Taiwanese Journal of Obstetrics & Gynecology 53 (2014) 252e255
Contents lists available at ScienceDirect
Taiwanese Journal of Obstetrics & Gynecology journal homepage: www.tjog-online.com
Case Report
Acute pancreatitis secondary to primary hyperparathyroidism in a postpartum patient: A case report and literature review Chia-Chieh Lee a, An-Shine Chao b, c, Yao-Lung Chang b, c, Hsiu-Huei Peng b, c, Tzu-Hao Wang b, c, Anne Chao d, * a
Department of Obstetrics and Gynecology, Cathay General Hospital, Taipei, Taiwan Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital at Linkou, Taoyuan, Taiwan Chang Gung University College of Medicine, Taoyuan, Taiwan d Department of Anesthesiology, National Taiwan University Hospital, Taipei, Taiwan b c
a r t i c l e i n f o
a b s t r a c t
Article history: Accepted 14 January 2013
Objective: Primary hyperparathyroidism (PHPT) is a rare clinical entity in reproductive women. Unusual hypercalcemia causing pancreatitis in the peripartum period carries significant morbidity to both the fetus and the mother. Case Report: A 38-year-old woman developed a morbid course of intractable intra-abdominal abscess by pancreatitis, hydronephrosis by renal lithiasis, and unusual neurological presentations soon after delivery. Serial serum calcium level and imaging studies lead to the final diagnosis of PHPT due to a parathyroid adenoma. Data on 14 patients who suffered from pancreatitis due to hyperparathyroidism were collected from a MEDLINE search. The reasons for delayed diagnosis and literature review of acute pancreatitis in PHPT are discussed. Conclusion: Hypercalcemia can be masked during pregnancy and in severe pancreatitis, as was detected in about half of the case series. Clinicians should have a high level of suspicion of parathyroid adenoma in cases with a profound pancreatitis. Timely diagnosis and early therapeutic intervention are important to resolve complications and improve the outcomes of mothers and fetuses. Copyright © 2014, Taiwan Association of Obstetrics & Gynecology. Published by Elsevier Taiwan LLC. All rights reserved.
Keywords: hypercalcemia pancreatitis primary hyperparathyroidism
Introduction The occurrence of acute pancreatitis during pregnancy is uncommon, with a reported incidence from 0.02% to 0.1% [1,2]. Acute pancreatitis secondary to primary hyperparathyroidism (PHPT) is even less common. When a MEDLINE search using the keywords “pancreatitis”, “hyperparathyroidism”, and “pregnancy” for articles published between January 1965 and December 2011 was performed, only 14 cases were identified [3e16]. Lack of awareness or underreporting of this disease is probably responsible for the low reported incidence during pregnancy and postpartum [17]. We describe a patient whose initial presentation of PHPT was acute postpartum pancreatitis. While managing the intractable pancreatic abscesses, this patient developed uncommon
* Corresponding author. Department of Anesthesiology, National Taiwan University Hospital, 7, Chung-Shan South Road, Taipei 100, Taiwan. E-mail address: [email protected] (A. Chao).
neurological symptoms and hypercalcemia, which eventually led to the diagnosis of a parathyroid adenoma after a tortuous clinical course.
Case presentation A previously healthy 38-year-old multiparous woman (gravida 8, para 3, induced abortion 4, spontaneous abortion 1) delivered a female infant at 37 weeks' gestation. The patient had no major medical history. She drank alcohol occasionally, and did not consume alcohol during this pregnancy. The neonate was in a healthy condition. The patient underwent a tubal ligation the day after her delivery. The next day she developed acute abdomen pain with sharp and persisting epigastric pain, which radiated to her back. An abdominal computed tomography (CT) scan revealed a swollen pancreas. The patient was diagnosed with acute pancreatitis (Fig. 1). Because of her hemodynamic instability, she was transferred to our hospital and admitted to the intensive care unit (ICU).
http://dx.doi.org/10.1016/j.tjog.2013.01.029 1028-4559/Copyright © 2014, Taiwan Association of Obstetrics & Gynecology. Published by Elsevier Taiwan LLC. All rights reserved.
C.-C. Lee et al. / Taiwanese Journal of Obstetrics & Gynecology 53 (2014) 252e255
Fig. 1. Abdominal computed tomography scan. A swollen pancreas with some peripancreatic fluid accumulation.
Upon ICU admission, her body temperature was 37.7 C, blood pressure was 110/76 mmHg, pulse rate was 135 beats per minute, respiratory rate was 32 breaths per minute, and oxygen saturation was 98% with a nonrebreathing oxygen mask. On physical examination, the patient had severe epigastric pain with diffuse tenderness over her abdomen. Laboratory test results showed leukocytosis (31,160/mm3), thrombocytopenia (44,000/mm3), high levels of serum amylase (3014 U/L), lipase (621 U/L), lactate dehydrogenase (LDH; 1297 U/L), impaired renal function (blood urea nitrogen 57 mg/dL, creatinine 3.2 mg/dL), and hypocalcemia (total calcium level 9.6 mg/dL, normal range 12e17 mg/dL). Arterial blood gas parameters were the following: pH 7.40, carbon dioxide partial pressure 28 mmHg, oxygen partial pressure 150 mmHg, bicarbonate level 108 mg/dL, and base excess 7.3 mEq/L. Analgesics, intravenous hydration, broad-spectrum antibiotics, and noninvasive positive pressure ventilation were provided. She improved gradually, and was transferred to the ward 7 days later with a normal calcium level. During her hospital stay, the patient underwent CT-guided percutaneous drainage of her pancreatic abscess. She complained of transient blurred vision, and visual field defects were detected. Before a definite diagnosis was reached, the patient's vision improved, and she was discharged. Despite repeated percutaneous drainage, her abdominal CT revealed the inadequacy of abscess drainage and the development of the bilateral hydronephrosis caused by renal stones. Bilateral ureteral double-J stents were placed. Because percutaneous drainage did not significantly improve the patient's clinical condition, a laparotomy for retroperitoneal necrosectomy was performed. On the 2nd postoperative day, the patient complained of headache and blurred vision with eyes deviated to the left side. Subsequently, a transient seizure-like attack and loss of consciousness occurred. Laboratory studies indicated hypercalcemia 18 mg/dL, and hyperparathyroidism was suspected. Brain magnetic resonance imaging revealed hyperintensity within the sulci of the bilateral occipital lobes with increased leptomeningeal enhancement (Fig. 2). The intact parathyroid hormone (iPTH) level was elevated (508 pg/dL, normal range: 13e54 pg/dL). Neck ultrasound
253
Fig. 2. Brain magnetic resonance imaging. T2-weighted imaging shows hyperintensity of the sulci of bilateral occipital lobes.
and technetium Tc99m sestamibi scintigraphy with single-photon emission CT indicated a tumor in the lower right portion of the parathyroid gland (Fig. 3). Despite hydration and the administration of diuretics and pamidronate disodium, the calcium levels still remained high. Right parathyroidectomy was performed 11 weeks after childbirth. Histological examination confirmed the diagnosis of a parathyroid adenoma. Calcium and iPTH levels returned to normal after surgery, and the visual problem improved. The ureteral double-J stents were removed, and the retroperitoneal abscess was drained. The patient has received regular follow-up
Fig. 3. Single-photon emission computed tomography. A strong signal (arrow) is noted at the right inferior thyroid bed.
254
C.-C. Lee et al. / Taiwanese Journal of Obstetrics & Gynecology 53 (2014) 252e255
Table 1 Cases of hyperparathyroidism and pancreatitis in pregnancy. Authorsa
GAb
Presenting symptoms and signs
Cac
Surgery
Maternal outcome
Neonatal outcome
Soyannwo et al 1968 [3]
16
19.0
1 d PP
Hypercalcemia, cardiac arrest, and death
Spontaneous abortion (16 wk)
Bronsky et al 1970 [4]
40
Foot pain, polyuria, hypotonia, vomiting, drowsy, conscious loss, and cramping abdominal pain Hypotension, obtunded, and unresponsive
>12.5
4 wk PP
Normocalcemia
Levine et al 1979 [5] Hess et al 1980 [6]
33 31
Right upper abdominal pain and vomiting Upper abdominal and left flank pain, vomiting, and conscious change
15 14.1
33 GA 31 GA
Thomason et al 1981 [7]
36
Vomiting and severe abdominal pain
12.9
5 wk PP
Hypocalcemia Received neck radiation, acute renal impairment, pancreatic pseudocyst medical treatment, hypocalcemia 6 m, and rib fractures Hypocalcemiac
Induced labor (40 wk) and hypocalcemiac Natural delivery (38 wk) Induced labor (32 wk), mild respiratory distress syndrome, and hypocalcemia 10 m
Ito et al 1985 [8] Fabrin et al 1986 [9]
40 36
12.4 14.8
10 wk PP 5 d PP
Hypocalcemia Hypocalcemia
Rajala et al 1987 [10] Kawamata et al 1987 [11]
36 33
Polyuria and severe abdominal pain Severe abdominal pain, vomiting, and conscious loss Severe upper abdominal pain, nausea Diffuse abdominal pain
12.3 16.8
2 wk PP 21 m PP
C/S (36 wk), normocalcemia C/S (33 wk), intrauterine death
Warneke et al 1988 [12]
19
16.8
26 GA
Normocalcemia Hypercalcemia with transient hypocalcemia Hypercalcemia with transient hypocalcemia
Torii et al 1988 [13] Inabnet et al 1996 [14]
20 17
High 10.5
PP 19 GA
Hypercalcemia Hypocalcemia
Spontaneous abortion (20 wk) Natural delivery (40 wk)
Kondo et al 1998 [15]
31
18.3
31 GA
C/S (31 wk), normocalcemia
Dahan et al 2001 [16]
32
11.6
13 wk PP
Hypercalcemia, brain damage, pneumonia, and death Normocalcemia
Present case
36
18
11 wk PP
Itching, vomiting, polyuria, tingling sensation over face and arms, and recurrence of pancreatitis Abdominal pain and threatened abortion Nausea, vomiting, weakness, and abdominal pain Lumbago, abdominal pain, and loss of consciousness Diffuse abdominal pain, nausea, and vomiting Severe diffuse abdominal pain, bilateral renal stones, eye deviation, visual field defect, and seizure
Hypercalcemia with transient hypocalcemia
Induced labor (38 wk), 2-d assisted ventilation, and hypocalcemiac C/S (42 wk), normocalcemia C/S (36 wk), hypocalcemiac
Natural delivery (39 wk), normocalcemia
Natural delivery (36 wk) and normocalcemia Natural delivery (36 wk) and normocalcemia
C/S ¼ cesarean section; PP ¼ postpartum. a All of the reported cases were parathyroid adenomas, except the case presented by Hess et al [6], which was carcinoma of the parathyroid. b GA ¼ gestational week. c Serum calcium concentration in mg/dL.
observations in the outpatient department for over 1 year. Her child has no symptoms of hypocalcemia and displays normal growth and development. Discussion PHPT is a rare clinical entity in women of childbearing age. Approximately 100 cases of hyperparathyroidism during pregnancy were reported from January 1966 to December 2000 [18]. The low reported incidence is probably due to the lack of awareness or the subtlety of clinical manifestations. The diagnosis of hyperparathyroidism is made based on the symptoms of hypercalcemia and the elevations of serum calcium and iPTH. Hypercalcemia can pose severe threats to both mother and fetus. Maternal morbidities, such as severe vomiting, hypertension, preeclampsia, eclampsia, renal stones, pancreatitis, bone disease, and lethal hypercalcemia crisis, may be encountered. Although some of the manifestations are common during pregnancy, obstetricians should take hyperparathyroidism into consideration. Fetal complications are spontaneous abortion, intrauterine growth restriction, intrauterine demise, and postpartum hypocalcemia-related tetany [2,18]. During pregnancy, several physiologic changes ameliorate maternal serum calcium levels, including physiologic hypoalbuminemia, increased renal excretion of calcium, and active transport of calcium from mother to fetus for growth and development. Therefore, acute pancreatitis can be induced when calcium level rises abruptly, once the protective effect of pregnancy has ceased in patients with PHPT [1,2].
Most of the cases (7/14) were accompanied by hypercalcemia, so hyperparathyroidism could be diagnosed in a more straightforward fashion [3e16]. However, the serum calcium levels in this patient remained low during acute pancreatitis. The hypocalcemia reflected the severity of the acute pancreatitis of the patient (six points of the Ranson criteria for acute pancreatitis were present: leukocytosis, high LDH, hypocalcemia, negative base excess, azotemia, and fluid sequestration) and hypocalcemia also delayed the diagnosis of hyperparathyroidism. In addition, the higher the serum calcium level, the more severe is the fetomaternal complications (Table 1). The common complaints of patients with acute pancreatitis due to PHPT were vomiting and abdominal pain. As the severity increased, conscious change and other central nervous symptoms appeared (Table 1). In this case, transient seizure and other less common neurological presentations, and eye deviation and impaired visual field were also noted. When the acute phase of pancreatitis of the patients had elapsed, it seemed that a smoldering process of chronic inflammation was still occurring, making it difficult to treat the retroperitoneal abscess [7,13]. Following the parathyroidectomy, the patient became eucalcemic, the pancreatic retroperitoneal abscess gradually resolved, the renal stones became smaller, and the patient did not complain of blurred vision. The patient's remarkable recovery after surgery highlights the importance of early diagnosis and surgery. In conclusion, although acute pancreatitis secondary to PHPT is uncommon, clinicians should have a high level of suspicion of PHPT
C.-C. Lee et al. / Taiwanese Journal of Obstetrics & Gynecology 53 (2014) 252e255
when the exact cause of pancreatitis is not found. Serial monitoring of serum calcium levels and iPTH should be performed, if hyperparathyroidism is suspected. Surgery is the primary treatment for symptomatic parathyroid adenoma. The remarkable recovery highlights the importance of early diagnosis and surgery in patients with PHPT. Conflicts of interest The authors have no conflicts of interest to declare. References [1] Hernandez A, Petrov MS, Brooks DC, Banks PA, Ashley SW, Tavakkolizadeh A. Acute pancreatitis and pregnancy: a 10-year single center experience. J Gastrointest Surg 2007;11:1623e7. [2] Tang SJ, Rodriguez-Frias E, Singh S, Mayo MJ, Jazrawi SF, Sreenarasimhaiah J, et al. Acute pancreatitis during pregnancy. Clin Gastroenterol Hepatol 2010;8: 85e90. [3] Soyannwo MA, Bell M, McGeown MG, Milliken TG. A case of acute hyperparathyroidism, with thyrotoxicosis and pancreatitis, presenting as hyperemesis gravidarum. Postgrad Med J 1968;44:861e78. [4] Bronsky D, Weisbery MG, Gross MC, Barton JJ. Hyperparathyroidism and acute postpartum pancreatitis with neonatal tetany in the child. Am J Med Sci 1970;296:160e4. [5] Levine G, Tsin D, Risk A. Acute pancreatitis and hyperparathyroidism in pregnancy. Obstet Gynecol 1979;54:246e8. [6] Hess HM, Dickson J, Fox HE. Hyperfunctioning parathyroid carcinoma presenting as acute pancreatitis in pregnancy. J Reprod Med 1980;25:83e7.
255
[7] Thomason JL, Sampson MB, Farb HF, Spellacy WN. Pregnancy complicated by concurrent primary hyperparathyroidism and pancreatitis. Obstet Gynecol 1981;57:34se6s. [8] Ito Y, Ishizuka T, Miura K, Tanaka A, Tanaka M, Tachibana S. A case of primary hyperparathyroidism complicated with acute exacerbation of chronic pancreatitis during pregnancy (in Japanese). Jpn J Gastroenterol 1985;82: 2651e6. [9] Fabrin B, Eldon K. Pregnancy complicated by concurrent hyperparathyroidism and pancreatitis. Acta Obstet Gynecol Scand 1986;64:651e2. [10] Rajala B, Abbasi RA, Hutchinson HT, Taylor T. Acute pancreatitis and primary hyperparathyroidism in pregnancy: treatment of hypercalcemia with magnesium sulphate. Obstet Gynecol 1987;70:460e2. [11] Kawamata O, Enomoto M, Watanabe T, Makabe M, Nakamura J, Sakaki A, et al. A case report of acute pancreatitis and hyperparathyroidism in pregnancy. J Jpn Soc Clin Surg 1987;48:562e6 [in Japanese, English abstract]. [12] Warneke G, Henning HV, Isemer FE, Muller HJ, Scheler F. Primary hyperparathyroidism with acute pancreatitis during pregnancy. Dtsch Med Wochenschr 1988;113:641e3. [13] Torii A, Suenaga H, Terashima Y, Okuda T, Kodera Y, Negita M, et al. A case of primary hyperparathyroidism diagnosed as acute pancreatitis during pregnancy (in Japanese). J Jpn Soc Clin Surg 1988;49:1643. [14] Inabnet WB, Baldwin D, Daniel RO, Staren ED. Hyperparathyroidism and pancreatitis during pregnancy. Surgery 1996;119:710e3. [15] Kondo Y, Nagai H, Kasahaa K, Kanazawa K. Primary hyperparathyroidism and acute pancreatitis during pregnancy. Int J Pancreatol 1998;24:43e7. [16] Dahan M, Chang RJ. Pancreatitis secondary to hyperparathyroidism during pregnancy. Obstet Gynecol 2001;98:23e5. [17] Schnatz PF, Curry SL. Primary hyperparathyroidism in pregnancy: evidencebased management. Obstet Gynecol Surv 2002;57:365e76. [18] Cheang CU, Ho SW, Tee YT, Su CF, Chen GD. Acute necrotizing pancreatitis complicating uteroplacental apoplexy. Taiwanese J Obstet Gynecol 2007;46: 64e7.
Contents lists available at ScienceDirect
Taiwanese Journal of Obstetrics & Gynecology journal homepage: www.tjog-online.com
Case Report
Acute pancreatitis secondary to primary hyperparathyroidism in a postpartum patient: A case report and literature review Chia-Chieh Lee a, An-Shine Chao b, c, Yao-Lung Chang b, c, Hsiu-Huei Peng b, c, Tzu-Hao Wang b, c, Anne Chao d, * a
Department of Obstetrics and Gynecology, Cathay General Hospital, Taipei, Taiwan Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital at Linkou, Taoyuan, Taiwan Chang Gung University College of Medicine, Taoyuan, Taiwan d Department of Anesthesiology, National Taiwan University Hospital, Taipei, Taiwan b c
a r t i c l e i n f o
a b s t r a c t
Article history: Accepted 14 January 2013
Objective: Primary hyperparathyroidism (PHPT) is a rare clinical entity in reproductive women. Unusual hypercalcemia causing pancreatitis in the peripartum period carries significant morbidity to both the fetus and the mother. Case Report: A 38-year-old woman developed a morbid course of intractable intra-abdominal abscess by pancreatitis, hydronephrosis by renal lithiasis, and unusual neurological presentations soon after delivery. Serial serum calcium level and imaging studies lead to the final diagnosis of PHPT due to a parathyroid adenoma. Data on 14 patients who suffered from pancreatitis due to hyperparathyroidism were collected from a MEDLINE search. The reasons for delayed diagnosis and literature review of acute pancreatitis in PHPT are discussed. Conclusion: Hypercalcemia can be masked during pregnancy and in severe pancreatitis, as was detected in about half of the case series. Clinicians should have a high level of suspicion of parathyroid adenoma in cases with a profound pancreatitis. Timely diagnosis and early therapeutic intervention are important to resolve complications and improve the outcomes of mothers and fetuses. Copyright © 2014, Taiwan Association of Obstetrics & Gynecology. Published by Elsevier Taiwan LLC. All rights reserved.
Keywords: hypercalcemia pancreatitis primary hyperparathyroidism
Introduction The occurrence of acute pancreatitis during pregnancy is uncommon, with a reported incidence from 0.02% to 0.1% [1,2]. Acute pancreatitis secondary to primary hyperparathyroidism (PHPT) is even less common. When a MEDLINE search using the keywords “pancreatitis”, “hyperparathyroidism”, and “pregnancy” for articles published between January 1965 and December 2011 was performed, only 14 cases were identified [3e16]. Lack of awareness or underreporting of this disease is probably responsible for the low reported incidence during pregnancy and postpartum [17]. We describe a patient whose initial presentation of PHPT was acute postpartum pancreatitis. While managing the intractable pancreatic abscesses, this patient developed uncommon
* Corresponding author. Department of Anesthesiology, National Taiwan University Hospital, 7, Chung-Shan South Road, Taipei 100, Taiwan. E-mail address: [email protected] (A. Chao).
neurological symptoms and hypercalcemia, which eventually led to the diagnosis of a parathyroid adenoma after a tortuous clinical course.
Case presentation A previously healthy 38-year-old multiparous woman (gravida 8, para 3, induced abortion 4, spontaneous abortion 1) delivered a female infant at 37 weeks' gestation. The patient had no major medical history. She drank alcohol occasionally, and did not consume alcohol during this pregnancy. The neonate was in a healthy condition. The patient underwent a tubal ligation the day after her delivery. The next day she developed acute abdomen pain with sharp and persisting epigastric pain, which radiated to her back. An abdominal computed tomography (CT) scan revealed a swollen pancreas. The patient was diagnosed with acute pancreatitis (Fig. 1). Because of her hemodynamic instability, she was transferred to our hospital and admitted to the intensive care unit (ICU).
http://dx.doi.org/10.1016/j.tjog.2013.01.029 1028-4559/Copyright © 2014, Taiwan Association of Obstetrics & Gynecology. Published by Elsevier Taiwan LLC. All rights reserved.
C.-C. Lee et al. / Taiwanese Journal of Obstetrics & Gynecology 53 (2014) 252e255
Fig. 1. Abdominal computed tomography scan. A swollen pancreas with some peripancreatic fluid accumulation.
Upon ICU admission, her body temperature was 37.7 C, blood pressure was 110/76 mmHg, pulse rate was 135 beats per minute, respiratory rate was 32 breaths per minute, and oxygen saturation was 98% with a nonrebreathing oxygen mask. On physical examination, the patient had severe epigastric pain with diffuse tenderness over her abdomen. Laboratory test results showed leukocytosis (31,160/mm3), thrombocytopenia (44,000/mm3), high levels of serum amylase (3014 U/L), lipase (621 U/L), lactate dehydrogenase (LDH; 1297 U/L), impaired renal function (blood urea nitrogen 57 mg/dL, creatinine 3.2 mg/dL), and hypocalcemia (total calcium level 9.6 mg/dL, normal range 12e17 mg/dL). Arterial blood gas parameters were the following: pH 7.40, carbon dioxide partial pressure 28 mmHg, oxygen partial pressure 150 mmHg, bicarbonate level 108 mg/dL, and base excess 7.3 mEq/L. Analgesics, intravenous hydration, broad-spectrum antibiotics, and noninvasive positive pressure ventilation were provided. She improved gradually, and was transferred to the ward 7 days later with a normal calcium level. During her hospital stay, the patient underwent CT-guided percutaneous drainage of her pancreatic abscess. She complained of transient blurred vision, and visual field defects were detected. Before a definite diagnosis was reached, the patient's vision improved, and she was discharged. Despite repeated percutaneous drainage, her abdominal CT revealed the inadequacy of abscess drainage and the development of the bilateral hydronephrosis caused by renal stones. Bilateral ureteral double-J stents were placed. Because percutaneous drainage did not significantly improve the patient's clinical condition, a laparotomy for retroperitoneal necrosectomy was performed. On the 2nd postoperative day, the patient complained of headache and blurred vision with eyes deviated to the left side. Subsequently, a transient seizure-like attack and loss of consciousness occurred. Laboratory studies indicated hypercalcemia 18 mg/dL, and hyperparathyroidism was suspected. Brain magnetic resonance imaging revealed hyperintensity within the sulci of the bilateral occipital lobes with increased leptomeningeal enhancement (Fig. 2). The intact parathyroid hormone (iPTH) level was elevated (508 pg/dL, normal range: 13e54 pg/dL). Neck ultrasound
253
Fig. 2. Brain magnetic resonance imaging. T2-weighted imaging shows hyperintensity of the sulci of bilateral occipital lobes.
and technetium Tc99m sestamibi scintigraphy with single-photon emission CT indicated a tumor in the lower right portion of the parathyroid gland (Fig. 3). Despite hydration and the administration of diuretics and pamidronate disodium, the calcium levels still remained high. Right parathyroidectomy was performed 11 weeks after childbirth. Histological examination confirmed the diagnosis of a parathyroid adenoma. Calcium and iPTH levels returned to normal after surgery, and the visual problem improved. The ureteral double-J stents were removed, and the retroperitoneal abscess was drained. The patient has received regular follow-up
Fig. 3. Single-photon emission computed tomography. A strong signal (arrow) is noted at the right inferior thyroid bed.
254
C.-C. Lee et al. / Taiwanese Journal of Obstetrics & Gynecology 53 (2014) 252e255
Table 1 Cases of hyperparathyroidism and pancreatitis in pregnancy. Authorsa
GAb
Presenting symptoms and signs
Cac
Surgery
Maternal outcome
Neonatal outcome
Soyannwo et al 1968 [3]
16
19.0
1 d PP
Hypercalcemia, cardiac arrest, and death
Spontaneous abortion (16 wk)
Bronsky et al 1970 [4]
40
Foot pain, polyuria, hypotonia, vomiting, drowsy, conscious loss, and cramping abdominal pain Hypotension, obtunded, and unresponsive
>12.5
4 wk PP
Normocalcemia
Levine et al 1979 [5] Hess et al 1980 [6]
33 31
Right upper abdominal pain and vomiting Upper abdominal and left flank pain, vomiting, and conscious change
15 14.1
33 GA 31 GA
Thomason et al 1981 [7]
36
Vomiting and severe abdominal pain
12.9
5 wk PP
Hypocalcemia Received neck radiation, acute renal impairment, pancreatic pseudocyst medical treatment, hypocalcemia 6 m, and rib fractures Hypocalcemiac
Induced labor (40 wk) and hypocalcemiac Natural delivery (38 wk) Induced labor (32 wk), mild respiratory distress syndrome, and hypocalcemia 10 m
Ito et al 1985 [8] Fabrin et al 1986 [9]
40 36
12.4 14.8
10 wk PP 5 d PP
Hypocalcemia Hypocalcemia
Rajala et al 1987 [10] Kawamata et al 1987 [11]
36 33
Polyuria and severe abdominal pain Severe abdominal pain, vomiting, and conscious loss Severe upper abdominal pain, nausea Diffuse abdominal pain
12.3 16.8
2 wk PP 21 m PP
C/S (36 wk), normocalcemia C/S (33 wk), intrauterine death
Warneke et al 1988 [12]
19
16.8
26 GA
Normocalcemia Hypercalcemia with transient hypocalcemia Hypercalcemia with transient hypocalcemia
Torii et al 1988 [13] Inabnet et al 1996 [14]
20 17
High 10.5
PP 19 GA
Hypercalcemia Hypocalcemia
Spontaneous abortion (20 wk) Natural delivery (40 wk)
Kondo et al 1998 [15]
31
18.3
31 GA
C/S (31 wk), normocalcemia
Dahan et al 2001 [16]
32
11.6
13 wk PP
Hypercalcemia, brain damage, pneumonia, and death Normocalcemia
Present case
36
18
11 wk PP
Itching, vomiting, polyuria, tingling sensation over face and arms, and recurrence of pancreatitis Abdominal pain and threatened abortion Nausea, vomiting, weakness, and abdominal pain Lumbago, abdominal pain, and loss of consciousness Diffuse abdominal pain, nausea, and vomiting Severe diffuse abdominal pain, bilateral renal stones, eye deviation, visual field defect, and seizure
Hypercalcemia with transient hypocalcemia
Induced labor (38 wk), 2-d assisted ventilation, and hypocalcemiac C/S (42 wk), normocalcemia C/S (36 wk), hypocalcemiac
Natural delivery (39 wk), normocalcemia
Natural delivery (36 wk) and normocalcemia Natural delivery (36 wk) and normocalcemia
C/S ¼ cesarean section; PP ¼ postpartum. a All of the reported cases were parathyroid adenomas, except the case presented by Hess et al [6], which was carcinoma of the parathyroid. b GA ¼ gestational week. c Serum calcium concentration in mg/dL.
observations in the outpatient department for over 1 year. Her child has no symptoms of hypocalcemia and displays normal growth and development. Discussion PHPT is a rare clinical entity in women of childbearing age. Approximately 100 cases of hyperparathyroidism during pregnancy were reported from January 1966 to December 2000 [18]. The low reported incidence is probably due to the lack of awareness or the subtlety of clinical manifestations. The diagnosis of hyperparathyroidism is made based on the symptoms of hypercalcemia and the elevations of serum calcium and iPTH. Hypercalcemia can pose severe threats to both mother and fetus. Maternal morbidities, such as severe vomiting, hypertension, preeclampsia, eclampsia, renal stones, pancreatitis, bone disease, and lethal hypercalcemia crisis, may be encountered. Although some of the manifestations are common during pregnancy, obstetricians should take hyperparathyroidism into consideration. Fetal complications are spontaneous abortion, intrauterine growth restriction, intrauterine demise, and postpartum hypocalcemia-related tetany [2,18]. During pregnancy, several physiologic changes ameliorate maternal serum calcium levels, including physiologic hypoalbuminemia, increased renal excretion of calcium, and active transport of calcium from mother to fetus for growth and development. Therefore, acute pancreatitis can be induced when calcium level rises abruptly, once the protective effect of pregnancy has ceased in patients with PHPT [1,2].
Most of the cases (7/14) were accompanied by hypercalcemia, so hyperparathyroidism could be diagnosed in a more straightforward fashion [3e16]. However, the serum calcium levels in this patient remained low during acute pancreatitis. The hypocalcemia reflected the severity of the acute pancreatitis of the patient (six points of the Ranson criteria for acute pancreatitis were present: leukocytosis, high LDH, hypocalcemia, negative base excess, azotemia, and fluid sequestration) and hypocalcemia also delayed the diagnosis of hyperparathyroidism. In addition, the higher the serum calcium level, the more severe is the fetomaternal complications (Table 1). The common complaints of patients with acute pancreatitis due to PHPT were vomiting and abdominal pain. As the severity increased, conscious change and other central nervous symptoms appeared (Table 1). In this case, transient seizure and other less common neurological presentations, and eye deviation and impaired visual field were also noted. When the acute phase of pancreatitis of the patients had elapsed, it seemed that a smoldering process of chronic inflammation was still occurring, making it difficult to treat the retroperitoneal abscess [7,13]. Following the parathyroidectomy, the patient became eucalcemic, the pancreatic retroperitoneal abscess gradually resolved, the renal stones became smaller, and the patient did not complain of blurred vision. The patient's remarkable recovery after surgery highlights the importance of early diagnosis and surgery. In conclusion, although acute pancreatitis secondary to PHPT is uncommon, clinicians should have a high level of suspicion of PHPT
C.-C. Lee et al. / Taiwanese Journal of Obstetrics & Gynecology 53 (2014) 252e255
when the exact cause of pancreatitis is not found. Serial monitoring of serum calcium levels and iPTH should be performed, if hyperparathyroidism is suspected. Surgery is the primary treatment for symptomatic parathyroid adenoma. The remarkable recovery highlights the importance of early diagnosis and surgery in patients with PHPT. Conflicts of interest The authors have no conflicts of interest to declare. References [1] Hernandez A, Petrov MS, Brooks DC, Banks PA, Ashley SW, Tavakkolizadeh A. Acute pancreatitis and pregnancy: a 10-year single center experience. J Gastrointest Surg 2007;11:1623e7. [2] Tang SJ, Rodriguez-Frias E, Singh S, Mayo MJ, Jazrawi SF, Sreenarasimhaiah J, et al. Acute pancreatitis during pregnancy. Clin Gastroenterol Hepatol 2010;8: 85e90. [3] Soyannwo MA, Bell M, McGeown MG, Milliken TG. A case of acute hyperparathyroidism, with thyrotoxicosis and pancreatitis, presenting as hyperemesis gravidarum. Postgrad Med J 1968;44:861e78. [4] Bronsky D, Weisbery MG, Gross MC, Barton JJ. Hyperparathyroidism and acute postpartum pancreatitis with neonatal tetany in the child. Am J Med Sci 1970;296:160e4. [5] Levine G, Tsin D, Risk A. Acute pancreatitis and hyperparathyroidism in pregnancy. Obstet Gynecol 1979;54:246e8. [6] Hess HM, Dickson J, Fox HE. Hyperfunctioning parathyroid carcinoma presenting as acute pancreatitis in pregnancy. J Reprod Med 1980;25:83e7.
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