Autopsy and Case Reports
https://www.autopsyandcasereports.org/article/doi/10.4322/acr.2023.440
Autopsy and Case Reports
Autopsy Case Report

Hypoplastic coronary artery disease, as a cause of sudden death

Moirangthem Sangita; Jayanthi Yadav; Jai Kumar Chaurasia; Arneet Arora; Afsar Jahan; Mrinal Patnaik

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Abstract

Hypoplastic coronary artery disease (HCAD) is a rare coronary artery anomaly that may be the cause of sudden death. It can involve a single or all coronary arteries. This anomaly may cause circulatory insufficiency leading to myocardial infarction. HCAD has no symptoms or may exhibit cardiovascular signs like syncope, dyspnea, chest discomfort, or dizziness. It is often diagnosed at autopsy, and early diagnosis is made with a coronary angiogram. We report HCAD as the cause of the sudden death of a 25-year-old female with a history of loss of consciousness following exertion. On autopsy, all the coronary arteries’ lumen was narrowed with thin vessel walls. Histopathological examination shows an underdeveloped and missing muscular layer of the left anterior descending and circumflex coronary arteries’ vascular wall. Many cases of HCAD diagnosed by radiographic imaging in living patients have been reported in the literature, but a structural anomaly of coronaries leading to HCAD has not been reported. We report a case of HCAD describing the histopathological examination findings of the vascular wall of coronary vessels illustrating the structural difference.

Keywords

Coronary Disease, Death, Sudden, Autopsy, Pathology

References

1 Aggrawal A. Textbook of forensic medine and toxicology. 2nd ed. Kala Amb: Avichal Publishing Company; 2021.

2 Hauser M. Congenital anomalies of the coronary arteries. Heart. 2005;91(9):1240-5. http://dx.doi.org/10.1136/hrt.2004.057299. PMid:16103577.

3 Ghosh P, Chauhan A. Rare variant of an anomalous left coronary artery: arising from the distal continuation of a single right coronary artery. Tex Heart Inst J. 2012;39(1):63-4. PMid:22412230.

4 Riede FN, Bulla S, Grundmann S, Werner M, Riede UN, Otto C. Isolated hypoplastic circumflex coronary artery: a rare cause of haemorrhagic myocardial infarction in a young athlete. Diagn Pathol. 2013;8(1):91. http://dx.doi.org/10.1186/1746-1596-8-91. PMid:23742172.

5 Wick R, Otto S, Byard RW. Is right coronary artery hypoplasia and sudden death an underdiagnosed association? Am J Forensic Med Pathol. 2007;28(2):128-30. http://dx.doi.org/10.1097/PAF.0b013e31805c93fd. PMid:17525562.

6 Rizzo S, Gaspari M, Frescura C, Padalino M, Thiene G, Basso C. Sudden death and coronary artery anomalies. Front Cardiovasc Med. 2021;8:636589. http://dx.doi.org/10.3389/fcvm.2021.636589. PMid:33869302.

7 Angelini P. Coronary artery anomalies: an entity in search of an identity. Circulation. 2007;115(10):1296-305. http://dx.doi.org/10.1161/CIRCULATIONAHA.106.618082. PMid:17353457.

8 McFarland C, Swamy RS, Shah A. Hypoplastic coronary artery disease: a rare cause of sudden cardiac death and its treatment with an implantable defibrillator. J Cardiol Cases. 2011;4(3):e148-51. http://dx.doi.org/10.1016/j.jccase.2011.08.005. PMid:30532882.

9 Feger J, Jones J, Hacking C. Coronary hypoplasia. Radiopaedia.org [Internet]. 2021. https://doi.org/10.53347/rID-86153.

10 Amabile N, Fraisse A, Quilici J. Hypoplastic coronary artery disease: report of one case. Heart. 2005;91(2):e12. http://dx.doi.org/10.1136/hrt.2004.047621. PMid:15657202.

11 Zugibe FT, Zugibe FT Jr, Costello JT, Breithaupt MK. Hypoplastic coronary artery disease within the spectrum of sudden unexpected death in young and middle age adults. Am J Forensic Med Pathol. 1993;14(4):276-83. http://dx.doi.org/10.1097/00000433-199312000-00002. PMid:8116584.

12 Molina DK, DiMaio VJ. Normal organ weights in women: part i-the heart. Am J Forensic Med Pathol. 2015;36(3):176-81. http://dx.doi.org/10.1097/PAF.0000000000000174. PMid:26153896.

13 Nasr AY, El Tahlawi M. Anatomical and radiological angiographic study of the coronary ostia in the adult human hearts and their clinical significance. Anat Cell Biol. 2018;51(3):164-73. http://dx.doi.org/10.5115/acb.2018.51.3.164. PMid:30310708.

14 Doi Y, Waki K, Ogino K, Hayashi T. Hypoplastic coronary artery disease and hypertension in a child: a case report. Eur Heart J Case Rep. 2022;6(1):ytab526. http://dx.doi.org/10.1093/ehjcr/ytab526. PMid:35106442.

15 Morales R, BokowskiJ, Aljemmali S, Murphy J, Bharati S, Abdulla RI. A rare report of hypoplastic coronary arteries and pulmonary veins: a case report and review of the literature. Pediatr Cardiol. 2020;41(6):1231-7. http://dx.doi.org/10.1007/s00246-020-02334-x. PMid:32361881.

16 Raut BK, Patil VN, Cherian G. Coronary artery dimensions in normal Indians.Indian Heart J. 2017;69(4):512-4. http://dx.doi.org/10.1016/j.ihj.2017.01.009. PMid:28822520.

17 Ogden JA. Congenital anomalies of the coronary arteries.Am J Cardiol. 1970;25(4):474-9. http://dx.doi.org/10.1016/0002-9149(70)90016-0. PMid:5438244.

18 Giorgio F, Abbate A, Stigliano E, Capelli A, Arena V. Hypoplastic coronary artery disease causing sudden death. Report of two cases and review of the literature. Cardiovasc Pathol. 2010;19(4):e107-11. http://dx.doi.org/10.1016/j.carpath.2009.05.002. PMid:19616973.

19 Smith ET Jr, Davis GJ. Sudden cardiac death associated with hypoplasia of the coronary arteries and conduction system alteration. Am J Forensic Med Pathol. 1997;18(2):189-93. http://dx.doi.org/10.1097/00000433-199706000-00017. PMid:9185940.

20 Hong YM. The diameter of coronary arteries in infants and children without heart disease. J Korean Pediatr Soc. 1990;33(10):1353-61.

21 Sim DS, Jeong MH, Choi S, et al. Myocardial infarction in a young man due to a hypoplastic coronary artery.Korean Circ J. 2009;39(4):163-7. http://dx.doi.org/10.4070/kcj.2009.39.4.163. PMid:19949606.

22 Artery, vein, nerve trichrome stain. 2022 [cited 2022 Oct 31]. Available from: https://histology.siu.edu/crr/CR020b.htm

23 Shi X, Liu J, Wu J, Hua Y, Zhou K, Li Y. Hypoplastic coronary arteries in a child with a mutation in Notch1: a case report. Medicine. 2020;99(33):e21355. http://dx.doi.org/10.1097/MD.0000000000021355. PMid:32871987.

24 Guo A, Bakhshi H, O’Hara J, et al. Hypoplastic coronary artery disease presenting with ventricular fibrillation cardiac arrest.Eur J Case Rep Intern Med. 2021;8(8):002736. PMid:34527617.
 


Submitted date:
10/31/2022

Accepted date:
06/20/2023

Publication date:
07/19/2023

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