Right ectopic paraesophageal parathyroid adenoma with refractory hypercalcemia in pregnancy: A case report and review of the literature

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ContentslistsavailableatScienceDirect

International

Journal

of

Surgery

Case

Reports

j o ur na l h o m e p a g e :w w w . c a s e r e p o r t s . c o m

Right

ectopic

paraesophageal

parathyroid

adenoma

with

refractory

hypercalcemia

in

pregnancy:

A

case

report

and

review

of

the

literature

Abdelrahman

Abusabeib

a

_,

_Harun

_Bhat

b

_,

_Walid

_El

_Ansari

c,d,e,∗

_,

_Mohamed

_S.

_Al

_Hassan

a

_,

Abdelrahman

Abdelaal

a

a_Department_of_General_Surgery,_Hamad_General_Hospital,_Hamad_Medical_Corporation,_Doha,_Qatar b_Weill_Cornell_Medicine_in_Qatar,_Doha,_Qatar

c_Department_of_Surgery,_Hamad_General_Hospital,_Hamad_Medical_Corporation,_Doha,_Qatar d_College_of_Medicine,_Qatar_University,_Doha,_Qatar

e_School_of_Health_and_Education,_University_of_Skovde,_Skovde,_Sweden

a

r

t

i

c

l

e

i

n

f

o

Articlehistory: Received2October2020 Accepted20October2020 Availableonline28October2020

Keywords:

Ectopicparathyroidadenoma Paraesophageal

Pregnancy

Primaryhyperparathyroidisminpregnancy

a

b

s

t

r

a

c

t

INTRODUCTION:Ectopicparathyroidadenomaisrareduringpregnancybutposesmultiplechallengesin treatment.Itpresentsasprimaryhyperparathyroidismwhichleadstosymptomsandcomplicationsof hypercalcemiainboththemotherandfetus.

PRESENTATIONOFCASE:A38-year-oldSudanesefemalepresentedwithdiffusebonepainandpolyuria. Laboratoryinvestigationsrevealedelevatedserumcalciumandparathyroidhormone.Ultrasoundofthe neckdidnotshowanyabnormallesion,however99mTc-sestamibiscanshowedarightsidedparathyroid adenoma,andanearlierCTscanshowedtheadenomatobeinanectopicparaesophagealposition. Focusedsurgicalneckexplorationwasdone,andtheectopicparathyroidadenomawasexcised. DISCUSSION:Preoperativelocalizationoftheectopicparathyroidadenomaallowsforafocusedsurgical procedure.Ultrasoundisthesafestduringpregnancy,but99mTc-sestamibiandCTscanmaybenecessary ifultrasoundorinitialbilateralneckexplorationdonotdetectanyadenoma.Mildelevationsinmaternal serumcalciumcanhavedetrimentaleffectsonthefetuswhichsuggeststhatasurgicalapproachmaybe necessaryinthemajorityofcases.

CONCLUSIONS:Ectopicparathyroidadenomaisrareduringpregnancyandisdetrimentaltoboththe motherandfetus.Preoperativelocalizationallowsforafocusedsurgerywhichisadeﬁnitivetreatment andcansafelybeperformedduringthe2ndtrimesterofpregnancy.

1. Background

Theparathyroidglands(PG)arefoursmallglandsonthe poste-rioraspectofthelaterallobesofthethyroidgland.Thesuperiorand inferiorglandsareusuallysymmetrical;however,theiranatomical positionsvariesduetotheirlinesofembryologicaldescentfrom the4thand3rdbranchialpouchesrespectively[1].PGwhich devi-atefromnormalpositionsareectopic,andtheirlocationisrelated tothesameoriginsoftheparathyroid,thyroid,andthymictissue [2].Theprevalenceofectopicparathyroidgland(EPG)rangesfrom 28–42.8%(autopsyseries)to6.3–16%(smallerseriesstudies)[1]. Forinstance,othersreporteda7%incidenceofparaesophagealEPG [3,4].

AprimarypathologyofthePGisaparathyroidadenoma(PA) resultinginprimaryhyperparathyroidismwithrefractory

hyper-∗ Correspondingauthorat:DepartmentofSurgery,HamadGeneralHospital, HamadMedicalCorporation,Doha,Qatar.

E-mailaddress:welansari9@gmail.com(W.ElAnsari).

calcemia. This is the third most common endocrine disorder worldwide[5], andfemales areaffected astwiceasmales. Pri-maryhyperparathyroidisminpregnancy,however, israre,with 0.15–1.4%prevalence[6].Theinitialpresentationofprimary hyper-parathyroidismincludesgeneralizedfatigue,proximalweakness andincreasedfrequencyofurination,allofwhichcanbemistaken tobepartofthenormaleffects ofpregnancy,thereforeleading toadelayeddiagnosis.Inaddition,maternalserumcalciummay notbesigniﬁcantlyelevatedduetothephysiologiceffectsof preg-nancythatincludedecreasedserumalbuminlevelsandincreased glomerularﬁltrationrate[7].

Duringpregnancy,primaryhyperparathyroidismneedstobe diagnosedasearlyaspossibleinordertoavoidthehighly delete-riouseffectsofhypercalcemiaonthemotheranddevelopingfetus [8].Fromthematernalside,veryhighserumcalciumlevelsmay present as hyperemesis,nephrolithiasis, recurrenturinary tract infection,andpancreatitis;asforthebaby,therecouldbepreterm delivery,lowbirthweightandfetaldemise,aswellashypocalcemia

https://doi.org/10.1016/j.ijscr.2020.10.093

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Fig.1.Timelineandsequenceofeventsover3years.

Normalvalues:Calcium(2.15–2.50mmol/L),PTH(10–65pg/mL),Albumin(35–50g/L),VitaminD(35–80ng/mL),GFR(>60mL/min),Creatinine(44–97␮mol/L),24-hurine

calcium(2.50–7.50mmol/Lper24h),TSH(0.4–4.0mIU/L).

andtetany(secondarytofetalparathyroidhormonesuppression) intheimmediateneonatalperiod[9,10].

Tothebestofourknowledge,thiscouldbetheseventhcase globally and the second reported case from the MENA region (MiddleEastandNorthAfrica)ofprimaryhyperparathyroidismin pregnancyduetoectopicparathyroidadenoma(EPA).Wereport thiscaseinlinewiththeupdatedconsensus-basedsurgicalcase report(SCARE)guidelines[11].Inaddition,weundertooka litera-turereviewofEPApresentingduringpregnancy.

2. Casepresentation

A38yearoldSudanesefemaleof15weeksgestation(gravida 9para4)cametoourThyroidSurgeryoutpatientclinicatHamad GeneralHospitalinDoha(largesttertiarycarefacilityinQatar), referredfromobstetrics,witha2yearhistoryofgeneralizedbone pain most prominent in the hands, palpitation, and increased frequencyofurination.Thesesymptomsgraduallyprogressedin severityandwereaffectingherqualityoflife.Shedeniedany nau-sea,vomiting,constipationorﬂankpain.

Pastmedicalhistory(Fig.1A)wassigniﬁcantforchronic gener-alizedlymphadenopathyandrecurrentlowerbilaterallowerlimb swelling(possiblelymphedema).Herlast5pregnanciesbeforethe current pregnancy resultedin 4 abortions anda miscarriage at 20-weekgestation,withnocleardiagnosis.Familyhistory,social history and review ofsystems wereunremarkable. Shedidnot

smoketobaccoandneverconsumedalcohol.Physicalexamination showedbilaterallowerlimbedema.Theremainderofthephysical examwasunremarkable.Onadmission,herpulse,bloodpressure andtemperaturewerewithinnormal.

On initial admission to the medical team, a hypercalcemia workupwasdone(Fig.1B).Serologyandlaboratorytestsshowed elevatedcorrectedcalciumandintactparathyroidhormone(PTH), andlowvitaminD.Hercompletebloodcountandliverenzymes ﬁndingswerewithinnormal limitsandurinalysis,microbiology andautoimmuneworkupswereunremarkable.Adiagnosisof pri-maryhyperparathyroidismwasmadewhichwarrantedimaging ofthePG(Fig.1B).Ultrasound(US)oftheneckdidnotidentify parathyroidlesions; anearlier99mTc-sestamibiscan(sestamibi scan)undertakenbeforehercurrentpregnancy(Fig.2)revealed activityatthelowerpoleoftherightthyroidlobesuggestingaright inferiorPA;and,herCTscantakenayearearlier(Fig.3)asworkup forgeneralizedlymphadenopathyshowedanectopicrightinferior paraesophagealparathyroidlesion(7mmdiameter)impressiveof rightinferiorPA.

Shewasreferred urgentlytoourthyroid surgery clinic,and was18weekspregnantwithhypercalcemiaduetoprimary hyper-parathyroidismassociatedwithsubclinicalhyperthyroidismwhich wasmanagedwithbetablockeronly(Fig.1C).Thepatientwasthen seenandadmittedtooursurgicalteam(indexadmission,Fig.1D). ApartfromherhighserumcalciumandPTH,laboratorytestsand renalfunctionwerewithinnormal,andbasedontheprevious

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ses-Fig.2. Earlyandlate99mTc-sestamibiscintigraphyparathyroidscanimagesofneckandmediastinumanteriorlyat20minand2hshowingincreasedfocaluptakesuggestive

ofrightinferiorparathyroidadenoma.

Fig.3. ACoronal,BaxialandCsagittalviewsCTscanoftheneckshowingtheectopicparathyroidadenoma(redarrow)locatedinaparaoesophagealposition,trachea(green

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Fig.4. ExcisedparaesophagealPA.

tamibi scanandCT(Fig.1B,andFigs. 2and3),thediagnosisof primaryhyperparathyroidismduetoEPA(paraoesophageal)was conﬁrmed.

The patient was admitted for urgent parathyroidectomy. Focusedneckexplorationbyanexperiencedseniorsurgeon con-firmedthatthePGwasnotatitsnormalanatomicalposition,and thatthePAwasectopic(paraesophageal),posteriortotherecurrent laryngealnerve.FrozensectionshistologicallyconfirmedPA,and intraoperativerapidPTHmonitoringbeforeexcisionand 10min after excisionofthe PA,showeda serumPTH decreaseby 98% (Fig.1E)confirmingtheremovalofthehypersecretinggland(Fig.4). Onthefirstpostoperativeday,thepatientdevelopedmild symp-tomsandsignsofhypocalcemia,thelaboratoryfindingsrevealed lowintactPTH(1pg/mL)andserumcalcium(1.9mmol/L),andshe wasgiventreatment(Fig.1F).Aweeklater,followupatour thy-roidsurgeryclinicrevealedthatthepatientdidnotcomplainofany ofthesymptomsofhypercalcemiaexperiencedpriortosurgery, orsymptomsofhypocalcemiathat developedintheearly post-operativeperiod,andshehadnormallaboratoryfindings(Fig.1G), andhistopathologyshowedfindingsconsistentwithhypercellular parathyroidtissue,compatiblewithPA(1.5×0.7×0.4cm) weight-ing0.1g.

3. Discussion

Calciumhomeostasisduringpregnancyavoidshypercalcemia and itscomplicationsinthemotherandfetus.Wereporta rare case ofapregnantfemalewithectopic rightparaesophageal PA leadingtohyperparathyroidismandhypercalcemia.Such hyper-calcemia mighthavebeenpossiblyresponsibleforherprevious foursuccessiveabortionsandonemiscarriageintheperiodprior tohercurrentpregnancy.FetalPGdevelopaftertheﬁrsttrimester, hencematernalserumcalciumlevelsdictatecalciumhomeostasis in thefetusduringthistime [12].Elevatedmaternalserum cal-ciumresultsinfetalhypercalcemia[13],andasfetalPGdevelop, fetalhypercalcemiasuppressesthefetalPGthatpredisposesthe fetustopostpartumhypocalcemiaoncecalciumdeliveryfromthe motherceasesafterbirthandthefetusisunabletomobilizecalcium fromthebones[14].Ourcasedemonstratesdysregulatedcalcium homeostasis evidenced by themother’s symptomatic presenta-tionandpreviousabortionsandmiscarriage,possiblyasaresult ofhypercalcemia.

Table 1 shows that ourpatient’s presentation withpolyuria agrees withcasesidentiﬁed in theliteraturereview we under-took[15],butshehadnorenalcomplicationse.g.,nephrolithiasis andpyelonephritis,contrarytootherreports[6,15,16].Her com-plaint of bone pain was alsonot commonly reported by other Table

1 Comparison of current case with other similar cases of ectopic parathyroid adenomas presenting in pregnancy identiﬁed in the literature review. Age Symptoms, M a Effects, F b S Ca S PTH MisC c Pre-op d Imaging Location e Surgical Procedure Trimester Excision f Current Case Qatar 36 Diffuse bone pain, polyuria — 3.03 117 1 Y US, CT Sestamibi R paraesophageal Focused NE 2 Y Triantafyllidou 2018 Switzerland 40 Nausea, hyperemesis Nil 3.04 120 0 N 18F-F PET/CT R middle Med TE after failed NE 2 N Horton 2017 USA 21 Headache, intractable vomiting, polydipsia Nil 3.37 191 0 N 4DCT Sestamibi L Med VATP after failed NE 2 Y Saad 2014 USA 18 Polyuria, weakness,

vomiting, nephrolithiasis, pyelonephritis

Nil 3.1 204 0 N US Half-dose Sestamibi S P Med VATP 2 Y McMullen 2010 Canada — Renal calculi Nil 3.5 236 0 N Sestamibi Med level of AA THc 2 Y — Renal calculi Nil 3.2 179 N Sestamibi Thymus — 2 Y Dinc ¸ er 2008 Turkey 29 Muscle weakness, bilateral flank pain/nephrolithiasis, alopecia, Abd pain Convulsive attacks 3.24 2490 0 N Sestamibi, SPECT, Spiral CT, MRI Med TE ** N Rooney 1998 Northern Ireland 31 Abd pain, nausea, vomiting Nil 2.93 38 0 N MRI, CT AA within thymus MS after failed CE 2 Y Due to space considerations, only first author is cited ; aSymptoms in mother; bEffect on fetus; cNumber of past miscarriages: dPre-op: Preoperative localization; eLocation of ectopic parathyroid gland; fExcision done during pregnancy; **Not applicable because surgery performed after delivery; AA: Aortic arch; Abd: abdominal; CE: cervical exploration; L: left; Med: mediastinum; MRI: magnetic resonance imaging; MS: median sternotomy; N: No; NE: Neck exploration; P: Posterior; PET/CT: Positron Emission Tomography R: Right; S Ca: Serum calcium (mmol/L); S PTH: Serum parathyroid hormone (pg/mL); S: superior; SPECT: Sestamibi: 99mTc-sestamibi scan; Single-photon emission computed tomography; TE: thoracoscopic exploration; THc: Thoracotomy; Trimester: Trimester of pregnancy when surgery was performed; US: ultrasound; VATP: video assisted thoracoscopic parathyroidectomy; Y: Yes; – Computed Tomography; 18F-F: 18F-fluorocholine; — not reported.

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authors(Table1).About67%ofwomenwithprimary hyperparathy-roidismhave symptomsofhypercalcemia,e.g.nausea,vomiting andconstipation,whicharenormalcomplaintsduringpregnancy [14,17]. In primary hyperparathyroidism duringpregnancy, the early mild hypercalcemia can be difficultto diagnose due two reasons: symptomscanbeoverlooked; and,normalphysiologic responses(increasedintravascularvolumeandglomerular filtra-tionthatleadtohemodilutionandgestationalhypoalbuminemia, hypercalciuria,and decreasedtotal bodycalcium) canmaskthe hypercalcemia[14,18,19].Despitesuchphysiologicresponses,our patienthadhighlevelofserumcalcium(3.03mmol/L).Asforthe investigations, ourcase demonstratestheimportance of identi-fyingEPApriortosurgeryinordertopreventunsuccessfulneck exploration and prolonged time of surgery. Preoperative local-ization ofEPAavoidsreoperationin>95%ofcases[20].CTand sestamibiareharmfultothefetus,butourpatienthadtheCTas aworkupforherlymphadenopathyandhadthesestamibiduring herinitialhypercalcemiaworkup,bothbeforepregnancy.USissafe duringpregnancybutisoperatordependent(possiblefalse nega-tivefindings),with27–89%sensitivityforEPAdetection[1].We encounteredsuchafalsenegativefinding(USdidnotdetectthe EPA),inagreementwithothers[12](Table1).LocalizationofEPA inpregnancyisdifficult,USistheimagingofchoice,butfurther imagingmayberequiredtolocalizetheEPA.Sestamibiscanis use-fulforpreoperativeidentificationofEPA(80–99%sensitivity)[21], anditsuccessfullyidentifiedourpatient’sPAbutdidnotlocalizeits ectopicposition.Inpregnancy,lowdosesestamibiscanisunlikely tobedetrimental[22];andCTiscontraindicatedinpregnancybut maybeusedwithappropriateabdominalshielding[6,23](Table1). Combinedimaging(sestamibiandCT)enhancethediagnosis, detectingthemostabnormalglands[6]. Suchcombinationafter failedneckexplorationcanensuresuccessofthesecondsurgery [6].18F-fluorocholinePET/CTmaylocalizeEPAinpregnancyafter negative/equivocalUSand/orscintigraphy/single-photonemission computedtomography(SPECT)[24].Amultidisciplinaryteamcan select theappropriateimaging withtheleastharmtoboth the motherandfetus[25].

The’true’EPAprevalenceremainsunconﬁrmed.Insmall stud-ies of patientsundergoing neckre-exploration, prevalencewas 29–45%[1].EPAcanlocatewithinthemediastinumasobserved inTable1[6,12,15,16,24]orthymus[23].

Nephrolithiasis, bone disease, and pancreatitis complicate hypercalcemia due to primary hyperparathyroidism [17]. Post-delivery,themothernolongerprovidesthefetus,whichexposes her to veryhigh calciumlevels (lifethreatening hypercalcemic crisis)[9,26].Postpartumfetalhypocalcemia(convulsiveattacks) withmaternal bilateralnephrolithiasis havebeenreported[16] (Table 1). Fetal complications include fetal demise, low birth weight,pretermdelivery,anddepressedfetalparathyroidleading topostpartumneonatalhypocalcemia/tetany[9,14,27].Ourcase had no complications:diagnosiswasdoneat theindex admis-sion,and withintwo weeks,urgent surgery(EPAexcision)was undertakeninthesecondtrimesterandfollowupbyobstetrician conﬁrmeduneventfulpost-operativeperiodformotherandfetus. Thisisinsupportoftheimportanceofearlysuspicionand diagno-sispriortoseverepresentations,increasedmorbidityandmortality [8].

As for miscarriage, hyperparathyroidism is associated with 3.5 timesincreaseinmiscarriage[8].Roughly>50%of primary hyperparathyroidisminpregnancyisundiagnosed;72%of preg-nantwomenhadhypercalcemiaatmiscarriage,suggestingdelayed diagnosis; and serumcalciumas lowas 10.7mg/dL ledto mis-carriage [8]. This warrants thorough investigations in order to excludePHA-primaryhyperparathyroidism.Duetoourpatient’s history of overlap ofpreviousabortions/miscarriageand symp-toms ofhypercalcemia,it is possiblethat thepatient’s primary

hyperparathyroidism(undiagnosedatthattime)couldhavebeen responsibleforhermiscarriageinSudanbeforeweencountered herinQatar.Weareunabletoconﬁrmsuchspeculationasthedata isnotavailable.

Asformanagement,surgicalproceduresdependontheEPA’s location(Table1).WelocalizedtheEPAbeforesurgery,and under-tookrightsidedfocusedparathyroidectomy,whichisaseffective asbilateralneckdissection,butassociatedwithbetterscarsand patientsatisfaction,andshorteroperativetimeandhospitalstay [28].Othertechniquesincludemediansternotomyformediastinal EPA[23],andless invasivevideoassistedthoracoscopic surgery [16].

Apregnantpatientraisesconcernsaboutthesafetyofgeneral anesthesiaandsurgery.Theargumentagainstparathyroidectomy duetoteratogeniceffectsofgeneralanesthesiamaybe overex-aggerated[6] as modern-day anesthesia can be used safely in pregnancy[29,30].Inaddition,surgery(parathyroidectomy)has much lower neonatal complications compared to conservative therapy[17,31].Parathyroidectomyis safewhencarried outby experiencedsurgeons[8],andsurgeryprovidesacurative treat-mentwhichremovestherisksofmaternalandfetalhypercalcemia as well as post-partum fetal hypocalcemia [31]. Conservative management(oralhydrationandbisphosphonates)issometimes recommended,althoughevenmildhypercalcemiacanleadto sig-nificantmaternal complicationsand neonataltetany[8,31]. The lackofatightlydefinedrelationshipbetweenserumcalciumlevel andtheabilitytopredictmaternalorfetalcomplicationssuggests thatmildhypercalcemiamaystillrequiredefinitivesurgical man-agement[6].

Thecurrentcasedemonstratedasuccessfulfocusedneck explo-rationundertaken duringthe2ndtrimester whichis anagreed optimal time for surgery [32]. For women diagnosed with PA andareplanningtogetpregnant,informationshouldbeprovided regardingthedifﬁcultyandrisksassociatedwiththemanagement ofprimaryhyperparathyroidismduringpregnancy[33].

4. Conclusion

EPAinpregnancyisrare,withchallengesindiagnosisand man-agement.A highindex ofsuspicion is useful,andas even mild elevationsof maternalserumcalciumcanexertpotential nega-tiveeffectsonthefetus,asurgicalapproachshouldbeconsidered overconservativetherapy.Amultidisciplinaryapproachisrequired inordertoselectthesafestimagingmodalitiesforpre-operative localizationoftheEPAinpregnancywhichwillallowforafocused, shorterandlessinvasivesurgicalprocedure.Theprocedureshould beperformedbyanexperiencedsurgeontakinginto considera-tiontheectopiclocationofthePAandthepotentialintraoperative technicaldifﬁcultiesandmorbiditiesrelatedtoboththemother andfetus.

DeclarationofCompetingInterest

Theauthorsreportnodeclarationsofinterest.

Funding

Nothingtodeclare.

Ethicalapproval

ApprovedbyMedicalResearchCenter,HamadMedical Corpo-rationreferencenumber(MRC-04-20-859).

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Consent

Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereportandaccompanyingimages.Acopy ofthewrittenconsentisavailableforreviewbytheEditor-in-Chief ofthisjournalonrequest.

Registrationofresearchstudies

NotﬁrstinMan.

Guarantor

ProfDrWalidElAnsari.

Provenanceandpeerreview

Notcommissioned,externallypeer-reviewed.

CRediTauthorshipcontributionstatement

Abdelrahman Abusabeib: Investigation,Writing - review &

editing.HarunBhat:Investigation,Writing-originaldraft, Writ-ing-review&editing.WalidElAnsari:Investigation,Supervision, Projectadministration,Writing-originaldraft,Writing-review &editing.MohamedS.AlHassan:Investigation,Project admin-istration, Writing - review & editing. Abdelrahman Abdelaal:

Investigation,Projectadministration,Writing-review&editing.

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