- Email: [email protected]
Hyperparathyrodism and pancreatitis during pregnancy
Brief clinical reports Hyperparathyroidism and pancreatitis during pregnancy William B. Inabnet, MD, David Baldwin, MD, Ronald O. Daniel, MD, and Edgar D. Staren, MD, Phi), Chicago, Ill. From the Departments of General Surgery, Internal Medicine, and Obstetrics and Gynecology, Rush-Presbyterian-St. Luke's Medical Center, Chicago, Ill.
PRIMARYHYPERPARATHYROIDISMIS rare d u r i n g pregnancy with slightly more than 100 cases reported in the English-language literature between 1930 a n d 1990.1 Associated fetal morbidity a n d mortality are high with a perinatal complication rate reaching 50%. 2, 3 Pancreatids is also u n c o m m o n d u r i n g pregnancy, occurring in less than 1% of all pregnancies. 4 Maternal mortality may be as high as 37%, a n d when occurring late in pregnancy, pancreatitis may lead to premature labor in up to 60% of patients. 4 The simultaneous occurrence of hyperparathyroidism a n d pancreatitis in pregnancy has only b e e n reported eight times. 5-12We present a case of c o n c u r r e n t hyperparathyroidism a n d pancreatitis in pregnancy masquerading as hyperemesis gravidarum. A review of the literature is provided, along with guidelines for diagnosis a n d treatment.
CASE R E P O R T The patient was a 27-year-old, gravida 1 para 0 woman who presented to her physician at 8-weeks' gestation with complaints of excessive nausea, vomiting, weakness, and a 14pound weight loss. She was diagnosed with hyperemesis gravidarum and treated initially as an outpatient with antiemetic therapy. Three weeks later she complained of new onset abdominal pain radiating to her back, and she was referred to our institution for admission. Admission laboratory studies were as follows: albumin, 2.4 units/L; calcium, 10.5 mg/dl; phosphorus, 2.1 mg/dl; amylase, 490 units/L; and lipase, 5517 units/L. An abdominal ultrasonogram showed an edematous, enlarged pancreas and gallbladder sludge, but no evidence of gallstones or bile duct dilatation was seen. The patient was diagnosed with pancreatitis and treated with bowel rest, vigorous hydration, and nutritional support. Her serum calcium level ranged from 9.8 to 10.5 mg/dl, with the ionized calcium level Accepted for publicationDec. 20, 1994. Reprint requests: Edgar D. Staren, MD, PhD, Departmentof General Surgery, Rush-Presbyterian-StLuke's Medical Center, 1653W. Congress Pkwy,Chicago, IL 60612. Surgery 1996;119:710-3. Copyright 9 1996 by Mosby-YearBook, Inc. 0039-6060/96/$5.00 + 0 11/57/72869 710
SURGERY
remaining poorly controlled despite medical therapy (1.47 to 1.83 mmol/L). An intact parathyroid hormone levelwas found to be 97 pg/ml (normal, 30 to 71 pg/ml). With the presumptive diagnosis of hyperparathyroidism, the patient underwent a parathyroidectomy for a parathyroid adenoma during her nineteenth week of pregnancy (Figure). Twelve hours after the procedure the ionized calcium level normalized at 1.2 mmol/L. Her amylase and lipase levels rapidly normalized during the next week, and the remainder of her postoperative course was unremarkable. She was discharged to home on a general diet, and after 40 weeks of pregnancy she delivered a healthy 7-pound boy with APGAR scores of 8 and 9, respectively. Two years after her initial presentation the patient again presented with abdominal pain that radiated to her back. Admitring laboratory data showed elevated amylase and lipase levels, but serum and ionized calcium levels were within normal limits. An abdominal ultrasonogram showed multiple gallstones. The patient underwent an unremarkable laparoscopic cholecystectomy with a negative intraoperative cholangiogram.
DISCUSSION Acute pancreatitis d u r i n g pregnancy was first reported in 1818. is Although the overall incidence remains unclear, 14q6 a recent report showed a 1 in 3000 incidence of pancreatitis complicating pregnancy, 17 an incidence similar to that occurring in n o n p r e g n a n t w o m e n of childbearing age. is Cholelithiasis is the leading cause of pancreatitis in p r e g n a n t patients, m Other less c o m m o n causes include alcohol ingestion, trauma, hypercalcemia, hyperlipidemia, viral infections, a n d drugs. 2~ The presentation of acute pancreatitis in the p r e g n a n t patient is essentially the same as in the n o n p r e g n a n t patient, is,2~ M a n a g e m e n t is primarily medical a n d includes bowel rest, intravenous fluid administration, a n d pain control. The occurrence of primary hyperparathyroidism during pregnancy was described by H u n t e r a n d Turnbul121 in 1931. It is estimated that for all w o m e n younger than the age of 40 years, 8 new cases per 100,000 p r e g n a n t w o m e n occur annually.22 The clinical presentation of hyperparathyroidism d u r i n g pregnancy includes mus-
Surgery Volume 119, Number 6
Inabnet et al.
711
Figure. Parathyroid adenoma. cle weakness (70%), arthralgia and myalgia (54%), and nephrolithiasis (30%). Less c o m m o n symptoms include peptic ulcer disease (12%), bone disease (2%), and pancreatitis (1%).93 As in n o n p r e g n a n t patients the diaguosis is made by the presence of an elevated serum calcium level in conjunction with a nonsuppressed serum parathyroid h o r m o n e level. Normal physiologic changes cause maternal serum calcium levels to decrease during pregnancy for several reasons. Because of the hypoalbuminemia of pregnancy, less protein is available to bind and carry calcium, thus lowering total but not free ionized serum calcium levels. An increase occurs in the renal excretion of calcium as a result of a pregnancy-induced increase in the glomerular filtration rate. Furthermore, maternal calcium is actively transported across the placenta to the fetus to provide calcium for fetal growth and development. The resulting hypocalcemia of pregnancy may induce a secondary physiologic hyperparathyroidism in the mother. 24 Maternal to fetal calcium shunting may render the hyperparathyroid m o t h e r eucalcemic or slightly hypercalcemic, 3 thereby confusing the diagnosis. Whereas calcium is actively transported to the fetus, parathyroid h o r m o n e is unable to cross the placental membrane. '25 Maternal hypercalcemia leads to fetal hypercalcemia, which suppresses fetal parathyroid development. Once the maternal source of calcium has been
removed after delivery, the fetus is unable to mobilize endogenous stores of calcium because of the longstanding parathyroid suppression. This leads to fetal hypocalcemia and possible tetany, which may be the first clinical clue to maternal hyperparathyroidism. 26 Maternal hypercalcemia may worsen once the protective effects of pregnancy are removed and could lead to hyperparathyroid crisis characterized by progressive muscle weakness, fatigue, nausea, vomiting, stupor, and coma. 3 The first step in the m a n a g e m e n t of hypercalcemia during pregnancy is to lower the serum calcium concentration with vigorous intravenous hydration. Oral phosphates, calcitonin, and loop diuretics have also been used with limited success. 27 Once the patient's medical condition has been optimized, definitive surgical therapy should be undertaken. 2s The second trimester is believed to be the optimal period for neck exploration.S, 18, 20, 26-28 Unless thd mother experiences life-threatening deterioration, surgery should be postp o n e d until the second trimester to allow completion of organogenesis in the f e t u s . 3'26-2s General anesthesia during the third trimester increases the risk of preterm laborZ9; nevertheless, surgery should not be postponed in the patient with symptoms. 1, 3, 28 The first case of pancreatitis and primary hyperparathyroidism d u r i n g pregnancy was reported in 19685; since that time there have been eight additional reports
712
Inabnet et al.
Surgery June 1996
T a b l e . All reported cases of hyperparathyroidism a n d pancreatitis in pregnancy
Author(s) Soyannwo et al.5 (1968) Bronsky et al.a (1970) Levine et al.7 (1979) Hess et al.8 (1980)
Thomason et al.~ (1981) Fabrin et a13~ (1986) Rajala et aLn (1986) Warneke et al.12 (1988) Present case
Presenting gestational age (wk)
Time of operation
Disease
16
Second trimester
40
4 Wk postpartum
33
Third trimester
Spontaneous abortion Hypercalcemia, tremors Not available
31
Third trimester
Parathyroid adenoma Parathyroid chief cell adenoma Parathyroid adenoma Parathyroid carcinoma
36
5 wk postpartum
36 36
Postpartum date not available 2 Wk postpartum
Transient hypocalcemia Transient hypocalcemia Normocalcemia
19
Second trimester
17
Second trimester
Parathyroid chief cell adenoma Parathyroid adenoma Parathyroid adenoma Parathyroid adenoma Parathyroid adenoma
Neonatal outcome
Respiratory distress syndrome, hypocalcemia
Normocalcemia Transient hypocalcemia
Maternal outs Cardiac arrest, death Normocalcemia, nol~nophosphatemia Transient hypocalcemia Pancreatic pseudocyst, hypocalcemia • 6 mo, ARF, rib fracture Transient hypocalcemia Transient hypocalcemia Normocalcemia Transient hypocalcemia Transient hypocalcemia
ARF,Acuterenalfailure.
including the present case (Table).612 Six of the reported cases were diagnosed d u r i n g the third trimester of pregnancy, of which four u n d e r w e n t neck exploration after delivery. The three cases diagnosed before the third trimester u n d e r w e n t s u r g e r y d u r i n g the second trimester. Parathyroid a d e n o m a was the underlying disease in eight cases; one case was caused by a parathyroid carcinoma. O n e maternal a n d one fetal death occurred, both in the first case. 5 The relationship between hyperparathyroidism a n d pancreatitis is c o n t r o v e r s i a l 9-32 As suggested by this case, a causal relationship appears to exist between these two disease processes. In our patient the diagnosis of pancreatitis subsequently led to the diagnosis of hyperparathyroidism a n d ultimate resection of a parathyroid adenoma. The second episode of pancreatitis, presumably caused by gallstones when the patient was not pregnant, suggests that more than one cause of pancreatitis may be present in any given patient at any point in time. The fact that our patient was initially diagnosed with hyperemesis gravidarnm shows the difficulty of accurate diagnosis d u r i n g pregnancy. Nausea a n d vomiting are quite c o m m o n in early pregnancy; however, when these symptoms persist beyond the first trimester, other causes should be considered a n d investigated.
SUMMARY
Although the simultaneous occurrence of hyperparathyroidism a n d pancreatitis d u r i n g pregnancy is rare, several points should be emphasized. Early recognition a n d treatment are essential. Pancreatitis should be kept in the differential diagnosis of u n e x p l a i n e d nausea, vomiting, a n d abdominal pain during pregnancy. Hyperparathyroidism should always be included in the differential diagnosis of pancreatitis. Finally, the second trimester is the optimal period for surgical intervention. REFERENCES
1. KellyTR. Primaryhyperparathyroidismduring pregnancy.Surgery 1991;110:1028-34. 2. LudwigG. Hyperpm'athyroidismin relationto pregnancy.N Engl J Med i962;267:637-42. 3. ShangoldMM, Dot N, Welt SI, FleischmanAR, Crenshaw CM. Hyperparathyroidism and pregnancy: a review. Obstet Gynecol Surv 1982;37:217-28. 4. Maj KR,YoungMB.Acutepancreatitisin pregnancy:two case reports. Obstet Gynecol 1982;60:653-7. 5. SoyannwoSM, McGowanM. A case of acute hyperthyroidism, with thyrotoxicosisand pancreatitis,presenting as hyperemesis gravidarum. Postgrad Med 1968;44:861-6. 6. BronskyD, Weisbely MG, Gross MC, BartonJJ. Hyperparathyroidisra and acute postpartum pancreatitiswith neonatal tetany in the child. AmJ Med Sci 1970;269:1604. 7. LevineG, Tsin D, RiskA. Acute pancreatitisand hyperparathyroidism in pregnancy.Obstet Gynecol1979;54:246-8.
Surgery Volume 119, Number 6
8. Hess HM, DicksonJ, Fox HE. Hyperfunctioning parathyroid carcinoma presenting as acute pancreatitis in pregnancy. J Reprod Med 1980;25:83-7. 9. Thomason JL, Sampson MB, Farb HF, Spellacy WN. Pregnancy complicated by concurrent primary hyperparathyroidism and pancreatitis. Obstet Gynecol 1981;57:34-6S. 10. Fabrin B, Eldon K. Pregnancy complicated by concurrent hyperparathyroidism and pancreatitis. Acta Obstet Gynecol Scand 1986;65:651-2. 11. Rajala B, Rafat AA, Hutchinson HT, Taylor T. Acute pancreatitis and primary hyperparathyroidism in pregnancy: treatment of hypercalcemia with magnesium sulfate. Obstet Gynecol 1987; 70:460-2. 12. Warneke G, Henning HV, Isemer FE, Muller HJ, Scheler F. Primary hyperparathyroidism with acute pancreatitis during pregnancy. Dtsch Med Wochenschr 1988;113:641-3. 13. Schmidt WJ. Sammlung-zweifelhafter schwangerschaft faille nebst einer kritischen einleitong. Viennna: F Wimmer, 1818:17280. 14. Langmade CF, Edmondson HA. Acute pancreatitis in pregnancy and the postpartum period. Surg Gynecol Obstet 1951;92:53-9. 15. Wilkinson EJ. Acute pancreatitis in pregnancy: a review of 98 cases and a report of 9 new cases. Obstet Gynecol Surv 1973;28: 281-7. 16. Jouppila P, Mokka R, Larmi TK. Acute pancreatitis in pregnancy. Surg Gynecol Obstet 1974;139:879-82. 17. Ramin K, Richey R, Ramin S, Cunningham FG. Acute pneumonitis in pregnancy [abstract]. Presented at the annual clinical meeting of the American College of Obstetrics and Gynecology, Washington, D.C., May 1993. 18. Cunningham FG, MacDonald PC, Gant NF, Leveno KJ, Gilstrap LC. Williams obstetrics-19th edition. Norwalk, Connecticut: Appleton and Lange, 1993:1165. 19. Block P, Kelly TR. Management of gallstone pancreatitis during
Inabnet et al.
713
pregnancy and the postpartum period. Surg Gynecol Obstet 1989;168:426-8. 20. Yeo CJ, CameronJL. The pancreas. In: Sabiston D, editor. Textbook of surgery. 14th ed. Philadelphia: WB Sanders, 1991:1080. 21. Hunter D, Turnbull H. Hyperparathyroidism: generalized osteitis fibrosawith observations upon bones, parathyroid tumors, a n d the normal parathyroid glands. BrJ Surg 1931;19:203-6. 22. Heath H, Hodgson SF, Kennedy MA. Primary hyperparathyroidism: incidence, morbidity, and potential economic impact in a community. N EnglJ Med 1980;302:189-93. 23. Wells S, Ashley S. The parathyroid glands. In: Sabiston D, editor. Textbook of surgery. 14th ed. Philadelphia: WB Saunders, 1991: 598-615. 24. Cushard W, Creditor M, Canterbury J, Reiss E. Physiological hyperparathyroidism in pregnancy. J Clin Endocrinol Metab 1972; 34:767-71. 25. Pitldn RM. Calcium metabolism in pregnancy: a review. Am J Obstet Gynecol 1975;121:724-37. 26. Delmonico FL, Neer RM, Cosimi AB, Barnes AB, Russell PS. Hyperparathyroidism during pregnancy. Am J Surg 1976;131:32837. 27. Carella M, Gossain V. Hyperparathyroidism and pregnancy: case report and review. J Gen Intern Med 1992;7:448-53. 28. Kristoffersson A, Dahlgren S, Litlmer F,JarhultJ. Primary hyperparathyroidism in pregnancy. Surgery 1985;97:326-30. 29. Schider SM, Webser GM. Maternal and fetal hazards of surgery during pregnancy. Am J Obstet Gynecol 1965;92:891-900. 30. Cope D, Culver PJ, Mixter CG, Nardi GL. Pancreatitis, a diagnostic clue to hyperparathyroidism. Ann Surg 1957;145:857-63. 31. Mixter CG, Keynes M, Cope O. Further experience with pancreatitis as a diagnostic clue to hyperparathyroidism. N EnglJ Med 1962;226:265-72. 32. Bess MA, Edis AJ, van Heerden JA. Hyperparathyroidism and pancreatitis: chance or acasual association?JAMA 1980;243:246-7.
PRIMARYHYPERPARATHYROIDISMIS rare d u r i n g pregnancy with slightly more than 100 cases reported in the English-language literature between 1930 a n d 1990.1 Associated fetal morbidity a n d mortality are high with a perinatal complication rate reaching 50%. 2, 3 Pancreatids is also u n c o m m o n d u r i n g pregnancy, occurring in less than 1% of all pregnancies. 4 Maternal mortality may be as high as 37%, a n d when occurring late in pregnancy, pancreatitis may lead to premature labor in up to 60% of patients. 4 The simultaneous occurrence of hyperparathyroidism a n d pancreatitis in pregnancy has only b e e n reported eight times. 5-12We present a case of c o n c u r r e n t hyperparathyroidism a n d pancreatitis in pregnancy masquerading as hyperemesis gravidarum. A review of the literature is provided, along with guidelines for diagnosis a n d treatment.
CASE R E P O R T The patient was a 27-year-old, gravida 1 para 0 woman who presented to her physician at 8-weeks' gestation with complaints of excessive nausea, vomiting, weakness, and a 14pound weight loss. She was diagnosed with hyperemesis gravidarum and treated initially as an outpatient with antiemetic therapy. Three weeks later she complained of new onset abdominal pain radiating to her back, and she was referred to our institution for admission. Admission laboratory studies were as follows: albumin, 2.4 units/L; calcium, 10.5 mg/dl; phosphorus, 2.1 mg/dl; amylase, 490 units/L; and lipase, 5517 units/L. An abdominal ultrasonogram showed an edematous, enlarged pancreas and gallbladder sludge, but no evidence of gallstones or bile duct dilatation was seen. The patient was diagnosed with pancreatitis and treated with bowel rest, vigorous hydration, and nutritional support. Her serum calcium level ranged from 9.8 to 10.5 mg/dl, with the ionized calcium level Accepted for publicationDec. 20, 1994. Reprint requests: Edgar D. Staren, MD, PhD, Departmentof General Surgery, Rush-Presbyterian-StLuke's Medical Center, 1653W. Congress Pkwy,Chicago, IL 60612. Surgery 1996;119:710-3. Copyright 9 1996 by Mosby-YearBook, Inc. 0039-6060/96/$5.00 + 0 11/57/72869 710
SURGERY
remaining poorly controlled despite medical therapy (1.47 to 1.83 mmol/L). An intact parathyroid hormone levelwas found to be 97 pg/ml (normal, 30 to 71 pg/ml). With the presumptive diagnosis of hyperparathyroidism, the patient underwent a parathyroidectomy for a parathyroid adenoma during her nineteenth week of pregnancy (Figure). Twelve hours after the procedure the ionized calcium level normalized at 1.2 mmol/L. Her amylase and lipase levels rapidly normalized during the next week, and the remainder of her postoperative course was unremarkable. She was discharged to home on a general diet, and after 40 weeks of pregnancy she delivered a healthy 7-pound boy with APGAR scores of 8 and 9, respectively. Two years after her initial presentation the patient again presented with abdominal pain that radiated to her back. Admitring laboratory data showed elevated amylase and lipase levels, but serum and ionized calcium levels were within normal limits. An abdominal ultrasonogram showed multiple gallstones. The patient underwent an unremarkable laparoscopic cholecystectomy with a negative intraoperative cholangiogram.
DISCUSSION Acute pancreatitis d u r i n g pregnancy was first reported in 1818. is Although the overall incidence remains unclear, 14q6 a recent report showed a 1 in 3000 incidence of pancreatitis complicating pregnancy, 17 an incidence similar to that occurring in n o n p r e g n a n t w o m e n of childbearing age. is Cholelithiasis is the leading cause of pancreatitis in p r e g n a n t patients, m Other less c o m m o n causes include alcohol ingestion, trauma, hypercalcemia, hyperlipidemia, viral infections, a n d drugs. 2~ The presentation of acute pancreatitis in the p r e g n a n t patient is essentially the same as in the n o n p r e g n a n t patient, is,2~ M a n a g e m e n t is primarily medical a n d includes bowel rest, intravenous fluid administration, a n d pain control. The occurrence of primary hyperparathyroidism during pregnancy was described by H u n t e r a n d Turnbul121 in 1931. It is estimated that for all w o m e n younger than the age of 40 years, 8 new cases per 100,000 p r e g n a n t w o m e n occur annually.22 The clinical presentation of hyperparathyroidism d u r i n g pregnancy includes mus-
Surgery Volume 119, Number 6
Inabnet et al.
711
Figure. Parathyroid adenoma. cle weakness (70%), arthralgia and myalgia (54%), and nephrolithiasis (30%). Less c o m m o n symptoms include peptic ulcer disease (12%), bone disease (2%), and pancreatitis (1%).93 As in n o n p r e g n a n t patients the diaguosis is made by the presence of an elevated serum calcium level in conjunction with a nonsuppressed serum parathyroid h o r m o n e level. Normal physiologic changes cause maternal serum calcium levels to decrease during pregnancy for several reasons. Because of the hypoalbuminemia of pregnancy, less protein is available to bind and carry calcium, thus lowering total but not free ionized serum calcium levels. An increase occurs in the renal excretion of calcium as a result of a pregnancy-induced increase in the glomerular filtration rate. Furthermore, maternal calcium is actively transported across the placenta to the fetus to provide calcium for fetal growth and development. The resulting hypocalcemia of pregnancy may induce a secondary physiologic hyperparathyroidism in the mother. 24 Maternal to fetal calcium shunting may render the hyperparathyroid m o t h e r eucalcemic or slightly hypercalcemic, 3 thereby confusing the diagnosis. Whereas calcium is actively transported to the fetus, parathyroid h o r m o n e is unable to cross the placental membrane. '25 Maternal hypercalcemia leads to fetal hypercalcemia, which suppresses fetal parathyroid development. Once the maternal source of calcium has been
removed after delivery, the fetus is unable to mobilize endogenous stores of calcium because of the longstanding parathyroid suppression. This leads to fetal hypocalcemia and possible tetany, which may be the first clinical clue to maternal hyperparathyroidism. 26 Maternal hypercalcemia may worsen once the protective effects of pregnancy are removed and could lead to hyperparathyroid crisis characterized by progressive muscle weakness, fatigue, nausea, vomiting, stupor, and coma. 3 The first step in the m a n a g e m e n t of hypercalcemia during pregnancy is to lower the serum calcium concentration with vigorous intravenous hydration. Oral phosphates, calcitonin, and loop diuretics have also been used with limited success. 27 Once the patient's medical condition has been optimized, definitive surgical therapy should be undertaken. 2s The second trimester is believed to be the optimal period for neck exploration.S, 18, 20, 26-28 Unless thd mother experiences life-threatening deterioration, surgery should be postp o n e d until the second trimester to allow completion of organogenesis in the f e t u s . 3'26-2s General anesthesia during the third trimester increases the risk of preterm laborZ9; nevertheless, surgery should not be postponed in the patient with symptoms. 1, 3, 28 The first case of pancreatitis and primary hyperparathyroidism d u r i n g pregnancy was reported in 19685; since that time there have been eight additional reports
712
Inabnet et al.
Surgery June 1996
T a b l e . All reported cases of hyperparathyroidism a n d pancreatitis in pregnancy
Author(s) Soyannwo et al.5 (1968) Bronsky et al.a (1970) Levine et al.7 (1979) Hess et al.8 (1980)
Thomason et al.~ (1981) Fabrin et a13~ (1986) Rajala et aLn (1986) Warneke et al.12 (1988) Present case
Presenting gestational age (wk)
Time of operation
Disease
16
Second trimester
40
4 Wk postpartum
33
Third trimester
Spontaneous abortion Hypercalcemia, tremors Not available
31
Third trimester
Parathyroid adenoma Parathyroid chief cell adenoma Parathyroid adenoma Parathyroid carcinoma
36
5 wk postpartum
36 36
Postpartum date not available 2 Wk postpartum
Transient hypocalcemia Transient hypocalcemia Normocalcemia
19
Second trimester
17
Second trimester
Parathyroid chief cell adenoma Parathyroid adenoma Parathyroid adenoma Parathyroid adenoma Parathyroid adenoma
Neonatal outcome
Respiratory distress syndrome, hypocalcemia
Normocalcemia Transient hypocalcemia
Maternal outs Cardiac arrest, death Normocalcemia, nol~nophosphatemia Transient hypocalcemia Pancreatic pseudocyst, hypocalcemia • 6 mo, ARF, rib fracture Transient hypocalcemia Transient hypocalcemia Normocalcemia Transient hypocalcemia Transient hypocalcemia
ARF,Acuterenalfailure.
including the present case (Table).612 Six of the reported cases were diagnosed d u r i n g the third trimester of pregnancy, of which four u n d e r w e n t neck exploration after delivery. The three cases diagnosed before the third trimester u n d e r w e n t s u r g e r y d u r i n g the second trimester. Parathyroid a d e n o m a was the underlying disease in eight cases; one case was caused by a parathyroid carcinoma. O n e maternal a n d one fetal death occurred, both in the first case. 5 The relationship between hyperparathyroidism a n d pancreatitis is c o n t r o v e r s i a l 9-32 As suggested by this case, a causal relationship appears to exist between these two disease processes. In our patient the diagnosis of pancreatitis subsequently led to the diagnosis of hyperparathyroidism a n d ultimate resection of a parathyroid adenoma. The second episode of pancreatitis, presumably caused by gallstones when the patient was not pregnant, suggests that more than one cause of pancreatitis may be present in any given patient at any point in time. The fact that our patient was initially diagnosed with hyperemesis gravidarnm shows the difficulty of accurate diagnosis d u r i n g pregnancy. Nausea a n d vomiting are quite c o m m o n in early pregnancy; however, when these symptoms persist beyond the first trimester, other causes should be considered a n d investigated.
SUMMARY
Although the simultaneous occurrence of hyperparathyroidism a n d pancreatitis d u r i n g pregnancy is rare, several points should be emphasized. Early recognition a n d treatment are essential. Pancreatitis should be kept in the differential diagnosis of u n e x p l a i n e d nausea, vomiting, a n d abdominal pain during pregnancy. Hyperparathyroidism should always be included in the differential diagnosis of pancreatitis. Finally, the second trimester is the optimal period for surgical intervention. REFERENCES
1. KellyTR. Primaryhyperparathyroidismduring pregnancy.Surgery 1991;110:1028-34. 2. LudwigG. Hyperpm'athyroidismin relationto pregnancy.N Engl J Med i962;267:637-42. 3. ShangoldMM, Dot N, Welt SI, FleischmanAR, Crenshaw CM. Hyperparathyroidism and pregnancy: a review. Obstet Gynecol Surv 1982;37:217-28. 4. Maj KR,YoungMB.Acutepancreatitisin pregnancy:two case reports. Obstet Gynecol 1982;60:653-7. 5. SoyannwoSM, McGowanM. A case of acute hyperthyroidism, with thyrotoxicosisand pancreatitis,presenting as hyperemesis gravidarum. Postgrad Med 1968;44:861-6. 6. BronskyD, Weisbely MG, Gross MC, BartonJJ. Hyperparathyroidisra and acute postpartum pancreatitiswith neonatal tetany in the child. AmJ Med Sci 1970;269:1604. 7. LevineG, Tsin D, RiskA. Acute pancreatitisand hyperparathyroidism in pregnancy.Obstet Gynecol1979;54:246-8.
Surgery Volume 119, Number 6
8. Hess HM, DicksonJ, Fox HE. Hyperfunctioning parathyroid carcinoma presenting as acute pancreatitis in pregnancy. J Reprod Med 1980;25:83-7. 9. Thomason JL, Sampson MB, Farb HF, Spellacy WN. Pregnancy complicated by concurrent primary hyperparathyroidism and pancreatitis. Obstet Gynecol 1981;57:34-6S. 10. Fabrin B, Eldon K. Pregnancy complicated by concurrent hyperparathyroidism and pancreatitis. Acta Obstet Gynecol Scand 1986;65:651-2. 11. Rajala B, Rafat AA, Hutchinson HT, Taylor T. Acute pancreatitis and primary hyperparathyroidism in pregnancy: treatment of hypercalcemia with magnesium sulfate. Obstet Gynecol 1987; 70:460-2. 12. Warneke G, Henning HV, Isemer FE, Muller HJ, Scheler F. Primary hyperparathyroidism with acute pancreatitis during pregnancy. Dtsch Med Wochenschr 1988;113:641-3. 13. Schmidt WJ. Sammlung-zweifelhafter schwangerschaft faille nebst einer kritischen einleitong. Viennna: F Wimmer, 1818:17280. 14. Langmade CF, Edmondson HA. Acute pancreatitis in pregnancy and the postpartum period. Surg Gynecol Obstet 1951;92:53-9. 15. Wilkinson EJ. Acute pancreatitis in pregnancy: a review of 98 cases and a report of 9 new cases. Obstet Gynecol Surv 1973;28: 281-7. 16. Jouppila P, Mokka R, Larmi TK. Acute pancreatitis in pregnancy. Surg Gynecol Obstet 1974;139:879-82. 17. Ramin K, Richey R, Ramin S, Cunningham FG. Acute pneumonitis in pregnancy [abstract]. Presented at the annual clinical meeting of the American College of Obstetrics and Gynecology, Washington, D.C., May 1993. 18. Cunningham FG, MacDonald PC, Gant NF, Leveno KJ, Gilstrap LC. Williams obstetrics-19th edition. Norwalk, Connecticut: Appleton and Lange, 1993:1165. 19. Block P, Kelly TR. Management of gallstone pancreatitis during
Inabnet et al.
713
pregnancy and the postpartum period. Surg Gynecol Obstet 1989;168:426-8. 20. Yeo CJ, CameronJL. The pancreas. In: Sabiston D, editor. Textbook of surgery. 14th ed. Philadelphia: WB Sanders, 1991:1080. 21. Hunter D, Turnbull H. Hyperparathyroidism: generalized osteitis fibrosawith observations upon bones, parathyroid tumors, a n d the normal parathyroid glands. BrJ Surg 1931;19:203-6. 22. Heath H, Hodgson SF, Kennedy MA. Primary hyperparathyroidism: incidence, morbidity, and potential economic impact in a community. N EnglJ Med 1980;302:189-93. 23. Wells S, Ashley S. The parathyroid glands. In: Sabiston D, editor. Textbook of surgery. 14th ed. Philadelphia: WB Saunders, 1991: 598-615. 24. Cushard W, Creditor M, Canterbury J, Reiss E. Physiological hyperparathyroidism in pregnancy. J Clin Endocrinol Metab 1972; 34:767-71. 25. Pitldn RM. Calcium metabolism in pregnancy: a review. Am J Obstet Gynecol 1975;121:724-37. 26. Delmonico FL, Neer RM, Cosimi AB, Barnes AB, Russell PS. Hyperparathyroidism during pregnancy. Am J Surg 1976;131:32837. 27. Carella M, Gossain V. Hyperparathyroidism and pregnancy: case report and review. J Gen Intern Med 1992;7:448-53. 28. Kristoffersson A, Dahlgren S, Litlmer F,JarhultJ. Primary hyperparathyroidism in pregnancy. Surgery 1985;97:326-30. 29. Schider SM, Webser GM. Maternal and fetal hazards of surgery during pregnancy. Am J Obstet Gynecol 1965;92:891-900. 30. Cope D, Culver PJ, Mixter CG, Nardi GL. Pancreatitis, a diagnostic clue to hyperparathyroidism. Ann Surg 1957;145:857-63. 31. Mixter CG, Keynes M, Cope O. Further experience with pancreatitis as a diagnostic clue to hyperparathyroidism. N EnglJ Med 1962;226:265-72. 32. Bess MA, Edis AJ, van Heerden JA. Hyperparathyroidism and pancreatitis: chance or acasual association?JAMA 1980;243:246-7.