Abstract
In this description of the clinical case, repair of the rarest pathology-the ring-sling complex in combination with a double outlet right
ventricle in a child of 3 months age old is presented. The ring-sling complex consists of complete cartilaginous rings together with an
abnormal origin of the left branch of the pulmonary artery from the right branch with a retro-tracheal its course and the formation of
a vascular loop or the so-called sling. In the world literature of this combination, including sliding tracheal plastic and radical correction
of the double outlet right ventricle has not been described yet. A feature of the presented observation is also the patient's early
age which is original for Russia at time of surgical operation. Interesting for the clinician, there are also sudden changes in the clinical
state of the patient as a result of the course of the disease, which ultimately required, in addition to reimplantation of the aberrant
left pulmonary artery, the performance of salvage tracheoplasty with the need for an extended seance of extracorporeal membrane
oxygenation procedure. It should also be noted that active cardiac surgery of patients with concomitant pathology of the trachea
under conditions of bypass circulation in our country appeared only in the last few years. In this regard, each such observation is valuable
for the accumulation of native experience which is still limited mainly as single case-reports.
References
1. Berdon W.E., Baker D.H., Wung J.T., Chrispin A.,
Kozlowski K., de Silva M. et al. Complete cartilage-ring tracheal
stenosis associated with anomalous left pulmonary
artery: the ring-sling complex. Radiology. 1984; 152: 57–64.
2. Weinberg P.M., Emmanouilides G., Reimenschneider T.,
Allen H. (Eds.) Moss and adams heart disease in infants, children
and adolscents. 5th ed. Baltimore: Williams and Wilkins;
1995: 810–37.
3. Arcieri L., Moschetti R., Assanta N., Cantinotti M. et al.
Successful simultaneous correction of complex congenital tracheal
stenosis and tetralogy of Fallot. World J. Pediatr.
Congenit. Heart Surg. 2012; 3 (4): 511–4.
4. Бокерия Л.А., Свободов А.А., Никифоров В.С., Адкин Д.В.,
Юрпольская Л.А. Случай успешной коррекции аномально-
го отхождения левой легочной артерии (слинг) с дефектом
межжелудочковой перегородки у ребенка в возрасте 6 ме-
сяцев. Детские болезни сердца и сосудов. 2010; 3: 68–9.
5. Разумовский А.Ю., Афуков И.И., Кулаев А.Д., Алха-
сов А.Б. и др. Скользящая трахеопластика у детей в усло-
виях экстракорпоральной мембранной оксигенации. Пер-
вый опыт в России. Хирургия. Журнал им. Н.И. Пирогова.
2015; 8: 4–13. DOI: 10.17116/hirurgia201584-13.
6. Шаталов К.В., Разумовский А.Ю., Арнаутова И.В., Турдие-
ва Н.С. Хирургическая коррекция слинга левой легочной ар-
терии в сочетании со стенозом трахеи у детей раннего воз-
раста. Детские болезни сердца и сосудов. 2015; 2: 34–8.
7. Chen Sh., Ting-Fang Shih T., Liu K., Chiu I., Wu M. et al.
Measurement of tracheal size in children with congenital heart
disease by computed tomography. Ann. Thorac. Surg. 2004;
77: 1216–21.
8. Santana P.R.P., Irion K.L., Escuissato D.L., da Silva Porto N.,
Fisher G.B. et al. Pulmonary artery sling: report of two cases
and review of the literature. Radiol. Bras. 2005; 38 (4)
(in Portuguese).
9. Glaevecke D., Doehle H.U. Uber eine selltene angeborene
Anomalie der Pulmonalarterie. München Med. Wochenschr.
1897; 44: 950–3.
10. Сontro S., Miller R.A., White H. and Potts A.J. Bronchial
obstruction due to pulmonary artery anomalies II. Pulmonary
artery aneurysm. Circulation. 1958; XVII: 424.
11. Yu J., Liao C., Ge S., Weng Z., Hsiung M., Chang J., Chen F.L.
The prevalence and clinical impact of pulmonary artery sling
on school-aged children: a large-scale screening study.
Pediatr. Pulmonol. 2008; 43: 656–61.
12. Xie J., Juan Y., Wang Q., Chen J., Zhuang J., Xie Z. et al.
Evaluation of left pulmonary artery sling, associated cardio-
vascular anomalies, and surgical outcomes using cardiovascular
computed tomography angiography. Scientific Reports.
2016; 7; article number: 40042.
13. Gregor K. Ein Fall von angeborener Missbildung der Luftrohre.
Jahrb. Kinderheilkd. 1899; 49: 123–6.
14. Procacci C., Residori E., Bertocco M., Di Benedeito P.,
Andreis I.A.B., D'Attoma N. Left pulmonary artery sling in the
adult: case report and review of the literature. Cardiovasc.
Interv. Radiol. 1993; 10: 388–91.
15. Backer C.L., Russel H.M., Kaushal S., Rastatter J.C.,
Rigsby C.K., Holinger L.D. Pulmonary artery sling: current
results with cardiopulmonary bypass. J. Thorac. Cardiovasc.
Surg. 2012; 143: 144–51.
16. Турдиева Н.С. Тактика хирургического лечения сосудис-
тых колец изолированной формы и в сочетании с другими
врожденными пороками сердца у детей грудного и раннего
возраста: Автореф. дис. ... канд. мед. наук. М.; 2017.
17. Kubota Y., Toyoda Y., Nagata N., Kubota H., Sawada S., Murakawa
M., Fujimori M. Tracheo-bronchial angles in infants and
children. Anesthesiology. 1986; 64: 374–6.
18. Ким А.И., Акатов Д.С., Макаренко В.Н., Мумладзе К.В., Ни-
кифоров В.С. Случай успешной коррекции аномального
отхождения левой легочной артерии от правой легочной
артерии с бужированием стеноза трахеи. Детские болез-
ни сердца и сосудов. 2012; 4: 68–9.
19. Potts W.J., Holinger P.H., Rosenblum A.H. Anomalous left pulmonary
artery сausing obstruction to right main bronchus:
report of a case. JAMA. 1954; 155: 1409–11.
20. Cantrell J.R., Guild H.G. Congenital stenosis of the trachea.
Am. J. Surg. 1964; 108: 297–305.
21. Hickey M.S.J., Wood A.E. Pulmonary artery sling with tracheal
stenosis: One-stage repair. Ann. Thorac. Surg. 1987; 44: 416–7.
22. Oshima Y., Yamaguchi M., Ohashi H., Yoshimura N et al.
Pulmonary artery sling with tracheal stenosis primary repair in
infancy. Japan. J. Thorac. Cardiovasc. Surg. 1998; 46 (4):
347–53.
23. Wijeweera O., Ng S.B. Retrospective review of tracheoplasty
for congenital tracheal stenosis. Singapore Med. J. 2011;
52 (10): 726–9.
24. Tsang V., Murday A., Gillbe C., Goldstraw P. Slide tracheoplasty
for congenital funnel-shaped tracheal stenosis. Ann.
Thorac. Surg. 1989; 48: 632–5.
25. Grillo H.C. Slide tracheoplasty for long-segment congenital
tracheal stenosis. Ann. Thorac. Surg. 1994; 58: 613–21.
About the authors
- Bazylev Vladlen Vladlenovich, Dr Med. Sc., Professor, Chief Physician, orcid.org/0000-0001-6089-9722;
- Chernogrivov Igor' Evgen'evich, Dr Med. Sc., Cardiovascular Surgeon, orcid.org/0000-0002-6619-3875;
- Gornostaev Aleksandr Aleksandrovich, Anesthesiologist-Intensivist, Head of Department, orcid.org/0000-0002-4477-0037;
- Paramonova Tat'yana Innokent'evna, Radiologist, Head of Department;
- Suleymanov Shakhriyar Fazailovich, Anesthesiologist-Intensivist;
- Toshchev Petr Nikolaevich, Anesthesiologist-Intensivist;
- Shiganov Aleksandr Vladimirovich, Endoscopist; Chernogrivov Aleksey Evgen'evich, Dr Med. Sc., Cardiovascular Surgeon, Head of Department, orcid.org/0000-0002-6207-4799