Perinatal results of conservative management in fetal

bronchopulmonary sequestration. Report of a case

Resultados perinatales del manejo prenatal conservador

en secuestro broncopulmonar fetal. Reporte de un caso

Revista Ecuatoriana de Pediatría

Editorial: Sociedad Ecuatoriana de Pediatría (Núcleo de Quito, Ecuador)

Tipo de estudio: Reporte de Caso

Área de estudio: Pediatría

Páginas: 37-45

Codígo DOI: https://doi.org/10.52011/RevSepEc/e228

URL: https://rev-sep.ec/index.php/johs/article/view/228

RESUMEN

Introduction: Congenital bronchopulmonary malformations are rare but potentially fatal anomalies in fetuses

and newborns. They require urgent diagnostic evaluation from the prenatal and neonatal period. In 5.9% of

cases, the lesion resolves spontaneously. The scarcity of evidence on its natural evolution has generated diver-

gences in the management strategies of prenatal cases and of asymptomatic neonates, which results in an

always controversial issue. Prenatal regression is common in cases diagnosed before birth, and most children

without symptoms beyond the neonatal period will remain symptom-free throughout infancy. The lack of sta-

tistical information in our country, and in our center, and the scarcity of comparative studies on the diﬀerent

therapeutic methods available, make it diﬃcult to formulate unique prenatal therapeutic protocols. We present

a clinical case attended from the prenatal period at the University Hospital of Guayaquil in the year 2023 with

expectant management of the fetus and resolution of maternal comorbidities with favorable perinatal outco-

me. Of note is the spontaneous regression from mild polyhydramnios to severe oligohydramnios in 4 weeks,

Reporte de Caso

Adriel Sarduy Sieres2, Joselyn Lissette Navarrete Moncayo1, Carolina Pedron Paiva2,

Maria Paz Valdivieso Uriguen2, Joffre Enrique Zavala Cárdenas3, Granizo Gabriela4

1. Postgraduate resident doctor in Gynecology and Obstetrics. Santiago de Guayaquil Catholic University.

2. Physician specialist in Gynecology and Obstetrics at the University Gynecological, Obstetric and Pediatric Hos-

pital of Guayaquil. Ministry of Public Health. Guayaquil. Ecuador.

3. Physician specializing in Pediatrics. Dr. Roberto Gilbert Elizalde Children’s Hospital. Charity Board. Guayaquil.

Ecuador.

4. Medical specialist in Medical Imaging. Guayaquil University Hospital. Ministry of Public Health. Ecuador.

Adriel Sarduy Sieres https://orcid.org/0000-0003-4146-7988

Joselyn Lissette Navarrete Moncayo https://orcid.org/0000-0003-1438-7720

Carolina Pedron Paiva https://orcid.org/0009-0004-3240-1180

Maria Paz Valdivieso Uriguen https://orcid.org/0000-0002-3842-1068

Joffre Enrique Zavala Cárdenas https://orcid.org/0000-0002-6883-4509

Granizo Gabriela https://orcid.org/0000-0003-1975-0883

Correspondencia: Catholic University of Santiago de Guayaquil. Guayaquil Gynecological, Obstetric and Pediatric

/ adriel.sarduy@gmail.com

Recibido: 10/feb/2024 - Aceptado: 12/abril/2024 - Publicado: 30/sep/2024

Revista Ecuatoriana de Pediatría | ISSNe: 2737-6494

Pagína 38 | VOL.25 N°3 (2024) Septiembre - Diciembre

simultaneous with the resolution of the fetal pleural eﬀusion and the rearrangement of the structures of the

fetal mediastinum, previously displaced by the tumor and the tension hydrothorax. Clinical case: A 32-year-

old multiparous patient detected late with a pregnancy of 30.1 weeks of gestation complicated by maternal

syphilis and fetal bronchopulmonary sequestration (BPS) with pleural eﬀusion and severe displacement of the

fetal mediastinum to the right, without fetal hemodynamic compromise. Diagnoses conﬁrmed by obstetric ul-

trasounds, serial fetal Doppler and fetal MRI. It evolves with mild polyhydramnios and is managed expectantly

until 37 weeks when, after spontaneous resolution of the hydrothorax, it progresses to severe oligohydramnios

and is completed by segmental cesarean section. The neonate receives a thoracotomy with resection of the

lung mass at 7 days after birth and the diagnosis is conﬁrmed by pathology. Conclusions: We present a cli-

nical case of a rare fetal pathology in all its types worldwide and underdiagnosed in our environment, treated

late at 30.1 weeks of gestation with multiple maternal-fetal comorbidities with spontaneous improvement in the

prenatal period, largely managed expectantly. due to the diagnostic and therapeutic challenges faced, in the

absence of a fetal medicine and surgery service in the area. Despite this, expectant fetal management is an

internationally valid option, with very favorable results in most cases without hydrops or other signs of fetal

heart failure. Comparative studies of diﬀerent prenatal management methods are lacking partly due to the

rarity of the pathology; In our environment, its dispersion and late prenatal diagnosis with poor accessibility to

subspecialists in maternal-fetal medicine and fetal surgery can negatively inﬂuence perinatal outcomes..

Keywords: Bronchopulmonary sequestration (BPS); Congenital adenomatous cystic airway malformation (CAM);

Congenital pulmonary malformation (CPM); preserved treatment; hybrid lesions; prenatal.

ABSTRACT

Introducción: Las malformaciones broncopulmonares congénitas son anomalías raras pero potencialmente

fatales en fetos y recién nacidos. Requieren evaluación diagnóstica urgente desde el período prenatal y neo-

natal. En el 5,9% de los casos, la lesión se resuelve espontáneamente. La escasez de evidencia sobre su evolu-

ción natural ha generado divergencias en las estrategias de manejo de los casos prenatales y de los neonatos

asintomáticos, lo que resulta en un tema siempre controversial. La regresión prenatal es común en los casos

diagnosticados antes del nacimiento, y la mayoría de los niños sin síntomas más allá del período neonatal

permanecerán asintomáticos durante toda la infancia. La falta de información estadística en nuestro país, y en

nuestro centro, y la escasez de estudios comparativos sobre los diferentes métodos terapéuticos disponibles,

diﬁcultan la formulación de protocolos terapéuticos prenatales únicos. Presentamos un caso clínico atendido

desde el período prenatal en el Hospital Universitario de Guayaquil en el año 2023 con manejo expectante

del feto y resolución de las comorbilidades maternas con resultado perinatal favorable. Destaca la regresión

espontánea de polihidramnios leve a oligohidramnios severo en 4 semanas, simultáneo con la resolución del

derrame pleural fetal y el reordenamiento de las estructuras del mediastino fetal, previamente desplazadas

por el tumor y el hidrotórax a tensión. Caso clínico: Paciente multípara de 32 años detectada tardíamente

con embarazo de 30.1 semanas de gestación complicado con síﬁlis materna y secuestro broncopulmonar

(SBP) fetal con derrame pleural y desplazamiento severo del mediastino fetal a la derecha, sin compromiso

hemodinámico fetal. Diagnósticos conﬁrmados por ecografías obstétricas, Doppler fetal seriado y resonancia

magnética fetal. Evoluciona con polihidramnios leve y se maneja expectante hasta las 37 semanas cuando,

tras resolución espontánea del hidrotórax, progresa a oligohidramnios severo y se completa mediante cesárea

segmentaria. El neonato recibe una toracotomía con resección de la masa pulmonar a los 7 días de nacido

y el diagnóstico se conﬁrma por patología. Conclusiones: Presentamos un caso clínico de una patología fetal

rara en todos sus tipos a nivel mundial y subdiagnosticada en nuestro medio, tratada tardíamente a las 30.1

semanas de gestación con múltiples comorbilidades materno-fetales con mejoría espontánea en el periodo

prenatal, manejada en gran medida de manera expectante debido a los retos diagnósticos y terapéuticos que

se enfrentan, en ausencia de un servicio de medicina y cirugía fetal en la zona. A pesar de ello, el manejo

fetal expectante es una opción válida a nivel internacional, con resultados muy favorables en la mayoría de

los casos sin hidrops ni otros signos de insuﬁciencia cardiaca fetal. Faltan estudios comparativos de diferentes

métodos de manejo prenatal en parte por la rareza de la patología; en nuestro medio, su dispersión y diag-

nóstico prenatal tardío con poca accesibilidad a subespecialistas en medicina materno-fetal y cirugía fetal

pueden inﬂuir negativamente en los resultados perinatales..

Palabras Clave: Secuestro Broncopulmonar (BPS), Malformación quística adenomatosa congénita de las

vías respiratorias (CPAM), Malformación Pulmonar Congénita (CPM), tratamiento preservado, lesiones híbridas,

prenatal.

Reporte de Caso

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Introduction

Congenital bronchopulmonary malforma-

tions are rare, but potentially fatal anomalies

in fetuses and newborns, and require urgent

diagnostic evaluation from the prenatal and

neonatal period, which is easily performed

by obstetric ultrasound in the ﬁrst trimester

as the method of choice1,2. Bronchopulmo-

nary sequestration (PS), congenital cystic

adenomatous malformation (CCAM), con-

genital lobar emphysema (CLE) and bron-

chogenic cyst (BC) are the four main con-

genital cystic lesions, but they share similar

embryological and clinical characteristics3,4.

Deﬁned as a heterogeneous group of solid

intrathoracic pulmonary masses that cause

lesions of dysfunctional lung tissue and that

in the case of bronchopulmonary seques-

tration will be supplied by an arterial vessel

branch of the thoracoabdominal aorta 5,

generally easy to visualize by color Doppler

and being more diﬃcult to detect . evaluate

its venous drainage even by fetal MRI. The

Great Ormond Street Hospital in London

described one of the largest series to date

on these pathologies in 2017. They analyzed

the long-term evolution of 119 cases trea-

ted at the institution (18 years). Of these,

43% (51 patients) underwent surgery, mainly

due to respiratory infections. 57% (68 pa-

tients) received conservative treatment for

an average of 9.9 years. In 5.9% (4 patients),

the lesion resolved spontaneously. 76% (52

patients) continue to be followed up and

remain asymptomatic, without presenting

malignancy6.

The scarcity of evidence on the natural

evolution of these congenital respiratory

tract malformations has resulted in a di-

vergence in the management strategy of

asymptomatic cases, making it undoubtedly

a controversial topic6,7. Prenatal regression

is common in cases diagnosed prenata-

lly, 5 and the majority of children who do

not present symptoms beyond the neonatal

period will remain symptom-free throughout

their childhood, with hemoptysis and respi-

ratory infections being the main causes of

active behaviors. Future analysis with lon-

ger follow-up could provide new insights to

identify children at risk of developing symp-

toms.

In our country, statistical information in this

regard is insuﬃcient and is often disper-

sed in small series, mostly postnatal studies

of pediatric surgery. In 2022, a group of

pediatric surgery residents at the Catholic

University of Santiago de Guayaquil men-

tion that they did not ﬁnd previous studies

that relate 2 therapeutic methods, such as

lesion resection vs. endovascular or percu-

taneous embolization 8 . Until 2019, Cruz et

al 5 carried out a review in which of 150

articles from various databases they chose

only 29 in which a combination of prenatal

diagnosis and fetal treatment was found,

which justiﬁes the diﬃculty in stating prena-

tal therapeutic protocols. unique.

Clinical case

Information of the Patiente

We present a 32-year-old, multiparous pa-

tient with 4 births and 3 previous abortions

and intergenic period of 4 years, with a his-

tory of syphilis since her second pregnancy,

having received incomplete treatment on 2

occasions. Received due to a clinical pictu-

re of threatened preterm labor secondary

to vaginosis and recurrent urinary tract in-

fection during pregnancy; At 30 weeks with

1/7 day of gestation, with insuﬃcient pre-

natal controls and obstetric ultrasounds, a

diagnosis of fetal left pleural eﬀusion was

recorded.

Clinical Findings

Afebrile, hydrated, hemodynamically sta-

ble patient given BP: 101/73 mmHg, HR: 90

bpm, RR: 21 rpm, O2 saturation: 99%, Tem-

perature: 36.3°C and alert level of cons-

ciousness. Size: 153 cm, weight: 50 kg. The

obstetric physical examination revealed ute-

rine height: 33 cm, single fetus, longitudinal

left dorsum, cephalic, FHR: 156 bpm, regular

uterine dynamics: 3/10 minutes/45 seconds/

baseline 10 mmHg/85 mmHg. Erythematous

external genitalia with yellow-green leucorr-

hoea, thick, ﬂuid, fetid, abundant, also con-

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ﬁrmed by speculoscopy where amniorrhea

did not appear with any maneuver through

the OCE of the cervix of the multiparous

woman presented to us.

Diagnostic Evaluation

Clinical laboratories:

Leukocytes 14300, neutrophils 78%. Reacti-

ve syphilis, VDRL positive with 1/16 dilutions,

non-reactive HIV. Pathological urinalysis

with abundant leukocytes (uncountable)

and positive nitrites with pH>7.3. Glucose: 73

mg/d . Direct examination of vaginal dis-

charge not available. Negative emergency

crystallography.

Emergency obstetric ultrasound:

Image 1. Solid mass in left fetal hemihtorax

in a sagital plane. B mode.

Single, longitudinal, live fetus, left anterior

dorsum, high and mobile head, active and

reactive fetal movements. FHR: 153 bpm.

Male sex. DBP: 62.4mm. DC: 294.9 mm.

DOF: 100.6 mm. AC: 298.3mm. LF: 62mm.

DTC: 42.5mm. CM: 8.3 mm.DC/AC: 0.99,

LF/AC: 20.78. PFE: 1957 grams. EG/US: 32

weeks and 6/7 days. ILA 21.8 cms that pro-

gresses to 24 cms at 2 weeks, and MBV:

9.1 cms. Ultrasound ﬁnding: a slightly hype-

rechogenic mass is seen that occupies the

basal 1/3 of the left hemithorax, with regular

borders of 43.9 mm by 42.0 mm (Image 1

and 2), without macroscopic cystic images

in its echo structure, which is irrigated by a

large-caliber nutrient vessel branch of the

descending thoracic aorta (Illustration 3).

Image 2. Solid mass in fetal left hemithorax.

Corte transversal. B mode.

Image 3. Central artery. Transvers plane.

Doppler color.

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Image 4. Fetal bronchopulmonary seques-

tration with pleural eﬀusion.

Image 5. Fetal MRI: Pleural eﬀusion in the

case of fetal bronchopulmonary sequestra-

tion (left: Transverse section), partial sponta-

neous reabsorption (sagittal section).

Therapeutic Intervention

We began prescribing empirical vaginosis

therapy with clindamycin 100 mg vaginally

(suppositories) QD, for 6 days followed by

lactobacilli acidophilus (Vaginal Tablets 0.03

Mg) for 6 days. Additionally, Fosfomycin 3

grams orally in a single dose is indicated

while awaiting urine culture results. Similarly,

nifedipine 10 mg PO TID for 72 hours and

progesterone 200 mg PO, QD, for 10 days

are prescribed. We evaluated the patient

weekly with partial improvement and even

without urine culture results, it was decided

to administer cephalexin 500 mg PO, QD

for 7 days. During this time, the patient’s

referral to the National Center for Maternal

Fetal Medicine in the city of Quito is coor-

dinated. The patient does not receive the

complete medication prescription despite

having it available, nor does she go to the

referral center, presenting irregular evolution

with recurrence of uterine dynamics. In ad-

dition, a complete induction scheme of fetal

lung maturation is placed with betametha-

sone 12 mg IM, QD for 2 doses.

At 33.1 weeks of gestation, she was admi-

tted to the emergency room with regular

intense uterine contractions that initially did

not improve with rehydration or the ad-

ministration of tocolytics at the emergency

therapeutic doses indicated, but then while

waiting for a surgical appointment, the dy-

namics subsided with nifedipine PO 3 doses.

(10mg/10mg/20mg) and magnesium sulfate

IV for 12 hours, which led to his transfer

to the High Obstetric Risk room, where he

received intravenous treatment with ampi-

cillin 1g QID for 7 days and clindamycin 100

mg vaginally for 6 days, improving substan-

tially. . Due to the obstetric, psychosocial

and perinatal risk, they remain hospitalized

awaiting referral to the National Center for

Fetal Medicine in the city of Quito. During

this time, the therapeutic regimen for syphi-

lis was completed with benzathine penicillin

2.4 million IM weekly for 3 weeks with sub-

sequent serological control reporting reacti-

ve syphilis, but with non-reactive VDRL and

dilutions of 1/6 (initially 1/16).

Termination of Pregnancy and Neona-

tal Evolution

At 37 weeks of gestation due to severe

oligohydramnios (ILA: 2 cm / MBV: 1 cm), it

was decided to terminate the pregnancy

electively via segmental cesarean section

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Image 6. Neonatal CT showing an intra-

thoracic mas with suspected bronchopul-

monary sequestration (Transverse section).

in which the absence of amniotic ﬂuid was

conﬁrmed, obtaining a live male newborn

with Apgar 8- 9 at one minute and 5 minu-

tes, does not require stimulation and tran-

sitions to attachment and breastfeeding in

the immediate postpartum period without

complications. During hospitalization, a sim-

ple chest tomography was performed on

the neonate, revealing a solid mass in the

posterior segments of the left lower lobe

(Illustration 5).

Due to bronchopulmonary infection and

acute respiratory distress, the neonate was

transferred to neonatal intensive care at the

Dr. Roberto Gilbert Elizalde Children’s Hos-

pital of the Guayaquil Charity Board. Af-

ter stabilizing the patient ventilatorily and

hemodynamically, it was decided to per-

form a thoracotomy and resection of the

mass, after which a successful recovery was

obtained and the patient was discharged

from the institution 7 days after the surgical

procedure without presenting other com-

plications (Illustration 6 and 7). And ﬁnally,

the result of the histopathological study is

obtained that conﬁrms the initial suspicion

of extralobar bronchopulmonary sequestra-

tion of the lower lobe of the left lung with

a single central nutrient vessel, bronchioli-

zation of the alveolar epithelium, lymphan-

giectasia and vascular congestion.

Reporte de Caso

Image 7. Anatomical piece, extralobar

bronchopulmonary sequestration.

Image 8. IPostoperative radiography.

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Discussion

Bronchopulmonary sequestration (BPS) is a

rare congenital malformation characterized

by a mass of nonfunctional lung tissue that

does not communicate with the tracheo-

bronchial tree and receives an abnormal

vascular blood supply from the systemic

circulation9.

There is a paucity of epidemiological studies

on SBP despite it ranking second as the most

common congenital pulmonary malforma-

tion, accounting for 0.15–8.3%; 0.42/10,000

live births in China between 2010-20199. Two

diﬀerent theories have been proposed to

explain the pathophysiological mechanisms

of congenital bronchopulmonary malfor-

mations. The environmental hypothesis su-

ggests that a persistent expression of early

markers of lung development, caused by

potential genetic defects, could lead to a

focal and temporal disruption of lung mor-

phogenesis. More recent histological studies

of lung malformations have suggested an

obstructive hypothesis: focal obstruction of

the respiratory tree, whether functional (pe-

ristalsis abnormality) or organic (bronchial

stenosis), would generate an increase in

mediators and cause CPAM abnormalities.

Currently, the modes and timing of obstruc-

tive events are poorly understood10.

Stocker et al. They initially identiﬁed three

stages through macroscopic and microsco-

pic description, which led to the ﬁrst clas-

siﬁcation (type 1, the most common form,

with large cysts containing mucous cells,

type 2 with multiple small cysts and possi-

bly other associated anomalies), type 3 with

bulky solid-appearing lesions and frequently

displaced mediastinum), which would later

be updated by adding type 0 or acinar

dysplasia, which is usually fatal, and type 4,

which is an acinar malformation characte-

rized by cysts of various sizes. , covered by

alveolar cells type 1 and 2 without mucous

cells10.

It generally occurs in early stages of life, al-

though cases have been reported in adul-

thood where it can manifest acutely and

with symptoms such as respiratory diﬃculty,

recurrent pneumonia, frequent infections,

presence of purulent sputum, hemopty-

sis and in cases unusual, hemothorax and

heart failure11.

Polyhydramnios may also occur secondary

to compression of the esophagus by the

intrathoracic mass and pleural eﬀusion (hy-

drothorax or fetal chylothorax), which usua-

lly resolves when the compression subsides,

either due to spontaneous involution, percu-

taneous chemical sclerosis, or laser sclerosis

of the nutrient vessel. Our case progressed

from mild polyhydramnios (MBV: 9.1 cm) to

severe oligohydramnios (MBV: 1 cm) spon-

taneously in 4 weeks with resorption of the

pleural eﬀusion.

Advances in ultrasound technology and

the widespread use of prenatal ultrasound

have resulted in increased identiﬁcation of

congenital pulmonary airway malforma-

tions during pregnancy. This has led to a

better understanding of how some of these

anomalies evolve naturally and has opened

up the possibility of making preparations

for childbirth and post-birth care, and even

in severe cases, providing prenatal treat-

ment12–14. In the literature related to surgery,

pathology, and imaging, various terms are

used to describe lung abnormalities present

at birth. For this reason Kellenberg et al. In

their 2020 study, they concluded and pro-

posed a classiﬁcation based on the ﬁndings

of specialized fetal lung MRI can accurately

identify, localize and categorize congenital

anomalies of the pulmonary digestive tract,

with high accuracy and very good agree-

ment between readers15, however this was

not used in our case.

In other studies, excellent agreement was

observed in relation to MRI results, even

between residents and radiologists with

more than 10 years of experience, which

shows reliable and comparable values for

each ﬁnding considered, with the exception

of identiﬁcation of venous drainage. Des-

pite the high diagnostic reliability of MRI

for bronchopulmonary sequestration, iden-

tifying the venous drainage branch repre-

sents the most diﬃcult challenge in diag-

nosis; However, it was recognized mainly

by the most experienced radiologists, who

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managed to correctly distinguish between

the intralobar and extralobar forms in a

higher percentage of cases than the other

groups16.

Some studies show that patients who pre-

sented symptoms had an unfavorable out-

come (longer stay in the intensive care

unit and higher incidence of postoperative

complications) compared to patients who

did not present symptoms, which raises

the question from the prenatal period. The

main limitation of most studies is that they

are carried out retrospectively and that the

number of patients included is generally li-

mited by their dispersion and/or diagnostic

limitations or access to subspecialists in ma-

ternal-fetal medicine and fetal surgery.

Conclusions

Prenatal diagnosis and treatment studies

are very limited in our country largely due

to the fact that there are only 2 Fetal The-

rapy centers in the country’s public sector

(both in the capital Quito), as well as sig-

niﬁcant diagnostic limitations in our center.

There is at least one study in the literatu-

re that compares two therapeutic options

in the neonatal period; however, we did

not ﬁnd any national prospective studies in

this regard. In our case, the main limitation

to achieving the desired goal of 39 weeks

and early hospitalization due to therapeutic

non-compliance on the part of the patient,

with no apparent impact on the outcome

of the pregnancy. It is recommended to

carry out prospective studies with a more

representative sample of patients to deter-

mine more clearly which is the best the-

rapeutic option8, as well as to streamline

the ﬂow of patients and their early access

to medical services subspecialized in fetal

medicine.

Patient perspective

The patient was always satisﬁed with the

diagnostic, therapeutic and follow-up plan

established and give us all permission to

use the information related to her son cli-

nical case. After almost having to end the

pregnancy at 33 weeks and keeping the

patient hospitalized for the next 4 weeks

with her consent and that of her closest

relatives, I managed to understand the

real life options of her future child, and the

possible complications. immediate neonatal

measures that included both a satisfactory

spontaneous evolution and neonatal sur-

gery since we did not have the option of

fetal surgery in the province. She expressed

gratitude at every moment of the care and

follow-up process and provided consent for

the use of all the information in the case,

including personal data, spontaneously and

in full use of her faculties.

Financing

The article is prepared without any external

ﬁnancing and without conﬂict of interest du-

ring writing. The patient did not request any

payment for the use and publication of the

clinical information about her pregnancy.

Reporte de Caso

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Para referenciar aplique esta cita:

Sarduy Sieres A, Navarrete Moncayo JL, Pedron Paiva C, Valdivieso Uriguen MP, Zavala Cárdenas

JE, Gabriela G. Resultados perinatales del manejo prenatal conservador en secuestro broncopulmonar fetal.

Reporte de un caso. REV-SEP [Internet]. 20 de noviembre de 2024; 25(3):37-45. Disponible en: https://rev-sep.

ec/index.php/johs/article/view/228

Reporte de Caso

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