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• W1587789142 abstract "HomeCirculationVol. 131, No. 19Ductus Arteriosus Aneurysm and Vocal Cord Paralysis Free AccessResearch ArticlePDF/EPUBAboutView PDFView EPUBSections ToolsAdd to favoritesDownload citationsTrack citationsPermissionsDownload Articles + Supplements ShareShare onFacebookTwitterLinked InMendeleyReddit Jump toSupplemental MaterialFree AccessResearch ArticlePDF/EPUBDuctus Arteriosus Aneurysm and Vocal Cord Paralysis Jennifer C. Walker, MD, Riksta Dikkers, MD, PhD, Gyorgy B. Halmos, MD, PhD, Rolf M.F. Berger, MD, PhD and Gideon J. du Marchie Sarvaas, MD Jennifer C. WalkerJennifer C. Walker From the Departments of Pediatric Cardiology (J.C.W., R.M.F.B., G.J.d.M.S.), Radiology (R.D.), and Otolaryngology, Head and Neck Surgery (G.B.H.), University Medical Center Groningen, University of Groningen, The Netherlands, Center for Congenital Heart Diseases, Beatrix Children’s Hospital. Search for more papers by this author , Riksta DikkersRiksta Dikkers From the Departments of Pediatric Cardiology (J.C.W., R.M.F.B., G.J.d.M.S.), Radiology (R.D.), and Otolaryngology, Head and Neck Surgery (G.B.H.), University Medical Center Groningen, University of Groningen, The Netherlands, Center for Congenital Heart Diseases, Beatrix Children’s Hospital. Search for more papers by this author , Gyorgy B. HalmosGyorgy B. Halmos From the Departments of Pediatric Cardiology (J.C.W., R.M.F.B., G.J.d.M.S.), Radiology (R.D.), and Otolaryngology, Head and Neck Surgery (G.B.H.), University Medical Center Groningen, University of Groningen, The Netherlands, Center for Congenital Heart Diseases, Beatrix Children’s Hospital. Search for more papers by this author , Rolf M.F. BergerRolf M.F. Berger From the Departments of Pediatric Cardiology (J.C.W., R.M.F.B., G.J.d.M.S.), Radiology (R.D.), and Otolaryngology, Head and Neck Surgery (G.B.H.), University Medical Center Groningen, University of Groningen, The Netherlands, Center for Congenital Heart Diseases, Beatrix Children’s Hospital. Search for more papers by this author and Gideon J. du Marchie SarvaasGideon J. du Marchie Sarvaas From the Departments of Pediatric Cardiology (J.C.W., R.M.F.B., G.J.d.M.S.), Radiology (R.D.), and Otolaryngology, Head and Neck Surgery (G.B.H.), University Medical Center Groningen, University of Groningen, The Netherlands, Center for Congenital Heart Diseases, Beatrix Children’s Hospital. Search for more papers by this author Originally published12 May 2015https://doi.org/10.1161/CIRCULATIONAHA.114.013568Circulation. 2015;131:1713–1714A 3-day-old full-term infant was transferred to our pediatric intensive care unit because of significant biphasic stridor with desaturation spells. The history revealed an uncomplicated pregnancy and normal spontaneous vaginal delivery. The mother was known to have hypothyroidism caused by Hashimoto thyroiditis (negative thyroid-stimulating immunoglobulin). Apgar scores were 6 and 9 after 1 and 5 minutes; no resuscitation was needed. Umbilical arterial and venous blood gas values showed marked acidosis (pH 6.81 and 6.88, respectively) without signs of encephalopathy. General and cardiovascular examinations were normal. In particular, there was no marfanoid habitus, heart sounds were normal, and peripheral pulses were preserved. Chest x-ray illustrated a pronounced cardiac silhouette with signs of a normal pulmonary circulation (Figure 1). Echocardiography showed a structurally normal heart, left aortic arch (without signs of airway compression), and no double aortic arch. However, the entrance orifice of the ductus arteriosus was enlarged, tapering to a small tortuous channel at the site of pulmonary artery insertion where the exit orifice was small, with restrictive left-to-right shunt (Figure 2). Hence, a diagnosis of ductus arteriosus aneurysm (DAA) was made. Computed tomography angiography scan confirmed this finding, with a ductal diameter of 9 mm and length of 11 mm (Figure 3). Fiberoptic laryngoscopy under general anesthesia and spontaneous breathing was performed and revealed left and partial right vocal cord paralysis, possibly resulting from compression of the left recurrent laryngeal nerve by the DAA (Movie I in the online-only Data Supplement). The patient was intubated and ventilated. Tracheotomy was performed, and ventilation was weaned. In the following weeks, echocardiography demonstrated closing of the ductus arteriosus and regression of the aneurysm size. Three months later, repeated laryngoscopy revealed normal movement of the vocal cords.Download figureDownload PowerPointFigure 1. Chest x-ray with a pronounced cardiac silhouette. There are no signs of a “ductal bump.”Download figureDownload PowerPointFigure 2. Echocardiography illustrating the enlarged entrance orifice of the ductus arteriosus aneurysm (DAA) with the small tortuous channel at the site of left pulmonary artery. DA indicates descending aorta; EO, exit orifice of the ductus aneurysm to the pulmonary artery; and LPA, left pulmonary artery.Download figureDownload PowerPointFigure 3. Computed tomographic angiography with 3-dimensional reconstruction (A), coronal (B), and sagittal (C) multiplanar reconstruction confirming the diagnosis of ductus arteriosus aneurysm (DAA).Congenital DAA, a rare condition caused by saccular dilatation of the ductus arteriosus, can potentially be fatal because of the risk of infection, dissection, rupture, or thrombus formation and embolization. It can also compress adjacent structures such as the recurrent laryngeal nerve. Signs and symptoms are usually scarce and discrete. If symptoms occur, the child usually presents before 2 months of age. The incidence of DAA is somewhat unclear, with a reported incidence of 0.3% to 1% in neonatal autopsies.1 Surgical resection could be considered if DAA remains patent beyond the neonatal period, if DAA is associated with connective tissue disease, if there is evidence of thromboembolism, or if significant compression of adjacent structures exists.2 Surgical excision, however, carries additional risk for (further) nerve damage. Ligation and decompression of DAA is another option.3 Spontaneous regression occurs, as seen previously4 and in the present case. Therefore, a “wait and see” approach can be a justified policy in some patients.DisclosuresNone.FootnotesThe online-only Data Supplement is available with this article at http://circ.ahajournals.org/lookup/suppl/doi:10.1161/CIRCULATIONAHA.114.013568/-/DC1.Correspondence to Gideon J. du Marchie Sarvaas, MD, Department of Pediatric Cardiology, University Medical Center Groningen, Hanzeplein 1, PO Box 30.001, 9700 RB, Groningen, The Netherlands. E-mail [email protected]References1. Cruickshank B, Marquis RM.Spontaneous aneurysm of the ductus arteriosus.Am J Med. 1958; 25:140–149.CrossrefMedlineGoogle Scholar2. Hornberger LK.Congenital ductus arteriosus aneurysm.J Am Coll Cardiol. 2002; 39:348–350.CrossrefMedlineGoogle Scholar3. Hornung TS, Nicholson IA, Nunn GR, Hawker RE.Neonatal ductus arteriosus aneurysm causing nerve palsies and airway compression: surgical treatment by decompression without excision.Pediatr Cardiol. 1999; 20:158–160.CrossrefMedlineGoogle Scholar4. Das BB, Solowiejczyk DE, Kamath V, Sharma J.Congenital ductus arteriosus aneurysm presenting with stridor in a newborn.J Perinatol. 2002; 22:179–180. doi: 10.1038/sj.jp.7210676.CrossrefMedlineGoogle Scholar Previous Back to top Next FiguresReferencesRelatedDetailsCited By (2019) Congenital Heart Disease (I) Pathology of Heart Disease in the Fetus, Infant and Child, 10.1017/9781316337073.004, (75-117) Wang J, Chen H, Su X and Zhang Z (2016) Aortic dissection manifesting as dysphagia and hoarseness: Ortner's syndrome, The American Journal of Emergency Medicine, 10.1016/j.ajem.2015.11.038, 34:6, (1185.e1-1185.e3), Online publication date: 1-Jun-2016. Sanchez-Jacob R, Cielma T and Mudd P (2021) Ultrasound of the vocal cords in infants, Pediatric Radiology, 10.1007/s00247-021-05235-0 May 12, 2015Vol 131, Issue 19 Advertisement Article InformationMetrics © 2015 American Heart Association, Inc.https://doi.org/10.1161/CIRCULATIONAHA.114.013568PMID: 25964282 Originally publishedMay 12, 2015 PDF download Advertisement SubjectsComputerized Tomography (CT)Echocardiography" @default.
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