An Unusual Cause of Hemoptysis in a Young Woman
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An Unusual Cause of Hemoptysis in a Young Woman Jia-Ming Chang, MD, Jen-Hsun Cheng, MD, and Wei Chen, MD Divisions of Thoracic Surgery and Pulmonary and Critical Care Medicine, Chia-Yi Christian Hospital, Chiayi; and Department of Respiratory Therapy, China Medical University, and Department of Life Science, National Chung Hsing University, Taichung, Taiwan A 19-year-old woman presented with a 5-year history of chest tightness and hemoptysis after light exer- cise. Chest radiograph showed a vague retrocardiac nod- ular shadow (Fig 1A, arrow). Computed tomography scan with reconstruction (Fig 1B) showed a complete lack of pulmonary arterial supply to the basal segments, and a large artery (arrows) arising from the descending thoracic aorta (blue color) supplying the basal segments of the left lower lobe. On thoracoscopic surgery, the left lower lobe was markedly congested (Fig 2, arrows), and lobectomy was performed with ligation division of the aberrant systemic artery. The patient’s symptoms completely re- solved after this treatment, and she has stayed symptom free until now (more than 19 months). Anomalous systemic arterial supply to the normal basal segments with normal bronchial connection of the lower lobe is a rare congenital anomaly [1]. The estimated incidence was never proposed in literature review, prob- ably owing to its rarity. It is easily misdiagnosed because the chest roentgenogram is invariably normal. A com- puted tomography scan remains the investigation of choice. It can demonstrate the anomalous systemic ar- tery, absence of pulmonary arterial supply, and normal bronchial system in the affected segments [2]. This entity was initially termed “Pryce type I pulmonary sequestra- tion,” as proposed by Pryce in 1946, and alternatively termed “anomalous systemic artery to the left lower lobe” for not fitting into the narrower definition of pulmonary sequestration (extralobar or intralobar type). In current classification, it is now generally accepted as a variant of “pulmonary sequestration spectrum,” includ- ing abnormalities from tracheobronchial structure, lung parenchyma, and arterial or venous structures. Surgical resection with ligation of the aberrant artery remained the treatment of choice. Recently, some authors have shown endovascular coil embolization to be an effective alternative management, although, theoretically, lung infarction might be a concern [3]. From learning perspec- tives, physicians should raise the clinical suspicion of the disease in young adults with recurrent hemoptysis or exertional dyspnea. References 1. Yamanaka A, Hirai T, Fujimoto T, et al. Anomalous systemic arterial supply to normal basal segments of the left lower lobe. Ann Thorac Surg 1999;68:332– 8. 2. Ashizawa K, Ishida Y, Matsunaga N, et al. Anomalous sys- temic arterial supply to normal basal segments of left lower lobe: characteristic imaging findings. J Comput Assist Tomogr 2001;25:764 –9. 3. Abe T, Mori K, Shiigai M, et al. Systemic arterial supply to the normal basal segments of the left lower lobe of the lung— treatment by coil embolization—and a literature review. Car- diovasc Intervent Radiol 2011;34(Suppl 2):117–21. Address correspondence to Dr Chen, Department of Respiratory Ther- apy, China Medical University, 539 Jhongsiao Rd, Chiayi, Taiwan; e-mail: [email protected]. Fig 1. Fig 2. © 2012 by The Society of Thoracic Surgeons Ann Thorac Surg 2012;93:1734 • 0003-4975/$36.00 Published by Elsevier Inc doi:10.1016/j.athoracsur.2011.10.002 FEATURE ARTICLES
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Transcript of An Unusual Cause of Hemoptysis in a Young Woman
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An Unusual Cause of Hemoptysis in a YoungWomanJia-Ming Chang, MD, Jen-Hsun Cheng, MD, and Wei Chen, MDDivisions of Thoracic Surgery and Pulmonary and Critical Care Medicine, Chia-Yi Christian Hospital, Chiayi; and Department of
Respiratory Therapy, China Medical University, and Department of Life Science, National Chung Hsing University, Taichung,TaiwanA19-year-old woman presented with a 5-year historyof chest tightness and hemoptysis after light exer-
cise. Chest radiograph showed a vague retrocardiac nod-ular shadow (Fig 1A, arrow). Computed tomography scanwith reconstruction (Fig 1B) showed a complete lack ofpulmonary arterial supply to the basal segments, and alarge artery (arrows) arising from the descending thoracicaorta (blue color) supplying the basal segments of the leftlower lobe. On thoracoscopic surgery, the left lower lobewas markedly congested (Fig 2, arrows), and lobectomywas performed with ligation division of the aberrantsystemic artery. The patient’s symptoms completely re-solved after this treatment, and she has stayed symptomfree until now (more than 19 months).
Anomalous systemic arterial supply to the normalbasal segments with normal bronchial connection of thelower lobe is a rare congenital anomaly [1]. The estimatedincidence was never proposed in literature review, prob-ably owing to its rarity. It is easily misdiagnosed becausethe chest roentgenogram is invariably normal. A com-puted tomography scan remains the investigation ofchoice. It can demonstrate the anomalous systemic ar-tery, absence of pulmonary arterial supply, and normalbronchial system in the affected segments [2]. This entitywas initially termed “Pryce type I pulmonary sequestra-tion,” as proposed by Pryce in 1946, and alternativelytermed “anomalous systemic artery to the left lower
Address correspondence to Dr Chen, Department of Respiratory Ther-apy, China Medical University, 539 Jhongsiao Rd, Chiayi, Taiwan; e-mail:
Fig 1.
© 2012 by The Society of Thoracic SurgeonsPublished by Elsevier Inc
lobe” for not fitting into the narrower definition ofpulmonary sequestration (extralobar or intralobar type).In current classification, it is now generally accepted as avariant of “pulmonary sequestration spectrum,” includ-ing abnormalities from tracheobronchial structure, lungparenchyma, and arterial or venous structures. Surgicalresection with ligation of the aberrant artery remainedthe treatment of choice. Recently, some authors haveshown endovascular coil embolization to be an effectivealternative management, although, theoretically, lunginfarction might be a concern [3]. From learning perspec-tives, physicians should raise the clinical suspicion of thedisease in young adults with recurrent hemoptysis orexertional dyspnea.
References
1. Yamanaka A, Hirai T, Fujimoto T, et al. Anomalous systemicarterial supply to normal basal segments of the left lowerlobe. Ann Thorac Surg 1999;68:332–8.
2. Ashizawa K, Ishida Y, Matsunaga N, et al. Anomalous sys-temic arterial supply to normal basal segments of left lowerlobe: characteristic imaging findings. J Comput Assist Tomogr2001;25:764–9.
3. Abe T, Mori K, Shiigai M, et al. Systemic arterial supply to thenormal basal segments of the left lower lobe of the lung—treatment by coil embolization—and a literature review. Car-
Fig 2.
diovasc Intervent Radiol 2011;34(Suppl 2):117–21.
Ann Thorac Surg 2012;93:1734 • 0003-4975/$36.00doi:10.1016/j.athoracsur.2011.10.002
