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Diagnosis and surgical treatment of primary aldosteronism in pregnancy: A case report
DIAGNOSIS
AND
TREATMENT
SURGICAL
OF PRIMARY
ALDOSTERONISM
IN PREGNANCY:
A
CASE REPORT
Racl\,q~zr~r~f: Aldosterone-produci1~g adrenal adenomas are rare, especially during pregnancy. We report a patient who presented in the early second trimester, was diagnosed with primary aldosteronism, and was treated successfully by adrenalectomy. Ctrsc~ A 77-year-old black nulliparous woman was found to have a blood pressure (BP) of 150/82 mmHg when she registered for prenatal care at 13 weeks’ gestation. Initial laboratory assessment revealed a markedly diminished serum potassium level of 2.1 mmolil. Further laboratory evaluation detected decreased random plasma renin activity and an elevated aldosteronc level. Magnetic resonance imaging revealed a 2-cm right adrenal lesion. She was diagnosed with an adrenal adenoma and successfully underwent an adrenalectomy at 17 weeks’ gestation. Postoperatively, her BP and serum potassium level normalized. She spontaneously delivered a normal male infant at term. Co~rrlrrsio~z: Although primary hyperaldosteronism is a rare clinical entity, it must be considered when hypertension and hvpokalemia are present concurrently. Antepartum medical management can be difficult, often resulting in poor obstetric outcome. Surgery in the second trimester is an effective option. COl?str’f Gl/~rc~~-~~l 199T;86:6N-5)
Sebtatlon. Initial laboratory assessment revealed hypokalemia, and she IVas admitted to our antepartum unit for further cvaluatiun. Before pregnancy, she had been told that she had nlild hvpertension, but she had never taken antihypertenslve medications. The patient M:as asymptomatic except for occa-ional mild headaches; she denied muscle weakness or fatigue. Her mother and maternal grandmother both have hypertcnilon, and the grandmother also has renal disease. On ,1dmission, her blood pressure (BP) was 150/82 mmHg, ,tnd her pulse rate was 94 beats per minute; she weighed 154 pounds. Funduscopic and cardiovascular examinations were normal L,terine size 1t.a~ appropriate for gestational age, and fet,ll heart tones were present. There was no peripheral edema. l,ahoratorv tests identified a low serum potassium level (2.1 mmol/l. 1 and mild (I+) proteinurla on urinalysis. Ilc>r 131’ M’~S measured frequently during her inpatient ~~~dlu,iti~ll~ , and it initially ranged between 120/80 and 15O/YO mmHg. The serum potassium level was resistant to ,Iggressivcn supplementation but was finally maintained at ,1bout 3.2 nimoI/L on a regimen of I20 mEq oral potassium l’c’r dnv. ;\ 24.hour urine collection showed elevations of protein (286 mg yer day, normal 10100); sodium (270 mEq per da\,, normal N-180); and potassium (68 mEq per day, normal -K-X0, but rzith Ic,~v serum potassium, normal less than 23). :\ 24.hour urlnarv determination of catecholamine c)utput and crcatinine clearance ~‘as normal. A morning ,erum corti\c)l concentration was 14.4 Fg/dL, which was in thr ntrrm~~l range. Random plasma renin activity was de~.rc~,l~t~d (I .O q/ml. per hour, normal pregnancy values 5.6 I?-), ,~nd her aldosterone level was elevated (90 ng/dL; norm,ll pregnancy values Ih-60’). A saline fluid challenge of 7 L Jminlbtcred intra\,enously over 4 hours failed to suppress the ,lld~l~tt~rone level; it lva? 13h.J ng/dL after the test. There 11 ai no change in aldosterane concentration or plasma renin ,iitivltv in response to change in posture from supine to sitting position. hlagnctic resonance imaging revealed a 2-cm right ,idrcnal I~YIOII, consistent with the clinical suspicion of adrenal ,ldcnom,~. Before surgery, the patient was treated with labetalol 100 mg orally everv 1 2 hours to control her worsening hvpcrtcnslon. Tht> patlent underwent a right adrenalectomy, without ~~omplicntlon, at 17 wrecks’ gestation. A 2 X 3-cm right adrenal ,ldenoma \\as identified. She did well postoperatively without I,lbt?alc~l; her BP normalized, and her serum potassium ler,el rose ~pont~~neously to 3.1 mmol/L. She was discharged on the tourth postoperative day. The remainder of her antepartum C‘OLI r\e \\.a5 unremarkable until 41 weeks’ gestation, at which time, iht‘ dt>veloped mild preeclampsin. Labor was induced, end she sp<>ntaneousl!, delivered a 3250-g male infant with t\pg~ir >cores of 9 at 1 and 5 minutes, respectively. The postpartum and netmatal courses were uncomplicated.
I’rimarv which fested mid.
aldosteronism autonomous
is a rare secretion
as hypertension, Svmptoms
include
of
clinical
syndrome
aldosterone
hypokalemia,
and
headache,
fatigue,
is manihyporenineweakness,
in
dizziness, and muscle c.ramps. The estimated pre\‘+ lence in hypertensive adults is about 1’7.” This syndrome has been di\+ded into several etiologies: aldosterone-producing adenoma; adrt>nc)cortical carcinoma; idiopathic hyperaldnstcr~)nislll (considered to be due to bilateral cortical hyperplasia); glucocorticoid-rcmudiable hyperaldosteronisIn; and “indeterminate hypernldosteronism.““ Ninety-ii\.c percent of primary hyperaldosteronism cases drt’ due t<> ;Ild,)ster,)ll,-rr’tducing adenomn or idiopathic h\~~~r~~ld~~sterc,nism. Each has a distinct therapy. Acieno~n~ responds well to surgical excision, but idiopathic hv~~t~r~~ldosteronism requlreb life-long medical therapy. The laborator\; diqnosis (,t prim~~ry aldosteronism is made bv finding high sccrt,tor\, rates of aldosteronc, together with 1~2. pl,isnia rcnln ~~alurs. Additional confirmation can l3e obtalncd bv saline loading to evaluate the suppressibillt\- c~f plasma aldosterone. To distinguish between ~i~cncmia’ diid h~pt~rpIa&i, pitstural response studies drc pt~riornied. Adenoma shoM3 a decreasein morning aldo5tcronc le\rcls in responseto upright posture, but aldostcront, Ie\~ls increase in upright patients with adrenal hvperplasia. Magnetic resoiiaiice imaging enables the localization of the tumor and provides an accurate LIihtinction het\\ven these tr1.o entities. The obstetric litcr~lturt~ on l~~~~~t~r~~lclostcronisrn conL?iris multiple
reporti
0i ddrt~n,iI
adcnomns
managed
conservativelv during pregnanc\i and followed by postpartum adrenalectomy. :\\ in liur case, hypcrtensicln and hypokalemia often become mc)redifficult to treat ‘1s the prcgnanq. progresses, ’ ” but in one case, the hypertension and hypokalemia improved during prqnancy, onlv to Lt’orsenagain postpartum.’ The mainstav of medical management is potasium supplrment,~tion with antitivrprtcnsivc mcdication5. Spironolactone or similar ,llclostcronc antagoni3ts art’ used preciominnntlv,’ ~” hut Idbt~talol,’ a mixed nlphr3and beeta-adrencrgicagent, ,lnd t~nalapril,” an inhibitc,r of the angiotensin con\,crting t’n/vmt’, ha1.ebeen us;cd to treat patients a> r\~ll. Ho\\t,\,er, the potential effects of medication on the dc>\t’loping fetus are a concern. Spironolactonc is known to h;lve antiandrogenic effects in humans and to caustx fcmIni/~ition in male rat fetuses.’ ’ Enalapril may be rc*sp~~nGblc for oligohydramnlos and neonatal renal iailurt~ Fetal morbidity and m(~I-talltv are OI utmost importance if conservati\,t> man~igtmt~nt is to btx considered. The first reported crlse at ?h r\,eeks’
Neerhof et al”’ reported a cesarean delivery for fetal distress from abruptio placentae at 26 weeks’ gestation. Surgery is the treatment of choice for adrenal adenoma. After unilateral adrenalectomy, excellent BP response has been reported; 70-89s of patients were normotensive, and the remainder showed improvement after surgery.“-‘” Thus, it would seemreasonable to consider proceeding directly with surgery in the late first or early second trimester, rather than to attempt medical management.
I. Craw MC;, Andeb JP, Harris JJ,Slate WC. Primary aldosteronism 11,t-‘regnancy. Obstet C;ynecol 1964;23:200-8. 2 Biglicri EC, Slaton PE. Pregnancy and primary aldosteronism. J Clin tndocrmol Metab 1967;2i:l628-32. 3 Eltcrman [J, Hagen AA. Aldosteronism in pregnancy: Association with \ irilization of fcmnle offsprmg. South Med J 1983;76:514+6. 4. Mcrrlll RH, Dombrowski RA. MacKenna JM. Primary hyperaldo,tcr(mism during pregnancy. Am 1 Obstet Gynecol 1984;350:786-7. ‘7. Lotgermg FK, Drrkx FCI, Wnllenburg HC. Primary hyperaldosteron~~m in pregnancy. Am J Obstet Gynecol 1986;155:986-8. 6 Gordon KD, FIshman LM, Liddle CW. Plasma renin activity and aldostrrone secretion m a pregnant woman with primary aldoster,,msm. J Clin Endocrmtrl Metab 1967;27:385-8. 7 iV11wn M, Morganti AA, Zervoudakis J. et al. Blood pressure, the r~nln-,lldosterone system and sex steroids throughout normal p”~“‘~““. Am 1 .bled 1Y80;68:Y7-104. 8. Brnxu EL. Primary aldosteronlsm. Ural Clin korth Am 1989;16: 4X1-6 Y. Row FW, Bosco I A, Graham CF, Milstien JB, Dreis M, Creamer J. Ueclnatal anuria with maternal angiotensin-converting enzyme Inhibition. Obstet Gynecol 3989;74:371-4. IO Newhot MG, Shlossman PA, Poll DS, Ludomirsky A, Weber S. Idlopathlc aldosteronism m pregnancy. Obstet Gynecol 1991;78: 484 -91 I I. \/kGna M, Kff~pandi I’, Ghiga E, ct al. Possible contraindication ,,f ~pironolactone during pregnancy. J Endocrinol Invest 1979;2: 222.-3 I2 Grant CS, Carpenter P. van Heerden JA, Hamberger B. Primary aldo~tcronisn~: Clinical management. Arch Surg 1984;11:585-90. 11 Lins PI:. Adamsot L. Pnmar~ aldvsteronism. A follow-up study of 78 cI1s,c> of surgically treated aldosterone-prc,dLlcing adenomas. Acta kltd Scam1 1987.22127582
Coyvrlght C IYYS by The CT\ niwlogi~ts.
American
College
of Obstetricians
and
AND
TREATMENT
SURGICAL
OF PRIMARY
ALDOSTERONISM
IN PREGNANCY:
A
CASE REPORT
Racl\,q~zr~r~f: Aldosterone-produci1~g adrenal adenomas are rare, especially during pregnancy. We report a patient who presented in the early second trimester, was diagnosed with primary aldosteronism, and was treated successfully by adrenalectomy. Ctrsc~ A 77-year-old black nulliparous woman was found to have a blood pressure (BP) of 150/82 mmHg when she registered for prenatal care at 13 weeks’ gestation. Initial laboratory assessment revealed a markedly diminished serum potassium level of 2.1 mmolil. Further laboratory evaluation detected decreased random plasma renin activity and an elevated aldosteronc level. Magnetic resonance imaging revealed a 2-cm right adrenal lesion. She was diagnosed with an adrenal adenoma and successfully underwent an adrenalectomy at 17 weeks’ gestation. Postoperatively, her BP and serum potassium level normalized. She spontaneously delivered a normal male infant at term. Co~rrlrrsio~z: Although primary hyperaldosteronism is a rare clinical entity, it must be considered when hypertension and hvpokalemia are present concurrently. Antepartum medical management can be difficult, often resulting in poor obstetric outcome. Surgery in the second trimester is an effective option. COl?str’f Gl/~rc~~-~~l 199T;86:6N-5)
Sebtatlon. Initial laboratory assessment revealed hypokalemia, and she IVas admitted to our antepartum unit for further cvaluatiun. Before pregnancy, she had been told that she had nlild hvpertension, but she had never taken antihypertenslve medications. The patient M:as asymptomatic except for occa-ional mild headaches; she denied muscle weakness or fatigue. Her mother and maternal grandmother both have hypertcnilon, and the grandmother also has renal disease. On ,1dmission, her blood pressure (BP) was 150/82 mmHg, ,tnd her pulse rate was 94 beats per minute; she weighed 154 pounds. Funduscopic and cardiovascular examinations were normal L,terine size 1t.a~ appropriate for gestational age, and fet,ll heart tones were present. There was no peripheral edema. l,ahoratorv tests identified a low serum potassium level (2.1 mmol/l. 1 and mild (I+) proteinurla on urinalysis. Ilc>r 131’ M’~S measured frequently during her inpatient ~~~dlu,iti~ll~ , and it initially ranged between 120/80 and 15O/YO mmHg. The serum potassium level was resistant to ,Iggressivcn supplementation but was finally maintained at ,1bout 3.2 nimoI/L on a regimen of I20 mEq oral potassium l’c’r dnv. ;\ 24.hour urine collection showed elevations of protein (286 mg yer day, normal 10100); sodium (270 mEq per da\,, normal N-180); and potassium (68 mEq per day, normal -K-X0, but rzith Ic,~v serum potassium, normal less than 23). :\ 24.hour urlnarv determination of catecholamine c)utput and crcatinine clearance ~‘as normal. A morning ,erum corti\c)l concentration was 14.4 Fg/dL, which was in thr ntrrm~~l range. Random plasma renin activity was de~.rc~,l~t~d (I .O q/ml. per hour, normal pregnancy values 5.6 I?-), ,~nd her aldosterone level was elevated (90 ng/dL; norm,ll pregnancy values Ih-60’). A saline fluid challenge of 7 L Jminlbtcred intra\,enously over 4 hours failed to suppress the ,lld~l~tt~rone level; it lva? 13h.J ng/dL after the test. There 11 ai no change in aldosterane concentration or plasma renin ,iitivltv in response to change in posture from supine to sitting position. hlagnctic resonance imaging revealed a 2-cm right ,idrcnal I~YIOII, consistent with the clinical suspicion of adrenal ,ldcnom,~. Before surgery, the patient was treated with labetalol 100 mg orally everv 1 2 hours to control her worsening hvpcrtcnslon. Tht> patlent underwent a right adrenalectomy, without ~~omplicntlon, at 17 wrecks’ gestation. A 2 X 3-cm right adrenal ,ldenoma \\as identified. She did well postoperatively without I,lbt?alc~l; her BP normalized, and her serum potassium ler,el rose ~pont~~neously to 3.1 mmol/L. She was discharged on the tourth postoperative day. The remainder of her antepartum C‘OLI r\e \\.a5 unremarkable until 41 weeks’ gestation, at which time, iht‘ dt>veloped mild preeclampsin. Labor was induced, end she sp<>ntaneousl!, delivered a 3250-g male infant with t\pg~ir >cores of 9 at 1 and 5 minutes, respectively. The postpartum and netmatal courses were uncomplicated.
I’rimarv which fested mid.
aldosteronism autonomous
is a rare secretion
as hypertension, Svmptoms
include
of
clinical
syndrome
aldosterone
hypokalemia,
and
headache,
fatigue,
is manihyporenineweakness,
in
dizziness, and muscle c.ramps. The estimated pre\‘+ lence in hypertensive adults is about 1’7.” This syndrome has been di\+ded into several etiologies: aldosterone-producing adenoma; adrt>nc)cortical carcinoma; idiopathic hyperaldnstcr~)nislll (considered to be due to bilateral cortical hyperplasia); glucocorticoid-rcmudiable hyperaldosteronisIn; and “indeterminate hypernldosteronism.““ Ninety-ii\.c percent of primary hyperaldosteronism cases drt’ due t<> ;Ild,)ster,)ll,-rr’tducing adenomn or idiopathic h\~~~r~~ld~~sterc,nism. Each has a distinct therapy. Acieno~n~ responds well to surgical excision, but idiopathic hv~~t~r~~ldosteronism requlreb life-long medical therapy. The laborator\; diqnosis (,t prim~~ry aldosteronism is made bv finding high sccrt,tor\, rates of aldosteronc, together with 1~2. pl,isnia rcnln ~~alurs. Additional confirmation can l3e obtalncd bv saline loading to evaluate the suppressibillt\- c~f plasma aldosterone. To distinguish between ~i~cncmia’ diid h~pt~rpIa&i, pitstural response studies drc pt~riornied. Adenoma shoM3 a decreasein morning aldo5tcronc le\rcls in responseto upright posture, but aldostcront, Ie\~ls increase in upright patients with adrenal hvperplasia. Magnetic resoiiaiice imaging enables the localization of the tumor and provides an accurate LIihtinction het\\ven these tr1.o entities. The obstetric litcr~lturt~ on l~~~~~t~r~~lclostcronisrn conL?iris multiple
reporti
0i ddrt~n,iI
adcnomns
managed
conservativelv during pregnanc\i and followed by postpartum adrenalectomy. :\\ in liur case, hypcrtensicln and hypokalemia often become mc)redifficult to treat ‘1s the prcgnanq. progresses, ’ ” but in one case, the hypertension and hypokalemia improved during prqnancy, onlv to Lt’orsenagain postpartum.’ The mainstav of medical management is potasium supplrment,~tion with antitivrprtcnsivc mcdication5. Spironolactone or similar ,llclostcronc antagoni3ts art’ used preciominnntlv,’ ~” hut Idbt~talol,’ a mixed nlphr3and beeta-adrencrgicagent, ,lnd t~nalapril,” an inhibitc,r of the angiotensin con\,crting t’n/vmt’, ha1.ebeen us;cd to treat patients a> r\~ll. Ho\\t,\,er, the potential effects of medication on the dc>\t’loping fetus are a concern. Spironolactonc is known to h;lve antiandrogenic effects in humans and to caustx fcmIni/~ition in male rat fetuses.’ ’ Enalapril may be rc*sp~~nGblc for oligohydramnlos and neonatal renal iailurt~ Fetal morbidity and m(~I-talltv are OI utmost importance if conservati\,t> man~igtmt~nt is to btx considered. The first reported crlse
Neerhof et al”’ reported a cesarean delivery for fetal distress from abruptio placentae at 26 weeks’ gestation. Surgery is the treatment of choice for adrenal adenoma. After unilateral adrenalectomy, excellent BP response has been reported; 70-89s of patients were normotensive, and the remainder showed improvement after surgery.“-‘” Thus, it would seemreasonable to consider proceeding directly with surgery in the late first or early second trimester, rather than to attempt medical management.
I. Craw MC;, Andeb JP, Harris JJ,Slate WC. Primary aldosteronism 11,t-‘regnancy. Obstet C;ynecol 1964;23:200-8. 2 Biglicri EC, Slaton PE. Pregnancy and primary aldosteronism. J Clin tndocrmol Metab 1967;2i:l628-32. 3 Eltcrman [J, Hagen AA. Aldosteronism in pregnancy: Association with \ irilization of fcmnle offsprmg. South Med J 1983;76:514+6. 4. Mcrrlll RH, Dombrowski RA. MacKenna JM. Primary hyperaldo,tcr(mism during pregnancy. Am 1 Obstet Gynecol 1984;350:786-7. ‘7. Lotgermg FK, Drrkx FCI, Wnllenburg HC. Primary hyperaldosteron~~m in pregnancy. Am J Obstet Gynecol 1986;155:986-8. 6 Gordon KD, FIshman LM, Liddle CW. Plasma renin activity and aldostrrone secretion m a pregnant woman with primary aldoster,,msm. J Clin Endocrmtrl Metab 1967;27:385-8. 7 iV11wn M, Morganti AA, Zervoudakis J. et al. Blood pressure, the r~nln-,lldosterone system and sex steroids throughout normal p”~“‘~““. Am 1 .bled 1Y80;68:Y7-104. 8. Brnxu EL. Primary aldosteronlsm. Ural Clin korth Am 1989;16: 4X1-6 Y. Row FW, Bosco I A, Graham CF, Milstien JB, Dreis M, Creamer J. Ueclnatal anuria with maternal angiotensin-converting enzyme Inhibition. Obstet Gynecol 3989;74:371-4. IO Newhot MG, Shlossman PA, Poll DS, Ludomirsky A, Weber S. Idlopathlc aldosteronism m pregnancy. Obstet Gynecol 1991;78: 484 -91 I I. \/kGna M, Kff~pandi I’, Ghiga E, ct al. Possible contraindication ,,f ~pironolactone during pregnancy. J Endocrinol Invest 1979;2: 222.-3 I2 Grant CS, Carpenter P. van Heerden JA, Hamberger B. Primary aldo~tcronisn~: Clinical management. Arch Surg 1984;11:585-90. 11 Lins PI:. Adamsot L. Pnmar~ aldvsteronism. A follow-up study of 78 cI1s,c> of surgically treated aldosterone-prc,dLlcing adenomas. Acta kltd Scam1 1987.22127582
Coyvrlght C IYYS by The CT\ niwlogi~ts.
American
College
of Obstetricians
and