TY - JOUR
T1 - Pregnancy and phaeochromocytoma/paraganglioma: clinical clues affecting diagnosis and outcome - a systematic review
AU - Langton, K.
AU - Tufton, N.
AU - Akker, S.
AU - Deinum, J.
AU - Eisenhofer, G.
AU - Timmers, H.J.L.M.
AU - Spaanderman, M.E.A.
AU - Lenders, J.W.M.
N1 - Funding Information:
J.W.M. Lenders and G. Eisenhofer were supported by the Deutsche Forschungsgemeinschaft (DFG) within the CRC/Transregio 205/1, project B12, ?The Adrenal: Central Relay in Health and Disease?. The authors thank Dr Sander M.?J. van Kuijk of the Department of Clinical Epidemiology and Medical Technology Assessment, Maastricht University Medical Centre, Maastricht, The Netherlands for his statistical advice. Open access funding enabled and organized by ProjektDEAL.
Funding Information:
J.W.M. Lenders and G. Eisenhofer were supported by the Deutsche Forschungsgemeinschaft (DFG) within the CRC/Transregio 205/1, project B12, ‘The Adrenal: Central Relay in Health and Disease’.
Publisher Copyright:
© 2020 The Authors. BJOG: An International Journal of Obstetrics and Gynaecology published by John Wiley & Sons Ltd
PY - 2021/7
Y1 - 2021/7
N2 - Background Phaeochromocytoma and paraganglioma (PPGL) in pregnancy, if not diagnosed antepartum, pose a high risk for mother and child.Objective To examine the clinical clues of antepartum and postpartum/postmortem diagnosis of PPGL.Search strategy Case reports on PPGL in pregnancy published between 1 January 1988 and 30 June 2019 in English, German, Dutch or French.Selection criteria Case reports containing a predefined minimum of clinical data on PPGL and pregnancy.Data collection and analysis Two authors independently performed data extraction and assessed data quality. We calculated odds ratios (OR) (with 95% confidence intervals) and used uni- and multivariable logistic regression analysis.Main results Maternal and fetal/neonatal mortalities were 9.0% (18/200) and 14.2% (29/204), respectively. Maternal mortality was 42-fold higher with PPGL diagnosed postpartum/postmortem (17/58; 29.3%) than antepartum (1/142; 0.7%) (adjusted OR 45.9, 95% CI 5.67-370, P = 0.0003). Offspring mortality was 2.6-fold higher with PPGL diagnosed postpartum/postmortem than antepartum (OR 3.1, 95% CI 1.38-6.91, P = 0.0044). Hypertension at admission (OR 2.29, 95% CI 1.12-4.68, P = 0.022), sweating (OR 3.14, 95% CI 1.29-7.63, P = 0.014) and a history of PPGL, a known PPGL-associated gene mutation or adrenal mass (OR 8.87, 95% CI 1.89-41.64, P = 0.0056) were independent factors of antepartum diagnosis. Acute onset of symptoms (OR 8.49, 95% CI 3.52-20.5, P < 0.0001), initial diagnosis of pre-eclampsia (OR 6.34, 95% CI 2.60-15.5, P < 0.0001), admission for obstetric care (OR 10.71, 95% CI 2.70-42.45, P = 0.0007) and maternal tachycardia (OR 2.72, 95% CI 1.26-5.85, P = 0.011) were independent factors of postpartum diagnosis.Conclusion Several clinical clues can assist clinicians in considering an antenatal diagnosis of PPGL in pregnancy, thus potentially improving outcome.Tweetable abstract Systematic review of 204 pregnant patients with phaeochromocytoma identified clinical clues for a timely antepartum diagnosis.
AB - Background Phaeochromocytoma and paraganglioma (PPGL) in pregnancy, if not diagnosed antepartum, pose a high risk for mother and child.Objective To examine the clinical clues of antepartum and postpartum/postmortem diagnosis of PPGL.Search strategy Case reports on PPGL in pregnancy published between 1 January 1988 and 30 June 2019 in English, German, Dutch or French.Selection criteria Case reports containing a predefined minimum of clinical data on PPGL and pregnancy.Data collection and analysis Two authors independently performed data extraction and assessed data quality. We calculated odds ratios (OR) (with 95% confidence intervals) and used uni- and multivariable logistic regression analysis.Main results Maternal and fetal/neonatal mortalities were 9.0% (18/200) and 14.2% (29/204), respectively. Maternal mortality was 42-fold higher with PPGL diagnosed postpartum/postmortem (17/58; 29.3%) than antepartum (1/142; 0.7%) (adjusted OR 45.9, 95% CI 5.67-370, P = 0.0003). Offspring mortality was 2.6-fold higher with PPGL diagnosed postpartum/postmortem than antepartum (OR 3.1, 95% CI 1.38-6.91, P = 0.0044). Hypertension at admission (OR 2.29, 95% CI 1.12-4.68, P = 0.022), sweating (OR 3.14, 95% CI 1.29-7.63, P = 0.014) and a history of PPGL, a known PPGL-associated gene mutation or adrenal mass (OR 8.87, 95% CI 1.89-41.64, P = 0.0056) were independent factors of antepartum diagnosis. Acute onset of symptoms (OR 8.49, 95% CI 3.52-20.5, P < 0.0001), initial diagnosis of pre-eclampsia (OR 6.34, 95% CI 2.60-15.5, P < 0.0001), admission for obstetric care (OR 10.71, 95% CI 2.70-42.45, P = 0.0007) and maternal tachycardia (OR 2.72, 95% CI 1.26-5.85, P = 0.011) were independent factors of postpartum diagnosis.Conclusion Several clinical clues can assist clinicians in considering an antenatal diagnosis of PPGL in pregnancy, thus potentially improving outcome.Tweetable abstract Systematic review of 204 pregnant patients with phaeochromocytoma identified clinical clues for a timely antepartum diagnosis.
KW - fetal
KW - maternal
KW - medical condition in pregnancy
KW - mortality
KW - paraganglioma
KW - phaeochromocytoma
KW - pregnancy
KW - signs
KW - symptoms
KW - Fetal
U2 - 10.1111/1471-0528.16635
DO - 10.1111/1471-0528.16635
M3 - (Systematic) Review article
C2 - 33342020
SN - 1470-0328
VL - 128
SP - 1264
EP - 1272
JO - Bjog-an International Journal of Obstetrics and Gynaecology
JF - Bjog-an International Journal of Obstetrics and Gynaecology
IS - 8
ER -