Guo R, Chang SH, Wang BQ, Zhang QG. Ipsilateral hemifacial microsomia with dextrocardia and pulmonary hypoplasia: A case report . World J Clin Cases 2022; 10(9): 2948-2953 [PMID: 35434090 DOI: 10.12998/wjcc.v10.i9.2948]
Corresponding Author of This Article
Bing-Qing Wang, MD, Attending Doctor, Department of Ear Reconstruction, Plastic Surgery Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, No. 33 Badachu Road, Shijingshan District, Beijing, 100000, China. plasticsurgern114@163.com
Research Domain of This Article
Otorhinolaryngology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Rui Guo, Shi-Hi Chang, Bing-Qing Wang, Qing-Guo Zhang, Department of Ear Reconstruction, Plastic Surgery Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100000, China
Author contributions: Guo R and Chang SH reviewed the literature and contributed to manuscript drafting; Wang BQ and Zhang QG were responsible for the revision of the manuscript for important intellectual content; all authors issued final approval for the version to be submitted.
Supported bythe National Natural Science Foundation of China, No. 81701930.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Bing-Qing Wang, MD, Attending Doctor, Department of Ear Reconstruction, Plastic Surgery Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, No. 33 Badachu Road, Shijingshan District, Beijing, 100000, China. plasticsurgern114@163.com
Received: October 11, 2021 Peer-review started: October 11, 2021 First decision: December 10, 2021 Revised: January 4, 2022 Accepted: February 20, 2022 Article in press: February 20, 2022 Published online: March 26, 2022 Processing time: 162 Days and 3.6 Hours
Abstract
BACKGROUND
Hemifacial microsomia (HFM) is a rare congenital malformation characterized by a combination of various anomalies, including the face, ears, eyes, and vertebrae. Prenatal diagnosis for HFM is possible, and quite accurate ultrasound can detect obvious defects. The etiology is still unknown, although some hypotheses have been proposed, including gene mutation, chromosome anomaly, and environmental risk factors. However, there are few reports of pulmonary hypoplasia and dextrocardia in HFM.
CASE SUMMARY
A 2-year-old boy presented to the ear reconstruction department of our hospital complaining of deviation of the face to the right side and auricular anomaly. Physical examination revealed facial asymmetry, preauricular skin tags, and concha-type microtia with stricture of the external auditory canal on the right side. Head magnetic resonance imaging showed bilateral semicircular canal dysplasia and bilateral internal auditory canals stenosis. Audiometric examination showed bilateral severe sensorineural hearing loss. Chest radiography and computed tomography showed dextrocardia and right pulmonary hypoplasia.
CONCLUSION
This case presented a rare finding and an unusual association of 3 malformations, ipsilateral HFM, pulmonary agenesis, and dextrocardia.
Core Tip: Hemifacial microsomia (HFM) is a rare congenital malformation characterized by a combination of various anomalies including face, ears, eyes, and vertebrae. We present a rare case of ipsilateral HFM with dextrocardia and pulmonary hypoplasia. Those malformations may compose a laterality syndrome or just an extension of an expanded spectrum of HFM.
