Intradiaphragmatic hybrid lesion in an infant: case report
Claudio Olivieri
a,
⁎
, Lorenzo Nanni
a
, Gaia Busato
a
, Guido Rindi
b
,
Riccardo Marano
c
, Claudio Pintus
a
a
Pediatric Surgery Unit, Policlinico “A. Gemelli,” Largo Agostino Gemelli, 8, 00168 Rome, Italy
b
Department of Pathology, Policlinico “A. Gemelli,” Rome, Italy
c
Department of Radiology, Policlinico “A. Gemelli,” Rome, Italy
Received 7 November 2011; revised 20 March 2012; accepted 2 April 2012
Key words:
Diaphragm;
Congenital cystic
adenomatoid
malformation;
Sequestration;
Hybrid lesion
Abstract Hybrid lesions have elements of both congenital cystic adenomatoid malformation and
bronchopulmonary sequestration. We report an unusual case of an infant treated for an intradiaphrag-
matic hybrid lesion. Although computed tomography remains the criterion standard imaging
examination for planning the operation, the exact localization of such lesions may be discovered
only at surgical exploration.
© 2012 Published by Elsevier Inc.
Congenital cystic adenomatoid malformation (CCAM)
and bronchopulmonary sequestration (BPS) belong to the
spectrum of congenital pulmonary malformations. Bronch-
opulmonary sequestration is defined as a mass of non-
functioning lung tissue that receives its vascular supply from
a systemic artery and is separated from the tracheobronchial
tree. Several cases of supradiaphragmatic or infradiaphrag-
matic extralobar sequestrations (ELSs) have been reported in
literature, whereas intradiaphragmatic ELS is an extremely
rare condition [1].
Congenital cystic adenomatoid malformation is consid-
ered a hamartomatous lesion of the bronchial tree; 3 different
types had been identified based on histologic characteristics
[2]. In contrast to ELS, CCAM is connected to the tracheo-
bronchial tree, and its vascular supply comes from the pul-
monary circulation.
Malformations with both of these components have been
recognized and defined as hybrid lesions [3].
We report a case of an infant with an intradiaphragmatic
hybrid lesion. To the best of our knowledge, such localization
has not been previously reported for hybrid lesions.
1. Case report
A 25-year-old prima gravida was referred to our hospital
after an ultrasound (US) examination at 24 weeks' gestation,
which showed a cystic lesion of the left lower lung. A 3.660-
g female infant was delivered vaginally at term, with normal
physical findings. At birth, the patient was initially inves-
tigated by a plain chest radiograph, which showed neither
cystic lesions nor displacement of the mediastinum. During
the first months of life, the patient remained asymptomatic;
follow-up chest radiographs at 3 and 7 months were con-
sidered normal. Chest computed tomography (CT) per-
formed at 8 months of age showed a mass in the left lower
lobe, measuring 28 × 11 × 32 mm, with an extension at the
posterior costophrenic angle. An aberrant arterial supply
from the celiac axis was identified. At age 11 months, the
patient underwent surgical exploration. After a muscle-
⁎
Corresponding author. Tel.: +39 06 3015 4032; fax: +39 06 3015 4363.
E-mail address: claudioolivieri@liberto.it (C. Olivieri).
www.elsevier.com/locate/jpedsurg
0022-3468/$ – see front matter © 2012 Published by Elsevier Inc.
doi:10.1016/j.jpedsurg.2012.04.010
Journal of Pediatric Surgery (2012) 47, E25–E28