Unilateral pulmonary vein atresia presenting with recurrent haemoptysis in a child: a case report
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CASE REPORT
Open Access
Unilateral pulmonary vein atresia presenting with recurrent haemoptysis in a child: a case report Martin Ngie Liong Wong1, Ing Ping Tang2*, Yek Kee Chor3, Kiew Siong Lau4, Anne Rachel John5, King Ching Hii2, Olive Pei Yi Lee2, Wooi Kok Lim1 and Hannah Pei Koon Tan6
Abstract Background: Haemoptysis is an uncommon presenting symptom in children and is usually caused by acute lower respiratory tract infection or foreign body aspiration. We report a rare case of right unilateral pulmonary vein atresia (PVA) as the underlying aetiology of recurrent haemoptysis in a child. Case presentation: A 4 years old girl presented with history of recurrent haemoptysis. Bronchoscopic evaluation excluded a foreign body aspiration but revealed right bronchial mucosal hyperaemia and varices. Diagnosis of right unilateral PVA was suspected on transthoracic echocardiography which demonstrated hypoplastic right pulmonary artery and non-visualization of right pulmonary veins. Final diagnosis was confirmed on cardiac CT angiography. A conservative treatment approach was opted with consideration for pneumonectomy in future when she is older. Conclusion: Rarer causes should be considered when investigating for recurrent haemoptysis in children. Bronchoscopy and cardiac imaging are useful tools to establish the diagnosis of unilateral PVA in our case. Keywords: Haemoptysis, Pulmonary vein atresia, Bronchial varices
Bacground Haemoptysis is an uncommon presenting symptom in children. The most common aetiologies are acute lower respiratory tract infection and foreign body aspiration [1, 2]. Diagnosis can be readily attained by history, physical examination and chest radiograph in most cases. With appropriate treatment, the clinical course is usually self-limiting. However, when the presentation is recurrent and initial assessment fails to establish the common causes, other rarer and potentially serious aetiologies should be explored. We report a rare case of right unilateral pulmonary vein atresia as the underlying aetiology of recurrent haemoptysis in a child. * Correspondence: [email protected] 2 Department of ORL HNS, Faculty of Medicine and Health Sciences, Universiti Malaysia Sarawak, Kota Samarahan, Sarawak, Malaysia Full list of author information is available at the end of the article
Case presentation A 4 years old girl was referred to our hospital for suspected foreign body aspiration. She presented with 3 days history of cough and haemoptysis. The expectorant consisted of fresh bright blood mixed with small clots and mucus (Fig. 1a). There were 2 previous similar episodes when she was 8 months and 3 years old. Both episodes were treated as Mallory-Weiss syndrome in another hospital and symptoms resolved without further investigation. On physical examination, she was pink and not tachypnoeic. Her oxygen saturation was 98% under the room air. A grade 2 systolic murmur was detected over the right upper sternal border. There was reduced breath sound over the right lung field. Other organ system examination was
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