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J Thorac Cardiovasc Surg 2006;132:1225-1226
© 2006 The American Association for Thoracic Surgery

Brief Communication

Platypnea–orthodeoxia syndrome combined with constrictive pericarditis after coronary artery bypass surgery

Masaki Hashimoto, MD, PhD*, Yasuhide Okawa, MD, Hiroshi Baba, MD, Yoshiyuki Nishimura, MD, Masakazu Aoki, MD

Division of Cardiovascular Surgery, Toyohashi Heart Center, Toyohashi, Aichi, Japan.

Received for publication May 24, 2006; revisions received June 21, 2006; accepted for publication July 12, 2006.

* Address for reprints: Masaki Hashimoto, MD, PhD, Division of Cardiovascular Surgery, Toyohashi Heart Center, 21-1 Gobudori, Oyama-cho, Toyohashi, 4418530, Japan. (Email: hashimoto{at}heart-center.or.jp).


Figure 1
Dr Hashimoto


Clinical Summary

A 61-year-old man presented with dyspnea 3 years after coronary artery bypass surgery. He had multiple cerebral infarctions 1 year after the operation. Because pulse oximetry was 92% in room air at his physician's consultation, he started home oxygen therapy. After 3 years, his dyspnea got worse. Redo work-up, including ventilation/perfusion (V/Q) scanning and computed tomographic scanning, was done. The result of the V/Q scan was normal. Computed tomographic scanning showed an abnormal cyst above the heart. The cyst contained liquid and showed a compressed right atrium and right ventricular outflow (Figure 1). He consulted with our institution to perform an extensive work-up.


Figure 1
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  		Figure 1. Preoperative computed tomography revealed an abnormal cyst above the heart. The cyst compressed the right atrium and right ventricular outflow.

 
Physical examination revealed a regular pulse, a blood pressure of 103/81 mm Hg, and no murmur in cardiac auscultation. Arterial blood gas analysis in a sitting posture showed a PaO 2 of 53.8 mm Hg and a PaCO 2 of 24.6 mm Hg with 15 L of oxygen supplementation by means of a face mask and a PaO 2 of 63.2 mm Hg and a PaCO 2 of 25.1 mm Hg in the supine position. Common blood count showed polycythemia, with a hematocrit value of 52.4%. Chest radiographic film showed any apparent pulmonary disease that could be a cause of the dyspnea.

Electrocardiography revealed sinus rhythm and left atrial load. Echocardiography was performed and revealed right atrial compression. Transesophageal echocardiography revealed an atrial septal aneurysm and patent foramen ovale and a right-to-left shunt through the foramen ovale. These shunts increased in a sitting posture.

Cardiac catheterization was performed and revealed a small right-to-left shunt (27.3%). A small portion of blood flowed from the superior vena cava influx directly into the inferior vena cava. Blood flowed from the inferior vena cava influx into the left atrium through the foramen ovale. The previous bypass graft (ie, the left internal thoracic artery to the left anterior descending artery) was patent. All of the right-sided intracardiac pressure measurements were normal (Table 1).


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  		TABLE 1. Cardiac catheterization parameters (supine)
 
Surgical removal of the cyst was performed. There were polytetrafluoroethylene* sheets surrounding the heart. There was a cyst surrounding the right atrium from the right side of the left atrium to the right ventricle. The cyst had a thick scar tissue and contained 300 mL of old, black serous liquid. Just after opening the cyst, the pulse oximetry improved, from 79% to 100%. The right atriotomy was performed during ventricular fibrillation with cardiopulmonary bypass. There was a patent foramen ovale, and the slit was closed in a continuous fashion. Because the right ventricle was covered with thick pericardium and expressed constrictive pericarditis, a complete pericardiectomy was performed.

The postoperative course was uneventful, and weaning from mechanical ventilation was quite easy. The patient could be withdrawn from oxygen supplementation and was discharged on postoperative day 15.

Discussion

Platypnea-orthodeoxia syndrome is infrequent, and the diagnosis can be difficult. Cheng1Go points out that two conditions must coexist to cause this syndrome. One is an anatomic component in the form of an interarterial communication, and the other is a functional component that produces a deformity in the atrial septal defect. In this case the former component is patent foramen ovale and the latter is the cyst above the heart and constrictive pericarditis. Constrictive pericarditis and the cyst above the heart can be a functional component of this syndrome but are rare in the literature.2,3Go In this case two functional components coexisted, and it was of concern in regard to selection of treatments. To close the shunt, some reports recommend transcatheter occlusion because it is less invasive.1Go In this case constrictive pericarditis might also contribute to the cause of this symptom, and therefore pericardiectomy was needed simultaneously. In addition, the pathology of the cyst was not unknown before treatment. These factors let us choose a surgical repair. The cyst was formed from the hematoma between a polytetrafluoroethylene sheet and the pericardium. One possible explanation why our patient presented with this symptom is that distortion of the right atrium and right ventricle by the cyst occurred, and as constrictive pericarditis was getting worse, he required high filling pressure to maintain an adequate cardiac output.4Go With the patient in the upright position, the right ventricular preload diminished, resulting in decreased right-sided cardiac output. As a result, there would be an increase in V/Q mismatch in zone I pulmonary circulation and increased dyspnea.

Although there is a high incidence of patent foramen ovale in the general population (ie, autopsy studies indicate an incidence of 27%), this kind of complication after surgical intervention is occurring more frequently but is not being recognized because of a lack of awareness.5Go The patient has a history of multiple cerebral infarctions and the characteristic pattern of occurrence of dyspnea, and these suggested this syndrome. It is important to note the pattern of occurrence of dyspnea for accurate diagnosis.

Footnotes

* Gore Preclude, registered trademark of W. L. Gore & Associates, Inc, Newark, Del. Back

References

  1. Cheng TO. Platypnea-orthodeoxia syndrome: etiology, differential diagnosis, and management. Catheter Cardiovasc Interv 1999;47:64-66.[Medline]
  2. Mashman WE, Silverman ME. Platypnea related to constrictive pericarditis. Chest 1994;105:636-637.[Abstract/Free Full Text]
  3. Miller DC, Ricks W, Oyer PE, Cipriano PR, Shumway NE. Localized tamponade of the right atrium and right ventricle: Induction of intracardiac right-to-left shunting after the use of a Gott shunt. Arch Surg 1978;113:764-766.[Abstract/Free Full Text]
  4. Zanchetta M, Rigatelli G, Ho YS. A mystery featuring right-to-left shunting despite normal intracardiac pressure. Chest 2005;128:998-1002.[Abstract/Free Full Text]
  5. Hussain SF, Mekan SF. Platypnea-orthodeoxia: report of two cases and review of the literature. South Med J 2004;97:657-662.[Medline]



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