Endoscopic Snapsho
GE Po J Gas oen e ol 2022;29:209–211
Idiopa hic Ch onic Pneuma osis Cys oids
In es inalis wi h Benign Pneumope i oneum:
A Ra e Endoscopic Finding
Cláudia Macedo
a Elisa G a i o-Soa es
a, b Ma a G a i o-Soa es
a, b
Ped o Ama o
a Rui Cae ano-Oli ei a
c Ped o Figuei edo
a, b
a Gas oen e ology Depa men , Cen o Hospi ala e Uni e si á io de Coimb a, Coimb a, Po ugal; b Facul y o
Medicine, Uni e si y o Coimb a, Coimb a, Po ugal; c Pa hology Depa men , Cen o Hospi ala e Uni e si á io de
Coimb a, Coimb a, Po ugal
Recei ed: No embe 22, 2020
Accep ed: Janua y 21, 2021
Published online: Ma ch 23, 2021
Cláudia Macedo
Gas oen e ology Depa men
Cen o Hospi ala e Uni e si á io de Coimb a
P ace a P o . Mo a Pin o, PT–3000-075 Coimb a (Po ugal)
claudia.macedo.07 @ ho mail.com
© 2021 Sociedade Po uguesa de Gas en e ologia
Published by S. Ka ge AG, Basel
ka ge @ka ge .com
www.ka ge .com/pjg
DOI: 10.1159/000514723
Keywo ds
Pneuma osis cys oids in es inalis · Colon · Endoscopy ·
Idiopa hic · Pneumope i oneum
Pneuma ose cís ica in es inal c ónica idiopá ica com
pneumope i oneu benigno: um achado endoscópico
a o
Pala as Cha e
Pneuma ose cís ica in es inal · Cólon · Endoscopia ·
Idiopá ica · Pneumope i oneu
A 51-yea -old man, o me smoke (20 pack-yea s),
wi h hemo hoidal disease, i i able bowel synd ome,
pulmona y emphysema and a mechanical p os he ic ao -
ic al e unde wa a in, p esen ed o pos -polypec omy
su eillance colonoscopy. He complained o equen e-
nesmus, dia hea (2–3 liquid dejec ions in he mo ning),
bloa ing and abdominal discom o . Se e al polypoid
g ape-like submucosal masses we e iden i ied in a 10-cm
segmen o he descenden colon (Fig.1a, b). Since he le-
sions we e easily inden ed wi h gen le p essu e and had a
bluish hue, pneuma osis cys oids in es inalis (PCI) was
suspec ed. Biopsy o one o hese masses caused immedi-
a e de la ion, con i ming he diagnosis. His opa hologi-
cal e alua ion showed no mal c yp a chi ec u e and a
conges i e edema ous lamina p op ia wi h minimal in-
lamma o y in il a e sugges ing eac i e changes (Fig.2).
The pa ien had no his o y o abdominal auma and no
pha macological (alpha-glucosidase inhibi o s, s e oids)
o in ec ious (Clos idioides di icile, ube culosis, human
immunode iciency in ec ion) causes. O e a long- e m
ollow-up o 12 yea s, he mild symp oms we e managed
conse a i ely wi h lope amide, spasmoly ics and p obi-
o ics, which showed an imp o emen . In his pe iod, wo
su eillance colonoscopies we e pe o med showing he
same unchanged endoscopic indings desc ibed abo e.
Recen ly, due o su eillance o an ascending ao ic aneu-
ysm, a con as ed ches compu ed omog aphy (CT)
iden i ied a small pneumope i oneum in he le sub-
ph enic egion. Abdominal CT ex ension showed mo e
ee pe i oneal ai in he le hypochond ium and ai bub-
bles adjacen o he lumen o he bowel, some o hem be-
ing subse osal (Fig.3a, b).
The p e alence o PCI is 0.03% in he gene al popula-
ion and i s pa hogenesis is poo ly unde s ood [1]. PCI is
cha ac e ized by he p esence o gas, o ming mul ilocu-
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NonComme cial-NoDe i a i es 4.0 In e na ional License (CC BY-
NC-ND) (h p://www.ka ge .com/Se ices/OpenAccessLicense).
Usage and dis ibu ion o comme cial pu poses as well as any dis-
ibu ion o modi ied ma e ial equi es w i en pe mission.
Macedo e al.
GE Po J Gas oen e ol 2022;29:209–211
210
DOI: 10.1159/000514723
la cys s, in he in es inal wall anging om an inciden al
inding o a li e- h ea ening in a-abdominal condi ion
[1, 2]. In 85% o cases, PCI is seconda y o se e al condi-
ions including immunological dis u bances, in ec ions,
pulmona y diso de s and diseases a ec ing gas oin es i-
nal mo ili y [3, 4]. In hese cases, he main symp oms and
also ea men s a e ela ed o he p ima y disease. In he
emaining 15%, PCI is idiopa hic [3]. Mos pa ien s a e
asymp oma ic o paucisymp oma ic and do no need
ea men [1, 2]. When symp oma ic, PCI can p esen
ab
ab
Fig. 1. a, b Colonoscopy showing polypoid g ape-like submucosal masses in he descenden colon.
Fig. 2. Pa hological indings wi h hema ox-
ylin and eosin s ain (×40): no mal c yp a -
chi ec u e and a conges i e edema ous
lamina p op ia wi h minimal in lamma o-
y in il a e sugges ing eac i e changes.
Fig. 3. a, b Abdominopel ic CT scan, axial
sec ion (a) and co onal sec ion (b). Pneu-
mope i oneum and ai bubbles adjacen o
he lumen o he bowel (whi e a ows),
some o hem in subse osal loca ion (yel-
low a ow).
Idiopa hic Ch onic wi h Benign
Pneumope i oneum
211
GE Po J Gas oen e ol 2022;29:209–211
DOI: 10.1159/000514723
wi h dia hea, hema ochezia, abdominal pain and/o dis-
en ion, cons ipa ion, la ulence, loss o appe i e and e-
nesmus [1, 2]. An ibio ics, elemen al die as well as inha-
la ional and hype ba ic oxygen he apy ha e been used,
al hough he e idence o suppo hei e icacy is limi ed
and he ecu ence a e is high. The diagnosis o PCI is
based on endoscopy o imageology [1, 2]. Endoscopically,
cys s ha e a pale o bluish appea ance and a y in size
om a ew millime e s o se e al cen ime e s, and when
biopsied, hey can apidly de la e wi h an audible hiss [1,
5]. Cha ac e is ic indings on abdominal CT a e collec-
ions o ai adjacen o bowel lumen, wi h subse osal cys s
commonly seen in small in es inal pneuma osis, bu ee
pe i oneal ai has a ely been epo ed [1]. Complica ions
associa ed wi h PCI occu in app oxima ely 16.3% o cas-
es and include in es inal obs uc ion o in es inal pe o a-
ion [2].
This case highligh s PCI as a benign en i y and ocus-
es on he ole o pa ien s’ clinical assessmen in he man-
agemen . I also emphasizes PCI as a cause o benign
pneumope i oneum. Al hough he pa ien did no mee
he diagnos ic c i e ia o ch onic obs uc i e pulmona y
disease ha has been associa ed wi h PCI, we ecognize
he po en ial ole o pulmona y emphysema in he e iol-
ogy.
S a emen o E hics
W i en in o med consen was gi en by he pa ien ha au ho-
ized publica ion o he case, including images.
Con lic o In e es S a emen
The au ho s ha e no con lic s o in e es o decla e.
Funding Sou ces
No unding was ecei ed.
Au ho Con ibu ions
Cláudia Macedo was esponsible o he da a acquisi ion and
edi ing, manusc ip w i ing and e iewed he li e a u e. Ped o
Ama o was esponsible o he da a acquisi ion and e iewed he
manusc ip . Rui Cae ano-Oli ei a was esponsible o he da a ac-
quisi ion. Elisa G a i o-Soa es, Ma a G a i o-Soa es and Ped o
Figuei edo e iewed he manusc ip . All au ho s app o ed he
published e sion o he manusc ip and ag eed o be accoun able
o all aspec s o he wo k, ensu ing ques ions ela ed o he accu-
acy o in eg i y o any pa o he wo k we e app op ia ely e alu-
a ed and esol ed.
Re e ences 1 Heng Y, Schu le MD, Haggi RC, Roh mann
CA. Pneuma osis in es inalis: a e iew. Am J
Gas oen e ol. 1995 Oc ; 90(10): 1747–58.
2 Wu LL, Yang YS, Dou Y, Liu QS. A sys em-
a ic analysis o pneuma osis cys oids in es i-
nalis. Wo ld J Gas oen e ol. 2013 Aug;
19(30): 4973–8.
3 Koss LG. Abdominal gas cys s (pneuma osis
cys oides in es ino um hominis); an analysis
wi h a epo o a case and a c i ical e iew o
he li e a u e. AMA A ch Pa hol. 1952 Jun;
53(6): 523–49.
4 Lommen MJ, Zineldine O, Meh a TI, Rad ke
LE, Se ano O. Pneuma osis Cys oides In es-
inalis Iden i ied on Sc eening Colonoscopy
Wi h Associa ed Pneumope i oneum. Cu e-
us. 2020 Aug; 12(8):e9512.
5 Miwa W, Hi a suka T, Sa o K, Ka o Y. Pneu-
ma osis cys oides in es inalis lesions chang-
ing in o yellowish plaque-like elas osis lesions
du ing healing. Clin J Gas oen e ol. 2020
Dec; 13(6): 1165–72.