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FRI196 Non-Hemorrhagic Adrenal Infarction in Pregnancy with Tuberous Sclerosis

Disclosure: A. Mahaldar: None. H. Al Jumaili: None. S.R. Campbell: None. M.Z. Baker: None. Background: Non hemorrhagic adrenal infarction is a rare diagnosis in pregnancy with less than 20 cases reported in literature so far. No cases have been reported in patients with tuberous sclerosis. Most pati...

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Detalles Bibliográficos
Autores principales: Mahaldar, Anshinee, Al Jumaili, Hamsa, Campbell, Spencer R, Baker, Mary Zoe
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10555813/
http://dx.doi.org/10.1210/jendso/bvad114.191
Descripción
Sumario:Disclosure: A. Mahaldar: None. H. Al Jumaili: None. S.R. Campbell: None. M.Z. Baker: None. Background: Non hemorrhagic adrenal infarction is a rare diagnosis in pregnancy with less than 20 cases reported in literature so far. No cases have been reported in patients with tuberous sclerosis. Most patients present with abdominal pain. Clinical Case: 29 year old woman G2P1 presented at 35 weeks gestation with right upper quadrant pain. She reported two episodes of dizziness in first trimester but otherwise had an uneventful pregnancy prior to presentation. First pregnancy was uncomplicated. Past medical history included asymptomatic tuberous sclerosis. No previous history of blood clots. Physical examination revealed a distended abdomen appropriate for gestational age and right upper quadrant tenderness to palpation. Initial laboratory data was unremarkable except for leukocytosis with WBC 16.6 k/mm3 (4-11 k/mm3). Normal electrolytes and liver function tests. Abdominal ultrasound revealed cholelithiasis without evidence of acute cholecystitis and unremarkable renal findings. CT abdomen pelvis with contrast showed thickening with surrounding stranding of right adrenal gland suggesting acute adrenal infarction and multiple pancreatic cysts. 8 am cortisol was 4.9 mcg/dl (4.1-16.6 mcg/dl) and ACTH was 23 pg/ml (7.2-63.3 pg/ml). Patient had received 2 doses of betamethasone due to concern for preterm delivery at 48 hours and 36 hours prior to this blood draw. Hypercoagulable panel was sent prior to starting enoxaparin. Elevated Factor VIII >150% (50 to 150%) was noted as expected in pregnancy. Lupus anticoagulant was weakly positive with LA sensitive PTT 50.1 (0 to 51.9 sec). Anticardiolipin antibody and beta 2 glycoprotein antibody were normal. Patient was treated with hydrocortisone 15 mg by mouth daily and full dose enoxaparin for anticoagulation. At one month follow up 8 am cortisol was 24 mcg/dl. Hydrocortisone replacement was discontinued. Labor was uncomplicated with vaginal delivery at 38 weeks gestation. Day 1 postpartum at 7:30 AM cortisol was 20.94 mcg/dl. Patient was discharged home in a stable condition with continued anticoagulation with enoxaparin and no further hydrocortisone treatment. Conclusion: We present a case of non hemorrhagic adrenal infarction in a pregnant patient with tuberous sclerosis. Adrenal infarction without hemorrhage is rare. Anatomy of adrenal gland makes it prone to microvascular thrombosis and infarction in a thrombotic state because of single draining vein for venous drainage. Pregnancy as a hypercoagulable state has been previously associated with non hemorrhagic adrenal infarction in few case reports. Whether tuberous sclerosis played any role in our patient’s pathogenesis remains unclear as there are no data or case reports linking the two. In conclusion, the diagnosis of non hemorrhagic adrenal infarction needs to be considered in hypercoagulable or pregnant patients presenting with abdominal pain. Presentation: Friday, June 16, 2023