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Jou nal o Su gical Case Repo s, 2024, 5, jae353
h ps://doi.o g/10.1093/jsc / jae353
Case Repo
Case Repo
Gian pleomo phic lipoma in pa ien wi h mul iple
os eochond oma osis
Benjamin Tho pe 1,*,Paloma Lage2,Ca olina Bei as1,Kelly Ba gas-Oso io3,F ancisco Canseco2
1Gene al Su ge y Depa men , Hospi al Clinico Uni e si a io de San iago de Compos ela, Rua Choupana s/n, San iago de Compos ela 15706, Spain
2Plas ic Su ge y Depa men , Hospi al Clinico Uni e si a io de San iago de Compos ela, Rua Choupana s/n, San iago de Compos ela 15706, Spain
3Pa hology Depa men , Hospi al Clinico Uni e si a io de San igao de Compos ela, Rua Choupana s/n, San iago de Compos ela 15706, Spain
*Co esponding au ho . Gene al su ge y depa men , Hospi al Clinico Uni e si a io de San iago de Compos ela, San iago de Compos ela, Rua Diego de Mu os 10,
1 izquie da, San iago de Comspo ela 15701, Spain. E-mail: Benjamin.Tho pe@ho mail.com, Benjaminal onso. ho pe@ ai.usc.es
Abs ac
Pleomo phic lipomas a e in equen so issue umou s wi h pseudosa coma ous beha iou . Thei polymo phism p o ides hem
ce ain cha ac e is ics ha may esemble malignancy, which may mislead he ini ial diagnosis. The p esen ed case epo is a 45-yea -
old man wi h a gian g owing umou on he le ce icoscapula egion ini ially ca ego ised as a liposa coma by magne ic esonance
wi h a inal con i med diagnosis o pleomo phic lipoma a e he su gical esec ion and he examina ion o he pa hologis . The pa ien
p esen ed impo an unc ional es ic ion o he uppe le ex emi y, which dec eased a e su gical esec ion, imp o ing he quali y
o li e.
Keywo ds:pleomo phic lipoma; gian lipoma; lipoblas s
In oduc ion
Pleomo phic lipomas (PLs) a e benign so issue umou s o adi-
pose o igin, clinically slow g owing and gene ally asymp oma ic,
which appea mo e equen ly on he shawl egion o middle-aged
men. Those acqui ing a size la ge han 10 cm o a weigh hea ie
han 1000 g (<1% o PL) can be ca ego ized as gian PL [1,2]. Gian
PL can cause symp oms such as pain o mo emen es ic ions
impai ing he quali y o li e o he pa ien [1–3].
De ini i e diagnosis is no always clea as PLs ha e a pseudosa -
coma ous beha iou ha can mimic malignancy e en hough
he e is no isk o me as asis o malignan ans o ma ion. Radi-
ologically, PLs lack pa hognomonic diagnos ic signs, esembling,
on ce ain occasions, agg essi e umou s and leading o an ini ial
misdiagnosis. Clinical p esen a ion and p eope a i e biopsies a e
c ucial o elucida e he diagnosis o he pa ien and a oid adi-
cal su ge y when no needed [4–6]. Pa hological analyses show-
cases lo e -like mul inuclea ed gian cells (CD34+and S100−),
lipoblas s, and ma u e adipocy es (S100+) in a myxoid s oma
wi h no signs o nec osis o mi o ic ac i i y sugges ing malignancy
[6–8]. Gene ic molecula s udies o MDMD2 and CDK4 in PLs a e
nega i e [2,8,9].
Case epo
A 45-yea -old man was admi ed o he eme gency depa men o
ou hospi al as a esul o a discon inuous bleeding in ela ion o
a g owing umou loca ed on his le shoulde (Fig. 1). The pa ien
desc ibed a lesion o a leas 20 yea s o e olu ion ha s a ed
g owing e y apidly o e he pas 12 mon hs. Recen ly, he pa ien
s a ed no icing equen blood spo ing on his clo hing, and o
his eason,he a ended he eme gency depa men .In spi e o he
size o he lesion and unc ional es ic ion o he le uppe limb,
he pa ien decla ed himsel capable o accomplishing his wo k
obliga ions and daily ac i i ies wi hou needing any help. The
pa ien p esen ed a p e ious diagnosis o os eochond oma osis
and emained o he wise heal hy.
Following a physical examina ion, he pa ien p esen ed no -
mal ensional alues, no achyca dia, and no signs o paleness o
he skin. An e iden 20 cm mass a ached o he ce icoscapula
egion o his le shoulde was ound hanging om a pedicle
∼10 cm in diame e . The lesion was dependen on he subcu-
aneous issue and showed ini ial signs o cu aneous p essu e
ulce s. Signs o skin in ec ion we e ound bu no ac i e bleeding
was p esen . The lesion limi ed he pa ien ’s mobili y o he
uppe le ex emi y when ying o aise he shoulde wi h an
ex ended a m; howe e , he emaining shoulde mo emen s we e
p ese ed.
An ini ial s udy was pe o med a he eme gency depa men
o dismiss ex ended disease in case o inal p o en malignancy.
The labo a o y esul s we e wi hin no mal alues; no anaemia
was de ec ed. Simple adiog aphy desc ibed a gian so issue
umou and bone de o mi ies compa ible wi h os eochond o-
ma osis (Fig. 2). The CT scan desc ibed a oluminous umou o
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2|Tho pe e al.
Figu e 1. Gian so issue umou on he le scapula egion; ini ial
signs o cu aneous p essu e ulce s.
Figu e 2. Simple adiog aphy: e iden so issue umou on he le
shoulde wi h mul iple bone de o mi ies compa ible wi h
os eochond oma osis.
adipose issue in he scapula egion o he le shoulde oge he
wi h o he bone lesions compa ible wi h mul iple os eochond o-
ma osis. No me as ases we e desc ibed in his s udy. The pa ien
was discha ged wi h o al an ibio ics and he use o a shoulde
sling o p e en u he complica ions due o he weigh o he
umou .
Du ing he ollow-up, a magne ic esonance (Figs 3 and 4)was
pe o med desc ibing a so issue lesion highly sugges i e o
liposa coma as a i s possibili y diagnosis, wi h an addi ion image
sugges ing me as a ic axilla y lymph node. A e he imaging
esul s, biopsies we e aken and analysed by he pa hologis wi h
a inal diagnosis o PL.
Immunohis ochemis y s udies e ealed a di use and in ense
CD34 ac i i y on he usocelula and pleomo phic componen and
S100 immuno eac i i y on he adipose componen . CK AE1/AE3,
EMA, AML, Desmin, SOX100, HMB45, CD31, STAT6, MDM2, and
Figu e 3. Magne ic esonance imaging T2 wi h con as : gian
exc escen lesion o 19 ×18 ×14.4 cm o diame e loca ed in he
subcu aneous issue o he pos e io aspec o he scapula di ided by
mul iple sep ums; axilla y lymph node o ∼0.9 cm; images compa ible
wi h liposa coma wi h a me as a ic axilla y lymph node.
Figu e 4. Magne ic esonance imaging T1: polylobula ed lesion wi h
mul iple sep a sepa a ing di e en a eas o a issue.
CDK4 we e also es ed bu esul ed nega i e. The FISH molecula
s udy o he gene MDM2 was no ampli ied.
A e he inal diagnosis, su ge y was scheduled in an ambula-
o y egime. Su gical excision o he umou was pe o med gen-
e a ing a skin a ea de ec on he le shoulde co e ed wi h a skin
g a om he la e al aspec o he le high o he pa ien . The
su gical piece p esen ed lipoma ous mac oscopic cha ac e is ics
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Gian pleomo phic lipoma |3
Figu e 5. Mac oscopic indings: solid lesion o 21 ×16 ×17cm o
lipoma ous cha ac e is ics wi h ocus o a nec osis compa ible
wi h PL.
Figu e 6. Mic oscopic indings (H & E ×40): well-ci cumsc ibed lesion a
he p o ound de mis, composed o ma u e adipocy es, wi h occasional
pleomo phic lipoblas s and spindle cells in he s oma some o which
p esen ed ‘ lo e ’-like mo phology; ocal a eas o nec osis and absence
o mi o ic ac i i y; in ense and di use esponse o CD34.
wi h a o al weigh o 3110 g (Fig. 5). Unde he mic oscope, lo e -
like spindle cells a e p esen ed, embedded oge he wi h ma u e
adipocy es (Fig. 6).
The pa ien was discha ged on he same day o he su ge y wi h
no immedia e su gical complica ions. The skin g a emained
wi h good ascula iza ion and cica isa ion (Fig. 7). Healing was
adequa e, wi h some a eas o skin hype ophy ha needed ea -
men wi h opic co icoids.
Rehabili a ion he apy was s a ed a e he su ge y o imp o e
he ange o mo emen o he shoulde ; howe e a e 1 yea , he
abduc ion achie ed on he le shoulde was a maximum o 90
deg ees. A Z - plas y- ype su ge y was pe o med o elonga e he
con ac ed sca and inc ease he pa ien ’s mo emen capabili y.
Discussion
PLs a e a sub ype o lipomas wi h a speci ic clinical p esen a ion
pa e n and mic oscopic cha ac e is ics ha gene a e an en i y
Figu e 7. Skin g a a e 3 week o he su ge y; co ec signs o healing,
good ascula iza ion, no nec osis.
by hemsel es, di e en o o he so issue umou s o adipose
o igin [8].
The mos common loca ion is he shoulde , he ce icoscapula
egion, and he uppe back, appea ing as a slow-g owing, painless,
mobile, and soli a y lesion dependen o he subcu aneous issue.
These umou s a e mo e equen in men in compa ison wi h
women in a a io o 9:1 and al hough hey can occu a any age,
hey end o appea a ound he i h and se en h decade o he
li e. Size a ies om 1.0 o 14.0 cm, being gene ally smalle han
5cm[
6,8]. Sanchez e al. ca ego ized gian lipomas as hose bigge
han 10 cm in a leas one dimension o hose hea ie han 1000 g
[1,2], as is he case in his epo wi h a o al weigh o mo e han
3000 g.
Gian lipomas a e in equen ep esen ing <1% o lipomas.
Besides causing cosme ic and physical de o mi ies, hey a ec
he quali y o li e o he pa ien by causing comp ession, pain,
and by diminishing hei mo emen capaci y [2,3]. Recen e iews
disca d PL me as a ic po en ial isk o malignan ans o ma ion
[6]. Howe e , Johnson e al. ecommend ha e e y so issue
umou la ge han 5 cm should be managed as malignan un il
p o en o he wise [10].
So issue umou s om benign and malignan o igin can
mimic clinical p esen a ion needing he mic oscopic analysis o
help cla i y he di e en ial diagnosis [6]. Unde he mic oscope,
PLs a e cha ac e ized o p esen ing ypical lo e -like mul inucle-
a ed gian cells (CD34+and S100−) embedded in a myxoid s oma
wi h a a iable dis ibu ion o ma u e adipocy es (S100+). No
signs o in il a i e g ow h, nec osis, o mi o ic ac i i y a e ound
[6–8]. The p esence o lipoblas s is no uncommon in PLs, being
p esen in a ound 66% o he cases; he e o e, i is no conside ed
a dis inguishable clinical ea u e o liposa coma [9,11].
Typical immunochemis y esul s p esen nega i e desmin al-
ues, posi i e CD34, and nega i e S100 (posi i e S100 in he p es-
ence o ma u e adipocy es). Molecula gene ic es ing is consid-
e ed he mos eliable diagnos ic ool [11],showingalacko
ampli ica ion o MDM2 o CDK4 in PLs, which helps di e en ia e
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4|Tho pe e al.
hem om o he so issue umou s such as a ypical lipoma ous
umou s o dedi e en ia ed liposa comas (DDLS), which ha e
posi i e nuclea exp ession o MDM2 o /and CDK4 in mos cases
[9,12,13].
To comp ehend he adiog aphic beha iou o PLs is impo -
an ; on ce ain occasions, supe imposable images o malignan
lesions can lead o he misdiagnosis o agg essi e umou s [5,
9]. In con as wi h an MRI, PL shows in ense enhancemen o
he nonadipose componen dis inguishing PL om con en ional
lipomas; howe e , simila beha iou is ound in so - issue sa -
coma, which can lead o an e oneous diagnosis [5,12,13]. The
adipose componen is e y a iable in PLs (25%–75%). Low a
PLs can esemble mo e agg essi e umou s like DDLS o non-
adipocy ic sa comas [4,6]. Vascula iza ion analysis is also key,
a ying om a disc ee ascula iza ion o an abundan plexi o m
ascula iza ion, which can simula e malignan umou s [4]. The e
a e no pa hognomonic signs o PLs on diagnos ic imaging, which
can delay he inal diagnosis, he eason is clinical p esen a ion
and p eope a i e biopsies a e i al o elucida e he diagnosis o
he pa ien and a oid adical su ge y when no needed [4,5].
Su ge y by simple excision wi h ee ma gins is conside ed he
gold s anda d wi h low a e o ecu ence. E en hough some
s udies decla e be e cosme ic ou comes and lesse mo bidi y
when lipomas a e ea ed by suc ion mechanisms, open su ge y
p o ides a smalle a e o complica ions, ne e damage, and a
lowe a e o ecu ence [5,14].
Conclusions
PLs can emula e malignan so issue umou s in e ms o clinical
p esen a ion, adiological imaging, and his opa hological ind-
ings, leading om he daily clinical p ac ice o an e oneous ini ial
diagnosis. I is impo an o es ablish a mul idisciplina y app oach
helped by an adequa e imaging and pa hology expe s ha may
help o es ablish a co ec inal diagnosis and hence plan he ype
o su gical in e en ion. F ee ma gins su ge y p o ides good and
sa e oncologic esul s wi h low ecu ency a es.
Con lic o in e es s a emen
None decla ed.
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