CORONARY PULMONARY FISTUL Текст научной статьи по специальности «Клиническая медицина»

https://doi.org/10.35805/BSK2022I044

Ormanov B.K.

orcid.org/0000-0002-8457-4911

Abilkhanov E.E.

orcid.org/0000-0002-3730-6164

Azizullayev I.Kh.

orcid.org/0000-0001-5631-6993

Sapunov A.V.

orcid.org/0000-0002-7125-8178

Kudaibergen A.B.

orcid.org/0000-0002-2725-7530

Corresponding author: Kudaibergen A.B. - Resident Cardiac Surgeon, "A.N. Syzganov National Scientific Center for Surgery" JSC, Almaty, Kazakhstan E-mail: kudaibergen_med@mail.ru

Conflict of interest

The authors declare that they have no conflicts of interest

Keywords

coronary fistula, vascular anomaly congenital defect

CORONARY PULMONARY FISTULA

Ormanov B.K., Abilkhanov E.E., Azizullayev I.Kh., Sapunov A.V., Kudaibergen A.B.

"A.N. Syzganov National Scientific Center of Surgery" JSC, Almaty, Kazakhstan

Abstract

Coronary arteriovenous fistula (CAF) is a rare form of congenital heart disease. However, it is the most common type of congenital anomaly of the coronary arteries. [1] When there is the connection between the coronary artery and the chambers of the heart, it is called a coronary chamber fistula. The fistula may also be between a coronary artery and another adjacent vessel from the pulmonary or systemic circulation. An open fistula provides low-resistance flow by directing blood from an artery into a vein, heart chamber, or other low-pressure vessel such as the pulmonary artery. Patients with CAF may develop symptoms at birth or later in life, depending on the type of fistula and the presence of collateral circulation. Studies have reported an association between ventricular arrhythmias and sudden cardiac death syndromes in young adults and athletes with certain types of coronary anomalies, such as anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) [2-5]. The most common symptom is a myocardial ischemia. The purpose of this article is to present a clinical case of endovascular treatment of coronary pulmonary fistula. As a result of a modern diagnostic methods, such as CT angiography with three-dimensional reconstruction, it is not difficult to assess the degree and nature of the pathology. Having assessed the tactics, modern doctors are able to cope with coronary arteriovenous fistulas with great success using minimally invasive X-ray endovascular technologies.

Коронарно-пульмональная фистула

Автор для корреспонденции: Кудайберген А.Б. - Резидент-кардиохирург, АО «Национальный научный центр хирургии им. А.Н. Сызганоеа», г. Алматы, Казахстан E-mail: kudaibergen_med@mail.ru

Орманов Б.К., Абилханов Е.Е., Азизуллаев И.Х., Сапунов А.В., Кудайберген А.Б.

АО «Национальный научный центр хирургии имени А.Н. Сызганова», г. Алматы, Казахстан

Конфликт интересов

Авторы заявляют об отсутствии конфликта интересов

Ключевые слова

коронарная фистула, аномалия сосудов, врожденный порок

Аннотация

Коронарная артериовенозная фистула (КАФ) является редкой формой врожденного порока сердца. Тем не менее, это наиболее распространенный тип врожденных аномалий коронарных артерий. Когда он находится между коронарной артерией и камерами сердца, он называется коронарно-камерным свищом. Свищ также может быть между коронарной артерией и другим соседним сосудом из легочного или большого круга кровообращения. Открытая фистула обеспечивает поток с низким сопротивлением, направляя кровь непосредственно из артерии в вену, сердечную камеру или другой сосуд с низким давлением, такой как легочная артерия. У пациентов с КАФ симптомы могут развиваться при рождении или в более позднем возрасте, в зависимости от типа фистулы и наличия коллатерального кровообращения. В исследованиях сообщалось о связи между желудочковыми аритмиями и синдромами внезапной сердечной смерти у молодых людей и спортсменов при определенных типах коронарных аномалий, таких как аномальное отхождение левой коронарной артерии от легочной артерии ^САРА). Одышка при физической нагрузке и стенокардия из-за ишемии миокарда являются самыми частыми симптомами. В данной статье представлен клинический случай эндоваскулярного лечения коронарно-пульмональной фистулы. На данный момент, благодаря современным методам диагностики, таких как КТ ангиография, с трехмерной реконструкцией, оценить степень и характер патологии не составляет труда. Оценив тактику, современные врачи с большим успехом способны справиться с коронарными артериовенозными фистулами с использованием малоинвазивных рентгенэндоваску-лярных технологий.

Коронарльщ екпе фистуласы

Орманов Б.К., Абилханов Е.Е., Азизуллаев И.Х., Сапунов А.В., Кудайберген А.Б.

«А.Н. Сызганов атындагы ¥лттьщ хирургия орталыгы» А^, Алматы ^аза^стан

Ацдатпа

Коронарлы; артериовенозды фистула (КАФ) туа бткен журек а;ауыныц сирек mypi болып табы-лады. Дегенмен, бул коронарлы; артериялардыц туа бткен аномалиясыныц ец квп таралган тур\. Коронарлы; артерия мен журек камералары арасында болса, оны коронарлы;-камералы; фистуласы деп атайды. Сондай-афистула коронарлы; артерия мен вкпе немесе жYйелi ;ан айналымынан бас;а iргелес тамыр арасында болуы мумкш. Ашы; фистула ;анды артериядан ткелей тамырга, журек ка-мерасына немесе вкпе артериясы сия;ты бас;а твмен ;ысымды тамырга багыттау ар;ылы твмен ;арсылы; агынын ;амтамасыз етедi. КАФ бар емделушлерде фистула турне жэне коллатеральды ;ан айналымыныц болуына байланысты туылганда немесе кей^ вм'рде сипаттамалар дамуы мумкш. Зерттеулер вкпе артериясынан сол жа; коронарлы; артерияныц аномалиялы; шыгу тег (ALCAPA) сия;ты коронарлы; аномалиялардыц белгiлi б'р тYрлерi бар жас ересектер мен спортшылардагы ;а-рыншалы; аритмиялар мен кенеттен журек влiмi синдромдары арасындагы байланыстарды хабар-лады. Миокард ишемиясыныц эсернен куш тyскендегi ентгу жэне стенокардия - ец жи кездесетн белглер. Бул ма;алада коронарлы; вкпе фистуласыныц эндоваскулярлы; емм'ц клиникалы; жагдайы берлген. К^аз'рг уа;ытта уш влшемд '1 реконструкциямен КТ ангиографиясы сия;ты заманауи диагно-стикалы; эдстердЦ ар;асында патологияныц дэрежес мен сипатын багалау ;иын емес. Тактиканы багалай отырып, заманауи дэргерлер минималды инвазивтi рентгендiк эндоваскулярлы; технологи-яларды ;олдана отырып, коронарлы; артериовенозды фистулаларды улкен табыспен жеце алады.

Хат алысатын автор: Кудайберген А.Б. - Резидент-кардиохирург, «А.Н.Сызганов атындагы ¥лттыц гылыми хирургия орталыгы» АК Алматы ц., Казахстан E-mail: kudaibergen_med@mail.ru

Мудделер ¡ацтыгысы

Аеторлар MYдделер цацтыгысыныц жоцтыгын мэлiмдейдi

Туйш сездер

коронарлын фистула, цан тамырлары аномалиясы, туа б!ткен ацау

Relevance

A coronary artery fistula or coronary arteriovenous fistula is a congenital or acquired defect characterized by an abnormal connection of a coronary artery with a pulmonayr or systemic circulation, as well as with one or more chambers of the heart, bypassing the capillary blood flow. Coronary arteriovenous fistulas occur in the population in 0.002% of cases, and in 0.25% of patients undergoing coronary angiography [6-9].

CAF was first described by Krause in 1865. [10], revealed by coronary angiography. Haller and Little described the characteristic clinical triad of CAF: murmurs, left-to-right atrial or ventricular shunt, and dilated, tortuous coronary arteries. CAF can lead to severe hemodynamic disorders such as myocardial ischemia, cardiac arrhythmias, heart failure, and infective endocarditis in adults [11]. The classification according to Sakakibara (12) is often used to classify CAF, according to which the division according to the type of vessel origin is important: from the proximal or distal segment of the coronary arteries.

The proximal type of fistula discharge is characterized by normal diameters of the involved coronary arteries. In the case of the distal type, the entire coronary artery often expands and often ends in the right heart.

The main pathogenetic mechanism is related to the drainage of blood from high pressure blood vessels into a low pressure system through a fistula.

Causes of CAF can be congenital or acquired. More than 90% of CAFs are congenital [13]. During

early fetal development, the sinusoids feed the primitive myocardium, which is associated with the primitive tubular heart. Later, in adulthood, sinusoids usually transform into Thebesian vessels and capillaries. Persistent sinusoids that do not regress may contribute to a fistulous connection between coronary arteries and heart chambers [14, 15]. There is also a residual primitive connection between the coronary arteries and other mediastinal vessels (eg, bronchial, pericardial, or mediastinal arteries) or the superior vena cava, which may contribute to the development of a coronary arteriovenous fistula [16]. Acquired CAF results from iatrogenic events such as coronary stenting, coronary artery bypass grafting, trauma, and chest radiation [17, 18]. Some diseases, such as coronary vasculitis and myocardial infarction, can lead to the development of CHF in the chronic phase [19].

Description of the clinical case

The purpose of this article is to present a clinical case of endovascular treatment of coronary pulmonary fistula. Patient M., 60 years old, a resident of the Almaty region, was admitted to JSC «NSC of surgery named after Syzganov A.N.'' with a diagnosis of Congenital heart disease. Coronary pulmonary fistula. Atrial fibrillation. Cryoablation in 2021. CH II FC by NYHA.

The first time patient was admitted to our center in September 2021. with complaints related to cardiac arrhythmias, interruptions in the work of the heart, the patient was additionally examined at the center. Due to the AF Cryoablation was performed. Selective polypositional coronary angiography re-

Figure 1.

Selective coronary angiography of the LCA, direct projection. The arrows indicate tortuous aberrant vessels originating from the CxA

Figure 2.

Selective coronary angiography of the RCA, direct projection.

Arrows indicate tortuous aberrant vessels originating from the RCA RCA CxA

vealed anomalies associated with the coronary arteries. Because of the low ejection fraction by echocardiography (29%), was made a decision to implant a cardioverter defibrillator. The patient was discharged with an improvement in his general condition, and inncreasing in EF up to 36% was noted during the control EchoCG.

In January 2022 the patient is re-admitted to the Department of Interventional Cardiology, Arrhythmology and Endovascular Surgery of our Center. According to EchoCG, LV EDV is noted: 151 ml, LV ESV: 74 ml, SV 77 ml, EF 51%. interventricular septum thickness 1.2 cm. RVSP 24 mm Hg. The valves are intact. According to the ECG: Horizontal position of the electrical axis. LV hypertrophy. Myocardialischemia of the posterior wall of the left ventricle. The patient complains of shortness of breath, mild angina pectoris, and general weakness. The patient was taken to the «cath lab»: after processing the surgical access was secured through the right radial artery using the Seldinger method. Selective polypositional coronary angiography revealed: LCA trunk - stenosis up to 30%, passable. LAD - stenosis in the proximal third up to 80%. There is an aberrant vessel in the CxA, which has a tortuous course with angiomatous nodes with a transition to an aneurysmal expansion in the final segment up to

3.0 mm and a fistula into the left pulmonary artery, the diameter of the fistula is 4.5-5.0 mm. C type by Sakakibara. RCA - in the proximal third there is an aberrant vessel with a tortuous course, expanded to 5.0 mm in the final segment and a fistula with a diameter of 4.5 mm communicating with the pulmonary artery trunk. A coronary guidewire was inserted into the coronary pulmonary fistula, into it is supply vessel, on which a microcatheter «Maestro Merit» was delivered. A Nester Embolization Coil was passed through the microcatheter and delivered. During embolization, the number of coils was not enough, and therefore it was decided to use a coronary stent Ultimaster 4.5-15 mm, which was implanted in the distal third of the OA at the site of the coronary-pulmonary fistula. During control coronary angiography, the discharge of contrasted blood through the coronary-pulmonary fistula was eliminated and completely occluded.

There were no complications during the operation. The patient was transferred to the ward for dynamic observation. The patient was discharged a few days later. Subsequently, the patient is scheduled to undergo embolization of the RCA fistula and stenting of the LAD of the LCA. As a result of treatment, the patient's general condition improved, chest pains ceased to bother, shortness of breath decreased.

Figure 3.

Selective coronary angiography of the CxA, direct projection. Final view after embolization of the aberrant vessels

Figure 4.

Classification of CAF according to the type of drainage [12] A - fistula with communication of the chamber of the right ventricle; B — fistula involving the right atrial chamber; C - fistula from the coronary artery to the pulmonary artery, with the presence of one large fistulous tract; D -coronary artery - pulmonary artery, fistula with multiple small fistulas; E - coronary artery fistula communicates with the coronary sinus; F - coronary artery-venous fistula; G - coronary artery-fistula of the bronchial artery.

Discussions

Unfortunately, at the moment there are no single accepted protocols for the postoperative management of patients with this pathology. According to some authors, 10% of cases of transcatheter or surgical treatment were subject to CAF recanalization [20].

Zhang and colleagues [21] reported that CAF recanalization occurs within 1 year, suggesting the importance of assessing the residual shunt in the early postoperative period. Although a residual shedding of less than 2-3 mm can be observed without further intervention, careful monitoring during the postoperative follow-up period is necessary [21, 22].

Other possible complications after coil embolization or surgical ligation of the coronary arteries include persistent dilatation or aneurysmal changes in the ostia and coronary arteries, as well as the formation of blood clots, which can lead to myocardial ischemia and even infarction [23].

According to some authors, ECG changes and arrhythmias may also occur during and after

References

transcatheter closure [24]. In CAF with a large fistulous tract, coil migration is possible [25]. On the other hand, in patients with small branching vessels, incomplete shunt occlusion may occur if coils are placed distal to the branching point [26]. Incomplete CAF occlusion or the presence of a foreign body increases the risk of infective endocarditis [17]. Conclusion

CAF is one of the rare pathologies with a high variability of clinical manifestations, which include such formidable complications as myocardial infarction and heart failure. As a result of a modern diagnostic methods, such as CT angiography with three-dimensional reconstruction, it is not difficult to assess the degree and nature of the pathology. Having assessed the tactics, modern doctors are able to cope with coronary arteriovenous fistulas with great success using minimally invasive X-ray endovascular technologies.

1. Fernandes ED, Kadivar H, Hallman GL, Reul GJ, Ott DA, Cooley DA. Congenital malformations of the coronary arteries: the Texas Heart Institute experience. Ann Thorac Surg. 1992 0ct;54(4):732-40.

2. Maron BJ, Shirani J, Poliac LC, Mathenge R, Roberts WC, Mueller FO. Sudden death in young competitive athletes. Clinical, demographic, and pathological profiles. JAMA. 1996 Jul 17;276(3):199-204. [PubMed]

3. Basso C, Maron BJ, Corrado D, Thiene G. Clinical profile of congenital coronary artery anomalies with origin from the wrong aortic sinus leading to sudden death in young competitive athletes. J Am Coll Cardiol. 2000 May;35(6):1493-501. [PubMed]

4. Cox ID, Bunce N, Fluck DS. Failed sudden

cardiac death in a patient with an anomalous origin of the right coronary artery. Circulation. 2000 Sep 19;102(12):1461-2. [PubMed]

5. Van Camp SP, Bloor CM, Mueller FO, Cantu RC, Olson HG. Nontraumatic sports death in high school and college athletes. Med Sci Sports Exerc. 1995 May;27(5):641-7. [PubMed]

6. McNamara JJ, Gross RE. Congenital coronary artery fistula. Surgery 1969;65(1):59-69.

7. Vavuranakis M, Bush CA, Boudoulas H. Coronary artery fistulas in adults: incidence, angiographic characteristics, natural history. Cathet Cardiovasc Diagn 1995;35(2):116-120.

8. Yamanaka O, Hobbs RE. Coronary artery anomalies in 126,595 patients undergoing

coronary arteriography. CathetCardiovasc Diagn 1990;21(1):28-40.

9. Kim MS, Jung JI, Chun HJ. Coronary to pulmonary artery fistula: morphologic features at multidetector CT. Int J Cardiovasc Imaging 2010;26(suppl 2):273-280.

10. Lim JJ, Jung JI, Lee BY, Lee HG. Prevalence and types of coronary artery fistulas detected with coronary CT angiography. AJR Am J Roentgenol 2014;203(3):W237-W243.

11. Krause W. Uber den ursprung einer accessorischen a. coronaria aus der a. pulmonalis. Z Ratl Med 1865;24:225-227.

12. Vitarelli A, De Curtis G, Conde Y, et al. Assessment of congenital coronary artery fistulas by transesophageal color Doppler echocardiography. Am J Med 2002;113(2):127-133.

13. Sakakibara S, Yokoyama M, Takao A, Nogi M, Gomi H. Coronary arteriovenous fistula: nine operated cases. Am Heart J 1966;72(3):307-314.

14. Challoumas D, Pericleous A, Dimitrakaki IA, Danelatos C, Dimitrakakis G. Coronary arteriovenous fistulae: a review. Int J Angiol 2014;23(1):1-10.

15. Ogden JA. Congenital anomalies of the coronary arteries. Am J Cardiol 1970;25(4):474-479.

16. Rittenhouse EA, Doty DB, Ehrenhaft JL. Congenital coronary artery-cardiac chamber fistula: review of operative management. Ann Thorac Surg 1975;20(4):468-485.

17. Baroldi G, Scomazzoni G. Coronary circulation in the normal and the pathologic heart. Washington, DC: Department of the

Army, United States Government Printing Office, 1967; 5-90.

18. Mangukia CV. Coronary artery fistula. Ann Thorac Surg 2012;93(6):2084-2092.

19. Luo L, Kebede S, Wu S, Stouffer GA. Coronary artery fistulae. Am J Med Sci 2006;332(2):79-84.

20. Yu R, Sharma B, Franciosa JA. Acquired coronary artery fistula to the left ventricle after acute myocardial infarction. Am J Cardiol 1986;58(6):557-558.

21. Armsby LR, Keane JF, Sherwood MC, Forbess JM, Perry SB, Lock JE. Management of coronary artery fistulae: patient selection and results of transcatheter closure. J Am Coll Cardiol 2002;39(6):1026-1032.

22. Zhang W, Hu R, Zhang L, Zhu H, Zhang H. Outcomes of surgical repair of pediatric coronary artery fistulas. J Thorac Cardiovasc Surg 2016;152(4):1123-1130.e1.

23. Hou B, Ma WG, Zhang J, et al. Surgical management of left circumflex coronary artery fistula: a 25-year single-center experience in 29 patients. AnnThorac Surg 2014;97(2):530-536.

24. Gowda RM, Vasavada BC, Khan IA. Coronary artery fistulas: clinical and therapeutic considerations. Int J Cardiol 2006;107(1):7-10.

25. Reddy G, Davies JE, Holmes DR, Schaff HV, Singh SP, Alli OO. Coronary artery fistulae. Circ Cardiovasc Interv 2015;8(11):e003062.

26. Liang CD, Ko SF. Midterm outcome of percutaneous transcatheter coil occlusion of coronary artery fistula. Pediatr Cardiol 2006;27(5):557-563.