Hostname: page-component-74656f9884-8r8nv Total loading time: 0 Render date: 2026-11-09T02:31:17.019Z Has data issue: false hasContentIssue false
  • English
  • Français

A rare cause of hemoptysis in childhood: peripheral pulmonary artery pseudoaneurysm to bronchial fistula in a case with tetralogy of Fallot

Published online by Cambridge University Press:  22 April 2022

Selin Guner Open the ORCID record for Selin Guner [Opens in a new window] ,
Turkay Saritas  and
Taliha Oner Open the ORCID record for Taliha Oner [Opens in a new window]
Selin Guner*
Affiliation:
University of Health Sciences, Umraniye Training and Education Hospital, Umraniye, Istanbul, Turkey
Turkay Saritas
Affiliation:
University of Health Sciences, Umraniye Training and Education Hospital, Umraniye, Istanbul, Turkey
Taliha Oner
Affiliation:
University of Health Sciences, Umraniye Training and Education Hospital, Umraniye, Istanbul, Turkey
*
Author for correspondence: Selin Guner, Umraniye Training and Education Hospital Elmalikent Mh, Adem Yavuz Cad. No: 1 Cocuk Kardiyolojisi Poliklinigi Umranıye, Istanbul, Turkey. Tel: +905398691121; Fax: +902166327124. E-mail: selinn.guneer@gmail.com
Rights & Permissions [Opens in a new window]

Abstract

We herein report a patient complaining from significant hemoptysis due to secondary fistulisation of the peripheral branch of the left pulmonary artery and the left bronchial tree, which was successfully treated with a transcatheter angiography.

Keywords

Hemoptysischildocclusion

Information

Type
Brief Report
Information
Cardiology in the Young , Volume 33 , Issue 1 , January 2023 , pp. 127 - 129
Check for updates
Copyright
© The Author(s), 2022. Published by Cambridge University Press

Hemoptysis is a rare but life-threatening symptom in childhood. Infections, particularly infections of the lower respiratory tract, foreign body aspirations, and tracheostomy complications are frequently cited as aetiological factors. Vascular pathologies are rare causes in aetiology. Reference Bannister1–Reference Patel, Uchida, Feola and Meier3

We herein report a patient complaining from significant hemoptysis due to secondary fistulisation of the peripheral pulmonary artery between the peripheral branch of the left pulmonary artery and the left bronchial tree, which was successfully treated with a transcatheter angiography.

Case report

A 6-year-old boy patient was admitted to our emergency department due to increased discharge of bloody sputum. The sputum had been extracted intermittently for the past 2 years and consisted of remnants of blood and mucus. On his medical records, it was discovered that he had a total correction surgery for tetralogy of Fallot performed 4 years ago. His post-operative period was uneventful, and he had used acetyl salicylic acid during this period. When he began to experience hemoptysis, an erythrocyte suspension was transfused due to iron deficiency anaemia. There was no history of chronic respiratory infections or a foreign body aspiration in the patient and had received all immunisations.

Physical examination of the patient, who presented with no genetic disease in his pedigree was conscious, with a withering appearance. Remnants of blood clots were detected in the oropharynx. The heart rate of the patient was 115 beats/min, with a blood pressure of 100/65 mmHg. While the cardiac examination showed 2–3/6º systolic murmur in the left 2.–3. intercostal range, there was no sign of a pathology detected in other organ system examinations, including the respiratory examination. The patient had a leucocyte count of 6470/mm3, haemoglobin 5.5 gr/dL, and a platelet count of 340,000/mm3 on complete blood count; liver–kidney function tests, and electrolytes and coagulation tests were normal. A ferritin level of 4.72 ng/mL was detected. A suspicious pericardiac infiltration was detected on chest X-ray.

The patient’s antiaggregant dose of acetyl salicylic acid was stopped, an erythrocyte suspension transfusion was conducted, an oral iron supplementation was begun, and the patient’s hemoptysis and hematemesis could not be cleared, thus a proton pump inhibitor was started. Nasolaryngoscopic examination indicated no signs of active bleeding. The upper gastrointestinal tract endoscopy was conducted to rule out possible gastrointestinal tract pathologies, and the findings were normal.

A thorax CT imaging to rule out possible vascular pathologies (aortic pulmonary collateral artery, etc.) and to display lung parenchyma in the patient who underwent full correction surgery due to tetralogy of Fallot was conducted. An aneurysmatic dilatation reaching 1.5 cm was observed in the lower lobe segmentary branch of the left pulmonary artery (Figure 1 a, b). No signs of haemorrhage were present on rigid bronchoscopy, and a catheter angiography was planned for the patient, to better visualise the aneurysmatic structure as well as for therapeutic purposes. Angiography showed extravasation of the contrast agent to the left main bronchus and trachea immediately after contrasting the aneurysm with an injection of radio-opaque material into the left pulmonary artery. The proximal part of the aneurysm was sealed with 10- and 12-mm vascular plugs. A control angiography conducted following the procedure indicated no sign of contrast material leakage into the aneurysm or the bronchus (Figure 2 a, b, c).

Fig. 1.

Aneurysmatic dilatation in left pulmonary artery brunch on computerized tomography (red stars).

Fig. 2.

Aneurysmatic dilatation in left pulmonary artery brunch on angiography (black star), filling with opaque substance and blood via pulmonary-bronchial connection to left bronchus (white stars) and occlusion of pulmonary aneursyms with vascular plug (white arrows).

The patient, who was stable after angiography, was discharged after 2 days. The patient did not present with a recurrence of hemoptysis, after a 3-month follow-up.

Discussion

Hemoptysis is a rare but life-threatening symptom in childhood. Infections, particularly infections of the lower respiratory tract, foreign body aspirations, and tracheostomy complications are frequently cited as aetiological factors. Other rare aetiological causes include chronic lung diseases (cystic fibrosis, bronchiectasis, tuberculosis, and pulmonary hemosiderosis), upper respiratory pathologies (tonsil haemorrhage, nose bleeds, and laryngitis), certain diseases of the gastrointestinal tract, CHD (tetralogy of Fallot especially accompanied by aortopulmonary collateral arteries), neoplasia, bleeding diathesis, and Munchausen’s syndrome and also include diseases such as Good Pasteur syndrome and Wegener’s granulomatosis, which are diseases of vascular origin and can infiltrate the lungs and kidneys. Reference Bannister1–Reference Saed, Potalivo, Panzini and Bisetti4

In the presented case, the anamnesis, physical examination, and basic laboratory tests rule out both the leading aetiological and other rare aetiological causes. Possible upper respiratory pathologies with ear, nose, and throat are ruled out with flexural nasopharyngoscopy, while gastrointestinal tract pathologies are also ruled out by an upper gastrointestinal tract endoscopy.

Pathologies of vascular origin, arteriovenous malformation, hereditary telangiectasia, tracheal hemangioma, bronchial artery or pulmonary artery pseudoaneurysm, direct pulmonary artery trauma, or lung injury are much rarer causes. Reference Batra and Holinger2,Reference Patel, Uchida, Feola and Meier3

The patient had a surgical correction procedure for tetralogy of Fallot 4 years prior to presentation. He underwent a contrasted tomography angiography of the thorax to investigate the pulmonary parenchyma in detail and to rule out any possible bronchial artery pseudoaneurysms which might originate from an aortopulmonary collateral artery. An aneurysm located at the distal segment of the left pulmonary artery was detected leading to bronchial erosion and hemoptysis. A pulmonary artery-bronchial fistulisation was confirmed with conventional catheter angiography, and pathology was treated successfully with percutaneous techniques.

These fistulisations can be occluded with different types of devices such as coils and vascular plugs. Reference Batra and Holinger2 In this case, we occluded the pulmonary artery-bronchial fistulisation with vascular plugs.

Secondary pulmonary artery injuries due to catheterisation or surgery and associated hemoptysis may develop. In addition, chronic lung infections, mushroom ball causes, etc.,or traffic accidents, stabbings, etc., and subsequent cases of hemoptysis due to lung injury have been reported. Reference Sokouti and Vahid Montazeri5–Reference Shin, Shin and Choi7 In addition, fine needle biopsy for the bronchial system, etc., and hemoptysis may develop after such invasive procedures. Reference Sabath8 In this case, in addition to the smooth post-operative period of the patient, no invasive interventions were made towards pulmonary arteries or the bronchial system during both the pre-operative and the post-operative period.

In conclusion, it is well known that sometimes hemoptysis may lead to life-threatening complications. The frequent causes are sought when investigating the aetiology of hemoptysis. On the other hand, although seldom, congenital cardiovascular pathologies (e.g., presence of aortopulmonary collateral arteries in tetralogy of Fallot) should be remembered as an aetiology for hemoptysis and ruled out with contrasted CT, angiography, or conventional angiography. When the cause is detected, it should be managed with appropriate measures to prevent bleeding and further consequences.

Acknowledgements

Authors convey sincere gratitude to Mr. Robert Moore for the linguistic revision of the manuscript.

Financial support

This research received no specific grant from any funding agency, commercial or not-for-profit sectors.

Conflicts of interest

None.

Ethical standards

This case report was appropriate by the Hospital Institutional Ethical Committee.

References

Bannister, M. Pediatric hemoptysis and the otorhinolaryngologist: systematic review. Int J Pediatric Otorhinolaryngology 2017; 92: 99–102.CrossRefGoogle ScholarPubMed
Batra, PS, Holinger, LD. Etiology and management of pediatric hemoptysis. Arch Otolaryngol Head Neck Surg 2001; 127: 377–382.CrossRefGoogle ScholarPubMed
Patel, R, Uchida, D, Feola, GP, Meier, JD. Bronchial artery pseudoaneurysm as an unsuspected cause of hemoptysis in a pediatric patient. Ann Otol Rhinol Laryngol 2014; 123: 591–595.CrossRefGoogle ScholarPubMed
Saed, G, Potalivo, S, Panzini, L, Bisetti, A. Munchausen’s syndrome. a case of factitious hemoptysis. Panminerva Med 1999; 41: 62–67.Google ScholarPubMed
Sokouti, M, Vahid Montazeri, V. Delayed massive hemoptysis 20 years after lung stabbing: an unusual presentation. Eur J Cardiothorac Surg 2007; 32: 679–681.CrossRefGoogle ScholarPubMed
Cerillo, AG, Barberi, E, Amoretti, F, Berti, S. Massive hemoptysis from a pulmonary artery pseudoaneurysm during cardiac surgery. Turk Thorac J 2018; 19: 94–96.CrossRefGoogle ScholarPubMed
Shin, S, Shin, T-B, Choi, H, et al. Peripheral pulmonary arterial pseudoaneurysms: therapeutic implications of endovascular treatment and angiographic classifications. Radiology 2010; 256: 656–664.CrossRefGoogle ScholarPubMed
Sabath, BF. Delayed massive hemoptysis after endobronchial ultrasound-guided transbronchial needle aspiration. Endosc Ultrasound 2020; 9: 419–420.CrossRefGoogle ScholarPubMed
Figure 0

Fig. 1. Aneurysmatic dilatation in left pulmonary artery brunch on computerized tomography (red stars).

Figure 1

Fig. 2. Aneurysmatic dilatation in left pulmonary artery brunch on angiography (black star), filling with opaque substance and blood via pulmonary-bronchial connection to left bronchus (white stars) and occlusion of pulmonary aneursyms with vascular plug (white arrows).