© 2014 Wiley Periodicals, Inc.
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REVIEW ARTICLE _______________________________________________________________
Lobar Torsion Following Bilateral Lung Transplantation Georgina Stephens,* Krishna Bhagwat,* Adrian Pick,* and David McGiffin*,y *Department of Cardiothoracic Surgery, Alfred Hospital, Melbourne, Victoria, Australia; and yMonash University, Melbourne, Victoria, Australia ABSTRACT Torsion of lobes of the lung is a rare complication following lung transplantation. We present a case of counterclockwise torsion of the right lung allograft and review of nine additional cases in the literature of lobar torsion following lung transplantation. A high degree of suspicion is needed for early diagnosis with rapid surgical intervention to preserve lung viability. doi: 10.1111/jocs.12476 (J Card Surg
2015;30:209–214)
Few lobar torsions following lung transplantation have been reported in the literature.1–7 Immediate surgical correction is required to correct the torsion and reduce the associated high mortality rate. We report a case of lobar torsion following lung transplantation and review the literature on this subject.
PATIENT PROFILE A 36-year-old female underwent bilateral sequential lung transplantation (BLTx) for cystic fibrosis–related bronchiectasis. There was a size mismatch with the donor lungs being significantly larger than the patient’s thoracic cavity, necessitating a lower lobectomy of the right donor lung. The operation was complicated by primary graft failure, requiring support with veno-venous extracorporeal membranous oxygenation (VV-ECMO). The ischemic time for the left lung was four hours, and five hours and fifteen minutes for the right lung. The postoperative chest radiograph (CXR) showed bilateral congestion of the lungs, a size mismatch with a large left allograft, and resultant mediastinal shift towards the right (Fig. 1). VV-ECMO was weaned, with decannulation occurring on postoperative day 4. The patient was extubated on day 5. Her stay in the intensive care unit was complicated by multiresistant Pseudomonas bacteraemia, which was treated with Cefepime and Ciprofloxacin.
Conflict of interest: The authors acknowledge no conflict of interest in the submission. Address for correspondence: Prof. David McGiffin, Department of Cardiothoracic Surgery, Alfred Health, 55 Commercial Road, Melbourne, Victoria 3004, Australia. Fax: þ61-3-9076-2317; e-mail: [email protected]
On postoperative day 10 routine flexible bronchoscopy showed that the right bronchial orifice had the appearance of a ‘‘fish mouth’’ slit opening with congested mucosa. The bronchus could not be engaged. A computed tomography (CT) high-resolution chest and pulmonary angiogram was performed. A partial torsion of the right hilum with severe compromise to both the right upper and middle lobe bronchi was identified. The right upper lobe pulmonary vein was occluded, resulting in secondary pulmonary venous hypertension (Fig. 2). Despite these radiological findings, the patient remained hemodynamically stable and clinically well. She was started on heparin for the pulmonary vein occlusion. The patient returned to the operating room on postoperative day 11 where the right-sided thoracotomy incision was opened. The hilum of the right lung was found to be rotated approximately 908 counterclockwise as seen from the right axilla, so that the right middle lobe (RML) was orientated antero-superiorly, and the right upper lobe (RUL) posterio-inferiorly. Detorsion was performed by clockwise rotation of the lobes. Both the RML and RUL were viable with no signs of necrosis or infarction. Intraoperative transesophageal echocardiogram showed no evidence of thrombus in the right pulmonary veins and demonstrated normal pulmonary venous flow. Abrasion pleurodesis was performed, as well as suturing of the visceral pleura of the RUL to the right side of the pericardium and to the parietal pleura posteriorly. Bronchoscopy confirmed that the RML and RUL bronchi were now patent. Postoperatively, the patient was successfully extubated. A postoperative CT scan demonstrated normal orientation of the right upper and middle lobe bronchi. Eight months after the transplant, the patient had no respiratory symptoms and her FEV1 was 2.31 L (83%
210
STEPHENS ET AL. LOBAR TORSION
J CARD SURG 2015;30:209–214
Figure 1. (A) Chest X-ray before transplant; (B) chest X-ray on day 1 post–lung transplant showing allograft size mismatch; (C) chest X-ray post-detorsion; (D) chest X-ray follow-up six months later.
of predicted). Normal orientation of the right upper and middle lobe was confirmed. DISCUSSION Lobar torsion is extremely rare after lung transplantation. This complication has a reported incidence of 0.089–0.3%, occurring most frequently after lung resection.1 It has also been reported after a transthoracic needle aspiration, in association with pneumonia, pleural effusions, abdominal surgery, traumatic pneumothorax, and a diaphragmatic hernia.2 A review of the literature shows that a delay in the diagnosis and treatment of this rare complication can have catastrophic consequences such as obstructive pneumonia, hemoptysis, hypoxemia, bronchorrhea, and persistent air leak. Interestingly, not many cases of lobar torsion following lung transplantation have been
reported; furthermore, this complication may be underrecognized and underreported. Previous reported cases of lung torsion after lung transplantation are outlined in Table 1. Lin et al.1 reported a case of RML torsion after BLTx for bronchiolitis obliterans due to paraneoplastic pemphigus. The right lower lobe of the donor lung was resected due to inflammatory change found during procurement. The postoperative CXR showed persisting RML infiltrates. RML lobectomy was performed after the reconstructed chest CT confirmed the diagnosis of RML torsion. Souilamas et al.4 reported four cases of acute lobar torsion in patients who had undergone BLTx. Bronchoscopy showed complete obstruction in only two of them. Multiplanar reformats from thin slice CT scan allowed diagnosing torsion, confirming vessels and bronchi orientation, course, and strictures. At
J CARD SURG 2015;30:209–214
STEPHENS ET AL. LOBAR TORSION
211
torsion. The diagnosis was confirmed by pulmonary angiography and later at thoracotomy. The presence of a complete left major fissure was identified, and it was speculated that this played a role in the development of torsion. Gilkeson et al.6 reported a case of right middle and lower lobe torsion following right SLTx for silicosis. This case took a benign course and did not require surgical correction. Despite the radiographic and bronchoscopic appearance of lung torsion, with the finding of a patent arterial and venous blood supply a nonoperative strategy was selected. These authors noted that the donor allograft was significantly smaller than the recipient’s right hemithorax, and that this may have contributed to the development of torsion. Collins et al.7,8 reported left upper lobe torsion in a 39-year-old man who developed shortness of breath, hemoptysis, and hypoxemia three days after BLTx for chronic obstructive pulmonary disease. A CT scan demonstrated displacement of the left major fissure, cut off of the left main-stem bronchus, abnormal orientation of the left pulmonary artery, and consolidation of the displaced left upper lobe. Torsion and hemorrhagic infarction were confirmed surgically at detorsion and left upper lobectomy five days after transplantation. Pathology Torsion occurs when a lobe, or the entire lung, rotates around the hilar structures. This can be complete (1808) or partial (908), creating compromise to the airway, arterial supply, and the venous and lymphatic drainage. The affected lobe often shows hemorrhagic infarction or necrosis. Figure 2. (A) CT Scan, Sagittal View showing torsion of RUL and RML with their respective pulmonary artery distorted; (B) CT Scan, Axial View showing interlobular septal thickening with venous congestion.
operation, one detorsion, and three lobectomies were performed. Shakoor et al.2 reported an unusual case of left lingular torsion following single lung transplantation (SLTx) performed for idiopathic pulmonary fibrosis. The patient experienced severe ventilatory compromise immediately after leaving the operating room, and a CXR revealed a well-demarcated area of consolidation involving the left mid- and lower lung zones. Lingular torsion was promptly diagnosed and corrected surgically. Grazia et al.5 reported left lower lobe torsion in a patient who had undergone BLTx for alpha-1-antitrypsin deficiency emphysema. The patient experienced acute pulmonary hypertension and hypoxemia on postoperative day 3. The CXR showed bilateral alveolar infiltrates and new focal consolidation of the left lower lobe. Fiberoptic bronchoscopy showed complete obstruction of the left lower lobe bronchus and abnormal rotation of the left upper lobe bronchus suggesting
Predisposing factors Felson9 has highlighted five risk factors leading to torsion as a result of trauma or as a complication following thoracic surgery: (i) lobar deflation (consolidation or atelectasis); (ii) a long lobar pedicle; (iii) presence of a complete fissure; (iv) pneumothorax or pleural effusion; and (v) mobilization of the inferior pulmonary ligament. The technique of lung transplantation together with these risk factors may increase the potential mobility of the lobe within the chest cavity.5,6 There is a higher incidence of torsion of the left lung lobes compared to right lung lobes seen in the reported cases, probably because the left lung has a higher incidence of complete fissures, found in as many as 27–82% of donors,2 and the relatively long pulmonary artery and main bronchus. It has been postulated that ischemia-reperfusion injury and the consequent increase in lung water and weight may also contribute to the development of consolidation and atelectasis.5 It has also been noted that repeated checking for hemostasis and reoperation for bleeding may fail to recognize a twisted lobe.4,10 As the most common indication for lung transplantation is emphysema, the recipients usually have very
2014
2013
2009
2009
2009
2009
2009
2003
2000
1998
Our case
Lin, M. W.
Shakoor, H.
Souilamas, R
Souilamas, R
Souilamas, R
Souilamas, R
Grazia, T. J.
Gilkeson, R. C.
Collins, J.
Author
1
1
1
4
3
2
1
1
1
1
59
34
46
40
30
15
37
62
30
36
Publication Patient Age Year Number (years)
M
M
M
Not mentioned Not mentioned Not mentioned Not mentioned
Not mentioned
F
F
Gender
End stage COPD
Idiopathic pulmonary fibrosis End-stage emphysema (alpha1- antitrypsin deficiency) End-stage silicosis
Cystic fibrosis
Cystic fibrosis
Cystic fibrosis
Idiopathic pulmonary fibrosis
Bronchiolitis obliterans
Cystic fibrosis
Disease
BLTx
SLTx
BLTx
BLTx
BLTx
BLTx
BLTx
SLTx
BLTx
BLTx
POD 5
POD 3
POD 3
POD 2
POD 1
POD 4
POD 1
POD 1
POD 8
POD 11
Transplant Day of Method Diagnosis
LLL
Size Incomplete discrepancy RML, RLL torsion LLL Donor lung Complete fissure
Donor lung Complete fissure
Size RUL and discrepancy, RML Complete fissure Donor lung RML Complete fissure Small donor Left lung lingula Complete fissure Not LLL mentioned Not LLL mentioned Not LUL mentioned Not LLL mentioned
Etiology
Torsion Lobe
Complete obstruction
Partial obstruction
Complete obstruction
Complete obstruction Complete obstruction Complete obstruction No obstruction
No obstruction
Partial obstruction
Partial obstruction
Alive 221 days
Alive at eight months Alive at two years Alive at one year Alive at five years Died POD 55 due to sepsis and severe ARDS Observation Survival after six months follow-up Left lower Died 16 mths, lobectomy of other complications
Left lower lobectomy
Left lower lobectomy
Left lower lobectomy Left lower lobectomy Detorsion
Right middle Survival after lobectomy two years’ follow-up Detorsion Discharged 11 days after
Detorsion
Not mentioned
Not mentioned
Not mentioned
0.65
0.62
0.58
0.52
Not mentioned
0.56
0.83
Predicted Bronchoscopy Follow-up/ Postoperative Finding Management Outcome FEV1 (%)
TABLE 1 Previous Reported Cases of Lung Torsion after Lung Transplantation
212 STEPHENS ET AL. LOBAR TORSION J CARD SURG 2015;30:209–214
J CARD SURG 2015;30:209–214
large chest cavities. Atelectatic lobes of the donor lung may increase mobility of the graft within the larger chest cavity resulting in partial or complete torsion. Additionally, the use of double lumen tubes for decompression of the lung can potentially predispose to the development of lobar torsion that may be overlooked upon reinflation.2 Contrary to oversized pleural space, in the undersized recipient chest cavity, the graft cannot be easily evaluated or manipulated after implantation. The compromised ability to visualize the graft while repeatedly checking for hemostasis can contribute to subsequent lobar torsion. The anterolateral incision could also contribute to this poor visualization of the graft.2
Clinical presentation The clinical presentation of lung torsion is usually acute, yet the rarity of this condition commonly results in a significant delay in diagnosis. Clinical presentation may vary depending on whether the torsion is partial or complete. Partial torsion can present more insidiously with obstructive pneumonia or partial collapse of the affected lobe(s). Complete torsion usually presents acutely with chest pain, hemoptysis, hypoxemia, bronchorrhea, and persistent air leak.6
Radiographic features On plain radiograph, features suspicious for torsion are a collapsed or consolidated lobe in an unusual position, rapid opacification of the lung following surgery, marked change in position of the opacified lobe on short interval serial radiographs, or an unusual position of a collapsed lobe may suggest the diagnosis. Other radiographic findings include an abnormal pulmonary vascular pattern, often with an inverted appearance at the hilum, bronchial cut-off or distortion, lobar air-trapping, a reticular pattern from venous congestion and displacement of the hilum in an inappropriate direction relative to the atelectatic lobe involved, and a nonaerated lobe in an unusual position, with or without accompanying compensatory hyperinflation of the surrounding lung.5,9 CT findings in postoperative lobar torsion include tapered obliteration of the proximal pulmonary artery and accompanying bronchus of the involved lobe, and amorphous soft-tissue attenuation at the hilum. The lobe can show poorly enhancing consolidation with increased volume, ground-glass attenuation, interlobular septal thickening, intralobular linear attenuation, and loss of parenchymal enhancement after contrast administration. The neofissure following a lobectomy may be edematous and appear in an unusual orientation. Bronchoscopy Direct visualization of the involved segment with fiberoptic bronchoscopy may demonstrate an occluded
STEPHENS ET AL. LOBAR TORSION
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or distorted bronchus without evidence of an endobronchial lesion or mucous plug, sometimes with a ‘‘fish mouth’’ appearance. The differential diagnosis includes other early or late postlobectomy complications such as pneumonia or vascular occlusion, which have a similar clinical presentation. Treatment and prognosis Lung torsion is considered a medical emergency. The optimal therapeutic approach remains controversial. The decisions to proceed with either detorsion or lobectomy usually depends on the viability of the torted lung, which is better appreciated after thoracotomy. Detorsion offers the possibility of lung salvage but lobectomy may be required if the lobe is nonviable. Velmahos et al.11 reported a case in which acute deterioration in the oxygen saturation and blood pressure was noted after detorsion before lobectomy. They postulated that the systemic release of toxic substances produced in the ischemic lung segment caused this. These authors recommended avoiding detorsion if at all possible before isolation and occlusion of the hilar structures prior to lobectomy, when the lobe is nonviable.4,5 The diagnosis is usually possible by imaging studies such as a CXR, chest CT scan or angiography, performed for symptoms suggestive of torsion. Flexible bronchoscopy demonstrates complete cut-off of the involved lobe. In our case, the patient developed primary graft failure developing in the operating room requiring VVECMO. The patient also had a reduction in the size of the right lung following lower lobectomy for size mismatch. The right allograft also had complete fissures. All these factors may lead to torsion. Surprisingly even after extubation, this patient remained well with no respiratory compromise. We postulate this may be due to compensation from the large left lung allograft. All the reported cases illustrate surgeons and transplant physicians should be aware of this rare complication of pulmonary surgery especially lung transplantation. Diagnosing lung torsion can be challenging. A high degree of suspicion is required in the postoperative lung transplant recipient who experiences acute respiratory insufficiency. The time to diagnosis is significant because delay may lead to pulmonary gangrene and sepsis. Onset can be deceptively insidious if the torsion is partial, but is usually quite abrupt in complete torsion. Lobectomy is indicated when the lobe is nonviable. Simple detorsion may be tried even in cases of late diagnosis. We recommend checking anatomical structures carefully before thoracotomy closure, especially during a second thoracotomy for hemostasis, and when the graft has a complete fissure. Surgical techniques, such as stapling or suturing of a freely mobile lobe in the presence of a complete major or minor fissure, should be considered as measures to assist in preventing lobar torsion. Adequate exposure
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and good visualization of the fully inflated graft with appropriate orientation prior to chest closure is important. A thorough intraoperative bronchoscopy to visualize all the bronchopulmonary segments is essential if such complications are to be avoided. Although torsion can be excluded at the completion of surgery, it can still occur anytime in the postoperative period. The importance of an early postoperative chest radiograph cannot be overstated. REFERENCES 1. Lin MW, Huang SC, Kuo SW, et al: Lobar torsion after lung transplantation. J Formos Med Assoc 2013;112(2):105– 108. 2. Shakoor H, Murthy S, Mason D, et al: Lobar torsion after lung transplantation—a case report and review of the literature. Artif Organs 2009;33(7):551–554. 3. Souilamas R, Saueressig M, Boussaud V, et al: Pulmonary resection after lung transplantation in cystic fibrosis patients. Asian Cardiovasc Thorac Ann 2011;19(3–4): 202–206.
J CARD SURG 2015;30:209–214
4. Souilamas R, Couchon S, Hernigou A, et al: Management of lobar torsion following lung transplantation. Asian Cardiovasc Thorac Ann 2009;17(2):196–198. 5. Grazia TJ, Hodges TN, Cleveland JC Jr, et al: Lobar torsion complicating bilateral lung transplantation. J Heart Lung Transplant 2003;22(1):102–106. 6. Gilkeson RC, Lange P, Kirby TJ: Lung torsion after lung transplantation: Evaluation with helical CT. AJR Am J Roentgenol 2000;174(5):1341–1343. 7. Collins J, Kuhlman JE, Love RB: Acute, life-threatening complications of lung transplantation. Radiographics 1998;18(1):21–43; discussion 43–7. 8. Stern EJ, Collins J, Love RB: Pulmonary torsion: Complication of lung transplantation. Clinical Pulmonary Medicine 1996;3(5):297. 9. Felson B: Lung torsion: Radiographic findings in nine cases. Radiology 1987;162(3):631–638. 10. Oddi MA, Traugott RC, Will RJ, et al: Unrecognized intraoperative torsion of the lung. Surgery 1981;89(3):390–393. 11. Velmahos GC, Frankhouse J, Ciccolo M: Pulmonary torsion of the right upper lobe after right middle lobectomy for a stab wound to the chest. J Trauma 1998;44(5):920– 922.
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209
REVIEW ARTICLE _______________________________________________________________
Lobar Torsion Following Bilateral Lung Transplantation Georgina Stephens,* Krishna Bhagwat,* Adrian Pick,* and David McGiffin*,y *Department of Cardiothoracic Surgery, Alfred Hospital, Melbourne, Victoria, Australia; and yMonash University, Melbourne, Victoria, Australia ABSTRACT Torsion of lobes of the lung is a rare complication following lung transplantation. We present a case of counterclockwise torsion of the right lung allograft and review of nine additional cases in the literature of lobar torsion following lung transplantation. A high degree of suspicion is needed for early diagnosis with rapid surgical intervention to preserve lung viability. doi: 10.1111/jocs.12476 (J Card Surg
2015;30:209–214)
Few lobar torsions following lung transplantation have been reported in the literature.1–7 Immediate surgical correction is required to correct the torsion and reduce the associated high mortality rate. We report a case of lobar torsion following lung transplantation and review the literature on this subject.
PATIENT PROFILE A 36-year-old female underwent bilateral sequential lung transplantation (BLTx) for cystic fibrosis–related bronchiectasis. There was a size mismatch with the donor lungs being significantly larger than the patient’s thoracic cavity, necessitating a lower lobectomy of the right donor lung. The operation was complicated by primary graft failure, requiring support with veno-venous extracorporeal membranous oxygenation (VV-ECMO). The ischemic time for the left lung was four hours, and five hours and fifteen minutes for the right lung. The postoperative chest radiograph (CXR) showed bilateral congestion of the lungs, a size mismatch with a large left allograft, and resultant mediastinal shift towards the right (Fig. 1). VV-ECMO was weaned, with decannulation occurring on postoperative day 4. The patient was extubated on day 5. Her stay in the intensive care unit was complicated by multiresistant Pseudomonas bacteraemia, which was treated with Cefepime and Ciprofloxacin.
Conflict of interest: The authors acknowledge no conflict of interest in the submission. Address for correspondence: Prof. David McGiffin, Department of Cardiothoracic Surgery, Alfred Health, 55 Commercial Road, Melbourne, Victoria 3004, Australia. Fax: þ61-3-9076-2317; e-mail: [email protected]
On postoperative day 10 routine flexible bronchoscopy showed that the right bronchial orifice had the appearance of a ‘‘fish mouth’’ slit opening with congested mucosa. The bronchus could not be engaged. A computed tomography (CT) high-resolution chest and pulmonary angiogram was performed. A partial torsion of the right hilum with severe compromise to both the right upper and middle lobe bronchi was identified. The right upper lobe pulmonary vein was occluded, resulting in secondary pulmonary venous hypertension (Fig. 2). Despite these radiological findings, the patient remained hemodynamically stable and clinically well. She was started on heparin for the pulmonary vein occlusion. The patient returned to the operating room on postoperative day 11 where the right-sided thoracotomy incision was opened. The hilum of the right lung was found to be rotated approximately 908 counterclockwise as seen from the right axilla, so that the right middle lobe (RML) was orientated antero-superiorly, and the right upper lobe (RUL) posterio-inferiorly. Detorsion was performed by clockwise rotation of the lobes. Both the RML and RUL were viable with no signs of necrosis or infarction. Intraoperative transesophageal echocardiogram showed no evidence of thrombus in the right pulmonary veins and demonstrated normal pulmonary venous flow. Abrasion pleurodesis was performed, as well as suturing of the visceral pleura of the RUL to the right side of the pericardium and to the parietal pleura posteriorly. Bronchoscopy confirmed that the RML and RUL bronchi were now patent. Postoperatively, the patient was successfully extubated. A postoperative CT scan demonstrated normal orientation of the right upper and middle lobe bronchi. Eight months after the transplant, the patient had no respiratory symptoms and her FEV1 was 2.31 L (83%
210
STEPHENS ET AL. LOBAR TORSION
J CARD SURG 2015;30:209–214
Figure 1. (A) Chest X-ray before transplant; (B) chest X-ray on day 1 post–lung transplant showing allograft size mismatch; (C) chest X-ray post-detorsion; (D) chest X-ray follow-up six months later.
of predicted). Normal orientation of the right upper and middle lobe was confirmed. DISCUSSION Lobar torsion is extremely rare after lung transplantation. This complication has a reported incidence of 0.089–0.3%, occurring most frequently after lung resection.1 It has also been reported after a transthoracic needle aspiration, in association with pneumonia, pleural effusions, abdominal surgery, traumatic pneumothorax, and a diaphragmatic hernia.2 A review of the literature shows that a delay in the diagnosis and treatment of this rare complication can have catastrophic consequences such as obstructive pneumonia, hemoptysis, hypoxemia, bronchorrhea, and persistent air leak. Interestingly, not many cases of lobar torsion following lung transplantation have been
reported; furthermore, this complication may be underrecognized and underreported. Previous reported cases of lung torsion after lung transplantation are outlined in Table 1. Lin et al.1 reported a case of RML torsion after BLTx for bronchiolitis obliterans due to paraneoplastic pemphigus. The right lower lobe of the donor lung was resected due to inflammatory change found during procurement. The postoperative CXR showed persisting RML infiltrates. RML lobectomy was performed after the reconstructed chest CT confirmed the diagnosis of RML torsion. Souilamas et al.4 reported four cases of acute lobar torsion in patients who had undergone BLTx. Bronchoscopy showed complete obstruction in only two of them. Multiplanar reformats from thin slice CT scan allowed diagnosing torsion, confirming vessels and bronchi orientation, course, and strictures. At
J CARD SURG 2015;30:209–214
STEPHENS ET AL. LOBAR TORSION
211
torsion. The diagnosis was confirmed by pulmonary angiography and later at thoracotomy. The presence of a complete left major fissure was identified, and it was speculated that this played a role in the development of torsion. Gilkeson et al.6 reported a case of right middle and lower lobe torsion following right SLTx for silicosis. This case took a benign course and did not require surgical correction. Despite the radiographic and bronchoscopic appearance of lung torsion, with the finding of a patent arterial and venous blood supply a nonoperative strategy was selected. These authors noted that the donor allograft was significantly smaller than the recipient’s right hemithorax, and that this may have contributed to the development of torsion. Collins et al.7,8 reported left upper lobe torsion in a 39-year-old man who developed shortness of breath, hemoptysis, and hypoxemia three days after BLTx for chronic obstructive pulmonary disease. A CT scan demonstrated displacement of the left major fissure, cut off of the left main-stem bronchus, abnormal orientation of the left pulmonary artery, and consolidation of the displaced left upper lobe. Torsion and hemorrhagic infarction were confirmed surgically at detorsion and left upper lobectomy five days after transplantation. Pathology Torsion occurs when a lobe, or the entire lung, rotates around the hilar structures. This can be complete (1808) or partial (908), creating compromise to the airway, arterial supply, and the venous and lymphatic drainage. The affected lobe often shows hemorrhagic infarction or necrosis. Figure 2. (A) CT Scan, Sagittal View showing torsion of RUL and RML with their respective pulmonary artery distorted; (B) CT Scan, Axial View showing interlobular septal thickening with venous congestion.
operation, one detorsion, and three lobectomies were performed. Shakoor et al.2 reported an unusual case of left lingular torsion following single lung transplantation (SLTx) performed for idiopathic pulmonary fibrosis. The patient experienced severe ventilatory compromise immediately after leaving the operating room, and a CXR revealed a well-demarcated area of consolidation involving the left mid- and lower lung zones. Lingular torsion was promptly diagnosed and corrected surgically. Grazia et al.5 reported left lower lobe torsion in a patient who had undergone BLTx for alpha-1-antitrypsin deficiency emphysema. The patient experienced acute pulmonary hypertension and hypoxemia on postoperative day 3. The CXR showed bilateral alveolar infiltrates and new focal consolidation of the left lower lobe. Fiberoptic bronchoscopy showed complete obstruction of the left lower lobe bronchus and abnormal rotation of the left upper lobe bronchus suggesting
Predisposing factors Felson9 has highlighted five risk factors leading to torsion as a result of trauma or as a complication following thoracic surgery: (i) lobar deflation (consolidation or atelectasis); (ii) a long lobar pedicle; (iii) presence of a complete fissure; (iv) pneumothorax or pleural effusion; and (v) mobilization of the inferior pulmonary ligament. The technique of lung transplantation together with these risk factors may increase the potential mobility of the lobe within the chest cavity.5,6 There is a higher incidence of torsion of the left lung lobes compared to right lung lobes seen in the reported cases, probably because the left lung has a higher incidence of complete fissures, found in as many as 27–82% of donors,2 and the relatively long pulmonary artery and main bronchus. It has been postulated that ischemia-reperfusion injury and the consequent increase in lung water and weight may also contribute to the development of consolidation and atelectasis.5 It has also been noted that repeated checking for hemostasis and reoperation for bleeding may fail to recognize a twisted lobe.4,10 As the most common indication for lung transplantation is emphysema, the recipients usually have very
2014
2013
2009
2009
2009
2009
2009
2003
2000
1998
Our case
Lin, M. W.
Shakoor, H.
Souilamas, R
Souilamas, R
Souilamas, R
Souilamas, R
Grazia, T. J.
Gilkeson, R. C.
Collins, J.
Author
1
1
1
4
3
2
1
1
1
1
59
34
46
40
30
15
37
62
30
36
Publication Patient Age Year Number (years)
M
M
M
Not mentioned Not mentioned Not mentioned Not mentioned
Not mentioned
F
F
Gender
End stage COPD
Idiopathic pulmonary fibrosis End-stage emphysema (alpha1- antitrypsin deficiency) End-stage silicosis
Cystic fibrosis
Cystic fibrosis
Cystic fibrosis
Idiopathic pulmonary fibrosis
Bronchiolitis obliterans
Cystic fibrosis
Disease
BLTx
SLTx
BLTx
BLTx
BLTx
BLTx
BLTx
SLTx
BLTx
BLTx
POD 5
POD 3
POD 3
POD 2
POD 1
POD 4
POD 1
POD 1
POD 8
POD 11
Transplant Day of Method Diagnosis
LLL
Size Incomplete discrepancy RML, RLL torsion LLL Donor lung Complete fissure
Donor lung Complete fissure
Size RUL and discrepancy, RML Complete fissure Donor lung RML Complete fissure Small donor Left lung lingula Complete fissure Not LLL mentioned Not LLL mentioned Not LUL mentioned Not LLL mentioned
Etiology
Torsion Lobe
Complete obstruction
Partial obstruction
Complete obstruction
Complete obstruction Complete obstruction Complete obstruction No obstruction
No obstruction
Partial obstruction
Partial obstruction
Alive 221 days
Alive at eight months Alive at two years Alive at one year Alive at five years Died POD 55 due to sepsis and severe ARDS Observation Survival after six months follow-up Left lower Died 16 mths, lobectomy of other complications
Left lower lobectomy
Left lower lobectomy
Left lower lobectomy Left lower lobectomy Detorsion
Right middle Survival after lobectomy two years’ follow-up Detorsion Discharged 11 days after
Detorsion
Not mentioned
Not mentioned
Not mentioned
0.65
0.62
0.58
0.52
Not mentioned
0.56
0.83
Predicted Bronchoscopy Follow-up/ Postoperative Finding Management Outcome FEV1 (%)
TABLE 1 Previous Reported Cases of Lung Torsion after Lung Transplantation
212 STEPHENS ET AL. LOBAR TORSION J CARD SURG 2015;30:209–214
J CARD SURG 2015;30:209–214
large chest cavities. Atelectatic lobes of the donor lung may increase mobility of the graft within the larger chest cavity resulting in partial or complete torsion. Additionally, the use of double lumen tubes for decompression of the lung can potentially predispose to the development of lobar torsion that may be overlooked upon reinflation.2 Contrary to oversized pleural space, in the undersized recipient chest cavity, the graft cannot be easily evaluated or manipulated after implantation. The compromised ability to visualize the graft while repeatedly checking for hemostasis can contribute to subsequent lobar torsion. The anterolateral incision could also contribute to this poor visualization of the graft.2
Clinical presentation The clinical presentation of lung torsion is usually acute, yet the rarity of this condition commonly results in a significant delay in diagnosis. Clinical presentation may vary depending on whether the torsion is partial or complete. Partial torsion can present more insidiously with obstructive pneumonia or partial collapse of the affected lobe(s). Complete torsion usually presents acutely with chest pain, hemoptysis, hypoxemia, bronchorrhea, and persistent air leak.6
Radiographic features On plain radiograph, features suspicious for torsion are a collapsed or consolidated lobe in an unusual position, rapid opacification of the lung following surgery, marked change in position of the opacified lobe on short interval serial radiographs, or an unusual position of a collapsed lobe may suggest the diagnosis. Other radiographic findings include an abnormal pulmonary vascular pattern, often with an inverted appearance at the hilum, bronchial cut-off or distortion, lobar air-trapping, a reticular pattern from venous congestion and displacement of the hilum in an inappropriate direction relative to the atelectatic lobe involved, and a nonaerated lobe in an unusual position, with or without accompanying compensatory hyperinflation of the surrounding lung.5,9 CT findings in postoperative lobar torsion include tapered obliteration of the proximal pulmonary artery and accompanying bronchus of the involved lobe, and amorphous soft-tissue attenuation at the hilum. The lobe can show poorly enhancing consolidation with increased volume, ground-glass attenuation, interlobular septal thickening, intralobular linear attenuation, and loss of parenchymal enhancement after contrast administration. The neofissure following a lobectomy may be edematous and appear in an unusual orientation. Bronchoscopy Direct visualization of the involved segment with fiberoptic bronchoscopy may demonstrate an occluded
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or distorted bronchus without evidence of an endobronchial lesion or mucous plug, sometimes with a ‘‘fish mouth’’ appearance. The differential diagnosis includes other early or late postlobectomy complications such as pneumonia or vascular occlusion, which have a similar clinical presentation. Treatment and prognosis Lung torsion is considered a medical emergency. The optimal therapeutic approach remains controversial. The decisions to proceed with either detorsion or lobectomy usually depends on the viability of the torted lung, which is better appreciated after thoracotomy. Detorsion offers the possibility of lung salvage but lobectomy may be required if the lobe is nonviable. Velmahos et al.11 reported a case in which acute deterioration in the oxygen saturation and blood pressure was noted after detorsion before lobectomy. They postulated that the systemic release of toxic substances produced in the ischemic lung segment caused this. These authors recommended avoiding detorsion if at all possible before isolation and occlusion of the hilar structures prior to lobectomy, when the lobe is nonviable.4,5 The diagnosis is usually possible by imaging studies such as a CXR, chest CT scan or angiography, performed for symptoms suggestive of torsion. Flexible bronchoscopy demonstrates complete cut-off of the involved lobe. In our case, the patient developed primary graft failure developing in the operating room requiring VVECMO. The patient also had a reduction in the size of the right lung following lower lobectomy for size mismatch. The right allograft also had complete fissures. All these factors may lead to torsion. Surprisingly even after extubation, this patient remained well with no respiratory compromise. We postulate this may be due to compensation from the large left lung allograft. All the reported cases illustrate surgeons and transplant physicians should be aware of this rare complication of pulmonary surgery especially lung transplantation. Diagnosing lung torsion can be challenging. A high degree of suspicion is required in the postoperative lung transplant recipient who experiences acute respiratory insufficiency. The time to diagnosis is significant because delay may lead to pulmonary gangrene and sepsis. Onset can be deceptively insidious if the torsion is partial, but is usually quite abrupt in complete torsion. Lobectomy is indicated when the lobe is nonviable. Simple detorsion may be tried even in cases of late diagnosis. We recommend checking anatomical structures carefully before thoracotomy closure, especially during a second thoracotomy for hemostasis, and when the graft has a complete fissure. Surgical techniques, such as stapling or suturing of a freely mobile lobe in the presence of a complete major or minor fissure, should be considered as measures to assist in preventing lobar torsion. Adequate exposure
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and good visualization of the fully inflated graft with appropriate orientation prior to chest closure is important. A thorough intraoperative bronchoscopy to visualize all the bronchopulmonary segments is essential if such complications are to be avoided. Although torsion can be excluded at the completion of surgery, it can still occur anytime in the postoperative period. The importance of an early postoperative chest radiograph cannot be overstated. REFERENCES 1. Lin MW, Huang SC, Kuo SW, et al: Lobar torsion after lung transplantation. J Formos Med Assoc 2013;112(2):105– 108. 2. Shakoor H, Murthy S, Mason D, et al: Lobar torsion after lung transplantation—a case report and review of the literature. Artif Organs 2009;33(7):551–554. 3. Souilamas R, Saueressig M, Boussaud V, et al: Pulmonary resection after lung transplantation in cystic fibrosis patients. Asian Cardiovasc Thorac Ann 2011;19(3–4): 202–206.
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4. Souilamas R, Couchon S, Hernigou A, et al: Management of lobar torsion following lung transplantation. Asian Cardiovasc Thorac Ann 2009;17(2):196–198. 5. Grazia TJ, Hodges TN, Cleveland JC Jr, et al: Lobar torsion complicating bilateral lung transplantation. J Heart Lung Transplant 2003;22(1):102–106. 6. Gilkeson RC, Lange P, Kirby TJ: Lung torsion after lung transplantation: Evaluation with helical CT. AJR Am J Roentgenol 2000;174(5):1341–1343. 7. Collins J, Kuhlman JE, Love RB: Acute, life-threatening complications of lung transplantation. Radiographics 1998;18(1):21–43; discussion 43–7. 8. Stern EJ, Collins J, Love RB: Pulmonary torsion: Complication of lung transplantation. Clinical Pulmonary Medicine 1996;3(5):297. 9. Felson B: Lung torsion: Radiographic findings in nine cases. Radiology 1987;162(3):631–638. 10. Oddi MA, Traugott RC, Will RJ, et al: Unrecognized intraoperative torsion of the lung. Surgery 1981;89(3):390–393. 11. Velmahos GC, Frankhouse J, Ciccolo M: Pulmonary torsion of the right upper lobe after right middle lobectomy for a stab wound to the chest. J Trauma 1998;44(5):920– 922.
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