An asymptomatic 16-year-old girl with anomalous left coronary artery from pulmonary artery (Bland-Withe-Garland syndrome)

Abstract

Pacjentka 16-letnia z bezobjawowym nieprawidłowym odejściem lewej tętnicy wieńcowej od pnia płucnego (zespół Blanda-White'a-Garlanda) G Gw wi id do on n P Po ol la ak k 1 1, ,2 2 , , T To om ma as sz z B Bi ia ał ło os sz zy yń ńs sk ki i 1 1 , , D Do or ro ot ta a M Mo ot ty yl l 2 2 , , A An nd dr rz ze ej j H Ho of ff fm ma an nn n A b s t r a c tA 16-year-old girl was referred to our hospital to perform cardiac multi-slice computed tomography (MSCT) due to an unclear view of coronary arteries in echocardiography performed during a routine visit to the cardiology clinic. As an infant she was hospitalized due to signs of severe heart failure, and while in hospital she underwent cardiac arrest. However, ever since this incident she has not experienced any cardiovascular symptoms, and still remains asymptomatic. On physical examination we found systolic murmur localized to the left sternal border. In echocardiography the dimensions of cardiac chambers and ventricular contractility were normal. Cardiac MSCT demonstrated markedly dilated tortuous right coronary artery (RCA), giving very well-developed collaterals crossing the interventricular septum toward the left coronary artery (LCA). The LCA was also dilated but not as much as the RCA. The images showed an anomalous origin of the LCA from the pulmonary artery trunk (ALCAPA) and suggested retrograde filling of the LCA through collaterals from the RCA and emptying into the pulmonary artery trunk (PA). Coronary angiography and aortography confirmed the diagnosis of ALCAPA (Bland-White-Garland syndrome). It showed a very big tortuous RCA, the LCA filling retrogradely through collaterals from the RCA and draining into the PA. Anomalous left coronary artery from pulmonary artery is a congenital anomaly associated with early infant mortality, very rarely diagnosed in asymptomatic adults, with uncertain but probably poor prognosis. MSCT is useful for diagnosis. The patient still remains asymptomatic and so far she and her parents have not agreed to surgical treatment. K Ke ey y w wo or rd ds s: : anomalous left coronary artery from pulmonary artery, Bland-White-Garland syndrome, cardiac multi-slice computed tomography S t r e s z c z e n i e Dziewczynka 16-letnia została skierowana do szpitala w celu wykonania wielorzędowej tomografii komputerowej (multi-slice computed tomography -MSCT) serca z powodu niejasnego obrazu tętnic wieńcowych w echokardiografii wykonanej podczas wizyty kontrolnej w poradni kardiologicznej. Jako niemowlę była ona hospitalizowana z powodu objawów ostrej niewydolności serca, przebyła wtedy zatrzymanie krążenia. Później rozwijała się dobrze, nie zgłasza dotąd żadnych dolegliwości ze strony układu krążenia. W badaniu przedmiotowym stwierdzono szmer skurczowy wzdłuż lewego brzegu mostka. W badaniu echokardiograficznym wymiary jam serca i kurczliwość mięśnia lewej komory były prawidłowe. W badaniu metodą MSCT uwidoczniono znacznie poszerzoną, krętą prawą tętnicę wieńcową (right coronary artery -RCA), dającą bardzo dobrze rozwinięte krążenie obocz...

Bland White Garland syndrome with aneurysm and collaterals of coronary arteries in an Afghan girl: a case report.

“…Therefore, BWG is the most appropriate diagnosis, and CAA may be associated with high blood flow between the RCA and LCA, as well as steal phenomenon. The existence of a left coronary artery aneurysm, an asymptomatic course, and normal heart function in an adolescent distinguish our case of BWGS from the previous published reports [ 1 , 4 , 6 , 8 , 9 , 11 , 13 ]. In this case, the patient was advised to undergo surgical management of BWGS in an advanced setting abroad.…”

“…Polak et al. reported a case of anomalous origin of the left coronary artery from the pulmonary artery in a 16-year-old girl with a history of heart failure in infancy [ 13 ]. An anomalous origin of the left coronary artery from the pulmonary artery was evaluated by Yazdi et al.…”