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The Radiologist's Tragedy, Or Bland-White

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The Radiologist's Tragedy, Or Bland-White HISTORY OF CARDIAC AND THORACIC SURGERY DOI: 10.5114/kitp.2014.43857 The radiologist’s tragedy, or Bland-White-Garland syndrome (BWGS). On the 80th anniversary of the first clinical description of ALCAPA (anomalous left coronary artery from the pulmonary artery) Magdalena Mazurak, Jacek Kusa Pediatric Cardiology Department, Regional Specialist Hospital, Wroclaw, Poland Kardiochirurgia i Torakochirurgia Polska 2014; 11 (2): 225-229 Abstract Streszczenie In 1933, three doctors from the Massachusetts General Hospi- W 1933 r. trzej lekarze ze szpitala w Bostonie: William Franklin tal in Boston, Paul Dudley White, William Franklin Bland, and Bland, Paul Dudley White i Joseph Garland, opisali przypadek Joseph Garland, described a case of an anomalous origin of the nieprawidłowego odejścia lewej tętnicy wieńcowej od tętnicy left coronary artery arising from the pulmonary artery (ALCAPA) płucnej (ALCAPA) u 3-miesięcznego chłopca. Dziecko zmarło in a three-month-old boy. The infant died following two weeks po 2 tygodniach hospitalizacji. Ojcem tego niemowlęcia był of hospitalization. The child’s father was Dr. Aubrey Hamp- dr Aubrey Hampton, radiolog, kolega z pracy White’a, Blanda ton, a radiologist and colleague of White, Bland, and Garland. i Garlanda. Niniejsze opracowanie jest spojrzeniem z perspek- The paper presents a perspective view on the occasion of the tywy 80. rocznicy pierwszego klinicznego opisu ALCAPA. 80th anniversary of the first clinical description of ALCAPA. Słowa kluczowe: zespół Blanda-White’a-Garlanda, ALCAPA, Key words: Bland-White-Garland syndrome, ALCAPA, history historia medycyny. of medicine. abnormality, and the curious effect which it has in produ- Introduction cing a cirsoid dilatation of the vessels in connection with In 1933, three doctors from the Massachusetts Gene- the anomalous arteries, have induced me to put on record ral Hospital (MGH) in Boston, William Franklin Bland, Paul the two cases in question. In the first case that was discove- Dudley White, and Joseph Garland, described a case of an red, the anomalous coronary artery was a vessel which (…) anomalous origin of the left coronary artery arising from sprang from the right anterior sinus of Valsalva of the pul- the pulmonary artery (ALCAPA) in a three-month-old boy. monary artery (…) [3]. Brooks’ conclusions included a very The infant died following two weeks of hospitalization. important observation. He stated that the flow of blood in The authors stressed the credibility of information obta- the right coronary artery is reverse, i.e. that arterial blood ined from the infant’s parents, i.e. the alarming symptoms flows from the normal left coronary artery arising from the they observed at home before leaving for hospital. The in- aorta through anastomoses to the right coronary artery le- fant’s father, Dr. Aubrey Hampton, a radiologist, was White, ading into the pulmonary artery. What is interesting, most Bland, and Garland’s colleague at MGH [1, 2]. contemporary authors referring to Brooks’ article misquote Anomalous origin of the coronary artery, a rare conge- it as a description of a case of anomalous left, rather than nital cardiac defect, has been known since 1866, when John right, coronary artery… Brooks described anomalous origin of right coronary artery The first report on anomalous left, rather than right, co- from the pulmonary artery (ARCAPA) in his post-mortem ronary artery from the pulmonary artery appeared in 1911. examination report for two patients. Brooks wrote: During In his extensive article illustrated with photos of autopsy the course of the Winter Session, 1884-5, two very remar- preparations and microscope sections, the Russian patho- kable instances of a coronary artery of the heart, springing logist Alexei Ivanovitch Abrikosov described left ventricu- from the pulmonary artery, occurred in the Practical Ana- lar aneurysm observed in the post-mortem examination of tomy Rooms of this University. The apparent rarity of this a 5-month-old girl, as well as the origin of the left coronary Address for correspondence: Magdalena Mazurak, MD, Oddział Kardiologii Dziecięcej, Wojewódzki Szpital Specjalistyczny, Kamieńskiego 73a, 51-124 Wrocław, tel. +48 71 327 01 40, e-mail: [email protected] Kardiochirurgia i Torakochirurgia Polska 2014; 11 (2) 225 The radiologist’s tragedy, or Bland-White-Garland syndrome (BWGS). On the 80th anniversary... artery from the pulmonary artery. The girl, named Marga- life the resistance in the pulmonary bed decreases, which rethe, was admitted to the hospital in a serious condition lowers the inflow of blood into the left ventricular artery in January 1901, with “an unknown history”. She was dia- and results in the myocardial perfusion disorder in the area gnosed with left-sided pneumonia and enlargement of the supplied by this artery. If the inflow of blood from the col- heart silhouette, suggesting exudative pericarditis. During laterals originating from the right coronary artery is insuffi- the night, the child passed away. Six hours later, Abrikosov cient, the patient shows the symptoms described by Bland, conducted a post-mortem examination of the infant’s body. White, and Garland: pallor of skin integuments, feeding dif- Some facts of Abrikosov’s later life are very interesting: in ficulties, and anxiety resembling baby colic. Chronic ische- January 1924, he spent three days embalming Lenin’s corp- mia results in endocardial fibroelastosis (EFE) and papillary se, the effects of which can still be viewed in the famo- muscle damage, which leads to mitral valve incompetence us mausoleum on Moscow’s Red Square. Abrikosov’s wife and progressing left ventricular dilation. In an acute form, was also an anatomical pathologist, while their son, Aleksei it is manifested as myocardial infarction. If collateral circu- Alekseyevitch, received a Nobel Prize in physics in 2003. lation is sufficient, the symptoms may be absent or weak, Abrikosov’s work met with almost no response, and which makes it possible to reach the adult age without any it was not until the article by Bland, White, and Garland medical intervention. Adults may also show no symptoms, was published in 1933, containing a description of the ECG, although they more often suffer from collapses or syncopes, chest X-ray, and a clinical picture, that serious interest was chest pains, arrhythmia, or sudden death. The predominant generated in this issue. The number of case descriptions symptoms observed in the echocardiographic picture inc- grew in subsequent years. Bland, White, and Garland wrote lude left ventricular dilation with myocardial contractility that on the day the infant was admitted to the hospital, disorders and mitral valve incompetence [2]. he was pale, groaning, and – according to the parents – he was sweating while eating. The physical examination of Diagnostics the lungs indicated dullness of the percussion sound. No Aubrey Hampton’s son diagnosis was not a survival dia- cyanosis was observed and there was no cardiac murmur. gnosis, but a post-mortem one. Eighty years after that case, The chest X-ray image showed enlargement of the heart we have a number of diagnostic (both invasive and non- silhouette, while the ECG registered reverse T waves with invasive) examinations, which make it possible to establish a low voltage of the QRS complex in leads I, II, and III. a correct diagnosis. In the majority of cases, an echocardio- th The infant’s condition was stable at first. On the 14 day after graphic examination proves sufficient to recognize the de­ admission to the hospital, the baby developed respiratory fect. It shows an expanded right coronary artery and a lack disorders and cyanosis, and soon the patient died. The ear- of origin of the left coronary artery from the aorta sinus and lier diagnosis of idiopathic cardiac hypertrophy. The heart its origin from the pulmonary artery. The use of the Dop- was significantly enlarged, with a large right ventricle and pler method marked with color enables visualization of the relatively small left ventricle. Atrioventricular and aortic reverse flow of blood from the left coronary artery to the valves had a proper structure and size. The examination pulmonary artery (a permanent inflow). Echocardiography of coronary arteries revealed a surprise: the right artery also makes it possible to evaluate the degree of dilatation was normal, while the left one originated from the pulmo- and dysfunction of the left ventricle. Mitral valve incompe- nary artery, providing vascularization of the left ventricle. tence is, besides left ventricular dilation, another important The microscopic examination indicated fibrosis, particularly symptom of ALCAPA. The underlying cause of that incom- of the left ventricle, with no sign of infarction. The authors petence is the dysfunction of papillary muscles and left ven- of the quoted article, Bland, White, and Garland, analyzed tricle dyskinesia accompanied by left ventricular dilation the existing literature on anomalous coronary arteries and and the expansion of the mitral valve ring. Ischemia can be concluded that such anomalies are associated with poor manifested through hyperechogenicity of papillary muscles prognosis [1, 2]. and left ventricular endocardial fibroelastosis (EFE). Until recently, patients with a dubious diagnosis of ALCAPA were Preface subjected to cardiac catheterization and angiocardiography. Anomalous left coronary artery from the pulmonary ar- This allows one to obtain an image of coexisting cardiac tery is a rare cardiac
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