Ann Thorac Surg 2001;72:608-609
© 2001 The Society of Thoracic Surgeons
Case report
A spontaneous ductal aneurysm presenting with left recurrent laryngeal nerve palsy
Jonathan R.S. Day, MRCSa,
Robin K. Walesby, FRCSa
a Department of Cardiothoracic Surgery, Middlesex Hospital, London, England, United Kingdom
Accepted for publication August 17, 2000.
Address reprint requests to Dr Day, Gruinart, Tullibardine Crescent, Auchterarder, Perthshire PH 3 ILY UK
e-mail: jrsday{at}hotmail.com
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Abstract
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A 76-year-old man was found to have a left recurrent laryngeal nerve palsy, due to a spontaneous patent ductus arteriosus aneurysm. Ortner’s syndrome (left recurrent laryngeal nerve palsy caused by cardiovascular pathology) secondary to patent ductus arteriosus aneurysm has not previously been described in the literature. We present a case, illustrated with surface rendering computer tomogram reconstructions.
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Introduction
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Ortner’s syndrome is the clinical entity of hoarseness due to left recurrent laryngeal nerve palsy caused by cardiovascular pathology. We present a case of patent ductus arteriosus aneurysm presenting with Ortner’s syndrome.
A 76-year-old man with severe rheumatoid arthritis and peripheral vasculitis presented to hospital with gradual onset hoarseness. Fiber optic laryngoscopy showed left-sided vocal cord palsy, and a chest radiograph revealed a mediastinal mass adjacent to the aortic knuckle. A contrast computed tomography (CT) scan of the thorax demonstrated a saccular aneurysm of a patent ductus arteriosus partially occluded by thrombus (Figs 1 and 2). The ductus aneurysm was compressing the left recurrent laryngeal nerve causing Ortner’s syndrome.
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Fig 1. Surface rendering computed tomographic reconstruction of the heart and great vessels, showing a saccular aneurysm of a patent ductus arteriosus.
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As a result of the underlying medical conditions it was decided that conservative management would be most appropriate.
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Comment
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One hundred and forty-four cases of aneurysms of the ductus arteriosus have been reported in the literature of which 106 appeared spontaneously and 38 followed surgical treatment of a patent ductus arteriosus [1].
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Fig 2. Dual-threshold computed tomographic reconstruction showing thrombus within the pulmonary artery and patent ductus arteriosus.
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In adults both aortography and CT are well-established diagnostic methods for identifying ductus aneurysms [1–3]. In neonates, transthoracic echocardiography gives good images of the ductus whereas in older children and adults transesophageal echocardiography is required [1]. Serious complications of spontaneous ductus arteriosus aneurysm are rupture, erosion, infection, and thromboembolism [1].
Histology of a ductal diverticular aneurysm resected from an adult was reported as showing focal necrosis and mucoid degeneration of the media, with intimal thickening and fibroelastosis [4]. Ohtsuka and associates [4] suggest that incomplete closure of the duct causes fragility of the ductal wall giving the potential for aneurysm formation. Ductal diverticular aneurysms have also been reported in association with systemic connective tissue disorders such as Ehlers-Danlos syndrome [5] and Marfan’s syndrome [6].
Lund and coworkers [1] suggest prompt surgical treatment of all spontaneous ductus arteriosus aneurysms in patients older than 2 months of age and in all patients with postoperative ductus arteriosus aneurysm. In infants, a ductus arteriosus aneurysm should be closely observed with echocardiography, as spontaneous regression has been reported in this age group. If no regression is seen within a few days, it should be surgically corrected [1].
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References
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Lund J.T., Jensen M.B., Hjelms E. Aneurysm of the ductus arteriosus. A review of the literature and the surgical implications. Eur J Cardiothorac Surg 1991;5:566-570.[Abstract/Free Full Text]
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Taneja K., Gulati M., Jain M., Saxena A., Das B., Rajani M. Ductus arteriosus aneurysm in the adult: role of computed tomography in diagnosis. Clin Radiol 1997;52:231-234.[Medline]
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Cohen B.A., Efremidis S.C., Dan S.J., Robinson B., Rabinowitz J.G. Case report. Aneurysm of the ductus arteriosus in an adult. J Comput Assist Tomogr 1981;5:421-423.[Medline]
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Ohtsuka S., Kakihana M., Ishikawa T., Noguchi Y., Kuga K., Ishimitsu T., et al. Aneurysm of patent ductus arteriosus in an adult case: findings of cardiac catheterization, angiography, and pathology. Clin Cardiol 1987;10:537-540.[Medline]
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Chang J.P., Chang C.H., Sheih M.J. Aneurysmal dilatation of patent ductus arteriosus in a case of Ehlers-Danlos syndrome. Ann Thorac Surg 1987;44:656-657.[Abstract/Free Full Text]
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Muller N.L., Mayo J., Culham J.A., Nichols D.M. Ductus arteriosus aneurysm in Marfan syndrome. Can Assoc Radiol J 1986;37:195-197.[Medline]
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Abstract
Introduction
