Cargando…
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...
Autores principales: | , , , |
---|---|
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 |
_version_ | 1785116741249007616 |
---|---|
author | Mahaldar, Anshinee Al Jumaili, Hamsa Campbell, Spencer R Baker, Mary Zoe |
author_facet | Mahaldar, Anshinee Al Jumaili, Hamsa Campbell, Spencer R Baker, Mary Zoe |
author_sort | Mahaldar, Anshinee |
collection | PubMed |
description | 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 |
format | Online Article Text |
id | pubmed-10555813 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | Oxford University Press |
record_format | MEDLINE/PubMed |
spelling | pubmed-105558132023-10-07 FRI196 Non-Hemorrhagic Adrenal Infarction in Pregnancy with Tuberous Sclerosis Mahaldar, Anshinee Al Jumaili, Hamsa Campbell, Spencer R Baker, Mary Zoe J Endocr Soc Adrenal (Excluding Mineralocorticoids) 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 Oxford University Press 2023-10-05 /pmc/articles/PMC10555813/ http://dx.doi.org/10.1210/jendso/bvad114.191 Text en © The Author(s) 2023. Published by Oxford University Press on behalf of the Endocrine Society. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs licence (https://creativecommons.org/licenses/by-nc-nd/4.0/), which permits non-commercial reproduction and distribution of the work, in any medium, provided the original work is not altered or transformed in any way, and that the work is properly cited. For commercial re-use, please contact journals.permissions@oup.com |
spellingShingle | Adrenal (Excluding Mineralocorticoids) Mahaldar, Anshinee Al Jumaili, Hamsa Campbell, Spencer R Baker, Mary Zoe FRI196 Non-Hemorrhagic Adrenal Infarction in Pregnancy with Tuberous Sclerosis |
title | FRI196 Non-Hemorrhagic Adrenal Infarction in Pregnancy with Tuberous Sclerosis |
title_full | FRI196 Non-Hemorrhagic Adrenal Infarction in Pregnancy with Tuberous Sclerosis |
title_fullStr | FRI196 Non-Hemorrhagic Adrenal Infarction in Pregnancy with Tuberous Sclerosis |
title_full_unstemmed | FRI196 Non-Hemorrhagic Adrenal Infarction in Pregnancy with Tuberous Sclerosis |
title_short | FRI196 Non-Hemorrhagic Adrenal Infarction in Pregnancy with Tuberous Sclerosis |
title_sort | fri196 non-hemorrhagic adrenal infarction in pregnancy with tuberous sclerosis |
topic | Adrenal (Excluding Mineralocorticoids) |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10555813/ http://dx.doi.org/10.1210/jendso/bvad114.191 |
work_keys_str_mv | AT mahaldaranshinee fri196nonhemorrhagicadrenalinfarctioninpregnancywithtuberoussclerosis AT aljumailihamsa fri196nonhemorrhagicadrenalinfarctioninpregnancywithtuberoussclerosis AT campbellspencerr fri196nonhemorrhagicadrenalinfarctioninpregnancywithtuberoussclerosis AT bakermaryzoe fri196nonhemorrhagicadrenalinfarctioninpregnancywithtuberoussclerosis |