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Platypnoea–orthodeoxia syndrome due to deformation of the patent foramen ovale caused by a dilated ascending aorta: a case report

Platypnoea–orthodeoxia syndrome due to deformation of the patent foramen ovale caused by a... Downloaded from https://academic.oup.com/ehjcr/advance-article-abstract/doi/10.1093/ehjcr/ytaa045/5770869 by guest on 03 March 2020 CASE REPORT European Heart Journal - Case Reports doi:10.1093/ehjcr/ytaa045 Heart failure Platypnoea–orthodeoxia syndrome due to deformation of the patent foramen ovale caused by a dilated ascending aorta: a case report Misaki Hasegawa, Tomoo Nagai *, Tsutomu Murakami, and Yuji Ikari Division of Cardiovascular Medicine, Department of Internal Medicine, Tokai University School of Medicine, Shimokasuya 143, Isehara-shi, Kanagawa 259-1193, Japan Received 9 October 2019; first decision 1 November 2019; accepted 28 January 2020 Background Platypnoea–orthodeoxia syndrome (POS) is characterized by dyspnoea and arterial desaturation in the sitting pos- ition. Although its pathophysiology is complex and still needed to be investigated, the disease is one of the clinical situations which should be immediately and adequately managed by health care workers from the initial presentation. ................................................................................................................................................................................................... Case summary A 66-year-old woman with a history of systemic lupus erythematosus, deep vein thrombosis, and lumbar compres- sion fracture was admitted for evaluation of the sudden onset of dyspnoea, while in the sitting position that was relieved on placing her in the supine position. Her transoesophageal echocardiogram did reveal a deformity in the patent foramen ovale (PFO) structure with a wide gap due to aortic compression, which was markedly different from that observed in the supine position, along with massive right-to-left shunting caused by redirected venous re- turn due to a persistent Eustachian valve. With the computed tomography and angiograms, POS was diagnosed. Then, the patient received aortic replacement and patch closure of PFO, and her symptoms were completely resolved. ................................................................................................................................................................................................... Discussion Platypnoea–orthodeoxia syndrome is a condition with quite unique features and needs multiple clinical measures for the diagnosis and medical management. For all health care workers, it is essential to have a high suspicion in order to detect POS in patients with unexplained dyspnoea. Echocardiography plays a major role in establishing the diagnosis and offering the choice of therapeutic options. Keywords Case report Echocardiography Patent foramen ovale Orthodeoxia � � � Learning points To show typical pathophysiology of platypnoea–orthodeoxia syndrome (POS) in the elderly. To highlight the role of echocardiography to detect and to manage POS. * Corresponding author. Tel: þ81 463 93 1121, Fax: þ81 463 93 6679, Email: nagait@tokai.ac.jp Handling Editor: Christoph Sinning Peer-reviewers: Cemil Izgi, Richard Alexander Brown and Georgia Daniel Compliance Editor: Carlos Minguito Carazo Supplementary Material Editor: Peregrine Green V The Author(s) 2020. Published by Oxford University Press on behalf of the European Society of Cardiology. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/ehjcr/advance-article-abstract/doi/10.1093/ehjcr/ytaa045/5770869 by guest on 03 March 2020 2 M. Hasegawa et al. upper mediastinum. However, in the sitting position, her blood gas Introduction analysis revealed marked hypoxaemia with low partial pressure of ar- Platypnoea–orthodeoxia syndrome (POS), which is characterized by . terial oxygen (PaO ) and oxygen saturation (54.1 mmHg and 75%, re- dyspnoea and arterial desaturation in the sitting position, was . spectively) in room air, which quickly recovered to the normoxic believed to be a rare clinical entity. The proposed pathophysiology . status with normal PaO and oxygen saturation (102.6 mmHg and is complex and involves the following two components: an anatomic- . 100%, respectively) in the supine position. While transthoracic echo- al component, such as an interatrial communication, and a secondary cardiogram showed preserved left ventricular ejection fraction (67%) or functional component that results in a deformation in the atrial and failed to detect any specific findings, her transoesophageal echo- 2 . septum with redirection of shunt flow in the upright posture. . cardiogram (TOE) revealed a patent foramen ovale (PFO) with a positive microbubble test but without obvious right-to-left shunting in the supine position (Figure 1A, Supplementary material online, Timeline Videos S1 and S2). In the sitting position, TOE further revealed a de- formity in the structure of the PFO with a wide gap due to aortic compression that was markedly different from that observed in the supine position. Massive right-to-left shunt flow caused by redirected Date Events ................................................................................................. venous return could be observed due to the persistent Eustachian 2001 Diagnosis of systemic lupus erythematosus . valve (Figure 1B and Supplementary material online, Video S3). June 2018 Admission to the prior medical institution due to . Therefore, POS was diagnosed. Latterly, the maximum diameter of bone fracture of the pelvis the ascending aorta was measured to be 57 mm on contrast- Progression of orthostatic dyspnoea enhanced computed tomography (Figure 1C). The elongated ascend- 2 July 2018 Transferred to our institution ing aorta expanded to pressurize the right atrium. Finally, the pres- Electrocardiogram, chest X-ray, and transthoracic ence of an intra-atrial shunt was confirmed on the right atrial echocardiography angiogram (Supplementary material online, Video S4). After the heart Not remarkable team conference, she received surgical prosthetic vascular graft re- 3 July 2018 Computed tomography placement of the ascending aorta and patch closure of PFO at the Ascending aorta dilation interatrial septum under cardiopulmonary bypass. Immediately after 4 July 2018 Comprehensive right heart catheterization including the surgery, her blood gas analysis normalized even in the sitting pos- right atrial angiogram ition, and her symptoms disappeared completely. A post-operative Existence of the right to left shunt at the level of TOE revealed no residual intracardiac shunt in the sitting position atriums . (Figure 1D and Supplementary material online, Video S5). She was dis- 6 July 2018 Transoesophageal echocardiography . charged without any events, and no recurrence occurred up to the De novo demonstration of the augmented right to . present. left shunt through patent foramen ovale (PFO) by sitting position 12 July 2018 Surgical repair of the PFO and resection of ascend- Discussion ing aortic aneurysm . . In this patient, the anatomical component was the PFO, and the sec- No relapse of the symptoms . . ondary component included both cardiac factors, such as the exist- 17 July 2018 Transoesophageal echocardiography . . ence of a persistent Eustachian valve, and vascular factors such as a No residual intracardiac shunt . . 3 . dilated aortic root. Agrawal et al. conducted a full review of the lit- 30 July 2018 Discharge . . erature from 1949 to 2016 regarding POS and reported PFO as the most common anatomical component. Since PFO was detected in 27% of the autopsy cases of the general population, it can be consid- ered a relatively common condition. Recently, several case reports Case presentation have documented the contribution of aortic atherosclerosis, which led to elongation of the ascending aorta or aortic root dilation as the A 66-year-old woman with a history of systemic lupus erythemato- 4–7 secondary component of POS in the elderly population. As the sus, deep vein thrombosis, and lumbar compression fracture was rate of atherosclerosis that induces aortic elongation or aortic root admitted for the evaluation of sudden onset dyspnoea, while in the . dilatation increases in the population annually, the combination of sitting position that was relieved when lying supine. She had become . congenital and acquired pathologies (PFO and aortic compression) frail as a result, having spent most of the day lying in bed due to the may now be considered one of the typical features of POS. above-mentioned symptoms. As long as she was recumbent, her con- For senior patients who are complicated due to multiple health dition was stable, and her physical examination did not reveal any car- . problems and who are otherwise prone to have a greater need for diopulmonary signs. Her general laboratory results were within the . bed rest, establishing the diagnosis of POS can be challenging. These normal limits, except for a relatively high brain natriuretic peptide . patients sometimes cannot explain or easily express their pattern of value which was 35.4 pg/mL (0–18.4 pg/mL). The electrocardiogram . symptoms, and extensive use of laboratory and diagnostic imaging was normal, although her chest X-ray revealed mild dilatation of the modalities is required to rule out several similar health conditions. Downloaded from https://academic.oup.com/ehjcr/advance-article-abstract/doi/10.1093/ehjcr/ytaa045/5770869 by guest on 03 March 2020 POS due to deformation of the PFO caused by a dilated AAo 3 Figure 1 (A) Transoesophageal echocardiogram with colour Doppler recorded with the patient in the supine position shows the existence of the patent foramen ovale without shunt flow. (B) Transoesophageal echocardiogram with colour Doppler recorded with the patient in the sitting pos- ition shows the wide patent foramen ovale with massive right-to-left shunt flow induced by the deformation. An arrow indicates the right-to-left shunt flow. (C) A computed tomography demonstrated that the maximum diameter of the ascending aorta was 57 mm. (D) A post-operative trans- oesophageal echocardiography revealed no residual intracardiac shunt in the sitting position. AAo, ascending aorta; LA, left atrium; RA, right atrium. Measuring arterial blood gases in different positions and obtaining the . Lead author biography expected results from positional change shall be the initial step for . Dr Misaki Hasegawa was born in the successful diagnosis of POS. Subsequently, contrast-enhanced Yokkaichi-shi, Japan in 1988. She transthoracic echocardiogram or TOE will confirm the existence of . received the MD degree from Tokai right-to-left intracardiac shunt and uncover the mechanism. Once . University School of Medicine the diagnosis is established, therapeutic options are usually deployed (Isehara-shi, Japan) in 2015. Since because surgical or percutaneous treatments have been already . 8 . 2018, she has been enrolled in the established. And, depending upon the patient’s overall health condi- . PhD programme of cardiovascular tion, the symptoms are then usually relieved. medicine (Tokai University Graduate School of Medicine, Isehara-shi, . Japan). Her research interest is main- Conclusion ly focused on cardiac imaging in cor- POS is a condition with quite unique features and needs multiple clin- . onary artery diseases and heart failure. ical measures for the diagnosis and medical management. Among . them, echocardiography plays a major role in establishing the diagno- sis and offering the choice of therapeutic options. As the number of Supplementary material POS cases is growing in the elderly population, POS may be one of Supplementary material is available at European Heart Journal - Case the emerging health risks of ageing in Western societies such as Reports online. Europe, the UK, the USA, and Japan. For physicians, it is essential to . have a high suspicion in order to detect POS in patients with unex- . Slide sets: A fully edited slide set detailing this case and suitable for plained dyspnoea, especially in the elderly population. local presentation is available online as Supplementary data. Downloaded from https://academic.oup.com/ehjcr/advance-article-abstract/doi/10.1093/ehjcr/ytaa045/5770869 by guest on 03 March 2020 4 M. Hasegawa et al. 3. Agrawal A, Palkar A, Talwar A. The multiple dimensions of platypnea-orthodeoxia Consent: The author/s confirm that written consent for submis- syndrome: a review. Respir Med 2017;129:31–38. sion and publication of this case report including image(s) and 4. Hagen PT, Scholz DG, Edwards WD. Incidence and size of patent foramen ovale associated text has been obtained from the patient in line with . during the first 10 decades of life: an autopsy study of 965 normal hearts. Mayo Clin Proc 1984;59:17–20. COPE guidance. 5. Medina A, de Lezo JS, Caballero E, Ortega JR. Platypnea-orthodeoxia due to aortic elongation. Circulation 2001;104:741. Conflict of interest: The authors declare no conflict of interest. 6. Shiraishi Y, Hakuno D, Isoda K, Miyazaki K, Adachi T. Platypnea-orthodeoxia syn- drome due to PFO and aortic dilation. JACC Cardiovasc Imaging 2012;5:570–571. References . 7. Chopard R, Meneveau N. Right-to-left atrial shunting associated with aortic root 1. Cheng TO. Platypnea-orthodeoxia syndrome: etiology, differential diagnosis, and aneurysm: a case report of a rare cause of platypnea-orthodeoxia syndrome. management. Catheter Cardiovasc Interv 1999;47:64–66. Heart Lung Circ 2013;22:71–75. 2. Cheng TO. Mechanisms of platypnea-orthodeoxia: what causes water to flow up- 8. Tobis JM, Abudayyeh I. Platypnea-orthodeoxia syndrome: an overlooked cause of hill? Circulation 2002;105:e47. . hypoxemia. JACC Cardiovasc Interv 2016;9:1939–1940. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png European Heart Journal - Case Reports Oxford University Press

Platypnoea–orthodeoxia syndrome due to deformation of the patent foramen ovale caused by a dilated ascending aorta: a case report

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Abstract

Downloaded from https://academic.oup.com/ehjcr/advance-article-abstract/doi/10.1093/ehjcr/ytaa045/5770869 by guest on 03 March 2020 CASE REPORT European Heart Journal - Case Reports doi:10.1093/ehjcr/ytaa045 Heart failure Platypnoea–orthodeoxia syndrome due to deformation of the patent foramen ovale caused by a dilated ascending aorta: a case report Misaki Hasegawa, Tomoo Nagai *, Tsutomu Murakami, and Yuji Ikari Division of Cardiovascular Medicine, Department of Internal Medicine, Tokai University School of Medicine, Shimokasuya 143, Isehara-shi, Kanagawa 259-1193, Japan Received 9 October 2019; first decision 1 November 2019; accepted 28 January 2020 Background Platypnoea–orthodeoxia syndrome (POS) is characterized by dyspnoea and arterial desaturation in the sitting pos- ition. Although its pathophysiology is complex and still needed to be investigated, the disease is one of the clinical situations which should be immediately and adequately managed by health care workers from the initial presentation. ................................................................................................................................................................................................... Case summary A 66-year-old woman with a history of systemic lupus erythematosus, deep vein thrombosis, and lumbar compres- sion fracture was admitted for evaluation of the sudden onset of dyspnoea, while in the sitting position that was relieved on placing her in the supine position. Her transoesophageal echocardiogram did reveal a deformity in the patent foramen ovale (PFO) structure with a wide gap due to aortic compression, which was markedly different from that observed in the supine position, along with massive right-to-left shunting caused by redirected venous re- turn due to a persistent Eustachian valve. With the computed tomography and angiograms, POS was diagnosed. Then, the patient received aortic replacement and patch closure of PFO, and her symptoms were completely resolved. ................................................................................................................................................................................................... Discussion Platypnoea–orthodeoxia syndrome is a condition with quite unique features and needs multiple clinical measures for the diagnosis and medical management. For all health care workers, it is essential to have a high suspicion in order to detect POS in patients with unexplained dyspnoea. Echocardiography plays a major role in establishing the diagnosis and offering the choice of therapeutic options. Keywords Case report Echocardiography Patent foramen ovale Orthodeoxia � � � Learning points To show typical pathophysiology of platypnoea–orthodeoxia syndrome (POS) in the elderly. To highlight the role of echocardiography to detect and to manage POS. * Corresponding author. Tel: þ81 463 93 1121, Fax: þ81 463 93 6679, Email: nagait@tokai.ac.jp Handling Editor: Christoph Sinning Peer-reviewers: Cemil Izgi, Richard Alexander Brown and Georgia Daniel Compliance Editor: Carlos Minguito Carazo Supplementary Material Editor: Peregrine Green V The Author(s) 2020. Published by Oxford University Press on behalf of the European Society of Cardiology. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/ehjcr/advance-article-abstract/doi/10.1093/ehjcr/ytaa045/5770869 by guest on 03 March 2020 2 M. Hasegawa et al. upper mediastinum. However, in the sitting position, her blood gas Introduction analysis revealed marked hypoxaemia with low partial pressure of ar- Platypnoea–orthodeoxia syndrome (POS), which is characterized by . terial oxygen (PaO ) and oxygen saturation (54.1 mmHg and 75%, re- dyspnoea and arterial desaturation in the sitting position, was . spectively) in room air, which quickly recovered to the normoxic believed to be a rare clinical entity. The proposed pathophysiology . status with normal PaO and oxygen saturation (102.6 mmHg and is complex and involves the following two components: an anatomic- . 100%, respectively) in the supine position. While transthoracic echo- al component, such as an interatrial communication, and a secondary cardiogram showed preserved left ventricular ejection fraction (67%) or functional component that results in a deformation in the atrial and failed to detect any specific findings, her transoesophageal echo- 2 . septum with redirection of shunt flow in the upright posture. . cardiogram (TOE) revealed a patent foramen ovale (PFO) with a positive microbubble test but without obvious right-to-left shunting in the supine position (Figure 1A, Supplementary material online, Timeline Videos S1 and S2). In the sitting position, TOE further revealed a de- formity in the structure of the PFO with a wide gap due to aortic compression that was markedly different from that observed in the supine position. Massive right-to-left shunt flow caused by redirected Date Events ................................................................................................. venous return could be observed due to the persistent Eustachian 2001 Diagnosis of systemic lupus erythematosus . valve (Figure 1B and Supplementary material online, Video S3). June 2018 Admission to the prior medical institution due to . Therefore, POS was diagnosed. Latterly, the maximum diameter of bone fracture of the pelvis the ascending aorta was measured to be 57 mm on contrast- Progression of orthostatic dyspnoea enhanced computed tomography (Figure 1C). The elongated ascend- 2 July 2018 Transferred to our institution ing aorta expanded to pressurize the right atrium. Finally, the pres- Electrocardiogram, chest X-ray, and transthoracic ence of an intra-atrial shunt was confirmed on the right atrial echocardiography angiogram (Supplementary material online, Video S4). After the heart Not remarkable team conference, she received surgical prosthetic vascular graft re- 3 July 2018 Computed tomography placement of the ascending aorta and patch closure of PFO at the Ascending aorta dilation interatrial septum under cardiopulmonary bypass. Immediately after 4 July 2018 Comprehensive right heart catheterization including the surgery, her blood gas analysis normalized even in the sitting pos- right atrial angiogram ition, and her symptoms disappeared completely. A post-operative Existence of the right to left shunt at the level of TOE revealed no residual intracardiac shunt in the sitting position atriums . (Figure 1D and Supplementary material online, Video S5). She was dis- 6 July 2018 Transoesophageal echocardiography . charged without any events, and no recurrence occurred up to the De novo demonstration of the augmented right to . present. left shunt through patent foramen ovale (PFO) by sitting position 12 July 2018 Surgical repair of the PFO and resection of ascend- Discussion ing aortic aneurysm . . In this patient, the anatomical component was the PFO, and the sec- No relapse of the symptoms . . ondary component included both cardiac factors, such as the exist- 17 July 2018 Transoesophageal echocardiography . . ence of a persistent Eustachian valve, and vascular factors such as a No residual intracardiac shunt . . 3 . dilated aortic root. Agrawal et al. conducted a full review of the lit- 30 July 2018 Discharge . . erature from 1949 to 2016 regarding POS and reported PFO as the most common anatomical component. Since PFO was detected in 27% of the autopsy cases of the general population, it can be consid- ered a relatively common condition. Recently, several case reports Case presentation have documented the contribution of aortic atherosclerosis, which led to elongation of the ascending aorta or aortic root dilation as the A 66-year-old woman with a history of systemic lupus erythemato- 4–7 secondary component of POS in the elderly population. As the sus, deep vein thrombosis, and lumbar compression fracture was rate of atherosclerosis that induces aortic elongation or aortic root admitted for the evaluation of sudden onset dyspnoea, while in the . dilatation increases in the population annually, the combination of sitting position that was relieved when lying supine. She had become . congenital and acquired pathologies (PFO and aortic compression) frail as a result, having spent most of the day lying in bed due to the may now be considered one of the typical features of POS. above-mentioned symptoms. As long as she was recumbent, her con- For senior patients who are complicated due to multiple health dition was stable, and her physical examination did not reveal any car- . problems and who are otherwise prone to have a greater need for diopulmonary signs. Her general laboratory results were within the . bed rest, establishing the diagnosis of POS can be challenging. These normal limits, except for a relatively high brain natriuretic peptide . patients sometimes cannot explain or easily express their pattern of value which was 35.4 pg/mL (0–18.4 pg/mL). The electrocardiogram . symptoms, and extensive use of laboratory and diagnostic imaging was normal, although her chest X-ray revealed mild dilatation of the modalities is required to rule out several similar health conditions. Downloaded from https://academic.oup.com/ehjcr/advance-article-abstract/doi/10.1093/ehjcr/ytaa045/5770869 by guest on 03 March 2020 POS due to deformation of the PFO caused by a dilated AAo 3 Figure 1 (A) Transoesophageal echocardiogram with colour Doppler recorded with the patient in the supine position shows the existence of the patent foramen ovale without shunt flow. (B) Transoesophageal echocardiogram with colour Doppler recorded with the patient in the sitting pos- ition shows the wide patent foramen ovale with massive right-to-left shunt flow induced by the deformation. An arrow indicates the right-to-left shunt flow. (C) A computed tomography demonstrated that the maximum diameter of the ascending aorta was 57 mm. (D) A post-operative trans- oesophageal echocardiography revealed no residual intracardiac shunt in the sitting position. AAo, ascending aorta; LA, left atrium; RA, right atrium. Measuring arterial blood gases in different positions and obtaining the . Lead author biography expected results from positional change shall be the initial step for . Dr Misaki Hasegawa was born in the successful diagnosis of POS. Subsequently, contrast-enhanced Yokkaichi-shi, Japan in 1988. She transthoracic echocardiogram or TOE will confirm the existence of . received the MD degree from Tokai right-to-left intracardiac shunt and uncover the mechanism. Once . University School of Medicine the diagnosis is established, therapeutic options are usually deployed (Isehara-shi, Japan) in 2015. Since because surgical or percutaneous treatments have been already . 8 . 2018, she has been enrolled in the established. And, depending upon the patient’s overall health condi- . PhD programme of cardiovascular tion, the symptoms are then usually relieved. medicine (Tokai University Graduate School of Medicine, Isehara-shi, . Japan). Her research interest is main- Conclusion ly focused on cardiac imaging in cor- POS is a condition with quite unique features and needs multiple clin- . onary artery diseases and heart failure. ical measures for the diagnosis and medical management. Among . them, echocardiography plays a major role in establishing the diagno- sis and offering the choice of therapeutic options. As the number of Supplementary material POS cases is growing in the elderly population, POS may be one of Supplementary material is available at European Heart Journal - Case the emerging health risks of ageing in Western societies such as Reports online. Europe, the UK, the USA, and Japan. For physicians, it is essential to . have a high suspicion in order to detect POS in patients with unex- . Slide sets: A fully edited slide set detailing this case and suitable for plained dyspnoea, especially in the elderly population. local presentation is available online as Supplementary data. Downloaded from https://academic.oup.com/ehjcr/advance-article-abstract/doi/10.1093/ehjcr/ytaa045/5770869 by guest on 03 March 2020 4 M. Hasegawa et al. 3. Agrawal A, Palkar A, Talwar A. The multiple dimensions of platypnea-orthodeoxia Consent: The author/s confirm that written consent for submis- syndrome: a review. Respir Med 2017;129:31–38. sion and publication of this case report including image(s) and 4. Hagen PT, Scholz DG, Edwards WD. Incidence and size of patent foramen ovale associated text has been obtained from the patient in line with . during the first 10 decades of life: an autopsy study of 965 normal hearts. Mayo Clin Proc 1984;59:17–20. COPE guidance. 5. Medina A, de Lezo JS, Caballero E, Ortega JR. Platypnea-orthodeoxia due to aortic elongation. Circulation 2001;104:741. Conflict of interest: The authors declare no conflict of interest. 6. Shiraishi Y, Hakuno D, Isoda K, Miyazaki K, Adachi T. Platypnea-orthodeoxia syn- drome due to PFO and aortic dilation. JACC Cardiovasc Imaging 2012;5:570–571. References . 7. Chopard R, Meneveau N. Right-to-left atrial shunting associated with aortic root 1. Cheng TO. Platypnea-orthodeoxia syndrome: etiology, differential diagnosis, and aneurysm: a case report of a rare cause of platypnea-orthodeoxia syndrome. management. Catheter Cardiovasc Interv 1999;47:64–66. Heart Lung Circ 2013;22:71–75. 2. Cheng TO. Mechanisms of platypnea-orthodeoxia: what causes water to flow up- 8. Tobis JM, Abudayyeh I. Platypnea-orthodeoxia syndrome: an overlooked cause of hill? Circulation 2002;105:e47. . hypoxemia. JACC Cardiovasc Interv 2016;9:1939–1940.

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Published: Jun 1, 2020

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