Bland-White-Garland syndrome or ALCAPA is an abnormality of birth of the left coronary artery from the pulmonary artery. It is a diagnostic and therapeutic emergency because it is a curable cause of hypokinetic dilated heart disease in infants. We report through this clinical case, the fourth case of infantile ALCAPA diagnosed in Senegal in a 7-month-old infant. The symptomatology began around the age of 2 months, with a grumpy state associated with more marked crying and moaning during feedings and bowel movements. The mother reported hospitalization for a severe lung infection when she was 6 months old. The examination noted an infant in poor general condition, retarded growth and weight, and a 3/6th holosystolic murmur at the apex. Troponinemia was positive at 43.90 ng/L. The electrocardiogram showed Q waves on the lower side, a sub endocardial lesion on the upper side and a ST segment elevation in aVR. Doppler echocardiography showed dilated cardiomyopathy with a mean alteration of systolic function of the left ventricle at 37%, a mean mitral insufficiency and a strong suspicion of a birth anomaly of the left coronary artery. The CT scan confirmed the diagnosis of ALCAPA. Surgicalreimplantation of the left coronary artery at the aortic level was performed at 10 months of life with a favourable outcome at D50 postoperative.
References
[1]
Leye, M. (2017) The Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery (ALCAPA) in Senegal. International Journal of Cardiovascular and Thoracic Surgery, 3, 9-13. https://doi.org/10.11648/j.ijcts.20170302.12
[2]
Heermann, P., Heindel, W. and Schülke, C. (2016) Coronary Artery Anomalies: Diagnosis and Classification based on Cardiac CT and MRI (CMR)—From ALCAPA to Anomalies of Termination. RoFo Fortschritte auf dem Gebiet der Rontgenstrahlen und der bildgebenden Verfahren, 189, 29-38.
https://doi.org/10.1055/s-0042-119452
[3]
Zhang, J.M., Ling, Y.F., Wang, Y. and Qian, Y.J. (2020) Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery (ALCAPA) Diagnosed in Children and Adolescents. Journal of Cardiothoracic Surgery, 15, Article No. 90.
https://doi.org/10.1186/s13019-020-01116-z
[4]
Dahle, G., Fiane, A.E. and Lindberg, H.L. (2007) ALCAPA, a Possible Reason for Mitral Insufficiency and Heart Failure in Young Patients. Scandinavian Cardiovascular Journal, 41, 51-58. https://doi.org/10.1080/14017430601050348
[5]
Pena, E., Nguyen, E.T., Merchant, N. and Dennie, C. (2009) ALCAPA Syndrome: Not Just a Pediatric Disease. RadioGraphics, 29, 553-565.
https://doi.org/10.1148/rg.292085059
[6]
Smail, H., Bouchart, F., Bessou, J.P. and Doguet, F. (2012) Cardiopathies congénitales de l’adulte Diversité anatomoclinique et particularités thérapeutiques. Chirurgie Thoracique Cardio-Vasculaire, 16, 147-156.
[7]
Pfannschmidt, J., Ruskowski, H. and Vivie, E.R. (1992) Das Bland-White-Garland- Syndrom (BWGS). Klinische Padiatrie, 204, 328-334.
https://doi.org/10.1055/s-2007-1025367
[8]
Dodge-Khatami, A., Mavroudis, C. and Backer, C.L. (2002) Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery: Collective Review of Surgical Therapy. The Annals of Thoracic Surgery, 74, 946-955.
https://doi.org/10.1016/S0003-4975(02)03633-0
[9]
Wesselhoeft, H. and Johnson, A.L. (1968) Anomalous Origin of the Left Coronary Artery from the Pulmonary Trunk. Circulation, 38, 403-425.
https://doi.org/10.1161/01.CIR.38.2.403
[10]
Krause, W. (1865) Ueber den Ursprung einer accessorischen a. coronaria cordis aus der a. pulmonalis. Rational Medicine, 24, 225.
[11]
Yau, J.M., Singh, R., Halpern, E.J. and Fischman, D. (2011) Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery in Adults: A Comprehensive Review of 151 Adult Cases and a New Diagnosis in a 53-Year-Old Woman. Clinical Cardiology, 34, 204-210. https://doi.org/10.1002/clc.20848
[12]
Bland, E.F., White, P.D. and Garland, J. (1933) Congenital Anomalies of the Coronary Arteries: Report of an Unusual Case Associated with Cardiac Hypertrophy. American Heart Journal, 8, 787-801.
https://doi.org/10.1016/S0002-8703(33)90140-4
[13]
Lotman, E.M., Karu, K., Mikkel, M. and Elmet, M. (2020) Late Adult Presentation of ALCAPA Syndrome: Need for a New Clinical Classification? A Case Report and Literature Overview. European Heart Journal—Case Reports, 4, 1-5.
https://doi.org/10.1093/ehjcr/ytaa318
[14]
Kayalar, N., Burkhart, H.M., Dearani, J.A., Cetta, F. and Schaff, H.V. (2009) Congenital Coronary Anomalies and Surgical Treatment. Congenital Heart Disease, 4, 239-251.
https://doi.org/10.1111/j.1747-0803.2009.00301.x
[15]
Pm, A. (2019) Naissance de la coronaire gauche à partir de l’artère pulmonaire révélée par une insuffisance cardiaque chez un nourrisson, à propos d’un cas au CNHU-HKM, Bénin, 8.
[16]
Moodie, D.S., Fyfe, D., Gill, C.C., Cook, S.A., Lytle, B.W., Taylor, P.C., et al. (1983) Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery (Bland-White-Garland Syndrome) in Adult Patients: Long-term Follow-Up after Surgery. American Heart Journal, 106, 381-388.
https://doi.org/10.1016/0002-8703(83)90207-7
[17]
Patrianakos, A.P., Hatzidakis, A., Marketou, M. and Parthenakis, F.I. (2018) Adult-Type ALCAPA Syndrome: A Rare Coronary Artery Anomaly. Echocardiography, 35, 1056-1059. https://doi.org/10.1111/echo.14013
[18]
Vizzuso, A., Righi, R., Zerbini, M., Gamanji, S., Cucchi, P., Gallo, F., et al. (2018) An Unusual Presentation of Anomalous Left Coronary Artery from the Pulmonary Artery (ALCAPA) Syndrome in a 70-Year-Old Man: A Case Report. Journal of Medical Case Reports, 12, Article No. 308. https://doi.org/10.1186/s13256-018-1851-4
[19]
Yew, K.L., Kang, Z. and Anum, A. (2016) Late Presentation of ALCAPA Syndrome in an Elderly Asian Lady. Medical Journal of Malaysia, 71, 217-219.
[20]
Holst, L.M.B., Helvind, M. and Orb, H. (2015) Diagnosis and Prognosis of Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery. Danish Medical Journal, 62, A5125.
[21]
Ramírez, S., Curi-Curi, P.J., Calderón-Colmenero, J., García, J., Britton, C., Erdmenger, J., et al. (2011) Outcomes of Coronary Reimplantation for Correction of Anomalous Origin of Left Coronary Artery from Pulmonary Artery. Revista Espanola de Cardiología (English Edition), 64, 681-687.
https://doi.org/10.1016/j.rec.2011.04.014
[22]
Edwards, J.E. (1964) Editorial: The Direction of Blood Flow in Coronary Arteries Arising from the Pulmonary Trunk. Circulation, 29, 163-166.
https://doi.org/10.1161/01.CIR.29.2.163
[23]
Backer, C.L., Stout, M.J., Zales, V.R., Muster, A.J., Weigel, T.J., Idriss, F.S., et al. (1992) Anomalous Origin of the Left Coronary Artery. The Journal of Thoracic and Cardiovascular Surgery, 103, 1049-1058. https://doi.org/10.1016/S0022-5223(19)34868-8
[24]
Agustsson, M.H., Gasul, B.M., Fell, E.H., Graettinger, J.S., Bicoff, J.P. and Waterman, D.F. (1962) Anomalous Origin of Left Coronary Artery from Pulmonary Artery: Diagnosis and Treatment of Infantile and Adult Types. JAMA, 180, 15-21.
https://doi.org/10.1001/jama.1962.03050140017004
[25]
Arciniegas, E., Farooki, Z.Q., Hakimi, M. and Green, E.W. (1980) Management of Anomalous Left Coronary Artery from the Pulmonary Artery. Circulation, 62, I180-I189.
[26]
Berdjis, F., Takahashi, M., Wells, W.J., Stiles, Q.R. and Lindesmith, G.G. (1994) Anomalous Left Coronary Artery from the Pulmonary Artery: Significance of Inter Coronary Collaterals. The Journal of Thoracic and Cardiovascular Surgery, 108, 17-20. https://doi.org/10.1016/S0022-5223(94)70212-8
[27]
Frapier, J.M., Leclercq, F., Bodino, M. and Chaptal, P.A. (1999) Malignant Ventricular Arrhythmias Revealing Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery in Two Adults. European Journal of Cardio-Thoracic Surgery, 15, 539-541. https://doi.org/10.1016/S1010-7940(99)00024-X
[28]
Nakle, N., Biscardi, S., Lambert, V., Sigal-Cinqualbre, A., Epaud, R. and Madhi, F. (2012) Anomalie de naissance de la coronaire gauche à partir de l’artère pulmonaire révélée par une bronchiolite. Revue des Maladies Respiratoires, 29, 912-915.
https://doi.org/10.1016/j.rmr.2012.03.006
[29]
Bilderling, G., Geurten, C., Evrard, J., Kanen, F., Houtekie, L., Poncelet, A., et al. (2017) Quand l’insuffisance cardiaque se cache derrière la bronchiolite. Louvain Médical, 136, 245-249.
[30]
Gribaa, R., Slim, M., Ben Salem, H., Neffati, E. and Boughzela, E. (2014) Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery Presenting as Dilated Cardiomyopathy: A Case Report. Journal of Medical Case Reports, 8, Article No. 170. https://doi.org/10.1186/1752-1947-8-170
[31]
Trabelsi Sahnoun, I., Abid, L., Abid, D., Triki, F., Sahnoun, M., Krichene, S., et al. (2008) La naissance anormale du tronc coronaire gauche à partir de l’artère pulmonaire: Etiologie de défaillance cardiaque chez le nourrisson. JIM Sfax, 15-16, 23-25.
[32]
Shivalkar, B., Borgers, M., Daenen, W., Gewillig, M. and Flameng, W. (1994) ALCAPA Syndrome: An Example of Chronic Myocardial Hypoperfusion? JACC: Journal of the American College of Cardiology, 23, 772-778.
https://doi.org/10.1016/0735-1097(94)90767-6
[33]
Kerut, E.K., Kogos, P.G. anderson, J.H., Turner, M., Ascuitto, R., Ross-Ascuitto, N., et al. (2018) Adult Presentation of ALCAPA: Echo and CT Diagnosis. Echocardiography (Mount Kisco, N.Y.), 35, 1045-1048. https://doi.org/10.1111/echo.13807
[34]
Vesterlund, T., Thomsen, P.E. and Hansen, O.K. (1985) Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery in an Adult. Heart, 54, 110-112.
https://doi.org/10.1136/hrt.54.1.110
[35]
Prandi, F.R., Zaidi, A.N., LaRocca, G., Hadley, M., Riasat, M., Anastasius, M.O., et al. (2022) Sudden Cardiac Arrest in an Adult with Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery (ALCAPA): Case Report. International Journal of Environmental Research and Public Health, 19, Article ID: 1554.
https://doi.org/10.3390/ijerph19031554
[36]
Alexi-Meskishvili, V., Berger, F., Weng, Y., Lange, P.E. and Hetzer, R. (1995) Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery in Adults. Journal of Cardiac Surgery, 10, 309-315.
https://doi.org/10.1111/j.1540-8191.1995.tb00617.x
[37]
Fernandes, E.D., Kadivar, H., Hallman, G.L., Reul, G.J., Ott, D.A. and Cooley, D.A. (1992) Congenital Malformations of the Coronary Arteries: The Texas Heart Institute Experience. The Annals of Thoracic Surgery, 54, 732-740.
https://doi.org/10.1016/0003-4975(92)91019-6
[38]
Silverman, N.H. (2015) Echocardiographic Presentation of Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery. Cardiology in the Young, 25, 1512-1523. https://doi.org/10.1017/S1047951115002565
[39]
Ghaderi, F., Gholoobi, A. and Moeinipour, A. (2014) Unique Echocardiographic Markers of Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery (ALCAPA) in the Adult. Echocardiography (Mount Kisco, N.Y.), 31, E13-E15.
https://doi.org/10.1111/echo.12378
[40]
Tan, X., Sun, K., Li, F., Zhang, Y. and Wu, L. (2008) Clinical Diagnosis of Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery in 22 Cases. Chinese Journal of Pediatrics, 46, 881-884.
[41]
Chang, R.R. and Allada, V. (2001) Electrocardiographic and Echocardiographic Features That Distinguish Anomalous Origin of the Left Coronary Artery from Pulmonary Artery from Idiopathic Dilated Cardiomyopathy. Pediatric Cardiology, 22, 3-10. https://doi.org/10.1007/s002460010142
[42]
Toussaint, M., Goube, P., Kuoch, V., Defrance, C., Amrar-Vennier, F., Nicollet, E., et al. (2011) Origine anormale de la coronaire gauche à partir de l’artère pulmonaire: Place du scanner 64 coupes. Journal of Radiology, 92, 1124-1127.
https://doi.org/10.1016/j.jradio.2011.05.015
[43]
Mesurolle, B., Qanadli, S.D., Mignon, F. and Lacombe, P. (2006) Anomalous Origin of the Left Coronary Artery Arising From the Pulmonary Trunk. The American Journal of Roentgenology, 186, 1202-1202. https://doi.org/10.2214/AJR.06.5029
[44]
Levin, S.E., Dansky, R. and Kinsley, R.H. (1990) Origin of Left Coronary Artery from Right Pulmonary Artery Co-Existing with Coarctation of the Aorta. International Journal of Cardiology, 27, 31-36.
https://doi.org/10.1016/0167-5273(90)90188-B
[45]
Wesselhoeft, H., Fawcett, J.S. and Johnson, A.L. (1968) Anomalous Origin of the Left Coronary Artery from the Pulmonary Trunk. Its Clinical Spectrum, Pathology, and Pathophysiology, Based on a Review of 140 Cases with Seven Further Cases. Circulation, 38, 403-425. https://doi.org/10.1161/01.CIR.38.2.403
[46]
Michielon, G., Di Carlo, D., Brancaccio, G., Guccione, P., Mazzera, E., Toscano, A., et al. (2003) Anomalous Coronary Artery Origin from the Pulmonary Artery: Correlation between Surgical Timing and Left Ventricular Function Recovery. The Annals of Thoracic Surgery, 76, 581-588.
https://doi.org/10.1016/S0003-4975(03)00344-8
[47]
Backer, C.L., Stout, M.J., Zales, V.R., Muster, A.J., Weigel, T.J., Idriss, F.S., et al. (1992) Anomalous Origin of the Left Coronary Artery. A Twenty-Year Review of Surgical Management. The Journal of Thoracic and Cardiovascular Surgery, 103, 1049-1057.
https://doi.org/10.1016/S0022-5223(19)34868-8
[48]
Laks, H., Ardehali, A., Grant, P.W. and Allada, V. (1995) Aortic Implantation of Anomalous Left Coronary Artery. An Improved Surgical Approach. The Journal of Thoracic and Cardiovascular Surgery, 109, 519-523.
https://doi.org/10.1016/S0022-5223(95)70283-0
[49]
Del Nido, P.J., Duncan, B.W., Mayer, J.E., Wessel, D.L., LaPierre, R.A. and Jonas, R.A. (1999) Left Ventricular Assist Device Improves Survival in Children with Left Ventricular Dysfunction after Repair of Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery. The Annals of Thoracic Surgery, 67, 169-172.
https://doi.org/10.1016/S0003-4975(98)01309-5
[50]
Jin, Z., Berger, F., Uhlemann, F., Schroder, C., Hetzer, R., Alexi-Meskhishvili, V., et al. (1994) Improvement in Left Ventricular Dysfunction after Aortic Reimplantation in 11 Consecutive Paediatric Patients with Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery. Early Results of a Serial Echocardiographic Follow-Up. European Heart Journal, 15, 1044-1049.
https://doi.org/10.1093/oxfordjournals.eurheartj.a060626
[51]
Alexi-Meskishvili, V., Hetzer, R., Weng, Y., Lange, P.E., Jin, Z., Berger, F., et al. (1994) Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery. Early Results with Direct Aortic Reimplantation. The Journal of Thoracic and Cardiovascular Surgery, 108, 354-362. https://doi.org/10.1016/S0022-5223(94)70018-4
[52]
Vouhé, P.R., Baillot-Vernant, F., Trinquet, F., Sidi, D., Geeter, B., Khoury, W., et al. (1987) Anomalous Left Coronary Artery from the Pulmonary Artery in Infants: Which Operation? When? The Journal of Thoracic and Cardiovascular Surgery, 94, 192-199.
https://doi.org/10.1016/S0022-5223(19)36281-6
[53]
Sauer, U., Stern, H., Meisner, H., Bühlmeyer, K. and Sebening, F. (1992) Risk Factors for Perioperative Mortality in Children with Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery. The Journal of Thoracic and Cardiovascular Surgery, 104, 696-705. https://doi.org/10.1016/S0022-5223(19)34739-7
[54]
Chan, R.K., Hare, D.L. and Buxton, B.F. (1995) Anomalous Left Main Coronary Artery Arising from the Pulmonary Artery in an Adult: Treatment by Internal Mammary Artery Grafting. The Journal of Thoracic and Cardiovascular Surgery, 109, 393-394. https://doi.org/10.1016/S0022-5223(95)70404-3
[55]
Wollenek, G., Domanig, E., Salzer-Muhar, U., Havel, M., Wimmer, M. and Wolner, E. (1993) Anomalous Origin of the Left Coronary Artery: A Review of Surgical Management in 13 Patients. The Journal of Cardiovascular Surgery (Torino), 34, 399-405.
[56]
Dc, S., Ca, N. and Hb, T. (1960) The Direction of Blood Flow in Anomalous Left Coronary Artery Arising from the Pulmonary Artery. Circulation, 22, 591-597.
https://doi.org/10.1161/01.CIR.22.4.591
[57]
Meyer, B.W., Stefanik, G., Stiles, Q.R., Lindesmith, G.G. and Jones, J.C. (1968) A Method of Definitive Surgical Treatment of Anomalous Origin of Left Coronary Artery. A Case Report. The Journal of Thoracic and Cardiovascular Surgery, 56, 104-107. https://doi.org/10.1016/S0022-5223(19)42879-1
[58]
Neches, W.H., Mathews, R.A., Park, S.C., Lenox, C.C., Zuberbuhler, J.R., Siewers, R.D., et al. (1974) Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery. A New Method of Surgical Repair. Circulation, 50, 582-587.
https://doi.org/10.1161/01.CIR.50.3.582
[59]
Takeuchi, S., Imamura, H., Katsumoto, K., Hayashi, I., Katohgi, T., Yozu, R., et al. (1979) New Surgical Method for Repair of Anomalous Left Coronary Artery from Pulmonary Artery. The Journal of Thoracic and Cardiovascular Surgery, 78, 7-11.
https://doi.org/10.1016/S0022-5223(19)38154-1
[60]
Lambert, V., Touchot, A., Losay, J., Piot, J.D., Henglein, D., Serraf, A., et al. (1996) Midterm Results after Surgical Repair of the Anomalous Origin of the Coronary Artery. Circulation, 94, II38-II43.
[61]
Bunton, R., Jonas, R.A., Lang, P., Rein, A.J. and Castaneda, A.R. (1987) Anomalous Origin Of Left Coronary Artery from Pulmonary Artery. Ligation versus Establishment of a Two Coronary Artery System. The Journal of Thoracic and Cardiovascular Surgery, 93, 103-108. https://doi.org/10.1016/S0022-5223(19)36481-5
[62]
Huddleston, C.B., Balzer, D.T. and Mendeloff, E.N. (2001) Repair of Anomalous Left Main Coronary Artery Arising from the Pulmonary Artery in Infants: Long-Term Impact on the Mitral Valve. The Annals of Thoracic Surgery, 71, 1985-1988.
https://doi.org/10.1016/S0003-4975(01)02518-8
[63]
Cabrera, A.G., Chen, D.W., Pignatelli, R.H., Khan, M.S., Jeewa, A., Mery, C.M., et al. (2015) Outcomes of Anomalous Left Coronary Artery from Pulmonary Artery Repair: Beyond Normal Function. The Annals of Thoracic Surgery, 99, 1342-1347.
https://doi.org/10.1016/j.athoracsur.2014.12.035
[64]
Ben Ali, W., Metton, O., Roubertie, F., Pouard, P., Sidi, D., Raisky, O., et al. (2009) Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery: Late Results with Special Attention to the Mitral Valve. European Journal of Cardio- Thoracic Surgery, 36, 244-248. https://doi.org/10.1016/j.ejcts.2009.03.014
[65]
Brown, J.W., Ruzmetov, M., Parent, J.J., Rodefeld, M.D. and Turrentine, M.W. (2008) Does the Degree of Preoperative Mitral Regurgitation Predict Survival or the Need for Mitral Valve Repair or Replacement in Patients with Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery? The Journal of Thoracic and Cardiovascular Surgery, 136, 743-748. https://doi.org/10.1016/j.jtcvs.2007.12.065
[66]
Caspi, J., Pettitt, T.W., Sperrazza, C., Mulder, T. and Stopa, A. (2007) Reimplantation of Anomalous Left Coronary Artery from the Pulmonary Artery without Mitral Valve Repair. The Annals of Thoracic Surgery, 84, 619-623.
https://doi.org/10.1016/j.athoracsur.2007.03.036
[67]
Ginde, S., Earing, M.G., Bartz, P.J., Cava, J.R. and Tweddell, J.S. (2012) Late Complications after Takeuchi Repair of Anomalous Left Coronary Artery from the Pulmonary Artery: Case Series and Review of Literature. Pediatric Cardiology, 33, 1115-1123.
https://doi.org/10.1007/s00246-012-0260-5