-
CASE REPORT Open Access
Giant parathyroid adenoma: a case reportand review of the
literatureMohamed S. Al-Hassan1, Menatalla Mekhaimar2, Walid El
Ansari3,4,5*, Adham Darweesh6 andAbdelrahman Abdelaal1
Abstract
Background: Giant parathyroid adenoma is a rare type of
parathyroid adenoma defined as weighing > 3.5 g. Theypresent as
primary hyperparathyroidism but with more elevated laboratory
findings and more severe clinicalpresentations due to the larger
tissue mass. This is the first reported case of giant parathyroid
adenoma from theMiddle East.
Case presentation: A 52-year-old Indian woman presented with a
palpable right-sided neck mass and generalizedfatigue.
Investigations revealed hypercalcemia with elevated parathyroid
hormone and an asymptomatic kidneystone. Ultrasound showed a
complex nodule with solid and cystic components, and Sestamibi
nuclear scanconfirmed a giant parathyroid adenoma. Focused surgical
neck exploration was done and a giant parathyroidadenoma weighing
7.7 gm was excised.
Conclusions: Giant parathyroid adenoma is a rare cause of
primary hyperparathyroidism and usually presentssymptomatically
with high calcium and parathyroid hormone levels. Giant parathyroid
adenoma is diagnosed byimaging and laboratory studies. Management
is typically surgical, aiming at complete resection. Patients
usuallyrecover with no long-term complications or recurrence.
Keywords: Giant parathyroid adenoma, Parathyroidectomy, Primary
hyperparathyroidism, Minimal invasiveparathyroidectomy, Atypical
parathyroid adenoma
BackgroundThe normal parathyroid gland weighs
approximately50–70mg. Parathyroid adenomas (PTAs) are usuallysmall,
measuring < 2 cm and weighing < 1 gm [1]. GiantPTAs (GPTAs),
although rare, are most commonly de-fined as weighing > 3.5 gm,
with some reports describingweights up to 110 gm [2, 3]. Both PTA
and GPTApresent with the syndrome of primary hyperparathyroid-ism
(PHPT), the third most common endocrine disorder[4]. The
pathophysiology of PHPT is autologous secre-tion of parathyroid
hormone (PTH) by one or more ofthe parathyroid glands [4]. Although
PHPT can becaused by parathyroid hyperplasia or carcinoma,
how-ever, around 85% of cases of PHPT are due to PTAs,
and the majority of these are because of solitary PTAs,of which
GPTA comprise a small number [5].To the best of our knowledge, this
case report de-
scribes the first case in the Middle East of a patient
withnon-ectopic GPTA presenting with visible neck swelling.This
case report also reviews the published literature toreport on the
clinical characteristics and typical presen-tation of GPTA as well
as diagnosis and treatment.
Case presentationA 52-year-old Indian woman was referred to our
SurgicalEndocrinology clinic at Hamad General Hospital in
Doha,Qatar. She complained of a neck swelling and
generalizedfatigue. Laboratory results showed hypercalcemia and
ele-vated PTH. Her past social, environmental, family,
andemployment history (housewife) were unremarkable. Shedid not
smoke tobacco and never consumed alcohol.There was no past history
of symptomatic kidney stones;however, a recent computed tomography
(CT) scan of herabdomen and pelvis showed a 2mm non-obstructing
© The Author(s). 2019 Open Access This article is distributed
under the terms of the Creative Commons Attribution
4.0International License
(http://creativecommons.org/licenses/by/4.0/), which permits
unrestricted use, distribution, andreproduction in any medium,
provided you give appropriate credit to the original author(s) and
the source, provide a link tothe Creative Commons license, and
indicate if changes were made. The Creative Commons Public Domain
Dedication
waiver(http://creativecommons.org/publicdomain/zero/1.0/) applies
to the data made available in this article, unless otherwise
stated.
* Correspondence: [email protected] of Surgery,
Hamad General Hospital, Hamad MedicalCorporation, Doha,
Qatar4College of Medicine, Qatar University, Doha, QatarFull list
of author information is available at the end of the article
Al-Hassan et al. Journal of Medical Case Reports (2019) 13:332
https://doi.org/10.1186/s13256-019-2257-7
http://crossmark.crossref.org/dialog/?doi=10.1186/s13256-019-2257-7&domain=pdfhttp://creativecommons.org/licenses/by/4.0/http://creativecommons.org/publicdomain/zero/1.0/mailto:[email protected]
-
calculus in the lower pole calyx of her right kidney withno
hydroureteronephrosis. Her past medical history indi-cated that she
had dyslipidemia, controlled with medica-tion; however, she was not
on any other medication. Onphysical examination, a right-sided neck
swelling was ob-vious on inspection; on palpation a mobile
non-tendernodule could be felt, approximately 3 cm in size. The
restof the physical examination was unremarkable. A neuro-logical
examination was unremarkable. On admission, herpulse, blood
pressure and temperature were normal.Serology laboratory tests
showed corrected calcium of
3.12 mmol/L, an intact PTH of 503 ng/L, vitamin D of19.97
nmol/L, and normal thyroid-stimulating hormone(TSH) level. Her
renal functions were within normallimits, serum creatinine was 67
μmol/L, and 24-hoururine calcium was 4.30mmol/L per 24 hours. Her
completeblood count (CBC) and liver laboratory findings werewithin
normal limits. Microbiology laboratory tests werenot deemed
necessary.Imaging investigations included an ultrasound of her
neck that showed a complex nodule (4.1 × 2.3 cm) withsolid and
cystic components, and vascularity was ob-served in the mid to
lower pole of her right thyroidgland (Fig. 1). A parathyroid
Sestamibi scan revealedtracer concentration in the thyroid tissues
with more in-tense focal uptake observed related to the lateral
side ofthe right thyroid lobe (Fig. 2). A delayed scan
revealedresidual persistent uptake, corresponding to the
initiallydescribed increased focal uptake seen on the early
im-ages. These Sestamibi findings were highly suggestive ofa PTA.
Ultrasound-guided fine-needle aspiration (FNA)was done but was
non-diagnostic.
Surgical techniqueThis patient fitted two of the criteria for
surgical man-agement of PTA: (1) she had renal involvement, and,
(2)serum calcium was 3.12 mmol/L (> 0.25 mmol/L fromthe upper
limit of normal) [6]. She underwent minimallyinvasive
parathyroidectomy (MIP) with focused explor-ation and excision of
the right PTA under general
anesthesia. A transverse collar incision was made, thesurgery
proceeded and the adenoma was identified andexcised (Fig. 3).
Intraoperative PTH (ioPTH) monitoringconfirmed the excision of the
adenoma as the PTHdropped from the initial pre-excision level of
546 ng/Lto 239 ng/L 10minutes after the excision, and then to161
ng/L 20 minutes after excision, a 70% drop. Intraop-erative frozen
sections were sent to the pathology la-boratory that confirmed PTA.
Surgery was concluded;our patient recovered without any
complications andwas discharged on the second postoperative day.
Finalhistopathology of the gland reported a nodule (4 × 2.5 ×1.5
cm) weighing 7.7 gm with histologic features consist-ent with a PTA
(Fig. 4). Our patient was followed for atotal of 3 years
postoperatively and she remained asymp-tomatic and normocalcemic,
without recurrence.
DiscussionA 52-year-old woman presented with a visible
palpableright-sided neck mass and generalized fatigue. Ultra-sound
showed a complex nodule with solid and cysticcomponents, and a
Sestamibi nuclear scan confirmed aGPTA. Focused surgical neck
exploration was done anda GPTA weighing 7.7 gm was excised. This
case of non-ectopic GPTA is unusual in that it presented mainlywith
visible palpable right-sided neck mass, which waslarge to the
extent that the initial ultrasound suggestedthat it could be a
thyroid nodule.PTAs are well-reported tumors that cause PHPT.
However, when their weight exceeds 3.5 gm, they areclassified as
GPTA [2]. Ours weighed 7.7 gm, and is con-sidered a smaller GPTA in
relation to other reportedcases [7–9].In terms of presentation, the
classic presentation of
PTA is with PHPT accompanied by recurrent kidneystones, and
psychiatric, bone, and gastrointestinal symp-toms [4]. However,
this full constellation of symptoms israrely seen nowadays due to
the more frequent routineassessments of blood chemistries of
patients presentingto hospital and clinics. Hence, such early
detection has
Fig. 1 Ultrasound of the neck showing complex nodule of the
right lobe with solid and cystic components (a) and vascularity
(b)
Al-Hassan et al. Journal of Medical Case Reports (2019) 13:332
Page 2 of 9
-
led to the majority of patients with PHPT now beingidentified
early in the asymptomatic stage [4, 10]. How-ever, our review of
cases of GPTA published during thelast 10 years (Table 1, 20 case
reports, 22 patients withGPTA) shows that only two out of the 22
publishedcases were completely free of signs and symptoms of
hy-percalcemia [15, 17]. On the contrary, Table 1 suggeststhat most
GPTAs presented symptomatically, rangingfrom vague bone/abdominal
pain [5, 11, 18, 24] to moresevere presentations, for example
chronic depression, re-current symptomatic kidney stones, and
severe gastro-intestinal symptoms [1, 9, 13, 14, 19–22]. Very
rarepresentations included hyperparathyroid crisis and
acutepancreatitis [1, 10, 16]. Our current patient presentedwith
neck swelling and generalized fatigue, and she hada silent kidney
stone that was identified by abdominalCT, suggesting that GPTA
generally presents symptom-atically or with signs of hypercalcemia.
This is probablyrelated to the higher levels of calcium produced by
thelarger tumor mass.In terms of physical examination, the majority
of
GPTAs in the neck had a visible and palpable mass inthe neck [7,
11, 13, 14, 17, 19, 20, 24, 25]. Their largesize is one of the
reasons a clinician may suspect thyroiddisease before reviewing the
laboratory results, as palp-able nodules are more common in the
thyroid. In ourcase, a swelling was readily visible on inspection
andpalpable on physical examination.As for diagnostic laboratory
studies, GPTA investiga-
tions start with serum calcium and PTH and proceed to
Fig. 2 Early and late 99mTc-sestamibi scintigraphy parathyroid
scan images of neck and mediastinum anteriorly at 20 minutes and 2
hoursshowing increased focal uptake suggestive of right giant
parathyroid adenoma
Fig. 3 Giant parathyroid adenoma identified intraoperatively
Al-Hassan et al. Journal of Medical Case Reports (2019) 13:332
Page 3 of 9
-
imaging for localization (Table 1). Hypercalcemia and el-evated
PTH are hallmarks of PHPT [4], in agreementwith our review where
all cases had elevated calciumand PTH laboratory values [18]. A
positive correlationbetween the size of a PTA and preoperative PTH
andcalcium levels has also been reported [2, 26, 27].
Calva-Cerqueira et al. (2007) concluded that if preoperativePTH is
> 232 ng/L, there is 95% likelihood of finding aPTA weighing
> 250mg [28]. This is valuable, as sur-geons can have an idea of
tumor size preoperatively. Asfor histopathology, although FNA
cytology (FNA-C) isincreasingly used in the diagnosis of
parathyroid pathology[29], its limitation is that FNA-C cannot
distinguish be-tween different types of parathyroid disease [30].
Our pre-operative FNA-C was non-diagnostic, similar to otherswhere
preoperative FNA-C was non-diagnostic [16].In terms of imaging,
localizing a GPTA is imperative to
guide management. The most commonly used method isa combination
of neck ultrasound and 99mTc-sestamibiscintigraphy (MIBI) scan. The
limitation of neck ultra-sound in GPTA is that it may not show the
extent of a le-sion; when the GPTA is ectopic, a neck ultrasound
willshow no finding [5]. In mediastinal GPTA, neck ultra-sound only
rules out a neck lesion but does not otherwiseaid in localization
[20–22]. In neck GPTA, the combin-ation of a MIBI scan and neck
ultrasound effectively local-izes the GPTA and allows for guided
neck exploration[27]. Ultrasound alone predicts GPTA location with
79%accuracy; combining ultrasound, MIBI, and CT increasesthe
accuracy of localization to 82% [2]. We agree, as inour patient,
that combining neck ultrasound and MIBIscan accurately localizes
the GPTA preoperatively. MIBIscans are more likely to localize GPTA
in patients withhigher preoperative PTH and larger GPTA size; an
aden-oma correctly localized by MIBI has a 95% likelihood
ofweighing > 5.5 gm [28]. As for the location, in agreementwith
most studies, our GPTA was not ectopic [2]; how-ever, Table 1 shows
that the mediastinum is a common lo-cation for ectopic GPTAs,
suggesting that such GPTAsarise in the inferior gland [31, 32].
For the management, the 2014 US National Institutesof Health
(NIH) guidelines for PTA management are ei-ther medical or surgical
if it fulfills specific criteria [6].As with all the cases in Table
1, our case fulfilled thesymptoms and laboratory criteria for
surgery. We under-took MIP, in agreement that it is the preferred
proced-ure [19], and ioPTH monitoring, to confirm removal ofthe PTA
before closure. Although the MIP/ioPTH com-bination is a
gold-standard treatment, comparable out-comes of MIP with and
without ioPTH monitoring havebeen reported [33], with some studies
suggesting thebenefit of ioPTH monitoring is only for patients
withequivocal imaging [34]. This might be an importantfeature to
consider when institutions seek resourceutilization and cost
savings. In agreement with the pointthat ioPTH might be beneficial
only in equivocal im-aging findings, ioPTH monitoring seems to have
limiteduse in GPTA. Only in five instances was ioPTH mon-itoring
undertaken [5, 13, 17, 19, 23] (Table 1), prob-ably because
preoperative imaging and intraoperativevisualization in GPTA leave
little doubt about the lo-cation [28], and because of a high
likelihood of singlegland disease [2]. At our institution, PTA
standard in-traoperative practice is ioPTH monitoring and frozen
sec-tion to confirm excision.Postoperatively, larger sized PTAs may
be associated
with a higher incidence of postoperative hypocalcemia[26]. Table
1 agrees with this, showing that hypocalcemiaoccurred in cases of
larger GPTA. Hungry bone syn-drome, a severe but rare form of
postoperative hypocalce-mia, occurred in four cases (Table 1), all
of which hadGPTAs weighing > 30 gm. Patients with smaller
GPTAswere less likely to have postoperative hypocalcemia.
Ourpatient presented with a 7.7 gm GPTA, considered asmaller GPTA
in relation to other reported cases [7–9],which could explain why
it had a less severe presentationas well as outcome after surgery
compared with the othercases. Postoperatively, our patient, due to
the smallerGPTA, became normocalcemic with normal PTH, andwas not
discharged on any calcium repletion therapy. Our
Fig. 4 Excised giant parathyroid adenoma (4 × 2.5 × 1.5 cm)
Al-Hassan et al. Journal of Medical Case Reports (2019) 13:332
Page 4 of 9
-
Table
1Literature
review
:Casestud
iesof
giantparathyroidaden
oma(2009–2019)
Stud
y*Sex
Age
,years
Side
Presen
tatio
nCa
(mmol/
L)/PTH
(ng/L)
Radiolog
yTreatm
ent
IPTH
Dim
ension
s(m
m)
Weigh
t(g)
Patholog
yPo
stop
erative
complications
a
Thyroidal
Agg
arwalet
al.,
2009
[11]
India
F33
LVisiblesw
elling,
palpable
nodu
le,
bone
pain,R
humerus
andR
pelvicfractures
2.65/762
US:well-d
efined
hypo
echo
iclesion
,posterio
rto
leftlobe
thyroid
Parathyroide
ctom
y(not
specified
)—
95×50
×35
102
Chief
cell
aden
oma
Symptom
atic
hypo
calcem
ia
Salehian
etal.,
2009
[12]
Iran
F53
RVisiblesw
elling,
bone
pain,
nausea,vom
iting
,weigh
tloss
3.65/
1624
USne
ck:h
eteroe
choicmass,inferio
rrig
htlobe
(2×4.8×3cm
);99mTc-M
IBI:
abno
rmalcollectionof
tracer
inR
side
ofne
ck
Neckexploration
andparathyroide
ctom
y(collarne
ckincision
)
—55
×35
×20
30PTA
Nil
Sisodiya
etal.,
2011
[13]
India
F52
RRecurren
tvomiting
4.25/598
US:largehypo
echo
iclesion
inrig
htparatrache
alregion
with
retrosternalextension
Parathyroide
ctom
y,low
anterio
rcervicalapproach
Men
tione
din discussion
39×20
×17
——
Hypocalcemia
Asgharet
al.,
2012
[14]
Pakistan
F55
LParathyroidcrisisb
Palpableno
dule
5.75/
1182
US:largecyst(6×3.7cm
)on
leftside
with
thrombo
sisof
IJV;M
IBI:cystic
lesion
inleftside
neck
displacing
the
thyroidglandon
therig
ht;C
T:large
hypo
denselesion
leftside
ofne
ckwith
perip
heralenh
ancemen
t,retrosternal
extensionandmasseffect
with
deviationof
trache
aandthrombo
sis
ofLIJV
Parathyroide
ctom
yT-shaped
incision
10suspicious-lo
oking
lymph
node
salso
removed
from
levels7and8(by
ENTandthoracic
surgeryteam
s)
—110×70
×60
—PTAwith
prom
inen
tcystic
dege
neratio
n;no
lymph
node
metastasis
Nil
Vilallong
aet
al.,
2012
[10]
Spain
F19
LParathyroidcrisis
3.55/
1207
US:47
×22
mm
nodu
lein
left
thyroidlobe
Hem
ithyroide
ctom
y(it
was
intrathyroidal)
Available,
notused
Max.
diam
eter
3070
Intrathyroidal
PTA
Non
eCalcium
IVd1
,orald2
Neago
eet
al.,
2014
[1]
Romania
(3cases)
M/
F/F
57/
60/
33
R/L/
RC1:Bo
nepain,
abdo
minalpain,
nausea,p
alpable
nodu
leC2:Parathyroidcrisis,
palpable
nodu
leC3:Recurren
tkidn
eyston
es,b
rowntumor
oftib
ia
C1:
3.54/
1780
C2:
4.04/863
C3:
3.15/
1174
MIBI:de
tected
aden
omas
inthe3cases
Bilateraln
eck
explorationand
parathyroide
ctom
y
Not
feasiblec
C1:50
×30
×20
C2:55
×40
×30
—
C1:
30.6
C2:
35.2
C3:>
30
2PTA;1
partially
cysticPTA
C1:Hun
gry
bone
ssynd
rome
C2:Mild
hypo
calcem
iaandhu
ngry
bone
ssynd
rome
C3:Mild
hypo
calcem
ia
Haldaret
al.,
2014
[15]
UK
F61
LAsymptom
atic
3.17/
179.2
US:6cm
massin
Linferio
rcervical
locatio
n;MIBI:pe
rsistent
activity
insamelocatio
n;SPEC
T:tubu
larstructure
insupe
riormed
iastinum
Parathyroide
ctom
y(
selective)
4cm
leftcollar
neck
incision
—65
×30
×15
12PTA
Nil
Garas
etal.,
2015
[5]
UK
F53
LBo
nepain,
palpable
nodu
le3.98/
4038
US:lobu
larwell-d
efined
hypo
echo
iclesion
behind
Llower
poleof
thyroid
gland;
MRI:leftinferio
rPTA,exten
dsde
epinto
med
iastinum
Parathyroide
ctom
y(transverse
cervicalincision
)
Don
e–
94%
redu
ction
in25
minutes
Max.
diam
eter
7027
Chief
cellPTA
Nil
Al-Hassan et al. Journal of Medical Case Reports (2019) 13:332
Page 5 of 9
-
Table
1Literature
review
:Casestud
iesof
giantparathyroidaden
oma(2009–2019)(Con
tinued)
Stud
y*Sex
Age
,years
Side
Presen
tatio
nCa
(mmol/
L)/PTH
(ng/L)
Radiolog
yTreatm
ent
IPTH
Dim
ension
s(m
m)
Weigh
t(g)
Patholog
yPo
stop
erative
complications
a
Rutledg
eet
al.,
2016
[7]
Ireland
F21
REnlargingne
ckmass,
constip
ation,
palpable
nodu
le
2.73/
1305.1
MIBI:lesion
posteriorto
right
lobe
ofthyroidwith
concen
trated
tracer
Rthyroid
lobe
ctom
yand
parathyroide
ctom
ywith
level6
neck
dissectio
n(suspe
cted
carcinom
a)
—80
×55
×30
58.8
AtypicalP
TASymptom
atic
hypo
calcem
ia,
hung
rybo
nesynd
rome
Krishn
amurthy
etal.,2016
[16]
India
M50
LRecurren
tattacksof
acutepancreatitis,
palpable
fullness
2.77/669
CT:6×4cm
massin
Lparatrache
alregion
with
extensionto
supe
rior
med
iastinum
;PET–C
T:isolated
uptake,
leftparatrache
alregion
;MIBI:localized
toLinferio
rparathyroidgland;
Preo
perativeFN
A-C
was
done
d
Parathyroide
ctom
yviatranscervicalapp
roach
—Max.
diam
eter
6020
PTA
Hypocalcemia
Castroet
al.,
2017
[17]
Spain
F40
LAsymptom
atic,
palpable
nodu
le3.35/825
US:solid
lesion
behind
Lthyroidlobe
;SPEC
T:intenseup
take,b
ackof
Lthyroidlobe
inearly
andlate
phases
Parathyroide
ctom
y(not
specified
)Don
e,90%
redu
ction
64×16
×20
10.8
PTA
Hypocalcemia
Sahsam
anis
etal.,2017
[18]
Greece
F42
LAbd
ominalpain
2.60/151
US:en
larged
parathyroidglandon
lower
side
ofcervicalregion
;MIBI:
largeconcen
trations
ofradiotracer
inthesamelocatio
n
Minim
allyinvasive
parathyroide
ctom
yNot
done
33×20
×14
5.39
PTA
Nil
Mantzoros
etal.,
2018
[19]
Greece
F73
RNecksw
elling,
bone
pain
3.63/
1629
US:hypo
echo
icno
duleat
inferio
rpo
leof
therig
htthyroid;
MIBI:hype
rfunctio
ning
rightlower
parathyroidgland
Minim
allyinvasive
parathyroide
ctom
yDon
e,95%
redu
ction
20minutes
after
removal
50×25
×25
30PTA
Hun
grybo
nesynd
rome
Med
iastinal
Migliore
etal.,
2013
[8]
Italy
F65
RPersistent
hype
rcalcemiae
Both
elevated
CT:7cm
massin
posterior
med
iastinum
;MIBI:confirm
edtheCTfinding
Vide
o-assisted
minith
oracotom
y—
—95
PTA
Nil
Tagh
aviK
ojidi
etal.,2016
[20]
Iran
M70
Mid
Ano
rexia,nausea,b
one
pain,con
stipation,
symptom
atickidn
eyston
es,p
olydipsia
3.60/930
US:multip
leisoe
choicno
dules,no
parathyroidglands
seen
;MIBI:focal
radiotraceraccumulation,
midline
anterio
rchestwall;CT:softtissue
density
mass,mild
enhancem
ent,
anterio
rmidline,xiph
oidlevel
Surgicalremoval
(not
specified
)f
——
75Active
parathyroid
lesion
Hypocalcemia
Pechevaet
al.,
2016
[21]
UK
F72
RDep
ression,
severe
osteop
orosis
(T=−3.2)
3.02/
250.8
US:no
parathyroidlesion
;MIBI:no
eviden
ceof
PTA;C
T:complex
cystic
solid
massin
themed
iastinum
Parathyroide
ctom
yviaVA
TSNot
used
,em
erge
ncy
—19
PTA
Hoarsen
ess,
bovine
coug
h
Talukder
etal.,
2017
[22]
India
F49
Mid
Brow
ntumor
14.07/
1000
US:no
abno
rmalparathyroidgland;
MIBI:tracer-avidlesion
inanterio
rmed
iastinum
;PET-CT:ectopic
parathyroidtissuein
anterio
rmed
iastinum
behind
manub
rium
sterni
Parathyroide
ctom
yviacervicalcollar
incision
and
hemisternotom
y
—40
×30
×20
12Neuroen
docrine
celltumor
Nil
Al-Hassan et al. Journal of Medical Case Reports (2019) 13:332
Page 6 of 9
-
Table
1Literature
review
:Casestud
iesof
giantparathyroidaden
oma(2009–2019)(Con
tinued)
Stud
y*Sex
Age
,years
Side
Presen
tatio
nCa
(mmol/
L)/PTH
(ng/L)
Radiolog
yTreatm
ent
IPTH
Dim
ension
s(m
m)
Weigh
t(g)
Patholog
yPo
stop
erative
complications
a
Garun
aMurthee
etal.,2018
[9]
UK
M72
Mid
Ano
rexia,lethargy,
abdo
minalcram
ps,
constip
ation,
weigh
tloss
15.19/
1867.1
CXR
:sizeablemed
iastinalmass;CT:
9cm
solid
cysticanterio
rmed
iastinal
tumor;M
IBI:he
teroge
neou
stracer
uptake
inthemed
iastinalmass
Med
ialsternotom
yandtotalthymectomy
—Maxim
umdiam
eter
78220
IntrathymicPTA
Nil
Miller
etal.,
2019
[23]
UK
M53
Mid
Asymptom
atic
renalstone
s11.22/
179.2
MIBI:linearregion
ofincreased
intensity
intheleftmed
iastinum
Parathyroide
ctom
yviatranscervicale
xcision
Don
e,81%
redu
ction
after10
minutes
80×30
×30
30.9
PTA
Nil
—no
trepo
rted
,can
notbe
inferred
,C1Case1,
C2Case2,
C3Case3,
CTcompu
tedtomog
raph
y,CX
RchestX-ray,EN
Totolaryn
gology
,Ffemale,FN
A-C
fine-ne
edle
aspiratio
ncytology
,IPTHintrao
perative
parathyroidho
rmon
e,IJVinternal
jugu
larvein,L
left,M
male,Mid
midlin
e,MIBIT
c99m-sestamibiscintigraph
yscan
,PET
positron
emission
tomog
raph
y,PTApa
rathyroidad
enom
a,PTHfin
e-ne
edle
aspiratio
ncytology
,Rrig
ht,SPECT
sing
leph
oton
emission
compu
tedtomog
raph
y,USultrasou
nd,V
ATS
vide
o-assisted
thoracoscopicsurgery
*Due
tospacelim
itatio
ns,o
nlythefirst
author
ismen
tione
daAllof
thecasesha
dasym
ptom
aticpa
tientswith
norm
alized
Caan
dfin
e-ne
edle
aspiratio
ncytology
onfollow
up(excep
tHalda
r+Sisody
a–Caon
ly)
bPa
rathyroidcrisiscomprises
anorexia,u
rinaryfreq
uency,severe
nausea,vom
iting
,con
stipation
cDon
e1ho
urpo
stop
erativefor2cases,foun
dto
beno
rmal
dPreo
perativ
efin
e-ne
edle
aspiratio
ncytology
show
edabe
nign
epith
eliallesionthat
couldno
tbe
furthe
rcharacterized
ePa
tient
hadprevious
totalthy
roidectomyforgo
iterassociated
with
hype
rcalcemicsynd
rome(exp
loratio
nha
dshow
edfour
norm
alpa
rathyroidglan
ds)
fPa
tient
hadprevious
totalp
arathy
roidectomy,thym
ectomy,an
drig
hthe
mith
yroide
ctom
y
Al-Hassan et al. Journal of Medical Case Reports (2019) 13:332
Page 7 of 9
-
patient was followed for a total of 3 years postoperativelyand
she remained asymptomatic and normocalcemic,without recurrence.
This fits with outcomes reported in astudy following patients for
an average of 40months,where all patients remained normocalcemic
and there wasno recurrence during this time, even in those with
suspi-cious histologic features [27].
ConclusionGPTA is a rare subset of PTAs that weigh > 3.5 gm,
it isbenign, but can manifest with the symptoms of
extremehypercalcemia. From our literature review, we concludethat
GPTA generally presents symptomatically, withhigh preoperative PTH
and serum calcium directly pro-portional to the adenoma weight. The
most accuratemethod for localizing a GPTA is a combination of
neckultrasound and MIBI scan. MIP with intraoperative PTHmonitoring
is the suggested management, although theneed for the ioPTH
monitoring is debatable in GPTAdue to their large size and accuracy
of preoperativeimaging.
AbbreviationsCBC: Complete blood count; CT: Computed tomography;
FNA: Fine-needleaspiration; FNA-C: Fine-needle aspiration cytology;
GPTA: Giant parathyroidadenoma; ioPTH: Intraoperative parathyroid
hormone; MIBI: 99mTc-sestamibiscintigraphy; MIP: Minimally invasive
parathyroidectomy; NIH: US NationalInstitutes of Health; PHPT:
Primary hyperparathyroidism; PTA: Parathyroidadenoma; PTH:
Parathyroid hormone; TSH: Thyroid-stimulating hormone
AcknowledgementsNot applicable.
Authors’ contributionsMM and WEA drafted the manuscript; AA
contributed to the writing of themanuscript. MM, AA, and MAH
acquired the clinical data. WEA, MM, and AAdeveloped the structure
and arguments of the paper. WEA, MM, AA, andMAH made important
revisions and approved the final version of themanuscript. All
authors agreed with the manuscript conclusions andreviewed and
approved the final manuscript.
FundingNot applicable
Availability of data and materialsData sharing is not applicable
to this article as no datasets were generatedor analyzed during the
current study.
Ethics approval and consent to participateEthics approval and
consent to publish provided: Medical Research Centrereview board,
Institutional Review Board (IRB), protocol #0419113, HamadMedical
Corporation, Doha, Qatar.
Consent for publicationEthics approval and consent to publish
provided (Medical Research Centrereview board, IRB, #0419113, Hamad
Medical Corporation, Doha, Qatar).Written informed consent was
obtained from the patient for publication ofthis case report and
any accompanying images. A copy of the writtenconsent is available
for review by the Editor-in-Chief of this journal.
Competing interestsThe authors declare that they have no
competing interests.
Author details1Department of General Surgery, Hamad General
Hospital, Hamad MedicalCorporation, Doha, Qatar. 2Weill Cornell
Medicine-Qatar, Doha, Qatar.3Department of Surgery, Hamad General
Hospital, Hamad MedicalCorporation, Doha, Qatar. 4College of
Medicine, Qatar University, Doha, Qatar.5School of Health and
Education, University of Skövde, Skövde, Sweden.6Department of
Medical Imaging, Hamad General Hospital, Hamad MedicalCorporation,
Doha, Qatar.
Received: 11 June 2019 Accepted: 10 September 2019
References1. Neagoe RM, Sala DT, Borda A, Mogoanta CA, Muhlfay
G. Clinicopathologic
and therapeutic aspects of giant parathyroid adenomas - three
case reportsand short review of the literature. Romanian J Morphol
Embryol. 2014;55(2Suppl):669–74.
2. Spanheimer PM, Stoltze AJ, Howe JR, Sugg SL, Lal G, Weigel
RJ. Do giantparathyroid adenomas represent a distinct clinical
entity? Surgery. 2013;154(4):714–8; discussion 8-9
3. Power C, Kavanagh D, Hill AD, O'Higgins N, McDermott E.
Unusualpresentation of a giant parathyroid adenoma: report of a
case. Surg Today.2005;35(3):235–7.
4. Madkhali T, Alhefdhi A, Chen H, Elfenbein D. Primary
hyperparathyroidism.Ulus Cerrahi Derg. 2016;32(1):58–66.
5. Garas G, Poulasouchidou M, Dimoulas A, Hytiroglou P, Kita M,
Zacharakis E.Radiological considerations and surgical planning in
the treatment of giantparathyroid adenomas. Ann R Coll Surg Engl.
2015;97(4):e64–6.
6. Bilezikian JP, Brandi ML, Eastell R, Silverberg SJ, Udelsman
R, Marcocci C,et al. Guidelines for the management of asymptomatic
primaryhyperparathyroidism: summary statement from the Fourth
InternationalWorkshop. J Clin Endocrinol Metab.
2014;99(10):3561–9.
7. Rutledge S, Harrison M, O'Connell M, O'Dwyer T, Byrne MM.
Acutepresentation of a giant intrathyroidal parathyroid adenoma: a
case report. JMed Case Rep. 2016;10(1):286.
8. Migliore M, Pulvirenti G, Okatyeva V, Cannizzaro MA.
Persistenthyperparathyroidism owing to a giant parathyroid adenoma
in posteriormediastinum. Surgery. 2013;154(1):132–3.
9. Garuna Murthee K, Tay WL, Soo KL, Swee DS. A Migratory
Mishap: GiantMediastinal Parathyroid Adenoma. Am J Med.
2018;131(5):512–6.
10. Vilallonga R, Zafon C, Migone R, Baena JA. Giant
intrathyroidal parathyroidadenoma. J Emerg Trauma Shock.
2012;5(2):196–8.
11. Aggarwal V, Mishra A, Bhargav PR, Ramakant P. Giant
parathyroid adenoma.ANZ J Surg. 2009;79(1–2):91.
12. Salehian M, Namdari O, Mohammadi SS, Feazli YH.
Primaryhyperparathyroidism due to a giant parathyroid adenoma: a
case report. IntJ Endocrinol Metabol. 2009;9(2):101–5.
13. Sisodiya R, Kumar S, Palankar N, BVD. Case report on giant
parathyroidadenoma with review of literature. Indian J Surg.
2011;75(Suppl 1):21–2.
14. Asghar A, Ikram M, Islam N. A case report: Giant cystic
parathyroid adenomapresenting with parathyroid crisis after Vitamin
D replacement. BMC EndocrDisord. 2012;12:14.
15. Haldar A, Thapar A, Khan S, Jenkins S. Day-case minimally
invasive excisionof a giant mediastinal parathyroid adenoma. Ann R
Coll Surg Engl. 2014;96(5):e21–3.
16. Krishnamurthy A, Raghunandan GC, Ramshankar V. A rare case
of giantparathyroid adenoma presenting with recurrent episodes of
pancreatitis.Indian J Nucl Med. 2016;31:36–8.
17. Castro MA, López AA, Fragueiro LM, García NP. Giant
parathyroid adenoma:differential aspects compared to parathyroid
carcinoma. EndocrinolDiabetes Metab Case Rep. 2017;2017:1.
18. Sahsamanis G, Gkouzis K, Samaras S, Pinialidis D,
Dimitrakopoulos G. Surgicalmanagement of a giant parathyroid
adenoma through minimal invasiveparathyroidectomy. A case report.
Int J Surg Case Rep. 2017;31:262–5.
19. Mantzoros I, Kyriakidou D, Galanos-Demiris K, Chatzakis C,
Parpoudi S,Sapidis N, et al. A Rare Case of Primary
Hyperparathyroidism Caused by aGiant Solitary Parathyroid Adenoma.
Am J Case Rep. 2018;19:1334–7.
20. Taghavi Kojidi H, Vagharimehr N, Mohseni S, Pajouhi M,
Mohajeri-TehraniMR. Unusual Ectopic Parathyroid Adenoma: A Case
Report. Acta Med Iran.2016;54(8):547–50.
Al-Hassan et al. Journal of Medical Case Reports (2019) 13:332
Page 8 of 9
-
21. Pecheva M, Mahendran K, Kadlec J, Lofthouse M, Van Tornout
F.Mediastinal giant parathyroid adenoma-a minimally invasive
mediastinalsurgical approach for an emergency presentation. Ann
CardiothoracSurg. 2016;5(1):70–3.
22. Talukder S, Behera A, Bhadada SK, Mitra S. Giant mediastinal
parathyroidadenoma presenting as bilateral brown tumour of
mandible: a rarepresentation of primary hyperparathyroidism. BMJ
Case Rep. 2017;2017(11):bcr–2017.
23. Miller BJ, Isaacs K, Khan E, Palazzo FF. Transcervical
excision of a giantmediastinal parathyroid adenoma. BMJ Case Rep.
2019;12(2) https://doi.org/10.1136/bcr-2018-228292.
24. Korukluoglu B, Ergul E, Yalcin S. Giant intrathyroidal
parathyroid cysticadenoma. J Pak Med Assoc. 2008;58(10):592.
25. Desigan S, Syed R, Conway GS, Kurzawinski TR, Bomanji JB.
Giant cervicalparathyroid adenoma mimicking a sternocleidomastoid
mass and presentingas a brown tumor of the mandible. Clin Nucl Med.
2007;32(4):306–8.
26. Zamboni WA, Folse R. Adenoma weight: a predictor of
transienthypocalcemia after parathyroidectomy. Am J Surg.
1986;152(6):611–5.
27. Abdel-Aziz TE, Gleeson F, Sadler G, Mihai R. Dwarfs and
Giants ofParathyroid Adenomas-No Difference in Outcome After
Parathyroidectomy.J Surg Res. 2019;237:56–60.
28. Calva-Cerqueira D, Smith BJ, Hostetler ML, Lal G, Menda Y,
O'Dorisio TM,et al. Minimally invasive parathyroidectomy and
preoperative MIBI scans:correlation of gland weight and
preoperative PTH. J Am Coll Surg. 2007;205(4 Suppl):S38–44.
29. Heo I, Park S, Jung CW, Koh JS, Lee SS, Seol H, et al. Fine
needle aspirationcytology of parathyroid lesions. Korean J Pathol.
2013;47(5):466–71.
30. Kumari N, Mishra D, Pradhan R, Agarwal A, Krishnani N.
Utility of fine-needle aspiration cytology in the identification of
parathyroid lesions. JCytol. 2016;33(1):17–21.
31. Phitayakorn R, McHenry CR. Incidence and location of ectopic
abnormalparathyroid glands. Am J Surg. 2006;191(3):418–23.
32. LoPinto M, Rubio GA, Khan ZF, Vaghaiwalla TM, Farra JC, Lew
JI. Location ofabnormal parathyroid glands: lessons from 810
parathyroidectomies. J SurgRes. 2017;207:22–6.
33. Mihai R, Palazzo FF, Gleeson FV, Sadler GP. Minimally
invasiveparathyroidectomy without intraoperative parathyroid
hormone monitoringin patients with primary hyperparathyroidism. Br
J Surg. 2007;94(1):42–7.
34. Khan AA, Khatun Y, Walker A, Jimeno J, Hubbard JG. Role of
intraoperativePTH monitoring and surgical approach in primary
hyperparathyroidism. AnnMed Surg (Lond). 2015;4(3):301–5.
Publisher’s NoteSpringer Nature remains neutral with regard to
jurisdictional claims inpublished maps and institutional
affiliations.
Al-Hassan et al. Journal of Medical Case Reports (2019) 13:332
Page 9 of 9
https://doi.org/10.1136/bcr-2018-228292https://doi.org/10.1136/bcr-2018-228292
AbstractBackgroundCase presentationConclusions
BackgroundCase presentationSurgical
techniqueDiscussionConclusionAbbreviationsAcknowledgementsAuthors’
contributionsFundingAvailability of data and materialsEthics
approval and consent to participateConsent for publicationCompeting
interestsAuthor detailsReferencesPublisher’s Note