© 2015 Wiley Periodicals, Inc.
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CASE REPORT ______________________________________________________________________
Aortic Valve Rupture During Isometric Muscle Contraction Darrin M. Payne, M.D., M.Sc., F.R.C.S.C.,*,y Tammy Richards, R.N.F.A.,y and Andrew Hamilton, M.D., F.R.C.S.C.*,y *Queens University, Kingston, Ontario, Canada; and yKingston General Hospital, Kingston, Ontario, Canada ABSTRACT Aortic valve rupture has been previously reported in association with blunt chest trauma as well as spontaneously in the setting of abnormal valves. We present a case of a patient who required emergent aortic valve replacement following rupture of his aortic valve during an isometric muscle contraction. doi: 10.1111/jocs.12519 (J Card Surg
2015;30:431–432) Aortic valve (AV) rupture is a rare, but well described event.1–10 It occurs most commonly in the setting of blunt chest trauma but can also occur spontaneously in the setting of structurally or molecularly abnormal valves. We present a case of a ruptured aortic valve occurring during straining with an isometric upper body contraction necessitating emergent surgery. Patient profile The patient provided informed consent for the publication of this report. A 52-year-old male arrived intubated and sedated. Aside from being a remote smoker, he had no significant recent or remote past medical history. Reports stated he had described feeling a ‘‘pop’’ in his chest while straining to close the rear doors of a semi-transport trailer. He began feeling unwell very shortly thereafter. He became increasingly short of breath and after an episode of hemoptysis, he presented to a local hospital. A computed tomography (CT) scan was performed to exclude pulmonary embolism and thoracic aortic dissection. He decompensated requiring intubation and ventilator support. He was then transferred to our tertiary care center, where physical examination revealed a diastolic murmur. Transthoracic echocardiogram (TTE) confirmed severe aortic insufficiency (AI), with a normal sized and contractile left ventricle (LV). He quickly deteriorated requiring escalating inotropic support. Due to his rapid
We have no sources of support or funding to acknowledge for this manuscript. Address for correspondence: Darrin M. Payne, M.D., M.Sc., F.R.C.S.C., Kingston General Hospital, 76 Stuart St, Kingston, Ontario, Canada, K7L 2V7. Fax: 613 548 2406; e-mail: [email protected]
and ongoing hemodynamic decline, we decided to forego a diagnostic coronary angiogram and proceed directly to the operating room (OR) for emergent surgery. Intraoperatively, a transesophageal echocardiogram (TEE) confirmed the severity of the AI, prolapsing AV and the absence of an aortic dissection (Fig. 1). Median sternotomy was performed and routine direct aortic and two-stage right atrial cannulation was performed. A retrograde coronary sinus catheter was placed and an LV vent inserted via the right superior pulmonary vein. The aorta was cross-clamped and retrograde cardioplegia administered to achieve arrest. The aorta was opened and the valve inspected. The AV was trileaflet. The left-right commissure was observed to be avulsed resulting in prolapse of both the right and left coronary cusps. Fenestrations were observed at the left-noncommissure. Both nonavulsed commissures appeared elongated and thinned. The free edges of the leaflets also appeared elongated and thinned out, as did the body of the leaflets themselves. It was felt that the valve could not be reliably repaired, thus the leaflets were excised and the valve was replaced with a size 27 St. Jude Epic biologic valve. Several extra minutes of reperfusion following valve implantation permitted the left ventricle to recover following the acute AI insult. Total cross-clamp and cardiopulmonary bypass times were 34 and 55 minutes, respectively. The patient was then weaned uneventfully from cardiopulmonary bypass. TEE confirmed a normal functioning prosthetic valve and normal LV function. Pathologic examination reported three aortic valve leaflets with moderate calcification that were grossly unremarkable. Unfortunately, sections were not taken for microscopic examination. The patient had an uneventful postoperative course and was discharged on postoperative day 5. Comment The majority of published reports of AV or commissural rupture have been related to traumatic events, although there have also been reports of spontaneous rupture occurring in the setting of valve abnormalities, such as fenestrations or abnormal fibrous bands.1–4 Blunt chest trauma is a well-described mechanism that can cause AV rupture and acute AI. Unal et al. and Taguchi et al. have both published on aortic commissural avulsion in normal structural aortic valves following motor vehicle collisions.5,6 Li et al. reported on a patient who, along with multiple other injuries, was found to have AI following a 10-meter fall.7 This patient was taken to the OR where a torn left coronary cusp was observed in an otherwise normal appearing valve. Sabzi et al. reported on a healthy 34-year-old man who fell from a height of 5 meters, rupturing both his aortic and tricuspid valves.8 This patient was found intraoperatively to have avulsed the right and non-coronary cusps of his aortic valve as well as ruptured the papillary
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PAYNE ET AL. AORTIC VALVE RUPTURE
J CARD SURG 2015;30:431–432
Figure 1. TEE showing prolapsing aortic valve and severe aortic insufficiency.
muscles in the right ventricle resulting in flail anterior and posterior tricuspid valve leaflets. He was successfully managed with AV repair and TV replacement. Not surprisingly, blunt chest trauma in patients with known connective tissue diseases, such as Marfan’s syndrome, can also predispose to AV rupture and likewise has been previously reported.9 In fact, there have been several reports of rupture of structurally abnormal aortic valves. Structural abnormalities of aortic valves that have led to acute AI have included rupture of anomalous fibrous bands connecting aortic cusp free edges to the aortic wall. This has been reported more commonly in bicuspid valves1–3 but has also been observed with tricuspid valves.3 Rupture of fenestrated aortic cusps has also been implicated to cause acute, severe AI.4 Connective tissue abnormalities that weaken aortic cusp tissue have also been implicated in AV spontaneous rupture, such as was reported by Hirata et al. in a patient with confirmed pseudoxanthoma elasticum.10 The underlying mechanism of AV rupture with trauma has been debated. The most likely mechanism is a rapid rise in intrathoracic pressure occurring during early diastole. This pressure rise is transmitted to the aorta. It is during early diastole when the LV has its lowest intracavitary pressure, thus a high pressure gradient over the AV occurs, resulting in cusp rupture or avulsion.6,9 This mechanism appears valid with our case as well, as the intrathoracic pressure would increase with straining and could thus be transmitted across the AV causing rupture. While the absolute pressure generated with straining and the rate at which that pressure developed would presumably be less than that of significant trauma, this potentially could still create enough of a pressure gradient to rupture a weakened or abnormal AV. The patient presented in this case report did have evidence of fenestrations in his aortic valve and
generally thinned-out and elongated leaflet tissue which may have predisposed him to valve rupture. REFERENCES 1. Vowels TJ, Gonzalez-Stawinski GV, Ko JM, et al: Anomalous cord from the raphe of a congenitally bicuspid aortic valve to the aortic wall producing either acute or chronic aortic regurgitation. J Am Coll Cardiol 2014;63:153–157. 2. Akiyama K, Taniyasu N, Iba Y, et al: Sudden deterioration of aortic regurgitation due to rupture of a raphal cord on the conjoined cusp. Jpn Circ J 2000;64:477–480. 3. Minami H, Asada T, Gan K, et al: Aortic regurgitation caused by rupture of the abnormal fibrous band between the aortic valve and aortic wall. Gen Thorac Cardiovasc Surg 2011;59:488–490. 4. Blaszyk H, Witkiewicz AK, Edwards WD: Acute aortic regurgitation due to spontaneous rupture of a fenestrated cusp: Report in a 65-year-old man and review of seven additional cases. Cardiovasc Pathol 1999;8:213–216. 5. Unal M, Demirsoy E, Gogus A, et al: Acute aortic valve regurgitation secondary to blunt chest trauma. Tex Heart Inst J 2001;28:312–314. 6. Taguchi S, Ueda T, Inoue Y, et al: Traumatic aortic regurgitation caused by detached commissure. Gen Thorac Cardiovasc Surg 2007;55:32–34. 7. Li W, Ni Y, Chen X, et al: Aortic valve tear with severe aortic regurgitation following blunt chest trauma. J Cardiothorac Surg 2011;6:84. 8. Sabzi F, Niazi M, Ahmadi A: Simultaneous aortic and tricuspid rupture after fall injury. J Inj Violence Res 2013;5:120–124. 9. Onorati F, Salvatore L, De Santo LS, et al: Marfan syndrome as a predisposing factor for traumatic aortic insufficiency. Ann Thorac Surg 2004;77:2194–2195. 10. Hirata K, Kazazu M, Wake M, et al: Acute aortic valvular regurgitation secondary to avulsion of aortic valve commissure in a patient with pseudoxanthoma elasticum. Intern Med 2000;39:940–942.
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CASE REPORT ______________________________________________________________________
Aortic Valve Rupture During Isometric Muscle Contraction Darrin M. Payne, M.D., M.Sc., F.R.C.S.C.,*,y Tammy Richards, R.N.F.A.,y and Andrew Hamilton, M.D., F.R.C.S.C.*,y *Queens University, Kingston, Ontario, Canada; and yKingston General Hospital, Kingston, Ontario, Canada ABSTRACT Aortic valve rupture has been previously reported in association with blunt chest trauma as well as spontaneously in the setting of abnormal valves. We present a case of a patient who required emergent aortic valve replacement following rupture of his aortic valve during an isometric muscle contraction. doi: 10.1111/jocs.12519 (J Card Surg
2015;30:431–432) Aortic valve (AV) rupture is a rare, but well described event.1–10 It occurs most commonly in the setting of blunt chest trauma but can also occur spontaneously in the setting of structurally or molecularly abnormal valves. We present a case of a ruptured aortic valve occurring during straining with an isometric upper body contraction necessitating emergent surgery. Patient profile The patient provided informed consent for the publication of this report. A 52-year-old male arrived intubated and sedated. Aside from being a remote smoker, he had no significant recent or remote past medical history. Reports stated he had described feeling a ‘‘pop’’ in his chest while straining to close the rear doors of a semi-transport trailer. He began feeling unwell very shortly thereafter. He became increasingly short of breath and after an episode of hemoptysis, he presented to a local hospital. A computed tomography (CT) scan was performed to exclude pulmonary embolism and thoracic aortic dissection. He decompensated requiring intubation and ventilator support. He was then transferred to our tertiary care center, where physical examination revealed a diastolic murmur. Transthoracic echocardiogram (TTE) confirmed severe aortic insufficiency (AI), with a normal sized and contractile left ventricle (LV). He quickly deteriorated requiring escalating inotropic support. Due to his rapid
We have no sources of support or funding to acknowledge for this manuscript. Address for correspondence: Darrin M. Payne, M.D., M.Sc., F.R.C.S.C., Kingston General Hospital, 76 Stuart St, Kingston, Ontario, Canada, K7L 2V7. Fax: 613 548 2406; e-mail: [email protected]
and ongoing hemodynamic decline, we decided to forego a diagnostic coronary angiogram and proceed directly to the operating room (OR) for emergent surgery. Intraoperatively, a transesophageal echocardiogram (TEE) confirmed the severity of the AI, prolapsing AV and the absence of an aortic dissection (Fig. 1). Median sternotomy was performed and routine direct aortic and two-stage right atrial cannulation was performed. A retrograde coronary sinus catheter was placed and an LV vent inserted via the right superior pulmonary vein. The aorta was cross-clamped and retrograde cardioplegia administered to achieve arrest. The aorta was opened and the valve inspected. The AV was trileaflet. The left-right commissure was observed to be avulsed resulting in prolapse of both the right and left coronary cusps. Fenestrations were observed at the left-noncommissure. Both nonavulsed commissures appeared elongated and thinned. The free edges of the leaflets also appeared elongated and thinned out, as did the body of the leaflets themselves. It was felt that the valve could not be reliably repaired, thus the leaflets were excised and the valve was replaced with a size 27 St. Jude Epic biologic valve. Several extra minutes of reperfusion following valve implantation permitted the left ventricle to recover following the acute AI insult. Total cross-clamp and cardiopulmonary bypass times were 34 and 55 minutes, respectively. The patient was then weaned uneventfully from cardiopulmonary bypass. TEE confirmed a normal functioning prosthetic valve and normal LV function. Pathologic examination reported three aortic valve leaflets with moderate calcification that were grossly unremarkable. Unfortunately, sections were not taken for microscopic examination. The patient had an uneventful postoperative course and was discharged on postoperative day 5. Comment The majority of published reports of AV or commissural rupture have been related to traumatic events, although there have also been reports of spontaneous rupture occurring in the setting of valve abnormalities, such as fenestrations or abnormal fibrous bands.1–4 Blunt chest trauma is a well-described mechanism that can cause AV rupture and acute AI. Unal et al. and Taguchi et al. have both published on aortic commissural avulsion in normal structural aortic valves following motor vehicle collisions.5,6 Li et al. reported on a patient who, along with multiple other injuries, was found to have AI following a 10-meter fall.7 This patient was taken to the OR where a torn left coronary cusp was observed in an otherwise normal appearing valve. Sabzi et al. reported on a healthy 34-year-old man who fell from a height of 5 meters, rupturing both his aortic and tricuspid valves.8 This patient was found intraoperatively to have avulsed the right and non-coronary cusps of his aortic valve as well as ruptured the papillary
432
PAYNE ET AL. AORTIC VALVE RUPTURE
J CARD SURG 2015;30:431–432
Figure 1. TEE showing prolapsing aortic valve and severe aortic insufficiency.
muscles in the right ventricle resulting in flail anterior and posterior tricuspid valve leaflets. He was successfully managed with AV repair and TV replacement. Not surprisingly, blunt chest trauma in patients with known connective tissue diseases, such as Marfan’s syndrome, can also predispose to AV rupture and likewise has been previously reported.9 In fact, there have been several reports of rupture of structurally abnormal aortic valves. Structural abnormalities of aortic valves that have led to acute AI have included rupture of anomalous fibrous bands connecting aortic cusp free edges to the aortic wall. This has been reported more commonly in bicuspid valves1–3 but has also been observed with tricuspid valves.3 Rupture of fenestrated aortic cusps has also been implicated to cause acute, severe AI.4 Connective tissue abnormalities that weaken aortic cusp tissue have also been implicated in AV spontaneous rupture, such as was reported by Hirata et al. in a patient with confirmed pseudoxanthoma elasticum.10 The underlying mechanism of AV rupture with trauma has been debated. The most likely mechanism is a rapid rise in intrathoracic pressure occurring during early diastole. This pressure rise is transmitted to the aorta. It is during early diastole when the LV has its lowest intracavitary pressure, thus a high pressure gradient over the AV occurs, resulting in cusp rupture or avulsion.6,9 This mechanism appears valid with our case as well, as the intrathoracic pressure would increase with straining and could thus be transmitted across the AV causing rupture. While the absolute pressure generated with straining and the rate at which that pressure developed would presumably be less than that of significant trauma, this potentially could still create enough of a pressure gradient to rupture a weakened or abnormal AV. The patient presented in this case report did have evidence of fenestrations in his aortic valve and
generally thinned-out and elongated leaflet tissue which may have predisposed him to valve rupture. REFERENCES 1. Vowels TJ, Gonzalez-Stawinski GV, Ko JM, et al: Anomalous cord from the raphe of a congenitally bicuspid aortic valve to the aortic wall producing either acute or chronic aortic regurgitation. J Am Coll Cardiol 2014;63:153–157. 2. Akiyama K, Taniyasu N, Iba Y, et al: Sudden deterioration of aortic regurgitation due to rupture of a raphal cord on the conjoined cusp. Jpn Circ J 2000;64:477–480. 3. Minami H, Asada T, Gan K, et al: Aortic regurgitation caused by rupture of the abnormal fibrous band between the aortic valve and aortic wall. Gen Thorac Cardiovasc Surg 2011;59:488–490. 4. Blaszyk H, Witkiewicz AK, Edwards WD: Acute aortic regurgitation due to spontaneous rupture of a fenestrated cusp: Report in a 65-year-old man and review of seven additional cases. Cardiovasc Pathol 1999;8:213–216. 5. Unal M, Demirsoy E, Gogus A, et al: Acute aortic valve regurgitation secondary to blunt chest trauma. Tex Heart Inst J 2001;28:312–314. 6. Taguchi S, Ueda T, Inoue Y, et al: Traumatic aortic regurgitation caused by detached commissure. Gen Thorac Cardiovasc Surg 2007;55:32–34. 7. Li W, Ni Y, Chen X, et al: Aortic valve tear with severe aortic regurgitation following blunt chest trauma. J Cardiothorac Surg 2011;6:84. 8. Sabzi F, Niazi M, Ahmadi A: Simultaneous aortic and tricuspid rupture after fall injury. J Inj Violence Res 2013;5:120–124. 9. Onorati F, Salvatore L, De Santo LS, et al: Marfan syndrome as a predisposing factor for traumatic aortic insufficiency. Ann Thorac Surg 2004;77:2194–2195. 10. Hirata K, Kazazu M, Wake M, et al: Acute aortic valvular regurgitation secondary to avulsion of aortic valve commissure in a patient with pseudoxanthoma elasticum. Intern Med 2000;39:940–942.
