Bilateral Communicating Intralobar Pulmonary Sequestration
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《交互式心脏血管和胸部手术医学期刊》
Department of Pediatric Surgery, Amrita Institute of Medical Sciences, Amrita Lake, Elamakkara, Kochi 682 026, Kerala, India.
A one-month-old female child presented with recurrent respiratory tract infections since birth. X-ray chest showed haziness of the right lower and middle zones and also of the left lower zone. Bronchoscopy revealed a normal tracheobronchial tree. Post bronchoscopy X-ray showed partial clearing of the opacities. Spiral CT scan showed intra lobar sequestration of right and left lower lobes. MRI angiogram showed aberrant arterial feeder from celiac trunk supplying both right and left sequestrations. Gastrograffin studies showed gastro-bronchial communication arising as a single trunk from lesser curvature of the stomach and dividing into two supplying both sequestrations (Fig.1).
Fig. 1. Gastrograffin study showing gastro bronchial fistula.
Laparotomy and division of the gastro-bronchial communication was done. Vascular supply was inacessible and so were not divided. Right thoracotomy showed right lower lobe and middle lobe sequestration. Both lobes were resected. Left lower lobe sequestration was left as such since resection of the left lower lobe along with right lower and middle lobes would have caused severe respiratory insufficiency. Vascular supply to the left side was identified and divided. Histopathological examination confirmed the diagnosis of intralobar sequestration. CT scan after 6 months showed that left lower lobe sequestration had disappeared.
This is a communicating type of sequestration where the sequestration communicates with the fore gut. Savic, et al.(1) reported that only 2.2% were in the middle or upper lobes. In our case both right and left lower lobes as well as middle lobe were involved making it an extremely rare presentation. Communication through the Pores of Kohn leading to partial aeration of the sequestration was responsible for the partial aeration seen after bronchoscopy. Srikanth, et al.(2) reviewed 57 cases and reported that bilateral communicating sequestrations occurred only in 7% of cases.
Treatment consists of lobectomy with division of fistulous communication. Embolisation has been tried with varying results. In our case bilateral lower lobectomies and middle lobectomy would not have been compatible with life. Spontaneous occlusion of the vascular supply has been reported with no untoward effect(3), which prompted us to ligate the blood supply to the left side along with ligation of the gastro-bronchial communication. This combined modality of resection of most infected side with ligation of fistula and vascular supply to the other side may be helpful in similar cases.
Acknowledgement
We thank Dr. P.K. Rajiv and his team for pre and post-operative medical management in the Department of Neonatology; and Dr. Lakshmi and Dr. Rekha for per-operative anesthetic support.
References
1. Savic B, Birtel FJ, Tholen W, Funke HD, Knoche R. Lung sequestration: Report of seven cases and review of 540 published cases. Thorax 1979; 34: 96-101.
2. Srikanth MS, Ford EG. Stanley P, Mahour GH. Communicating bronchopulmonary foregut malformation: Classification and embryogenesis. J Pediatr Surg 1992; 27: 732-736.
3. Lababidi Z, Dyke PC, Angiographic demonstration of spontaneous occlusion of systemic arterial supply in pulmonary sequestration. Pediatr Cardiol 2003; 24: 406-408.(Joy M.G., Mohan K. Abraham,)
A one-month-old female child presented with recurrent respiratory tract infections since birth. X-ray chest showed haziness of the right lower and middle zones and also of the left lower zone. Bronchoscopy revealed a normal tracheobronchial tree. Post bronchoscopy X-ray showed partial clearing of the opacities. Spiral CT scan showed intra lobar sequestration of right and left lower lobes. MRI angiogram showed aberrant arterial feeder from celiac trunk supplying both right and left sequestrations. Gastrograffin studies showed gastro-bronchial communication arising as a single trunk from lesser curvature of the stomach and dividing into two supplying both sequestrations (Fig.1).
Fig. 1. Gastrograffin study showing gastro bronchial fistula.
Laparotomy and division of the gastro-bronchial communication was done. Vascular supply was inacessible and so were not divided. Right thoracotomy showed right lower lobe and middle lobe sequestration. Both lobes were resected. Left lower lobe sequestration was left as such since resection of the left lower lobe along with right lower and middle lobes would have caused severe respiratory insufficiency. Vascular supply to the left side was identified and divided. Histopathological examination confirmed the diagnosis of intralobar sequestration. CT scan after 6 months showed that left lower lobe sequestration had disappeared.
This is a communicating type of sequestration where the sequestration communicates with the fore gut. Savic, et al.(1) reported that only 2.2% were in the middle or upper lobes. In our case both right and left lower lobes as well as middle lobe were involved making it an extremely rare presentation. Communication through the Pores of Kohn leading to partial aeration of the sequestration was responsible for the partial aeration seen after bronchoscopy. Srikanth, et al.(2) reviewed 57 cases and reported that bilateral communicating sequestrations occurred only in 7% of cases.
Treatment consists of lobectomy with division of fistulous communication. Embolisation has been tried with varying results. In our case bilateral lower lobectomies and middle lobectomy would not have been compatible with life. Spontaneous occlusion of the vascular supply has been reported with no untoward effect(3), which prompted us to ligate the blood supply to the left side along with ligation of the gastro-bronchial communication. This combined modality of resection of most infected side with ligation of fistula and vascular supply to the other side may be helpful in similar cases.
Acknowledgement
We thank Dr. P.K. Rajiv and his team for pre and post-operative medical management in the Department of Neonatology; and Dr. Lakshmi and Dr. Rekha for per-operative anesthetic support.
References
1. Savic B, Birtel FJ, Tholen W, Funke HD, Knoche R. Lung sequestration: Report of seven cases and review of 540 published cases. Thorax 1979; 34: 96-101.
2. Srikanth MS, Ford EG. Stanley P, Mahour GH. Communicating bronchopulmonary foregut malformation: Classification and embryogenesis. J Pediatr Surg 1992; 27: 732-736.
3. Lababidi Z, Dyke PC, Angiographic demonstration of spontaneous occlusion of systemic arterial supply in pulmonary sequestration. Pediatr Cardiol 2003; 24: 406-408.(Joy M.G., Mohan K. Abraham,)