Ann Thorac Surg 2009;88:1358-1360. doi:10.1016/j.athoracsur.2008.11.065
© 2009 The Society of Thoracic Surgeons
Case Reports
Anomalous Systemic Arterial Supply to Separate Lingular and Basal Segments of the Lung: An Anatomic Consideration
Miyako Hiramatsu, MD,
Masayuki Iwashita, MD,
Takuya Inagaki, MD,
Hideki Matsudaira, MD,
Jun Hirano, MD,
Makoto Odaka, MD,
Kozo Nakanishi, MD,
Masataka Okabe, MD, PhD,
Toshiaki Morikawa, MD*
Department of Chest Surgery and Department of Anatomy, Tokyo Jikei University School of Medicine, Tokyo, Japan
Accepted for publication November 24, 2008.
* Address correspondence to Dr Morikawa, Department of Thoracic Surgery, Tokyo Jikei University School of Medicine, 3-25-8 Nishi-shimbashi, Minato-ku, Tokyo, 105-8461, Japan (Email: tmorikawa{at}jikei.ac.jp).
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Abstract
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A 22-year-old man was referred for hemoptysis and general fatigue after exercise. Arteriography demonstrated an anomalous artery arising from the descending aorta supplying the lingular and all of the basal segments of the left lung. The feeding areas of the pulmonary and anomalous arteries were mutually exclusive. He underwent division of the anomalous artery and combined resection of the diseased segments. The upper division of the upper lobe and the superior segment of the lower lobe were spared. His symptoms were greatly improved postoperatively. The preoperative anatomic evaluation of anomalous vessels is crucial in surgical management.
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Introduction
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Anomalous systemic arterial supply to the lung without an associated lack of airway development (pulmonary sequestration) is rare. In most patients, the anomalous artery arises from the thoracic descending aorta and usually supplies only the left basal segments. Surgical management by lower lobectomy has been usually indicated for this anomaly. We report a patient with a unique anomalous artery that fed not only the basal but also the lingular segments of the left lung.
A 22-year-old man presented with a 7-year history of recurrent episodes of hemoptysis. He also complained of general fatigue after exertion. A computed tomography scan demonstrated a large anomalous systemic artery arising from the descending aorta at the level of the eighth vertebral body. Descending aortography demonstrated a prominent anomalous artery that fed the lingular and all of the basal segments, separately (Fig 1B). Pulmonary arteriograms did not show these segments, but were normal within the upper division of the upper lobe and superior segment of the lower lobe (Fig 1A).
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Fig 1. (A) Lateral view of pulmonary arteriography. (B) Lateral view of arteriography of the anomalous artery. (The dashed line designates the interlobar fissure.)
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These results indicated that the blood supplies from the normal pulmonary artery and anomalous systemic artery to the left lung parenchyma were mutually exclusive and that the venous flow returned to the pulmonary venous system rather than to the systemic venous system. A perfusion scan supported these findings, showing a perfusion defect in the lower part of the left lung. The results of a virtual bronchoscopy and a ventilation scan showed normal branching of the bronchial tree. The diagnosis was anomalous systemic arterial supply to separate segments (lingular and basal) of the left lung, and he underwent an operation to eliminate the symptoms that were related to this left-to-left circulatory shunt.
The operation was performed with an anterolateral incision under video assistance. Marked capillary dilatation was seen on the surface of the basal and lingular segments, and it was easy to distinguish diseased parts from healthy lung. The anomalous artery originated from the anterolateral aspect of the descending aorta and went into the left lower lobe in parallel with the left inferior pulmonary vein. Reactive lymph node swelling was prominent around the anomalous artery.
Upon dissection of the major fissure, an anomalous artery that crossed the fissure from the basal to lingular segments was seen (Fig 2), which was temporarily occluded with a clamp at its origin. After we confirmed no remarkable changes had occurred in hemodynamics or blood gases, the artery was divided with a vascular stapler. The basal and lingular segments were removed by combined segmentectomies, and the upper division of the upper lobe and superior segments of the lower lobe were spared.
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Fig 2. Diagram shows the pathologic anatomy. (PA = left pulmonary artery; ASA = anomalous systemic artery; LUL = left upper lobe; LLL = left lower lobe.)
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The patient's postoperative recovery was uneventful, and he was discharged from the hospital early. The complaints of hemoptysis and general fatigue disappeared.
Histopathologic examination of the resected specimen showed that the wall of the anomalous artery was rich in elastic fibers and thickened with atherosclerotic change. Obstructive change of the small pulmonary artery in the peripheral bronchopulmonary bundle was seen. Also present were scattered hemosiderin-laden macrophages that reflected chronic hemorrhage; but otherwise, the lung parenchyma was normal.
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Comment
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In this patient an anomalous systemic artery arising from the descending thoracic aorta fed separate segments of both the upper and lower lobes of the left lung. This type of arterial configuration through the interlobar fissure into both the upper and lower lobes is unusual compared with previous reports of anomalous systemic arterial supply to the lung. The most common pattern of anomalous systemic artery to the lung arises from the descending thoracic aorta and feeds the basal segments of the left lower lobe. More than 50 patients with this pattern have been reported in Japan [1], and 15 have been described in the English literature [2]. While other patterns of anomalous artery have also been sporadically reported, an anomalous artery that supplied part of the left upper lobe together with the basal segment, as in the current case, has not been reported, previously.
The systemic arterial supply to the lung was first described by Huber in 1777 [3]. Since then, several articles have reported this congenital anomaly with various patterns of arterial origin and feeding area. Pryce and associates [4] described three variants of systemic arterial supply to the lower lung tissue and identified pulmonary intralobar sequestration as a clinical and pathologic entity. In this, systemic anomalous arterial supply without bronchial dislocation was classified as Pryce type I sequestration [4]. This arterial anomaly is now considered to be different from true sequestration [5] because it has normal bronchial connections and the involved lung is not sequestrated. Some synonyms for this clinical entity, such as "arterial pulmonary malinosculation" and "systemic arterializations of lung without sequestration," have been used recently, but the most widely accepted term is "anomalous systemic arterial supply (to the basal segments of the lung)." It is essential that a normal airway is confirmed to make a diagnosis of this anomaly, and therefore, virtual bronchoscopy and ventilation scan were performed in the current patient.
A surgical procedure for this anomaly is indicated only to reduce symptoms such as hemoptysis or dyspnea on exertion. Anastomosis of the divided systemic artery to the pulmonary artery has been reported in a few instances, but the results were successful only in a pediatric patient [6]. Simple ligation of the anomalous artery has been reported to be effective for eliminating the symptoms [7], probably by releasing systemic arterial pressure in lung parenchyma and by cutting off the left-to-left circulatory shunt. As a characteristic of this anomaly, however, the areas perfused by the pulmonary and anomalous arteries have been reported to be mutually exclusive, and a perfusion–ventilation mismatch would not be improved by this procedure. In most reported cases, left lower lobectomy with dissection of the anomalous vessel was performed [2], which permitted over-removal of normal portion in the superior segment of the lower lobe. In the current patient, preoperative arteriography showed that the anomalous artery also fed lingular segments and involved both the upper and lower lobes; therefore, combined resection of only the involved segments, not simple lower lobectomy, was indicated.
Segmentectomy has been reported to be technically feasible, without any additional morbidity, in 15% to 20% of sequestration cases [8], but this is the first report of anomalous arterial supply to the lung that was treated successfully by combined-segmentectomies over the major fissure. In the surgical management of anomalous systemic arterial supply to the lung, a rigorous preoperative evaluation of the perfusion area of the anomalous artery is crucial to delineate the appropriate anatomic components of the lung that should be removed.
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References
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- Hiromitsu M, Tatsuo K, Miwako N, et al. A case of pulmonary sequestration (Pryce Type-I) detected during a routine infant examination for cardiac murmur Jpn J Pediatr Surg 2003;39:4607-4612.
- 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;68:332-338.[Abstract/Free Full Text]
- Huber JJ. Observations aliquod de arteria sinngulari pulmoni. concessa Acta Helvet 1777;8:85.
- Pryce DM. Lower accessory pulmonary artery with intralobar sequestration of lung: a report of seven cases J Pathol 1946;58:457-467.
- Sade RM, Clouse M, Ellis Jr FH. The spectrum of pulmonary sequestration Ann Thorac Surg 1974;18:644-658.[Abstract/Free Full Text]
- Hessel II EA, Boyden EA, Stamm SJ, et al. High systemic origin of the sole artery to the basal segments of the left lung: findings, surgical treatment, and embryologic interpretation Surgery 1970;67:624-632.[Medline]
- Pernot C, Simon P, Hoeffel JC, et al. Systemic artery-pulmonary vein fistula without sequestration Pediatr Radiol 1991;21:158-159.[Medline]
- White JJ, Donahoo JS, Ostrow PT, et al. Cardiovascular and respiratory manifestations of pulmonary sequestration in childhood Ann Thorac Surg 1974;18:286-294.[Abstract/Free Full Text]
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Abstract
Introduction
