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Plication of the Diaphragm in Postpneumectomy Right-to-Left Shunting
Plication of the Diaphragm in Postpneumectomy Right-to-Left Shunting Joyce-Manyi Bakia, MS, Jeroen M. A. M. Retera, MD, Roland P. van Ieperen, MD, Robbert J. de Winter, MD, and Joël van der Niet, MD Departments of Surgery, Pulmonology, and Anaesthesiology, Elisabeth-Tweesteden Hospital, Tilburg, Department of Cardiology, Amsterdam Medical Center, Amsterdam, and Department of Surgery, Elisabeth-Tweesteden Hospital, Tilburg, The Netherlands
A rare adverse event of a right-sided pneumectomy with an elevated hemidiaphragm is right-to-left shunting through a patent foramen ovale. In this case report we describe our experience with plication of the right hemidiaphragm with instantaneous hemodynamic results and pain relief, followed by secondary closure of the foramen ovale. (Ann Thorac Surg 2017;104:e181–3) Ó 2017 by The Society of Thoracic Surgeons
P
neumectomies account for grossly 20% of all lung resections for cancer. The hemodynamic consequences are rare and mostly short term. However, certain adverse events can arise up to years after the procedure. One of the more rarely described adverse events after this procedure is right-to-left shunting through a patent foramen ovale (PFO), causing dyspnea. This usually arises in the presence of an elevated right hemidiaphragm, often related to phrenic nerve injury. Most reports detailing this adverse event have described primary closure of the PFO to treat the patient [1–3]. A single case series has been published describing plication of the diaphragm as a treatment of right-to-left shunting after a right pneumectomy, without percutaneous closure of the PFO [4]. In this case report we describe our experience with a patient with right-to-left shunting through the foramen ovale who underwent plication of the right hemidiaphragm with instant hemodynamic results, with secondary closure of the foramen ovale.
lobectomy. Fibrosis of the lung at the right hilum resulted in conversion to a pneumectomy. The phrenic nerve could not be spared. After initial recovery, the patient experienced progressive exertional dyspnea 3 months after the procedure. The patient also experienced rightsided dull abdominal pain. A CT scan showed an elevation of the right hemidiaphragm with compression of the right ventricle and atrium (Fig 1). The result of a hyperoxia test suggested the presence of right-to-left shunting with a calculated shunt of 22%, not present before the pneumectomy. With normal atrial pressures during initial cardiac ultrasonography, it was proved only during subsequent ultrasonography with intravenous contrast medium that there was a large PFO. Our hypothesis was that by alleviating the pressure on the right ventricle and atrium, the PFO would spontaneously close. A right-sided thoracotomy was performed with plication of the right hemidiaphragm. An instant effect was observed. Without a change in other perioperative circumstances, the patient’s oxygen requirement dropped from 60% to 40%. The postoperative position of the diaphragm is shown in Figure 2. The patient made a swift postoperative recovery. The exertional dyspnea she reported preoperatively was greatly reduced. The abdominal pain was no longer present. One month postoperatively, cardiac ultrasonography with intravenous contrast medium showed reduced shunting. In a bid to further decrease shunting, the patient underwent uncomplicated percutaneous closure of the PFO under transesophageal echocardiographic guidance. At a follow-up visit 6 months after operation and 4 months after closure of the PFO, the calculated shunting with a hyperoxia test was reduced to
A 52-year old woman experienced dyspnea after a rightsided pneumectomy. Two years earlier the patient had received a diagnosis of right-sided T2N2M0 squamous cell lung carcinoma. She underwent combined chemotherapy and radiotherapy. Cancer remission was achieved until September 2014, when a local recurrence was diagnosed on a computed tomography (CT) scan. After multidisciplinary review, the patient’s good clinical condition led to a departure from protocol. The decision was made to strive for a curative resection with a
Accepted for publication Feb 28, 2017. Address correspondence to Dr Bakia, Radboudumc, Department of Surgery, Geert Grooteplein Zuid 10a, 6525 GA Nijmegen, NL; email: [email protected].
Ó 2017 by The Society of Thoracic Surgeons Published by Elsevier Inc.
Fig 1. Coronal view of the thorax before plication of the right hemidiaphragm. 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2017.02.080
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CASE REPORT BAKIA ET AL DIAPHRAGM PLICATION IN RIGHT-TO-LEFT SHUNT
Ann Thorac Surg 2017;104:e181–3
Fig 2. Roentgenogram of the thorax after plication of the right hemidiaphragm.
11%. Unfortunately, a CT scan revealed a new lesion in the right kidney, histologically confirmed to be a metastasis. The patient is now being treated with a protein kinase inhibitor.
Comment In this case we demonstrate our experience with plication of the diaphragm as a technique in a patient with a PFO in the presence of an elevated hemidiaphragm after a right-sided pneumectomy with phrenic nerve injury. This case serves to illustrate that by relieving the mechanical pressure on the right atrium and ventricle by plication of the diaphragm, improvement of a hemodynamically significant right-left shunt was achieved. In contrast with the majority of previous reports, this did not require simultaneous closure of the reopened foramen ovale [1, 3]. It is noteworthy that the plication provided immediate results. In our patient, symptoms developed 3 months after the initial procedure. Several case series have demonstrated a large variety in the onset of symptoms (1 day to 10 years) [1, 4, 5]. This might be caused by intrathoracic remodeling over the years and by low awareness of these potential long-term developments, leading to a delay in diagnosis. In a study describing patients with pulmonary hypertension, as many as 45% of study patients with a PFO had right-to-left shunting [6]. As hypothesized by Welvaart et al [4], the increased pressure induced by an elevated right hemidiaphragm might lead to similar sequelae. This suggests that in a subset of the population, there is a functional rather than an anatomic closure of the foramen ovale. The hypothesis is that mediastinal contortion with
Fig 3. Simplified depiction of the reopening of the foramen ovale after intrathoracic remodeling.
a shift of the right atrium, with a concurrent fixed position of the inferior vena cava, resulted in increased pressure. This increased pressure then led to the reopening of the (functionally closed) foramen ovale (Fig 3) [7]. The elevated diaphragm, both through phrenic nerve injury and volume loss, was deemed to be the cause of this intrathoracic remodeling, leading to eventual reopening of the foramen ovale. That is why we chose treatment of the diaphragm itself, instead of primary percutaneous closure of the PFO. Moreover, the elevation of the diaphragm also had the potential to cause circulatory problems (such as decreased filling pressures of the right atrium), again prompting primary plication of the diaphragm. In contrast to previous reports, the PFO could not be reversed by plication of the diaphragm alone [4]. The plication of the diaphragm did seem to be sufficient to let the heart return to its natural position (Fig 2). Why the PFO could not be fully reversed with plication alone is unknown. In conclusion, right-to-left shunting through a PFO is a relatively rare adverse event that can arise secondary to increased mechanical pressure resulting from an elevated right hemidiaphragm after phrenic nerve injury and volume loss after a pneumectomy. Plication of the diaphragm can give instant hemodynamic results with concurrent relief of respiratory symptoms and, in our patient, of abdominal pain. However, reversal of the shunting is not always possible with isolated plication of the diaphragm. The authors wish to thank Milo van der Maaden for creating Figure 3.
Ann Thorac Surg 2017;104:e181–3
References 1. Ghamande R, Ramsey R, Rhodes JF, Stoller JK. Right hemidiaphragmatic elevation with a right-to-left shunt through a patent foramen ovale: a case report and literature review. Chest 2001;120:2094–6. 2. Murray KD, Kalanges LK, Weiland JE, et al. Platypneaorthodeoxia: an unusual indication for surgical closure of a patent foramen ovale. J Card Surg 1991;6:62–7. 3. Perkins LA, Costa SM, Boethel CD, Lawrence ME. Hypoxemia secondary to right-to-left interatrial shunt through a patent foramen ovale in a patient with elevated right hemidiaphragm. Respir Care 2008;53:462–5.
CASE REPORT BAKIA ET AL DIAPHRAGM PLICATION IN RIGHT-TO-LEFT SHUNT
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4. Welvaart WN, Becker A, Marcus JT, Vonk-Noordegraaf A, Oosterhuis JW, Paul MA. Unusual treatment of patent foramen ovale after pneumonectomy. Eur Respir J 2010;35:929–30. 5. Aigner C, Lang G, Taghavi S, et al. Haemodynamic complications after pneumonectomy: atrial inflow obstruction and reopening of the foramen ovale. Eur J Cardiothorac Surg 2008;33:268–71. 6. Sun XG, Hansen JE, Oudiz RJ, Wasserman K. Gas exchange detection of exercise-induced right-to-left shunt in patients with primary pulmonary hypertension. Circulation 2002;105:54–60. 7. Zanchetta M, Rigatelli G, Ho SY. A mystery featuring right-toleft shunting despite normal intracardiac pressure. Chest 2005;128:998–1002.
A rare adverse event of a right-sided pneumectomy with an elevated hemidiaphragm is right-to-left shunting through a patent foramen ovale. In this case report we describe our experience with plication of the right hemidiaphragm with instantaneous hemodynamic results and pain relief, followed by secondary closure of the foramen ovale. (Ann Thorac Surg 2017;104:e181–3) Ó 2017 by The Society of Thoracic Surgeons
P
neumectomies account for grossly 20% of all lung resections for cancer. The hemodynamic consequences are rare and mostly short term. However, certain adverse events can arise up to years after the procedure. One of the more rarely described adverse events after this procedure is right-to-left shunting through a patent foramen ovale (PFO), causing dyspnea. This usually arises in the presence of an elevated right hemidiaphragm, often related to phrenic nerve injury. Most reports detailing this adverse event have described primary closure of the PFO to treat the patient [1–3]. A single case series has been published describing plication of the diaphragm as a treatment of right-to-left shunting after a right pneumectomy, without percutaneous closure of the PFO [4]. In this case report we describe our experience with a patient with right-to-left shunting through the foramen ovale who underwent plication of the right hemidiaphragm with instant hemodynamic results, with secondary closure of the foramen ovale.
lobectomy. Fibrosis of the lung at the right hilum resulted in conversion to a pneumectomy. The phrenic nerve could not be spared. After initial recovery, the patient experienced progressive exertional dyspnea 3 months after the procedure. The patient also experienced rightsided dull abdominal pain. A CT scan showed an elevation of the right hemidiaphragm with compression of the right ventricle and atrium (Fig 1). The result of a hyperoxia test suggested the presence of right-to-left shunting with a calculated shunt of 22%, not present before the pneumectomy. With normal atrial pressures during initial cardiac ultrasonography, it was proved only during subsequent ultrasonography with intravenous contrast medium that there was a large PFO. Our hypothesis was that by alleviating the pressure on the right ventricle and atrium, the PFO would spontaneously close. A right-sided thoracotomy was performed with plication of the right hemidiaphragm. An instant effect was observed. Without a change in other perioperative circumstances, the patient’s oxygen requirement dropped from 60% to 40%. The postoperative position of the diaphragm is shown in Figure 2. The patient made a swift postoperative recovery. The exertional dyspnea she reported preoperatively was greatly reduced. The abdominal pain was no longer present. One month postoperatively, cardiac ultrasonography with intravenous contrast medium showed reduced shunting. In a bid to further decrease shunting, the patient underwent uncomplicated percutaneous closure of the PFO under transesophageal echocardiographic guidance. At a follow-up visit 6 months after operation and 4 months after closure of the PFO, the calculated shunting with a hyperoxia test was reduced to
A 52-year old woman experienced dyspnea after a rightsided pneumectomy. Two years earlier the patient had received a diagnosis of right-sided T2N2M0 squamous cell lung carcinoma. She underwent combined chemotherapy and radiotherapy. Cancer remission was achieved until September 2014, when a local recurrence was diagnosed on a computed tomography (CT) scan. After multidisciplinary review, the patient’s good clinical condition led to a departure from protocol. The decision was made to strive for a curative resection with a
Accepted for publication Feb 28, 2017. Address correspondence to Dr Bakia, Radboudumc, Department of Surgery, Geert Grooteplein Zuid 10a, 6525 GA Nijmegen, NL; email: [email protected].
Ó 2017 by The Society of Thoracic Surgeons Published by Elsevier Inc.
Fig 1. Coronal view of the thorax before plication of the right hemidiaphragm. 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2017.02.080
e182
CASE REPORT BAKIA ET AL DIAPHRAGM PLICATION IN RIGHT-TO-LEFT SHUNT
Ann Thorac Surg 2017;104:e181–3
Fig 2. Roentgenogram of the thorax after plication of the right hemidiaphragm.
11%. Unfortunately, a CT scan revealed a new lesion in the right kidney, histologically confirmed to be a metastasis. The patient is now being treated with a protein kinase inhibitor.
Comment In this case we demonstrate our experience with plication of the diaphragm as a technique in a patient with a PFO in the presence of an elevated hemidiaphragm after a right-sided pneumectomy with phrenic nerve injury. This case serves to illustrate that by relieving the mechanical pressure on the right atrium and ventricle by plication of the diaphragm, improvement of a hemodynamically significant right-left shunt was achieved. In contrast with the majority of previous reports, this did not require simultaneous closure of the reopened foramen ovale [1, 3]. It is noteworthy that the plication provided immediate results. In our patient, symptoms developed 3 months after the initial procedure. Several case series have demonstrated a large variety in the onset of symptoms (1 day to 10 years) [1, 4, 5]. This might be caused by intrathoracic remodeling over the years and by low awareness of these potential long-term developments, leading to a delay in diagnosis. In a study describing patients with pulmonary hypertension, as many as 45% of study patients with a PFO had right-to-left shunting [6]. As hypothesized by Welvaart et al [4], the increased pressure induced by an elevated right hemidiaphragm might lead to similar sequelae. This suggests that in a subset of the population, there is a functional rather than an anatomic closure of the foramen ovale. The hypothesis is that mediastinal contortion with
Fig 3. Simplified depiction of the reopening of the foramen ovale after intrathoracic remodeling.
a shift of the right atrium, with a concurrent fixed position of the inferior vena cava, resulted in increased pressure. This increased pressure then led to the reopening of the (functionally closed) foramen ovale (Fig 3) [7]. The elevated diaphragm, both through phrenic nerve injury and volume loss, was deemed to be the cause of this intrathoracic remodeling, leading to eventual reopening of the foramen ovale. That is why we chose treatment of the diaphragm itself, instead of primary percutaneous closure of the PFO. Moreover, the elevation of the diaphragm also had the potential to cause circulatory problems (such as decreased filling pressures of the right atrium), again prompting primary plication of the diaphragm. In contrast to previous reports, the PFO could not be reversed by plication of the diaphragm alone [4]. The plication of the diaphragm did seem to be sufficient to let the heart return to its natural position (Fig 2). Why the PFO could not be fully reversed with plication alone is unknown. In conclusion, right-to-left shunting through a PFO is a relatively rare adverse event that can arise secondary to increased mechanical pressure resulting from an elevated right hemidiaphragm after phrenic nerve injury and volume loss after a pneumectomy. Plication of the diaphragm can give instant hemodynamic results with concurrent relief of respiratory symptoms and, in our patient, of abdominal pain. However, reversal of the shunting is not always possible with isolated plication of the diaphragm. The authors wish to thank Milo van der Maaden for creating Figure 3.
Ann Thorac Surg 2017;104:e181–3
References 1. Ghamande R, Ramsey R, Rhodes JF, Stoller JK. Right hemidiaphragmatic elevation with a right-to-left shunt through a patent foramen ovale: a case report and literature review. Chest 2001;120:2094–6. 2. Murray KD, Kalanges LK, Weiland JE, et al. Platypneaorthodeoxia: an unusual indication for surgical closure of a patent foramen ovale. J Card Surg 1991;6:62–7. 3. Perkins LA, Costa SM, Boethel CD, Lawrence ME. Hypoxemia secondary to right-to-left interatrial shunt through a patent foramen ovale in a patient with elevated right hemidiaphragm. Respir Care 2008;53:462–5.
CASE REPORT BAKIA ET AL DIAPHRAGM PLICATION IN RIGHT-TO-LEFT SHUNT
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4. Welvaart WN, Becker A, Marcus JT, Vonk-Noordegraaf A, Oosterhuis JW, Paul MA. Unusual treatment of patent foramen ovale after pneumonectomy. Eur Respir J 2010;35:929–30. 5. Aigner C, Lang G, Taghavi S, et al. Haemodynamic complications after pneumonectomy: atrial inflow obstruction and reopening of the foramen ovale. Eur J Cardiothorac Surg 2008;33:268–71. 6. Sun XG, Hansen JE, Oudiz RJ, Wasserman K. Gas exchange detection of exercise-induced right-to-left shunt in patients with primary pulmonary hypertension. Circulation 2002;105:54–60. 7. Zanchetta M, Rigatelli G, Ho SY. A mystery featuring right-toleft shunting despite normal intracardiac pressure. Chest 2005;128:998–1002.