposure

Primary adrenal insufficiency due to bilateral adrenal infarction in COVID-19: a case report

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.

Left flank pain during pregnancy with an unpredictable etiology: think of nonhemorrhagic adrenal infarction

Abdominal pain is the most presenting complaint during pregnancy with multiple etiologies. The diagnosis could be unpredictable. We present a case of 36-year-old pregnant woman gravida 10 para 7 abortus 2 at 36 + 5 weeks of gestation presenting twice for an increasing left abdominal pain, not relieved despite analgesics. She was delivered for severe oligohydramnios. After delivery, she was found to have a left adrenal infarction on computed tomography scan. She was found to have two mutations of the gene MTHFR 677CC. Our presented case should remind physicians to consider the presence of thromboembolic state during pregnancy. The diagnosis of adrenal infarction should be among the differentials of an ambiguous flank pain that is resilient to medical therapy. Diagnosis in a pregnant patient can be easily confirmed with MRI, after which anticoagulation should be started and the workup for hypercoagulable state investigated.

Acute Adrenal Infarct in a Pregnant Female With Severe Abdominal Pain

Background: The differential diagnosis for pregnant patient presenting with abdominal pain is broad. Adrenal infarction is a rare cause of such presentation. Clinical Case: A 24-year-old woman, 30 weeks pregnant, presented to the emergency room with severe, sharp, left upper quadrant abdominal pain that radiated to the back and was associated with nausea and vomiting. Abdominal exam showed tenderness to light palpation in the left upper area with voluntary guarding. Genitourinary exam revealed a closed cervical os. Abdominal MRI noted a T2 hypointense left adrenal gland with loss of normal diffusion restriction. Edematous changes were identified within the left perinephric and anterior pararenal spaces. Left adrenal gland non-hemorrhagic infarction with necrosis was diagnosed, and patient was started on enoxaparin. Patient was admitted for hydration and pain control. A morning serum cortisol was equivocal at 10.3 µg/dL with a mid-normal ACTH of 38.9 pg/mL. ACTH stimulation noted a suboptimal cortisol response with a serum cortisol at baseline of 9.1 µg/dL, at 30 minutes 10.7 µg/dL, and at 60 minutes 11.0 µg/dL. Patient was started on stress dose intravenous hydrocortisone. Evaluation for thrombophilia was undertaken and patient initiated on therapeutic weight-based enoxaparin. Investigations for etiology included normal factor V leiden, proteins C and S activity, antithrombin III antigen, antiphospholipid screening, C3 and C4 complement, anti-double stranded DNA antibody, anti-neutrophil cytoplasmic antibody, cryoglobulin, homocysteine, and factor II mutation. Factor VIII activity was increased >200%, which is within expected range during pregnancy. Patient was heterozygous for MTHFR C677T and A1298C mutations. Hydrocortisone was tapered and patient discharged on a physiologic dose, in addition to enoxaparin. Conclusion: Unilateral adrenal infarction is an uncommon entity in general, and even more uncommon during pregnancy (1). Thrombophilia evaluation is warranted as antiphospholipid syndrome and MTHFR gene mutations have been associated with this condition. Given the rarity of this disease, it may not be included in the differential diagnosis of pregnant patients who present with acute abdominal pain. MRI is superior to other imaging modalities due to its sensitivity in detecting adrenal infarct and even hemorrhage, if present. Unfortunately, there are no guideline on managing such patients, but the focus is usually placed on anticoagulation to prevent the infarction of the contralateral adrenal gland, with cautious monitoring as hemorrhagic conversion is possible. Screening patient for adrenal insufficiency for prompt supplementation of corticosteroids if needed is necessary, while attempting to find the underlying cause. Reference: (1) Green P., et al. Unilateral adrenal infarction in pregnancy. BMJ Case reports. 2013; bcr2013009997

659 Unilateral Infarction of Adrenal Gland During Pregnancy: A Case Report and Important Learning Point

Background Abdominal pain in pregnancy offers a challenge for the General surgeon. Unilateral adrenal infarction is an extremely rare event. Case presentation: We report the case of an obese 23-year-old pregnant female G4 P1 + 2 diagnosed with unilateral adrenal infarction and hemorrhage. She presented at 34 + 5 weeks with severe RUQ and flank pain under the obstetric team. After initially being discharged, she later returned with intractable pain. Examination and initial investigations (USS KUB and abdomen) were inconclusive. She was empirically treated for pylonephritis. During the night she underwent emergency c-section due to uncontrollable pain. Subsequently, a CT abdomen demonstrated a unilateral right adrenal infarct with hemorrhage. Discussion: Unilateral adrenal infarction often leads to a delay in diagnosis and should be part of differential diagnosis in patients with severe RUQ pain (1.3%). It is a rare event diagnosed generally by CT or MRI scan showing attenuation or swelling of adrenal glands. Physiologically it is due to the state of venous circulation during pregnancy alongside others coagulation risks factor. Learning point: To consider adrenal infarction with severe RUQ pain when first line diagnosis normal. Key Investigations: MRI during pregnancy and CT AP after. Discussion in MDT and screen for coagulation disorder.