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An Unusual Case of Arterial Embolism in an Adolescent With a Mitral Valve Repair With a Ring
An Unusual Case of Arterial Embolism in an Adolescent With a Mitral Valve Repair With a Ring Christos Tourmousoglou, MD, Christos Karkos, MD, Theodoros Fidanis, MD, Efstratios K. Theofilogiannakos, MD, Prodromos Hytiroglou, MD, and Antonis Pitsis, MD Thessaloniki Heart Institute, St. Luke’s Hospital, Thessaloniki; Vascular Unit, 5th Department of Surgery, Hippokratio Hospital, Aristotle University of Thessaloniki, Thessaloniki; Departments of Radiology and Cardiology, St. Luke’s Hospital, Thessaloniki; and Department of Pathology, Aristotle University of Thessaloniki, Thessaloniki, Greece
We report a case of an adolescent young man who presented with embolism of both lower legs. The patient had undergone mitral valve repair with a Kalangos biodegradable ring (Bioring SA, Lonay, Switzerland) 9 months earlier. Bilateral embolectomy was performed. Histopathologic examination revealed minute fragments of synthetic material within the embolus, which otherwise consisted of recent thrombus. (Ann Thorac Surg 2017;104:e315–7) Ó 2017 by The Society of Thoracic Surgeons
T
hromboembolic complications, including systemic emboli, occur at a rate of 0.7% to 6% patient-years in thrombosis of mechanical valves. Thrombosis of a bioprosthetic valve is a rare occurrence when it is compared with mechanical prostheses. There is also the possibility of ring thrombosis after mitral valve repair even in patients without thrombotic risk factors. Knots and sutures that are left too long can favor the formation of thrombi. A 19-year-old man was admitted with a 48-hour-long history of left leg ischemia. He was complaining of pain along his lower leg and a feeling of coldness and numbness. On examination in our unit all 6 Ps of acute limb ischemia were present (ie, pain, pallor, pulselessness, paralysis, paresthesia, and perishing cold). There were no palpable pulses either in the symptomatic left lower limb or in the right leg, which was asymptomatic. There was no evidence of arrhythmia on examination and on electrocardiography. The patient was afebrile and the serum inflammatory markers were within normal values. The patient was not taking aspirin or any anticoagulation when he was admitted to the hospital 9 months after mitral valve surgery. This patient’s past medical history was remarkable for mitral valve repair 9 months previous to the current presentation because of severe mitral valve regurgitation at another hospital. During the operation, a cleft in A2
Accepted for publication April 19, 2017. Address correspondence to Dr Tourmousoglou, 48 Proxenou Koromila St, 54622, Thessaloniki, Greece; email: [email protected].
Ó 2017 by The Society of Thoracic Surgeons Published by Elsevier Inc.
was discovered. Closure of the cleft with Prolene suture (Ethicon, Somerville, NJ) was decided and a Kalangos biodegradable ring No. 32 mm (Bioring SA, Lonay, Switzerland) was inserted. The recovery of the patient was uneventful. He underwent an emergency digital subtraction angiography, which suggested an embolic occlusion of the left femoral bifurcation, as well as a further embolic occlusion of the midpopliteal artery (Figs 1A, 1B). Additional emboli were present in the right common and external iliac arteries. Attempts for percutaneous thrombus aspiration yielded so-called thrombotic material of unusual appearance and the patient was taken to the operating room for a formal embolectomy rather than continuing with catheter-directed thrombolysis. Under general anesthesia, both groins were exposed and the femoral bifurcation was dissected and slung; 5.000 units of heparin were administered intravenously. Femoral embolectomy was performed through a transverse arteriotomy. A 4F Fogarty embolectomy catheter (Edwards Lifesciences, Irvine, CA) was advanced both proximally and distally, retrieving atypical embolic material, which was whitebrown in color and rock hard in texture. A very small amount of fresh thrombus was also removed. Due to the patient’s history of recent cardiac surgery and the atypical appearance of the emboli, these were sent for histopathology. Following arteriotomy closure and completion of embolectomy, all peripheral pulses were restored. His postoperative course was uneventful. The patient was anticoagulated with Coumadin and was discharged home 3 days later in a satisfactory condition. Histopathologic examination of the surgical specimen showed recent thrombi consisting of a fibrin mesh that contained red and white blood cells (Fig 2A). Focally, minute fragments of synthetic material were seen (Fig 2B). These were birefringent on examination under polarized light. Because of his recent history of mitral annuloplasty, transesophageal echocardiography was performed. Transesophageal echocardiography showed normal left ventricular size and systolic function with mild-tomoderate mitral regurgitation. Most importantly, multiple hyperechogenic masses attached at the anterior leaflet of the mitral valve were observed. These masses did not have independent motion of the valve and did not cause mitral stenosis (Figs 1C, 1D). These masses might be thrombi that were attached to the ring and they contained minute fragments of synthetic material. The possibility of reoperative mitral valve surgery was considered but the parents did not consent, and they preferred to visit the previous hospital where their child had the mitral valve repair.
Comment We report a rare presentation of thrombotic emboli containing minute fragments of synthetic material in a young man, who had mitral valve repair with a Kalangos biodegradable ring. 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2017.04.051
e316
CASE REPORT TOURMOUSOGLOU ET AL ARTERIAL EMBOLISM IN MITRAL VALVE REPAIR
Ann Thorac Surg 2017;104:e315–7
Fig 1. Digital subtraction angiography revealing abrupt cutoff in (A) the left common femoral artery and (B) the left popliteal artery (B) in keeping with the diagnosis of an embolic occlusion. (C, D) Transesophageal echocardiography showing normal left ventricular size and systolic function with mild-to-moderate mitral regurgitation. Most importantly, multiple hyperechogenic masses attached at the anterior leaflet of the mitral valve are observed.
Kalangos annuloplasty ring is an annuloplasty ring made of a biodegradable polymer, poly-1,4 dioxanone. The ring has a curved C segment. Suture material (30 monofilament polypropylene) is in continuity over the entire central portion of the ring with interruption in the midportion of the ring (Figs 3A, 3B). It is implanted subendocardially into the mitral or tricuspid annulus and is completely degraded within 6 months after implantation, leaving a fibrous tissue behind. Both the ring itself Fig 2. Hematoxylin and eosin–stained sections of the embolic material: (A) low-power view; (B) high-power view revealing minute fragments of synthetic material (arrows).
and the replacing fibrous band provide adequate support to the valve and preserve annular contraction during systole. It is a partial ring and it allows native annular growth corresponding to somatic growth. The lack of contact between the ring and blood circulation as it is implanted subendocardially and the lack of need for anticoagulation are the other advantages of this ring [1, 2]. This report is of interest not only because synthetic material is not usually observed in thrombotic material,
Ann Thorac Surg 2017;104:e315–7
CASE REPORT TOURMOUSOGLOU ET AL ARTERIAL EMBOLISM IN MITRAL VALVE REPAIR
e317
We hypothesize that this complication happened because the ring might not be implanted in the correct position and there was contact between the ring and blood circulation. The masses that were attached to the ring might be thrombi and they contained minute fragments of synthetic material. Ring thrombosis may occur in patients who were operated for mitral valve surgery with or without predisposing factors, such as atrial fibrillation or thrombophilic disorders [4].
References
Fig 3. Kalangos Bioring in its commercial form (Bioring SA, Lonay, Switzerland). The body of the mitral Bioring (A) differs from that of the tricuspid Bioring (B) because of different anatomic structures of the native mitral and tricuspid valve.
but also especially because arterial embolism is an unreported potentially lethal complication of this device [3].
1. Pektok E, Sierra J, Cikirikcioglu M, Muller H, Myers P, Kalangos A. Midterm results of valve repair with a biodegradable annuloplasty ring for acute endocarditis. Ann Thorac Surg 2010;89:1180–6. 2. Yakub M, Sivalingam S, Dillon J, Matsuhama M, Latiff H, Ramli M. Mitral valve repair for congenital mitral valve disease: impact of the use of a biodegradable annuloplasy ring. Ann Thorac Surg 2015;99:884–90. 3. Bautista-Hernandez V, Myers P, Loyola H, et al. Atrioventricular valve annular remodeling with a bioabsorbable ring in young children. J Am Coll Cardiol 2012;60:2255–60. 4. Sponga S, Auci E, Gianfagna P, Livi U. Ring thrombosis following mitral valve repair. Eur J Cardiothorac Surg 2014;45: 762.
We report a case of an adolescent young man who presented with embolism of both lower legs. The patient had undergone mitral valve repair with a Kalangos biodegradable ring (Bioring SA, Lonay, Switzerland) 9 months earlier. Bilateral embolectomy was performed. Histopathologic examination revealed minute fragments of synthetic material within the embolus, which otherwise consisted of recent thrombus. (Ann Thorac Surg 2017;104:e315–7) Ó 2017 by The Society of Thoracic Surgeons
T
hromboembolic complications, including systemic emboli, occur at a rate of 0.7% to 6% patient-years in thrombosis of mechanical valves. Thrombosis of a bioprosthetic valve is a rare occurrence when it is compared with mechanical prostheses. There is also the possibility of ring thrombosis after mitral valve repair even in patients without thrombotic risk factors. Knots and sutures that are left too long can favor the formation of thrombi. A 19-year-old man was admitted with a 48-hour-long history of left leg ischemia. He was complaining of pain along his lower leg and a feeling of coldness and numbness. On examination in our unit all 6 Ps of acute limb ischemia were present (ie, pain, pallor, pulselessness, paralysis, paresthesia, and perishing cold). There were no palpable pulses either in the symptomatic left lower limb or in the right leg, which was asymptomatic. There was no evidence of arrhythmia on examination and on electrocardiography. The patient was afebrile and the serum inflammatory markers were within normal values. The patient was not taking aspirin or any anticoagulation when he was admitted to the hospital 9 months after mitral valve surgery. This patient’s past medical history was remarkable for mitral valve repair 9 months previous to the current presentation because of severe mitral valve regurgitation at another hospital. During the operation, a cleft in A2
Accepted for publication April 19, 2017. Address correspondence to Dr Tourmousoglou, 48 Proxenou Koromila St, 54622, Thessaloniki, Greece; email: [email protected].
Ó 2017 by The Society of Thoracic Surgeons Published by Elsevier Inc.
was discovered. Closure of the cleft with Prolene suture (Ethicon, Somerville, NJ) was decided and a Kalangos biodegradable ring No. 32 mm (Bioring SA, Lonay, Switzerland) was inserted. The recovery of the patient was uneventful. He underwent an emergency digital subtraction angiography, which suggested an embolic occlusion of the left femoral bifurcation, as well as a further embolic occlusion of the midpopliteal artery (Figs 1A, 1B). Additional emboli were present in the right common and external iliac arteries. Attempts for percutaneous thrombus aspiration yielded so-called thrombotic material of unusual appearance and the patient was taken to the operating room for a formal embolectomy rather than continuing with catheter-directed thrombolysis. Under general anesthesia, both groins were exposed and the femoral bifurcation was dissected and slung; 5.000 units of heparin were administered intravenously. Femoral embolectomy was performed through a transverse arteriotomy. A 4F Fogarty embolectomy catheter (Edwards Lifesciences, Irvine, CA) was advanced both proximally and distally, retrieving atypical embolic material, which was whitebrown in color and rock hard in texture. A very small amount of fresh thrombus was also removed. Due to the patient’s history of recent cardiac surgery and the atypical appearance of the emboli, these were sent for histopathology. Following arteriotomy closure and completion of embolectomy, all peripheral pulses were restored. His postoperative course was uneventful. The patient was anticoagulated with Coumadin and was discharged home 3 days later in a satisfactory condition. Histopathologic examination of the surgical specimen showed recent thrombi consisting of a fibrin mesh that contained red and white blood cells (Fig 2A). Focally, minute fragments of synthetic material were seen (Fig 2B). These were birefringent on examination under polarized light. Because of his recent history of mitral annuloplasty, transesophageal echocardiography was performed. Transesophageal echocardiography showed normal left ventricular size and systolic function with mild-tomoderate mitral regurgitation. Most importantly, multiple hyperechogenic masses attached at the anterior leaflet of the mitral valve were observed. These masses did not have independent motion of the valve and did not cause mitral stenosis (Figs 1C, 1D). These masses might be thrombi that were attached to the ring and they contained minute fragments of synthetic material. The possibility of reoperative mitral valve surgery was considered but the parents did not consent, and they preferred to visit the previous hospital where their child had the mitral valve repair.
Comment We report a rare presentation of thrombotic emboli containing minute fragments of synthetic material in a young man, who had mitral valve repair with a Kalangos biodegradable ring. 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2017.04.051
e316
CASE REPORT TOURMOUSOGLOU ET AL ARTERIAL EMBOLISM IN MITRAL VALVE REPAIR
Ann Thorac Surg 2017;104:e315–7
Fig 1. Digital subtraction angiography revealing abrupt cutoff in (A) the left common femoral artery and (B) the left popliteal artery (B) in keeping with the diagnosis of an embolic occlusion. (C, D) Transesophageal echocardiography showing normal left ventricular size and systolic function with mild-to-moderate mitral regurgitation. Most importantly, multiple hyperechogenic masses attached at the anterior leaflet of the mitral valve are observed.
Kalangos annuloplasty ring is an annuloplasty ring made of a biodegradable polymer, poly-1,4 dioxanone. The ring has a curved C segment. Suture material (30 monofilament polypropylene) is in continuity over the entire central portion of the ring with interruption in the midportion of the ring (Figs 3A, 3B). It is implanted subendocardially into the mitral or tricuspid annulus and is completely degraded within 6 months after implantation, leaving a fibrous tissue behind. Both the ring itself Fig 2. Hematoxylin and eosin–stained sections of the embolic material: (A) low-power view; (B) high-power view revealing minute fragments of synthetic material (arrows).
and the replacing fibrous band provide adequate support to the valve and preserve annular contraction during systole. It is a partial ring and it allows native annular growth corresponding to somatic growth. The lack of contact between the ring and blood circulation as it is implanted subendocardially and the lack of need for anticoagulation are the other advantages of this ring [1, 2]. This report is of interest not only because synthetic material is not usually observed in thrombotic material,
Ann Thorac Surg 2017;104:e315–7
CASE REPORT TOURMOUSOGLOU ET AL ARTERIAL EMBOLISM IN MITRAL VALVE REPAIR
e317
We hypothesize that this complication happened because the ring might not be implanted in the correct position and there was contact between the ring and blood circulation. The masses that were attached to the ring might be thrombi and they contained minute fragments of synthetic material. Ring thrombosis may occur in patients who were operated for mitral valve surgery with or without predisposing factors, such as atrial fibrillation or thrombophilic disorders [4].
References
Fig 3. Kalangos Bioring in its commercial form (Bioring SA, Lonay, Switzerland). The body of the mitral Bioring (A) differs from that of the tricuspid Bioring (B) because of different anatomic structures of the native mitral and tricuspid valve.
but also especially because arterial embolism is an unreported potentially lethal complication of this device [3].
1. Pektok E, Sierra J, Cikirikcioglu M, Muller H, Myers P, Kalangos A. Midterm results of valve repair with a biodegradable annuloplasty ring for acute endocarditis. Ann Thorac Surg 2010;89:1180–6. 2. Yakub M, Sivalingam S, Dillon J, Matsuhama M, Latiff H, Ramli M. Mitral valve repair for congenital mitral valve disease: impact of the use of a biodegradable annuloplasy ring. Ann Thorac Surg 2015;99:884–90. 3. Bautista-Hernandez V, Myers P, Loyola H, et al. Atrioventricular valve annular remodeling with a bioabsorbable ring in young children. J Am Coll Cardiol 2012;60:2255–60. 4. Sponga S, Auci E, Gianfagna P, Livi U. Ring thrombosis following mitral valve repair. Eur J Cardiothorac Surg 2014;45: 762.