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Department of Pathology,Grant Medical College,Mumbai, Maharashtra,lndia

Address for correspondence:Dr. Shubhangi NarayanraoJibh kate,

E-mail: shubhanish@yahoo.com

Received : 31-10-2012

Review completed : 28-02-2013

Accepted : f6-02-2074

Ari^ary hyperparathyroidism (PHPT) is a rare etiology of<-/ hypercalcemiainduced pancreatitis, accountingfor 0.47o to1.5% of cases. To date, less than 200 cases with PHPT diagnosedduring pregnancy have been described.llt Asymptomaticpresentation and rarity of PHPT in the reproductive age groupmakes its diagnosis during pregnancy challenging.ll[ Laboratoryfindings are also masked by pregnancy-induced changes in calciumhomeostasis.Fl Pregnant females with PHPT often experience a

clinically overt disease u,hich makes correct diagnosis imperative.If left untreated, it may pose a sigaificant risk to the mother andfetus in the form of postpartum matemal hypercalcemia, pretermdeliverv and fetal hypocalcemia that develops late.ll,I

Case Report

A Z'l-vear woman was referred on Day 15 postpartum to ourhospital u.ith complaints of pain in abdomen, vonriting, feverand decreased urine output. She had delivered a preterm 1.9-kgfemale child in a referral hospital and her previous delivery was

'uneventful. During her pre-, intra- and postnatal period, shehad history of repeated upper respiratory tract infections. Onexamination, she was febrile (103"F), pale, with a dry tongue,tachycardia andblood pressure of 140/60 mmHg. Right-sidedlingcrepitations were present. Tendemess and guarding were presentin the epigastric region. The uterus was bulky (12 weeks) andanteverted on per vaginal examination. Ultrasonography (USG)revealed increased echogenicig in kidneys and a slightly enlargedpancreas. A USC on Day 2 of admission showed an enlarged liver,bilateral enlarged kidneys with a poor corticomedullary ratio.On Day 6 of admission, a CT scan revealed enlarged pancreasconsistent with acute pancreatitis, cortical nephrocalcinosis withacute renal failure. She had no history of gestational diabetesmellitus, hypo/hyperthyroidism, alcohol use, OC pill use,gallstones or drug intake. Initial laboratory investigations (Day2) revealed amylasaemia (Zz}Ullnormal values < I50 Ul) andlipasaemia (646 Ul; normal values < I 50 U/1, increased leucocytecount ( 1 4,3 00/mm-,) and a hem atocit of 23.4%. Hemoglobin was7.9 gm% and platelet count was 249 x l07cmm. Lipid profilewas normal, serum creatinine was 8 mg/dl and ureawas 133 ntg%o.

Calcium level was I1.56 mgldl (normal value 8.5-10.1 mg/dl).Ionized calciumwas 1.60 mmoVl (1.12-1.32 mmoll). Phosphoruswas 7.5 mg%. (2-+.5 mg%). Prothrombin time (PT) - 22.1 sec(normal-I2.5 sec), Intemational normalized ratio (lNR) - 1.76,activated partial thromboplastin time (APTT) - >60 sec (normalvalue 22-30) . Acute pancreatitis with chronic renal disease withsepsis with disseminated intravascular coagulation (DIC) wasdiagnosed. On 17'h postpartum day parathyroid hormone wasdone and found tobe raised at a value of 581 pglml (normal:15-65pglml). Also herlipase was raised to l0l8 Ul. And platelet countr.vas reduced 56,000/cmm (on l7,h day itself). On Day 20, her PT

Hyperparathyroidism complicating pregna:A diagnostic challenge?Jibhkate SN, Valand AG, Ansari S, Bharambe BM

ABSTRACTPrimary hyperparathyroidism (PHPTI is a rare etiology of hypercalcemia-induced pancreatitis, contributingabout 0.4% to 1 .50/o of cases in the general population and up to 1 37o of cases during pregnancy. pHpT thatoccurs during pregnancy is a challenging diagnosis as the physiological changes in calcium homeostasis maskthe symptoms of hypercalcemia. PHPT during pregnancy often remain undiagnosed and untreated, and mayresult in serious clinical implications for the mother and fetus. Most clinicians consider surgery within thesecond trimester 0f pregnancy as the treatment of choice in this group 0f patients. This article refers to acase of a 24-year manied woman in whom PHPT was diagnosed for the first time in postpartum period. Shesuccumbed to complications on Day 20 postpartum.Pathological findings revealed metastatic calcification inlungs, pancreas and uterine vessels, chronic pancreatitis and renal cortical necrosis.

KEY W0BDS: Hypercalcemia, pregnancy, primary hyperparathyroidism

Introduction

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Jibhkate, ii.:i.: Hyperparathyroidism complicating pregnancy

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was 101.5 and INRwas 7.8 and she died the same day. Postmorten'rexamination revealed bilateral renal cortical necrosis [Figures Iand 2], few whitish firnr areas in lungs and chalky white fimrareas in pancreas [Figure 3]. Histopathologi revealed extensivecalcification in lungs [Figure 4], pancreas [Figure 5] and uterinevessels [Figure 6]. Pancreas also showed interstitial fibrosis,inflamrnation [Figure 7] and peripancreatic fat necrosis.

Discussion

Gestational PHPT often goes undetected, and by the timeof diagnosis, a n'rajority of rvomen have endured one ormore failed pregnancies. This is because of the physiologicalchanges in calciunr homeostasis during pregnancl, like

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Figure 1: Kidney showing iortical necrosis

Figure 3: Pancreas showing features ol chronic pancreatitis withfibrosis and calcification

Figure 5: Pancreatic lat necrosis with calcification

Figure 2: Kidney showing cortical necrosis with calcification

Figure 4: Lung showing metastatic calcification

Figure 6: Uterine vessel showing medial calcification

Journal of Postgraduate Medicine ?? 2014 Vol ?? Issue ?

Jibhkate, eii),.: Hyperparathyroidism complicating pregnancy

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Figure 7: Pancreas showing features of acute on chronic pancreatitisin the form ot interstitial fibrosis and mixed inflammation

maternal blood volume expansion, hypoalbuminemia andincreased fetal calcium requirements mask the symptoms ofhypercalcemia. This happened with our patient. Maternal andfetal complications include nephrolithiasis, osteitis fibrosacystica, pancreatitis, hypercalcaemic crisis,l0i spontaneousmiscarriage, intrauterine growth retardation, premature deliveryneonatal hypocalcemia,16l fetal polyhydramniollland fetal death.Incidence of n.raternal and fetal complication is 67% and80%,respectively. Interestingly, the frequency of pancreatitis inpregnancy-related hyperparathyroidism is higher (7 -13%) thanin nonpregnant (\-Z%)EI PHPT causes pancreatitis'by severalmechanisms but most probably through hypercalcemia. In fact,persistent hypercalcemia might increase calcium concentrationin pancreatic juice, and activate trypsinogen to trypsincausing pancreatic ductal and parenchymal damage leadingto pancreatitis.fl Hypercalcemia also decreases the volumeof pancreatic juice and causes protein plugs in the pancreaticduct which obstruct the flow leading to pancreatitis.IlglHypercalcemia causes vasoconstriction and pancreatic ductnarrowing. Some believe that parathyroid hormone itself acts

as toxin causing local thromboendarteritis.Ll0 All these mightresult in pancreatic tissue necrosis.l?l Our patient developedcomplications like pancreatitis and renal failure, due tohypercalcemia as evinced by higher serum calcium levels andmetastatic calcification in lung, pancreas and uterine vessels,and DIC in the postpartum period. Suspicion of PHPT and anearly diagnosis can thus greatly benefit and help avert maternaland fetal morbidity.

References

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1.

Murray JA, Newman WA 3d, Dacus JV Hyperparathyroidism inpregnancy: Diagnostic dilemma? Obsrer Gynecol Surv 1 997;52:202-5.

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Journal ol Postgraduate Med;cine ?? 2014 Vol ?? Issue ?

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