Skip to main content
Erschienen in: Italian Journal of Pediatrics 1/2023

Open Access 01.12.2023 | Research

Chronic respiratory disorders due to aberrant innominate artery: a case series and critical review of the literature

verfasst von: Adele Corcione, Melissa Borrelli, Leonardo Radice, Oliviero Sacco, Michele Torre, Francesco Santoro, Gaetano Palma, Eleonora Acampora, Francesca Cillo, Pietro Salvati, Angelo Florio, Francesca Santamaria

Erschienen in: Italian Journal of Pediatrics | Ausgabe 1/2023

Abstract

Background

Tracheal compression (TC) due to vascular anomalies is an uncommon, but potentially serious cause of chronic respiratory disease in childhood. Vascular slings are congenital malformations resulting from abnormal development of the great vessels; in this group of disorders the most prevalent entity is the aberrant innominate artery (AIA). Here we provide a report on diagnosis and treatment of AIA in nine children with unexplained chronic respiratory symptoms. We describe the cases, perform a literature review, and provide a discussion on the diagnostic workup and treatment that can help manage AIA.

Methods

Clinical history, diagnostic procedures and treatment before and after the AIA diagnosis were retrospectively reviewed in nine children (5 boys and 4 girls), who were referred for recurrent-to-chronic respiratory manifestations over 10 years (2012–2022). We performed a comprehensive report on the ongoing clinical course and treatment as well as an electronic literature search on the topic.

Results

Diagnoses at referral, before AIA was identified, were chronic dry barking cough associated with recurrent pneumonia (n = 8, 89%), lobar/segmental atelectasis (n = 3, 33%), atopic/non atopic asthma (n = 3, 33%); pneumomediastinum with subcutaneous emphysema complicated the clinical course in one case. When referred to our Unit, all patients had been previously treated with repeated antibiotic courses (n = 9, 100%), alone (n = 6, 67%) or combined with prolonged antiasthma medications (n = 3, 33%) and/or daily chest physiotherapy (n = 2, 22%), but reported only partial clinical benefit. Median ages at symptom onset and at AIA diagnosis were 1.5 [0.08–13] and 6 [4–14] years, respectively, with a relevant delay in the definitive diagnosis (4.5 years). Tracheal stenosis at computed tomography (CT) was ≥ 51% in 4/9 cases and ≤ 50% in the remaining 5 subjects. Airway endoscopy was performed in 4 cases with CT evidence of tracheal stenosis ≥ 51% and confirmed CT findings. In these 4 cases, the decision of surgery was made based on endoscopy and CT findings combined with persistence of clinical symptoms despite medical treatment. The remaining 5 children were managed conservatively.

Conclusions

TC caused by AIA may be responsible for unexplained chronic respiratory disease in childhood. Early diagnosis of AIA can decrease the use of expensive investigations or unsuccessful treatments, reduce disease morbidity, and accelerate the path toward a proper treatment.
Hinweise

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Abkürzungen
TC
Tracheal compression
AIA
Aberrant innominate artery
CT
Computed tomography
RRI
Recurrent respiratory infections
CF
Cystic fibrosis
PCD
Primary ciliary dyskinesia
PID
Primary immunodeficiency
ICS
Inhaled corticosteroids
MRI
Magnetic resonance imaging

Introduction

Upper and lower airways chronic diseases are increasing in prevalence everywhere, particularly among children and elderly people. In children and adolescents, chronic cough can be a major manifestation of several recurrent-to-chronic respiratory diseases [1]. In many cases the etiology of the disease remains elusive, and a misidentification of the underlying disorder results in failure to start effective treatment [2].
Tracheal compression (TC) from congenital vascular anomalies is an uncommon, but potentially serious, cause of chronic respiratory disease in childhood. Although compression of the upper airway is typically associated with vascular abnormalities, other conditions may be responsible for TC. Among these, abnormal thoracic configuration as pectus excavatum, narrow chest diameter, scoliosis, rib anomalies and small left hemithorax have been described [3]. TC can also occur by space occupying lesions such as anterior mediastinal masses like Hodgkin or non-Hodgkin lymphomas, neurofibromas occurring in neurofibromatosis [4, 5], goiter, and cysts [6]. A case of airway compression by a large osteochondroma, arising from chest wall and sternum as a part of hereditary multiple exostoses, has also been described [7].
Stridor with cyanosis and apnea may be a presenting feature of TC in infancy, while incessant dry croup-like “seal-bark” cough, which sometimes is misdiagnosed as asthma, is commonly reported in older children [8]. Vascular slings are congenital malformations resulting from abnormal development of the great vessels; in this group of disorders the most prevalent entity is the aberrant innominate artery (AIA). Although accurate epidemiological data are lacking, AIA is an extremely common congenital vascular disorder, with 3% incidence [9, 10]. In normal individuals, the innominate artery crosses the trachea anteriorly after arising from the left side of the aortic arch. In AIA patients, the origin of innominate artery from the left side of the aortic arch is more distal than in normal conditions, thus causing TC. Severity of airway symptoms depends on the rate of external compression of the airway lumen and reflects secondary tracheomalacia. The patient’s ability to clear secretions from the distal airways is often impaired, and recurrent respiratory infections (RRI) may occur [11]. Importantly, a late AIA diagnosis increases the risk of prolonged damage to the airways [10]. Moreover, a severe TC demands a surgical repair [12], but patients should be thoroughly investigated before deciding whether operative or conservative treatment should be performed.
We herein retrospectively describe a case series of children with recurrent-to-chronic respiratory manifestations who underwent repeated investigations and ineffective therapies before TC due to AIA was demonstrated. We also carried out an electronic keyword-based literature search for English articles published on this topic that could improve the diagnostic path and influence the choice of treatment in children with AIA.

Methods

This is a retrospective case series of 9 pediatric patients referred to our Unit for recurrent-to-chronic respiratory symptoms. For each patient, we described the initial clinical manifestations and the winding path prior the diagnosis of AIA was confirmed. Finally, once AIA was diagnosis, we reported the treatment choice (i.e., operative, or conservative) and commented on the current clinical course. We also carried out an electronic keyword-based literature search for English original articles and/or case series ever published on this topic up to December 31, 2022, in the Scopus, Web of Science, PubMed, and MEDLINE databases. Studies conducted exclusively on adults and anecdotal single case reports were excluded. The terms “aberrant innominate artery” AND dry cough OR bark cough OR barking cough OR chronic cough OR recurrent respiratory infections OR recurrent pneumonia OR diagnosis OR treatment OR complications were used as keywords in combination. The identified studies were further evaluated to select only relevant literature, and, in addition, a manual search was conducted to evaluate references from review articles.

Results

Case series

The charts of 9 children (5 boys; 4 girls) admitted to the Pediatric Pulmonology, Department of Translational Medical Sciences, Federico II University, Naples, over a 10-year period (2012–2022) were reviewed. All were living in Campania (Southern Italy). Table 1 summarizes the clinical manifestations and the history of the study population, including the diagnostic work-up and treatment either before or after the diagnosis of AIA. The therapeutic approach to AIA, conservative or operative, and the current outcome are also reported. Diagnoses at referral were chronic dry cough (100% of the cases) that was associated with recurrent pneumonia (n = 8; 89%) or lobar/segmental atelectasis (n = 3; 33%) or atopic/non atopic asthma (n = 3, 33%), and included pneumomediastinum as additional complication in one case (11%). Treatment at referral prior to AIA diagnosis included prolonged repeated antibiotic courses (n = 9; 100%), alone or combined with prolonged antiasthma medications (n = 3; 33%) and/or daily chest physiotherapy in cases with recurrent pneumonia and lobar atelectasis (n = 2; 22%), with only partial clinical benefit. Median age at symptoms onset and at AIA diagnosis were 1.5 [range, 0.08–13] and 6 [range, 4–14] years, respectively, with a noticeable delay in the definitive diagnosis (4.5 years).
Table 1
Clinical characteristics, diagnostic work-up and treatment of the study population
 
Case 1
(male)
Case 2
(male)
Case 3
(female)
Case 4
(female)
Case 5
(male)
Case 6
(male)
Case 7
(male)
Case 8
(female)
Case 9
(female)
Diagnosis at referral
Chronic dry cough
Atopic asthma
RP
ML atelectasis
Chronic dry cough
RP
Chronic dry cough
RP
ML atelectasis
Chronic dry cough
RP
Segmental atelectasis
Chronic dry cough
RP
Chronic dry cough
RP
Chronic dry cough
Non atopic asthma
Chronic dry cough
RP
Chronic dry cough
Atopic asthma
RP
PM
Treatment at referral
Antibiotics
Chest PT
Antiasthma
Antibiotics
Antibiotics
Chest PT
Antibiotics
Antibiotics
Antibiotics
Antibiotics
Antiasthma
Antibiotics
Antibiotics
Antiasthma
Age at onset (years)
0.6
1.5
3
1
2
0.08
13
0.4
4
Age at AIA diagnosis
(years)
6
4
7
4
5
8
13
6
14
Whole diagnostic work-upa
Spirometry; FeNO; nNO/NB; ST/CFTR; sIg/vaccines response; Chest X-ray/CT AE
NB; ST/CFTR; sIg/vaccines response; Chest X-ray/CT
Spirometry; FeNO; nNO/NB; ST/CFTR; sIg/vaccines response; Chest X-ray/CT AE
NB; ST/CFTR; sIg/vaccines response; Chest X-ray/CT AE
NB; ST/CFTR; sIg/vaccines response; Chest X-ray/CT MII-pH
Spirometry; FeNO; nNO/NB; ST/CFTR; sIg/vaccines response; Chest X-ray/CT
Spirometry FeNO; nNO
ST/CFTR;sIg/vaccines response; MII-pH Chest MRI
Spirometry; FeNO; nNO/NB; ST/CFTR; sIg/vaccines response; Chest X-ray/CT
Spirometry; FeNO; nNO/NB; ST/CFTR; sIg/vaccines response; Chest X-ray/CT MII-pH AE
AIA treatment
Aortopexy
Conservative
Aortopexy
Tracheopexy
Conservative
Conservative
Conservative
Conservative
Tracheopexy
Current symptoms
None
None
None
None
None
None
None
None
None
Abbreviations: RP recurrent pneumonia, ML middle lobe, PM pneumomediastinum, AIA aberrant innominate artery, AE airway endoscopy, MII-pH multichannel intraluminal impedance-pH, PT physiotherapy, FeNO fractional exhaled nitric oxide, nNO nasal nitric oxide, NB nasal brush, ST sweat test, CFTR cystic fibrosis transmembrane regulator, sIg serum immunoglobulins levels, CT computed tomography, MRI Magnetic Resonance Imaging
aIncluding findings collected over either pre- or post-AIA diagnosis period
Once admitted at our Unit, an internal diagnostic protocol was applied to investigate children with chronic cough and RRI. Sweat chloride test and CFTR analysis; nasal nitric oxide plus transmission electron microscopy and beat analysis of ciliary ultrastructure and motility, respectively, on nasal brushing; and immune status assessment (including serum total immunoglobulins levels and response to immunizations) for ruling out cystic fibrosis (CF), primary ciliary dyskinesia (PCD) and primary immunodeficiency (PID), respectively, were negative or normal.
All cases underwent routine echocardiography as a standard of care to evaluate suspected vascular abnormality and myocardial function, and results excluded a coexisting heart disease. Multichannel intraluminal impedance-pH monitoring, obtained in cases of reported gastrointestinal disturbances (such as troublesome heartburn and/or vomiting) (cases 2 and 7), ruled out gastroesophageal reflux disease. A computed tomography (CT) scan with and without contrast medium was performed in all patients without general anesthesia to confirm the diagnosis and measure the percentage of tracheal obstruction. Grading system of tracheal stenosis was used to stratify tracheal stenosis in four grades (grade I: stenosis up to 50%; grade II: stenosis between 51 and 70%; grade III: stenosis > 70%; grade IV: no lumen visualized at the narrowest point) [13]. Image analysis was performed on an offline workstation (Multimodality Workplace, Toshiba Healthcare). CT tracheal stenosis was ≥ 51% (grade II) in 4 cases (44%) and up to 50% (grade I) in the remaining 5 subjects (55%). We chose to perform airway endoscopy (AE) only in those 4 patients with CT evidence of tracheal stenosis ≥ 51%. Based on tracheal compression degree and on severity of clinical course, cases were either managed conservatively (and monitored in follow-ups) or surgically treated. Surgery was performed if tracheal stenosis was ≥ 51% and was associated with persistent clinical symptoms unresponsive to medical or supportive medical treatment (i.e., recurrent pneumonia associated or not with complications such as lobar atelectasis and/or chronic asthma).
We herein briefly describe the clinical course either prior or after the diagnosis of AIA of two cases who were surgically or conservatively treated.

Case 1

The boy was born at term after an uneventful pregnancy. Family history revealed season allergic rhinitis in the father. Environmental history excluded parental cigarette smoking inside or outside home. Clinical well-being was reported from birth up to age 8 months, when recurrent preschool wheezing episodes due to viral infection associated with dry, barking cough were reported. At 4-year-old age, atopic bronchial asthma was diagnosed (skin tests positive to Dermatophagoides spp and Parietaria). Maintenance treatment with inhaled corticosteroids (ICS) combined with rescue inhaled albuterol was prescribed, with partial clinical benefit. At age 18 months, recurrent upper and lower respiratory tract infections started, and from 2- to 4-year-old age 3 episodes of pneumonia were documented at chest X-ray, which required hospitalization and antibiotic treatment. Middle lobe atelectasis was documented at chest CT without contrast media, demanding repeated antibiotic treatments and daily chest physiotherapy. However, dry barking cough frequently recurred at any upper or lower respiratory tract infection. At age 6, a more severe pneumonia event required hospital admission. Once ruled out CF, PCD and PID, we obtained a spirometry that showed a plateau in both expiratory and inspiratory phase (Fig. 1). The chest CT with contrast showed the anomalous course of the innominate artery and an anterior compression on the right tracheal wall inducing a tracheal stenosis quantified as grade II (Fig. 2A, B). Bronchoscopy showed an extrinsic pulsatile compression of the anterior wall of the trachea (between the middle and the distal part) with a lumen reduction > 50% (Fig. 2C). After cardiothoracic surgical consultation, aortopexy was made without any complication. The follow-up showed complete disappearance of the symptoms.

Case 2

The boy was born preterm at 36 weeks due to premature rupture of membranes, but neither birth complications nor need for ventilatory support were reported. Family history was negative for allergy and there was no passive exposure to smoke cigarette at home. The child was healthy until the age of 18 months, when pneumonia with lower right lobe consolidation was documented and oral antibiotics were administered. Since then, recurrent upper and lower airways infections with persistent dry, barking cough occurred. At least 2 episodes of pneumonia localized at the right lower lobe were reported. At age 4 years, the patient was referred to our Unit, and we ruled out atopy, PID, CF and PCD. The chest CT with contrast medium showed the anomalous course of the innominate artery with mild anterolateral compression on the right tracheal wall (grade I tracheal stenosis). Given the slight extent of the restriction of the tracheal lumen, we decided not to perform the airway endoscopy. At moment, the patient (age 5 years) is in good clinical conditions. As there was a progressive, substantial reduction of the frequency of the airways infections and cough has been rarely reported in the last 2 years, we decided not to refer the boy to the surgeon, but rather continue the monitoring of the clinical course.

Review of the literature

Table 2 summarizes the main findings from 20 original articles reporting data on 2166 patients with several vascular anomalies, including 1092 patients with AIA. Patients were followed at 5 EU, 7 US and 1 Canada centers. Of all studies (3 prospective [1416] and 17 retrospective [1012, 1730]), the oldest was dated 1963 [17] and the most recent was published in 2015 [10]. The patients’ age range was 0 to 17 years. Prevalence of presenting features was extremely variable, likely because different criteria for the enrollment were adopted along studies. Stridor (up to 100% [18]) or apnea (up to 60%; [15]) or barking (up to 100% [10]) or chronic cough (up to 75% [15]) were more frequently reported, while less cases had RRI as the main manifestation (up to 56% [15]). The most prevalent diagnoses at admission were asthma (14% [11]) or atopy (18% [10]) or laryngomalacia (7% [10]), yet the diagnosis at admission was available only in 42% of the studies. In 2 retrospective studies, 100% of the patients were admitted because of “symptomatic compression of the trachea or esophagus” [19]. The most used diagnostic procedures were chest imaging studies (including conventional X-ray and/or CT and/or magnetic resonance imaging (MRI) and/or angiography; 90% of the studies), bronchoscopy (80% of the studies) and esophagography (70% of the studies, with apnea and/or or gastrointestinal complaints such as dysphagia or suspected gastroesophageal reflux as main indications). It is worth noting that, when the indication was the “symptomatic compression of the trachea or esophagus”, the preferred diagnostic approach usually combined chest imaging studies, esophagography and bronchoscopy [12, 19]. Based on the results of the diagnostic work-up, the most prevalent final diagnoses were AIA (from 18 to 100% of the cases) or double aortic arch (from 2 to 54% of the cases) or right aortic arch (from 3 to 31% of the cases). Data on treatment (available in 18 of 20 studies) showed that the approach was solely operative in 9/20 studies (45%) or mixed (i.e., operative, and conservative) in the remaining 11 (55%). In 13/20 studies, in which the most prevalent cause of tracheal compression was AIA [20], surgery was adopted in a highly variable proportion of cases (14% to 100%), whereas the conservative approach (with symptomatic medical treatment and observation on an out-patient basis) was undertaken in less cases but with the same variable proportion (3% to 86%). Overall, indication for surgery (available in all except 5 studies) was the persistence of moderate to-severe symptoms (such as apnea and/or > 2 episodes of tracheobronchitis or pneumonia and/or severe stridor and/or cyanosis and/or dyspnea and/or failure to thrive) that were judged unresponsive to conservative treatment. A tracheal compression > 50% was the main reason for deciding surgery only in one study [18]. Overall, data on long-term follow-up of patients undergoing surgical correction of AIA showed complete resolution of symptoms in a proportion of cases ranging from 40 to 100%, while less cases showed persistence of symptoms [21]. Postsurgical complications, including pericardial effusion or pneumonia or surgical wound infection, were rarely reported and all recovered after causal treatment [11]. Conversely, data on the conservative approach indicated that improvement or remission of symptoms occurred slowly, after different period (up to 14 months). Importantly, the observation of improvements depended not only on the degree of TC, but also on the severity, duration and number of cough episodes before diagnosis, as well as on comorbidities [10].
Table 2
Main findings from studies on vascular anomalies including aberrant innominate artery
Author
[Reference]
N of cases
Age
Presenting
features
(% of cases)
Diagnosis at
admission
(%)
Diagnostic
procedures
Final
diagnosis
(%)
Therapeutic
approach
(%)
Indications
for surgery
Long-term
follow-up
(%)
Fearon
[17]
104 cases
0–11
yrs
Cyanosis
Stridor
Apnea
Tracheoesophageal
fistula Thymus
enlargement
Laryngomalacia
Cystic fibrosis
Hiatus hernia
Foreign body
Asthma; RRI
Esophagography
Bronchoscopy
Chest x-ray
Angiogram
AIA (66%)
DAA (11%)
RAA (3%)
PAS (3%)
ARSA (2%)
Others (7%)
Unknown
(8%)
Operative
Conservative
NA
NA
Mustard
[20]
285 cases
0–3 yrs
NA
NA
NA
AIA
(100%)
Operative
(14%)
Conservative
(86%)
Apnea and/or > 2 episodes of tracheabronchitis or pneumonia
Resolution (60%)a or persistence of symptoms (25%)a Poor outcome (15%)a
Eklof
[21]
30 cases
0–5
yrs
Stridor; Wheezing
Belly cough
Hoarse voice
Respiratory distress
RRI; Dysphagia
Cyanosis
NA
Esophagography
Chest x-ray
Angiogram
AIA (13%)
DAA (47%)
RAA (17%)
ARSA (13%)
PAS (10%)
Operative
(97%)
Conservative
(3%)
Stridor
RRI
Cyanosis
Dysphagia
Complete (40%) or partial resolution (20%)
Persistence of symptoms (13%) Death (27%)
Moes
[27]
90 cases
NA
Stridor (83%)
Apnea (26%)
RRI (32%)
NA
Esophagography
Tracheography
Bronchoscopy
Chest x-ray
Angiogram
AIA
(100%)
Operative
(67%)
Conservative
(33%)
Apnea and/or respiratory distress with severe tracheal narrowing
Complete
resolution (57%)a partial resolution (28%) or persistence of symptoms (15%)a
Welz
[26]
16 cases
0–1
yrs
Stridor (75%)
RRI (50%)
Apnea (44%)
NA
Tracheography
Bronchoscopy
Angiograms
AIA (100%)
Operative
(37.5%)
Conservative
(62.5%)
Apnea
Resolution of symptoms (100%)
Marmon
[14]
54 cases
0–10
yrs
Dysphagia
Wheezing
Stridor
Apnea
RRI
NA
Esophagography
Bronchoscopy
Angiogram
AIA (18%)
DAA (44%)
RAA (31%)
PAS (5%)
Operative
(100%)
NA
Resolution (87%) or persistence of symptoms (2%)
Death (2%)
Lost on follow up
(9%)
Strife
[28]
936 cases
0–17
yrs
Normal population (n = 
807)
Congenital heart
disease (n = 129)
NA
Chest X-ray
Angiogram
AIA
(30%)b
NA
NA
NA
Ardito
[29]
78 cases
0–8
yrs
Apnea (36%)
Stridor (32%)
Cough (14%)
RRI (14%)
NA
Esophagography
Bronchoscopy
Chest X-ray
Angiogram
AIA (100%)
Operative
(42%)
Conservative
(58%)
Apnea
Resolution (85%) or persistence of symptoms (15%)a
Hawkins
[25]
29 cases
0–15
yrs
Apnea (59%)
Stridor (24%)
RRI (14%)
Exercise-asthma and
stridor (3%)
NA
Esophagography
Bronchoscopy
Chest MRI
Chest CT
AIA (100%)
Operative
(100%)
Apnea and/or > 2 tracheobronchitis or pneumonia and/or severe stridor
Resolution (93%)
or persistence of
symptoms (7%)
Anand
[30]
41 cases
0–3
yrs
NA
NA
NA
AIA (19%)
DAA (44%)
RAA (27%)
PAS (10%)
Operative
(100%)
NA
Resolution (70%) or persistence of symptoms (30%)
Adler
[18]
25 cases
0–8
yrs
Stridor (100%)
Apnea (32%)
RRI (28%)
Cyanosis (24%)
Asthma (24%)
Esophagography
Bronchoscopy
Chest x-ray
Angiogram
AIA
(100%)
Operative
(100%)
Tracheal compression  > 50%
Stridor apnea
Recurrent
pneumonia
Uncontrolled asthma
Resolution (96%) or persistence of symptoms (4%)
Jones
[16]
12 cases
0–3
yrs
Stridor (100%)
Feeding troubles (75%)
Cyanosis (25%)
Apnea (8%)
Respiratory arrest
(33%)
NA
Esophagography
Bronchoscopy
Chest x-ray
Chest CT
AIA
(100%)
Operative
(100%)
Cyanosis
Apnea
Feeding
troubles
Resolution
(100%)
Erwin
[23]
45 cases
0–11
yrs
Stridor (73%)
Apnea (47%)
Bark cough (31%)
Retractions (24%)
RRI (20%)
Airway anomaly (20%)
Dysphagia (16%)
Tracheoesophageal
fistula (16%)
Asthma (4%)
Subglottic stenosis (2%)
Esophagography
Bronchoscopy
Angiogram
Chest MRI
AIA (75%)
LAV (22%)
ARSA (2%)
Operative
(100%)
Moderate tosevere
symptoms
Resolution (87%) or persistence of symptoms (9%)
Death (2%)
Tracheotomy (2%)
McLaughling
[19]
35 cases
0–17
yrs
Stridor or wheezing
(100%)
RRI (47%)
Apnea (20%) Dysphagia
(14%) Chronic cough
(14%)
Failure to thrive (11%)
Aspiration pneumonia
(6%) Vomiting (6%)
DiGeorge syndrome
(3%) VATER (3%)
Pectus excavatum (3%)
Subglottic hemangioma
(3%)
Atrial septal defect
(3%) Incarcerated
hernia (3%)
Gastroesophageal
reflux (3%)
Down syndrome (3%)
Cerebral palsy (3%)
Esophagography
Bronchoscopy
Chest x-ray
Chest MRI
Chest CT
AIA (9%)
DAA (54%)
RAA (31%)
LAV (3%)
PAS (3%)
Operative
(100%)
NA
Resolution (71%) or persistence of symptoms (20%)
Lost on follow-up (9%)
Gormley
[15]
16 cases
0–10
yrs
Stridor (100%) Chronic
cough (75%)
Dyspnea (75%) Apnea
(60%)
RRI (56%)
Dysphagia (25%)
Laryngomalacia (12%)
Asthma (12%)
Recurrent croup (12%)
Sleep apnea (6%)
Esophagography
Bronchoscopy
Fluoroscopy
Chest X-ray
Angiogram
Chest CT
AIA (94%)
DAA (6%)
Operative
(75%)
Conservative
(25%)
Apnea
Exercise
intolerance
RRI
unresponsive
to treatment
Resolution of
symptoms (75%)
Mild/residual
stridor (25%)
Woods
[12]
82 cases
0–12
yrs
Stridor (46%)
RRI (35%)
Feeding difficulty
(15%)
Suspected
tracheoesophageal
compression
Esophagography
Bronchoscopy
Chest X-ray
Angiogram
Chest MRI
Chest CT
AIA (24%)
DDA (38%)
RAA (27%)
ARSA (5%)
PAS (4%)
ALSA (2%)
Operative
(100%)
Persistent
symptoms of
tracheo
esophageal
compression
Complete
resolution (70%)
partial resolution
(19%) or symptoms
of complications
(11%)
Malik
[22]
29 cases
0–5
yrs
Stridor (69%) Cyanosis
(31%) Apnea (21%)
Choking episodes (14%)
RRI (7%)
NA
Esophagography
Bronchoscopy
Angiogram
Chest MRI
AIA (38%)
DAA (10%)
RAA (7%)
Others
(17.5%)
None
(27.5%)
Operative
(24%)
Conservative
(76%)
Severe or complicated# cases
NA
Grimmer
[24]
22 cases
0–7
yrs
Stridor (86%) Cyanosis
(50%) Apnea (41%)
Cough (41%) Intubation
(9%)
Failure to thrive (9%)
Dysphagia (9%)
Ventilator need (4%)
NA
Bronchoscopy
Angiogram
Chest MRI
Chest CT
AIA
(100%)
Operative
(100%)
Apnea
Cyanosis
Dyspnea
Failure to
thrive
Oxygen/
Ventilator
dependence
Complete/partial resolution (95%) or persistence of symptoms (5%)
Gardella
[11]
28 cases
0–13
yrs
Apnea (53%)
RRI (50%)
Chronic cough (46%)
Dyspnea (32%) Stridor
(21%) Wheezing (14%)
GER (35%)
Overweight (21%)
Atopy (14%)
Asthma (14%)
Laryngomalacia (7%)
Emotional paroxysm
(3%)
Esophagography
Bronchoscopy
Chest MRI
Chest CT
AIA
(100%)
Operative
(57%)
Conservative
(43%)
Severe symptoms, also including QoL
No improvement after conservative treatment
Resolution of symptoms (100%)
Ghezzi
[10]
209 cases
6.4
yrs
Barking cough (100%)
RRI (20%)
Exercise induced cough
(17%)
Dysphagia (6%)
Stridor (4%)c
Bronchial obstruction
(14%)
Atopy (18%)
GER disease (22%)c
Bronchoscopy
Chest CT
AIA (25%)
RAA (4%)
DAA (2%)
ARSA (1%)
None (67%)
Operative
(20%)
Conservative
(80%)c
Severe symptoms, also including QoL
No improvement after conservative treatmentd
Faster improvement of symptoms in the operative versus the conservative groupd
Abbreviations: N number, Yrs years, RRI Recurrent respiratory tract infections, AIA Anomalous Innominate Artery, DAA Double Aortic Arch, RRA Right Aortic Arch, PAS Pulmonary Artery Sling, ARSA Aberrant Right Subclavian Artery, NA Not Available, CVR Complete Vascular Ring, VATER Vertebral defects, Anus defects, Tracheoesophageal fistula, and Radial and Renal dysplasia, ALSA Aberrant Left Subclavian Artery, GER gastroesophageal reflux, LAV Left Arch Variant, MRI magnetic resonance imaging, CT computed tomography, QoL quality of life
aPercentage referred to surgery group
bPercentage referred to the group of children younger than 2 yrs (n = 508)
cPercentage referred to 68 cases with evidence of tracheal compression
dReferred to anomalous innominate artery group

Discussion

It has been reported that the most severe forms of TC caused by AIA are usually reported in infants with stridor or episodic apnea or even “near-miss” life-threatening events [10]. Older children with less severe symptoms may be identified late or remain undiagnosed, when persistent unexplained barking cough and RRI, often wrongly treated, lead to re-consider the case and the diagnostic work-up [8]. However, determining which subject should undergo invasive diagnostic procedures for confirming AIA is a challenging task, thus hampering the recognition of approximately 2/3 of the affected cases [31]. Finally, once TC from AIA is demonstrated, patients should be addressed to either a surgical procedure or a conservative treatment, a hard-to-take decision, ideally assessed by a multidisciplinary team, including pediatric pulmonologists, chest radiologists, bronchoscopists and cardiac surgeons.
Starting from these considerations, we retrospectively evaluated a small cohort of children with unexplained persistent respiratory symptoms who eventually received an AIA diagnosis. Several findings from the current case series deserve further comments. In our AIA patients, the age of symptoms onset was significantly lower than the age at diagnosis (1.5 versus 6 years), thus confirming a large diagnostic delay as previously reported [10]. Yet, all patients underwent several investigations to rule out the most common causes of chronic cough and/or recurrent pneumonia, such as PCD [32], CF and PID [33], and were prescribed multiple medical therapies, which were only partially effective (if not completely useless). Three patients had been previously diagnosed as asthmatics and received prolonged antiasthma medications, mainly ICS [34], which ultimately proved to be ineffective.
As known, the delay in establishing treatment of TC indeed increases the risk of long-term lung obstructive disease [35]. Moreover, while in normal subjects a small number of pathogens may invade the airways without causing a local colonization, subjects with extrinsic airways compression have an increased mucus production and impaired mucociliary clearance, which in turn favor bacteria airway colonization [36]. This may lead to recurrent lung infectious exacerbations and secondary segmental-to-lobar atelectasis. If the airways are not cleared of obstructing mucus by removing the primary cause (in our cases TC from AIA), then a vicious circle of persistent airway obstruction and bacterial airways colonization sets up, thus increasing the risk of protracted bacterial bronchitis and bronchiectasis [8]. In these circumstances, lobar collapse associated with hypoventilation and impaired gas exchange may develop [37]. All the above events occurred in at least 3 current patients with AIA who had marked TC associated with recurrent pneumonia and lobar atelectasis. Finally, frequent (and expensive) investigations have an impact on the costs sustained by the health care system, as well as the repeated (and often non definitive) referral to care centers may increase the psychological burden to families experiencing AIA cases [38].
Over the years, literature has highlighted many controversies about the choice of the diagnostic procedures for confirming an AIA diagnosis. AE is considered by several authors the best means of showing a narrowed trachea and a pulsatile compression on its wall from outside [10, 11]. The endoscopic diagnosis of TC and an estimation of tracheal stenosis severity is based on subjective evaluation by the bronchoscopist during the procedure. Patients undergoing AE should be breathing spontaneously for proper assessment of trachea dynamics, but deep sedation or general anesthesia is often necessary [39], thus requiring a positive-pressure ventilation. The latter might contribute to a missed diagnosis of airway collapse at the time of AE procedure [40]. Tracheal narrowing and tracheomalacia can be indeed evaluated by a skilled AE team if the patient is slightly sedated or in the awakening stage of the procedure, when cough reflex finds out trachea collapse [10]. This represents a potential, albeit relevant, drawback of relying only on the AE for the assessment of an AIA condition, since AE can record only the existing pulsatile compression on the tracheal wall. Conversely, chest imaging with contrast medium, including MRI and CT (or CT angiography), are effective modalities to identify the vessel compressing the airways and quantify tracheal stenosis by standardized measurement [35]. They both have advantages and disadvantages. Chest MRI effectively images and accurately characterizes the thoracic vascular abnormalities and serves to exclude AIA-mimicking conditions, such as other vascular disorders, mediastinal masses, intrinsic upper airways or upper gastrointestinal tract abnormalities [41]. Despite the long acquisition time and the need that patients are either cooperative or sedated, MRI avoids radiation exposure and is ideal when follow-up imaging is required [42]. Regrettably, the equipment is not universally available, and results require expertise in interpretation. Nevertheless, in case of AIA chest MRI is also used as a pre-operation procedure, either to define accurately the vascular anatomy or plan the surgical intervention [19, 22]. Chest CT is recognized as the gold standard modality for demonstrating densities due to lobar-to-segmental pneumonia or atelectasis or interstitial disease or malformations or bronchiectasis [22, 42, 43] or the pulmonary vascular structures (the latter best visualized by CT angiography) [44]. Compared to MRI, CT is cheaper and almost universally available, and provides high-spatial resolution images with fast acquisition times. Exposure to ionizing radiation is still a matter of concern in pediatric patients, but newer CT equipment, even using angiography, allows for significant radiation dose reduction [44]. Given that all current patients (except for one) had also recurrent pneumonia, obtaining details of lung parenchyma and vessel anatomy was mandatory. The choice of chest imaging (whether CT or MRI) should be tailored to the individual patient, accounting for clinical circumstances, parental/clinician preferences, need for and risks of sedation, imaging equipment and expertise available. Chest images at MRI or CT / CT angiography can be later reviewed and measured to determine the relationship between the trachea and the innominate artery. Difficulties in differentiating a TC due to AIA from a TC due to external masses using AE have been reported [23], unless an obvious vessel-caused extrinsic pulsatile compression is clearly visualized on the trachea. For this reason, AE is usually followed by chest imaging to confirm the AE findings [11, 16, 19, 24, 25]. In cooperating subjects, the flow-volume loop is an additional and effective method to document affected airways in suspected TC patients [22].
A relevant point to be discussed is the choice of treatment of AIA cases. Of the current series, 4 patients underwent surgery as all experienced serious unremitting events, namely chronic dry cough, recurrent pneumonia, lobar atelectasis and spontaneous pneumomediastinum and subcutaneous emphysema (the latter only in case 4), with monthly exacerbations and very short intercritical symptom-free periods. These findings, combined with the demonstration of TC > 50% at CT, led to conclude that surgery was mandatory. Conversely, in 55% of subjects (cases 5 to 9) surgery was not deemed necessary, given the non-severity of the clinical course. Even though symptoms started early, these patients progressively improved, did not report complications and had less infectious exacerbations, fewer episodes of barking cough and progressive extension of the well-being period, as case 9 report has shown. These findings, combined with TC < 50% at chest imaging, led to a conservative management decision. Follow-up confirmed that the clinical course is currently uneventful. Even though patients from previous studies have been addressed either to surgery or conservative treatment, in presence of severe symptoms due to high-grade tracheal stenosis, or if the symptoms do not regress upon medical treatment, surgical treatment is always recommended.
In our case series, surgical interventions consisted of aortopexy (cases 1 and 3) or tracheopexy (cases 2 and 4). Aortopexy is considered the preferred approach in patients with AIA [5, 45]. For aortopexy, pledgeted non resorbable sutures are placed in the adventitia of the ventral surface of the aortic arch without entering the aortic lumen at the origin of the innominate artery. Sutures are placed transternally and transcartilaginously, and tightened and secured to achieve anterior displacement of the aortic arch and the innominate artery [45]. In 2 children with AIA, the major contribution to airway compression was the hypermobility of the pars membranacea protruding into the tracheal lumen during cough, as shown by AE. In these cases, posterior tracheopexy consisting of suture of the posterior tracheal membrane to the anterior longitudinal ligament of the spine through a posterior right thoracotomy was considered necessary [46].
We retrospectively described a small cohort of children with chronic respiratory disorders which were ultimately attributed TC from AIA and managed through a multidisciplinary approach. We also provided a review of the relevant literature on the topic. Based on current findings, we believe that the diagnostic work up in cases presenting with symptoms or signs of airway compression must include a list of procedures aimed at confirming the suspected abnormality and defining the best therapeutic option as early as possible. We propose a management algorithm which may be helpful for clinicians dealing with infants and children with respiratory symptoms suspected to be secondary to AIA (Fig. 3).

Conclusions

TC caused by AIA may be a serious cause of chronic respiratory disease in childhood. Early diagnosis and prompt decision of treatment can reduce the risk of long-term airway obstructive disease and improve patients’ daily life. Data from this report may help in addressing the diagnostic work-up and the choice of treatment. A management algorithm of patients suspected of AIA based on the evidence from literature review is proposed. Like all algorithms, it is not meant to replace clinical judgment, but it should rather drive physicians to adopt a systematic approach to the disease.

Acknowledgements

Not applicable.

Declarations

In view of the retrospective nature of the study, the need for Ethics Committee approval was not considered.
Written informed consent for publication was obtained.

Competing interests

The authors declare that they have no competing interests.
Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://​creativecommons.​org/​licenses/​by/​4.​0/​. 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 in a credit line to the data.

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Literatur
1.
Zurück zum Zitat Shields MD, Doherty GM. Chronic cough in children. Paediatr Respir Rev. 2013;14:100–5; quiz 106, 137–8. Shields MD, Doherty GM. Chronic cough in children. Paediatr Respir Rev. 2013;14:100–5; quiz 106, 137–8.
2.
Zurück zum Zitat Kantar A, Marchant JM, Song W-J, Shields MD, Chatziparasidis G, Zacharasiewicz A, et al. History taking as a diagnostic tool in children with chronic cough. Front Pediatr. 2022;10:850912. Kantar A, Marchant JM, Song W-J, Shields MD, Chatziparasidis G, Zacharasiewicz A, et al. History taking as a diagnostic tool in children with chronic cough. Front Pediatr.  2022;10:850912.
3.
Zurück zum Zitat Donnelly LF, Bisset GS 3rd. Airway compression in children with abnormal thoracic configuration. Radiology. 1998;206:323–6. Donnelly LF, Bisset GS 3rd. Airway compression in children with abnormal thoracic configuration. Radiology. 1998;206:323–6.
4.
Zurück zum Zitat Kirks DR, Fram EK, Vock P, Effmann EL. Tracheal compression by mediastinal masses in children: CT evaluation. AJR Am J Roentgenol. 1983;141:647–51.CrossRef Kirks DR, Fram EK, Vock P, Effmann EL. Tracheal compression by mediastinal masses in children: CT evaluation. AJR Am J Roentgenol. 1983;141:647–51.CrossRef
5.
Zurück zum Zitat Perger L, Lee EY, Shamberger RC. Management of children and adolescents with a critical airway due to compression by an anterior mediastinal mass. J Pediatr Surg United States. 2008;43:1990–7.CrossRef Perger L, Lee EY, Shamberger RC. Management of children and adolescents with a critical airway due to compression by an anterior mediastinal mass. J Pediatr Surg United States. 2008;43:1990–7.CrossRef
6.
Zurück zum Zitat Hysinger EB, Panitch HB. Paediatric Tracheomalacia Paediatr Respir Rev. 2016;17:9–15. Hysinger EB, Panitch HB. Paediatric Tracheomalacia Paediatr Respir Rev. 2016;17:9–15.
7.
Zurück zum Zitat Sharma AK, Patel S, Meena D, Sahu RK, Goyal A, Garg PK, et al. Large sternal exostoses presenting as stridor: a surgical and anesthetic challenge. J Card Surg. 2020; 35: 2388–91. Sharma AK, Patel S, Meena D, Sahu RK, Goyal A, Garg PK, et al. Large sternal exostoses presenting as stridor: a surgical and anesthetic challenge. J Card Surg.  2020; 35: 2388–91.
8.
Zurück zum Zitat Ruiz-Solano E, Mitchell M. Rings and slings: not such simple things. Curr Cardiol Rep. 2022;24:1495–503.CrossRef Ruiz-Solano E, Mitchell M. Rings and slings: not such simple things. Curr Cardiol Rep. 2022;24:1495–503.CrossRef
9.
Zurück zum Zitat Kussman BD, Geva T, McGowan FX. Cardiovascular causes of airway compression. Paediatr Anaesth. 2004;14:60–74.CrossRef Kussman BD, Geva T, McGowan FX. Cardiovascular causes of airway compression. Paediatr Anaesth. 2004;14:60–74.CrossRef
10.
Zurück zum Zitat Ghezzi M, Silvestri M, Sacco O, Panigada S, Girosi D, Magnano GM, et al. Mild tracheal compression by aberrant innominate artery and chronic dry cough in children. Pediatr Pulmonol. 2016;51:286–94.CrossRef Ghezzi M, Silvestri M, Sacco O, Panigada S, Girosi D, Magnano GM, et al. Mild tracheal compression by aberrant innominate artery and chronic dry cough in children. Pediatr Pulmonol. 2016;51:286–94.CrossRef
11.
Zurück zum Zitat Gardella C, Girosi D, Rossi GA, Silvestri M, Tomà P, Bava G, et al. Tracheal compression by aberrant innominate artery: clinical presentations in infants and children, indications for surgical correction by aortopexy, and short- and long-term outcome. J Pediatr Surg. 2010;45:564–73.CrossRef Gardella C, Girosi D, Rossi GA, Silvestri M, Tomà P, Bava G, et al. Tracheal compression by aberrant innominate artery: clinical presentations in infants and children, indications for surgical correction by aortopexy, and short- and long-term outcome. J Pediatr Surg. 2010;45:564–73.CrossRef
12.
Zurück zum Zitat Woods RK, Sharp RJ, Holcomb GW 3rd, Snyder CL, Lofland GK, Ashcraft KW, et al. Vascular anomalies and tracheoesophageal compression: a single institution’s 25-year experience. Ann Thorac Surg. 2001;72:434–9.CrossRef Woods RK, Sharp RJ, Holcomb GW 3rd, Snyder CL, Lofland GK, Ashcraft KW, et al. Vascular anomalies and tracheoesophageal compression: a single institution’s 25-year experience. Ann Thorac Surg. 2001;72:434–9.CrossRef
13.
Zurück zum Zitat Myer CM 3rd, O’Connor DM, Cotton RT. Proposed grading system for subglottic stenosis based on endotracheal tube sizes. Ann Otol Rhinol Laryngol. 1994;103:319–23.CrossRef Myer CM 3rd, O’Connor DM, Cotton RT. Proposed grading system for subglottic stenosis based on endotracheal tube sizes. Ann Otol Rhinol Laryngol. 1994;103:319–23.CrossRef
14.
Zurück zum Zitat Marmon LM, Bye MR, Haas JM, Balsara RK, Dunn JM. Vascular rings and slings: long-term follow-up of pulmonary function. J Pediatr Surg. 1984;19:683–92.CrossRef Marmon LM, Bye MR, Haas JM, Balsara RK, Dunn JM. Vascular rings and slings: long-term follow-up of pulmonary function. J Pediatr Surg. 1984;19:683–92.CrossRef
15.
Zurück zum Zitat Gormley PK, Colreavy MP, Patil N, Woods AE. Congenital vascular anomalies and persistent respiratory symptoms in children. Int J Pediatr Otorhinolaryngol. 1999;51:23–31.CrossRef Gormley PK, Colreavy MP, Patil N, Woods AE. Congenital vascular anomalies and persistent respiratory symptoms in children. Int J Pediatr Otorhinolaryngol. 1999;51:23–31.CrossRef
16.
Zurück zum Zitat Jones DT, Jonas RA, Healy GB. Innominate artery compression of the trachea in infants. Ann Otol Rhinol Laryngol. 1994;103:347–50.CrossRef Jones DT, Jonas RA, Healy GB. Innominate artery compression of the trachea in infants. Ann Otol Rhinol Laryngol. 1994;103:347–50.CrossRef
17.
Zurück zum Zitat Fearon B, Shortreed R. Tracheobronchial compression by congenital cardiovascular anomalies in children. Syndrome of apnea. Ann Otol Rhinol Laryngol. 1963;72:949–69. Fearon B, Shortreed R. Tracheobronchial compression by congenital cardiovascular anomalies in children. Syndrome of apnea. Ann Otol Rhinol Laryngol. 1963;72:949–69.
18.
Zurück zum Zitat Adler SC, Isaacson G, Balsara RK. Innominate artery compression of the trachea: diagnosis and treatment by anterior suspension. A 25-year experience. Ann Otol Rhinol Laryngol. 1995;104:924–7. Adler SC, Isaacson G, Balsara RK. Innominate artery compression of the trachea: diagnosis and treatment by anterior suspension. A 25-year experience. Ann Otol Rhinol Laryngol. 1995;104:924–7.
19.
Zurück zum Zitat McLaughlin RBJ, Wetmore RF, Tavill MA, Gaynor JW, Spray TL. Vascular anomalies causing symptomatic tracheobronchial compression. Laryngoscope. 1999;109:312–9.CrossRef McLaughlin RBJ, Wetmore RF, Tavill MA, Gaynor JW, Spray TL. Vascular anomalies causing symptomatic tracheobronchial compression. Laryngoscope. 1999;109:312–9.CrossRef
20.
Zurück zum Zitat Mustard WT, Bayliss CE, Fearon B, Pelton D, Trusler GA. Tracheal compression by the innominate artery in children. Ann Thorac Surg. 1969;8:312–9.CrossRef Mustard WT, Bayliss CE, Fearon B, Pelton D, Trusler GA. Tracheal compression by the innominate artery in children. Ann Thorac Surg. 1969;8:312–9.CrossRef
21.
Zurück zum Zitat Eklöf O, Ekström G, Eriksson BO, Michaëlsson M, Stephensen O, Söderlung S, et al. Arterial anomalies causing compression of the trachea and-or the oesophagus. A report of 30 symptomatic cases. Acta Paediatr Scand. 1971;60:81–9. Eklöf O, Ekström G, Eriksson BO, Michaëlsson M, Stephensen O, Söderlung S, et al. Arterial anomalies causing compression of the trachea and-or the oesophagus. A report of 30 symptomatic cases. Acta Paediatr Scand. 1971;60:81–9.
22.
Zurück zum Zitat Malik TH, Bruce IA, Kaushik V, Willatt DJ, Wright NB, Rothera MP. The role of magnetic resonance imaging in the assessment of suspected extrinsic tracheobronchial compression due to vascular anomalies. Arch Dis Child. 2006;91:52–5.CrossRef Malik TH, Bruce IA, Kaushik V, Willatt DJ, Wright NB, Rothera MP. The role of magnetic resonance imaging in the assessment of suspected extrinsic tracheobronchial compression due to vascular anomalies. Arch Dis Child. 2006;91:52–5.CrossRef
23.
Zurück zum Zitat Erwin EA, Gerber ME, Cotton RT. Vascular compression of the airway: indications for and results of surgical management. Int J Pediatr Otorhinolaryngol Ireland. 1997;40:155–62.CrossRef Erwin EA, Gerber ME, Cotton RT. Vascular compression of the airway: indications for and results of surgical management. Int J Pediatr Otorhinolaryngol Ireland. 1997;40:155–62.CrossRef
24.
Zurück zum Zitat Grimmer JF, Herway S, Hawkins JA, Park AH, Kouretas PC. Long-term results of innominate artery reimplantation for tracheal compression. Arch Otolaryngol Head Neck Surg. 2009;135:80–4. Grimmer JF, Herway S, Hawkins JA, Park AH, Kouretas PC. Long-term results of innominate artery reimplantation for tracheal compression. Arch Otolaryngol Head Neck Surg. 2009;135:80–4.
25.
Zurück zum Zitat Hawkins JA, Bailey WW, Clark SM. Innominate artery compression of the trachea. Treatment by reimplantation of the innominate artery. J Thorac Cardiovasc Surg. 1992;103:678–82. Hawkins JA, Bailey WW, Clark SM. Innominate artery compression of the trachea. Treatment by reimplantation of the innominate artery. J Thorac Cardiovasc Surg. 1992;103:678–82.
31.
Zurück zum Zitat Akyüz M, Işık O, Duman Şenol H, Bakiler AR. A diagnostic challenge for the clinicians: Aberrant innominate artery. Turk gogus kalp damar cerrahisi Derg. 2019;27:130–1. Akyüz M, Işık O, Duman Şenol H, Bakiler AR. A diagnostic challenge for the clinicians: Aberrant innominate artery. Turk gogus kalp damar cerrahisi Derg. 2019;27:130–1.
32.
Zurück zum Zitat Ullmann N, Santamaria F, Allegorico A, Fainardi V, Borrelli M, Ferraro VA, et al. Primary ciliary dyskinesia: A multicenter survey on clinical practice and patient management in Italy. Pediatr Pulmonol. 2023; Ullmann N, Santamaria F, Allegorico A, Fainardi V, Borrelli M, Ferraro VA, et al. Primary ciliary dyskinesia: A multicenter survey on clinical practice and patient management in Italy. Pediatr Pulmonol. 2023;
33.
Zurück zum Zitat Romano R, Borrelli M, Cirillo E, Giardino G, Spadaro G, Crescenzi L, et al. Respiratory Manifestations in primary immunodeficiencies: findings from a pediatric and adult cohort. Arch Bronconeumol. 2021. p. 712–4. Romano R, Borrelli M, Cirillo E, Giardino G, Spadaro G, Crescenzi L, et al. Respiratory Manifestations in primary immunodeficiencies: findings from a pediatric and adult cohort. Arch Bronconeumol. 2021. p. 712–4.
34.
Zurück zum Zitat Duse M, Santamaria F, Verga MC, Bergamini M, Simeone G, Leonardi L, et al. Inter-society consensus for the use of inhaled corticosteroids in infants, children and adolescents with airway diseases. Ital J Pediatr. 2021;47:97. Duse M, Santamaria F, Verga MC, Bergamini M, Simeone G, Leonardi L, et al. Inter-society consensus for the use of inhaled corticosteroids in infants, children and adolescents with airway diseases. Ital J Pediatr.  2021;47:97.
35.
Zurück zum Zitat Columbo C, Landolfo F, De Rose DU, Massolo AC, Secinaro A, Santangelo TP, et al. The role of lung function testing in newborn infants with congenital thoracic arterial anomalies. Front Pediatr. 2021;9:682551. Columbo C, Landolfo F, De Rose DU, Massolo AC, Secinaro A, Santangelo TP, et al. The role of lung function testing in newborn infants with congenital thoracic arterial anomalies. Front Pediatr. 2021;9:682551.
36.
Zurück zum Zitat Calabrese C, Corcione N, Di Spirito V, Guarino C, Rossi G, Domenico Gargiulo G, et al. Recurrent respiratory infections caused by a double aortic arch: the diagnostic role of spirometry. Respir Med Case Rep. 2013;8:47–50. Calabrese C, Corcione N, Di Spirito V, Guarino C, Rossi G, Domenico Gargiulo G, et al. Recurrent respiratory infections caused by a double aortic arch: the diagnostic role of spirometry. Respir Med Case Rep. 2013;8:47–50.
37.
Zurück zum Zitat Marini JJ. Acute Lobar Atelectasis Chest. 2019;155:1049–58. Marini JJ. Acute Lobar Atelectasis Chest. 2019;155:1049–58.
38.
Zurück zum Zitat Nir V, Bentur L, Zucker-Toledano M, Gur M, Adler Z, Hanna M, et al. Functional capacity and quality of life in patients with vascular ring. Pediatr Pulmonol. 2022;57:2946–53.CrossRef Nir V, Bentur L, Zucker-Toledano M, Gur M, Adler Z, Hanna M, et al. Functional capacity and quality of life in patients with vascular ring. Pediatr Pulmonol. 2022;57:2946–53.CrossRef
39.
Zurück zum Zitat Jaggar SI, Haxby E. Sedation, anaesthesia and monitoring for bronchoscopy. Paediatr Respir Rev. 2002;3:321–7. Jaggar SI, Haxby E. Sedation, anaesthesia and monitoring for bronchoscopy. Paediatr Respir Rev. 2002;3:321–7.
40.
Zurück zum Zitat Okata Y, Hasegawa T, Bitoh Y, Maeda K. Bronchoscopic assessments and clinical outcomes in pediatric patients with tracheomalacia and bronchomalacia. Pediatr Surg Int. 2018;34:55–61.CrossRef Okata Y, Hasegawa T, Bitoh Y, Maeda K. Bronchoscopic assessments and clinical outcomes in pediatric patients with tracheomalacia and bronchomalacia. Pediatr Surg Int. 2018;34:55–61.CrossRef
41.
Zurück zum Zitat Newman B. Magnetic resonance imaging for congenital lung malformations. Pediatr Radiol. 2022;52:312–22.CrossRef Newman B. Magnetic resonance imaging for congenital lung malformations. Pediatr Radiol. 2022;52:312–22.CrossRef
42.
Zurück zum Zitat Montella S, Maglione M, Bruzzese D, Mollica C, Pignata C, Aloj G, et al. Magnetic resonance imaging is an accurate and reliable method to evaluate non-cystic fibrosis paediatric lung disease. Respirology. 2012;17:87–91.CrossRef Montella S, Maglione M, Bruzzese D, Mollica C, Pignata C, Aloj G, et al. Magnetic resonance imaging is an accurate and reliable method to evaluate non-cystic fibrosis paediatric lung disease. Respirology. 2012;17:87–91.CrossRef
43.
Zurück zum Zitat Borrelli M, Corcione A, Castellano F, Fiori Nastro F, Santamaria F. Coronavirus disease 2019 in children. Front Pediatr. 2021;9:668484. Borrelli M, Corcione A, Castellano F, Fiori Nastro F, Santamaria F. Coronavirus disease 2019 in children. Front Pediatr. 2021;9:668484.
44.
Zurück zum Zitat Rapp JB, Ho-Fung VM, Ramirez KI, White AM, Otero HJ, Biko DM. Dual-source computed tomography protocols for the pediatric chest - scan optimization techniques. Pediatr Radiol. 2022;1–12. Rapp JB, Ho-Fung VM, Ramirez KI, White AM, Otero HJ, Biko DM. Dual-source computed tomography protocols for the pediatric chest - scan optimization techniques. Pediatr Radiol. 2022;1–12.
45.
Zurück zum Zitat Isik O, Akyuz M, Ozcifci G, Durak F, Mercan I, Anıl AB. Role of aortopexy in the treatment of aberrant innominate artery in children. Pediatr Surg Int. 2022;39:47. Isik O, Akyuz M, Ozcifci G, Durak F, Mercan I, Anıl AB. Role of aortopexy in the treatment of aberrant innominate artery in children. Pediatr Surg Int. 2022;39:47.
46.
Zurück zum Zitat Torre M, Carlucci M, Speggiorin S, Elliott MJ. Aortopexy for the treatment of tracheomalacia in children: review of the literature. Ital J Pediatr. 2012;38:62. Torre M, Carlucci M, Speggiorin S, Elliott MJ. Aortopexy for the treatment of tracheomalacia in children: review of the literature. Ital J Pediatr. 2012;38:62.
Metadaten
Titel
Chronic respiratory disorders due to aberrant innominate artery: a case series and critical review of the literature
verfasst von
Adele Corcione
Melissa Borrelli
Leonardo Radice
Oliviero Sacco
Michele Torre
Francesco Santoro
Gaetano Palma
Eleonora Acampora
Francesca Cillo
Pietro Salvati
Angelo Florio
Francesca Santamaria
Publikationsdatum
01.12.2023
Verlag
BioMed Central
Erschienen in
Italian Journal of Pediatrics / Ausgabe 1/2023
Elektronische ISSN: 1824-7288
DOI
https://doi.org/10.1186/s13052-023-01473-0

Weitere Artikel der Ausgabe 1/2023

Italian Journal of Pediatrics 1/2023 Zur Ausgabe

Ähnliche Überlebensraten nach Reanimation während des Transports bzw. vor Ort

29.05.2024 Reanimation im Kindesalter Nachrichten

Laut einer Studie aus den USA und Kanada scheint es bei der Reanimation von Kindern außerhalb einer Klinik keinen Unterschied für das Überleben zu machen, ob die Wiederbelebungsmaßnahmen während des Transports in die Klinik stattfinden oder vor Ort ausgeführt werden. Jedoch gibt es dabei einige Einschränkungen und eine wichtige Ausnahme.

Alter der Mutter beeinflusst Risiko für kongenitale Anomalie

28.05.2024 Kinder- und Jugendgynäkologie Nachrichten

Welchen Einfluss das Alter ihrer Mutter auf das Risiko hat, dass Kinder mit nicht chromosomal bedingter Malformation zur Welt kommen, hat eine ungarische Studie untersucht. Sie zeigt: Nicht nur fortgeschrittenes Alter ist riskant.

Begünstigt Bettruhe der Mutter doch das fetale Wachstum?

Ob ungeborene Kinder, die kleiner als die meisten Gleichaltrigen sind, schneller wachsen, wenn die Mutter sich mehr ausruht, wird diskutiert. Die Ergebnisse einer US-Studie sprechen dafür.

Bei Amblyopie früher abkleben als bisher empfohlen?

22.05.2024 Fehlsichtigkeit Nachrichten

Bei Amblyopie ist das frühzeitige Abkleben des kontralateralen Auges in den meisten Fällen wohl effektiver als der Therapiestandard mit zunächst mehrmonatigem Brilletragen.

Update Pädiatrie

Bestellen Sie unseren Fach-Newsletter und bleiben Sie gut informiert.