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Fetal cardiac tamponade due to an intrapericardial teratoma Article in The Annals of Thoracic Surgery · September 1998 DOI: 10.1016/S0003-4975(98)00508-6 · Source: PubMed
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Fetal cardiac tamponade due to an intrapericardial teratoma Tim Tollens, Filip Casselman, Hugo Devlieger, Marc H. Gewillig, Kamiel Vandenberghe, Toni E. Lerut and Willem J. Daenen Ann Thorac Surg 1998;66:559-560
The online version of this article, along with updated information and services, is located on the World Wide Web at: http://ats.ctsnetjournals.org/cgi/content/full/66/2/559
The Annals of Thoracic Surgery is the official journal of The Society of Thoracic Surgeons and the Southern Thoracic Surgical Association. Copyright © 1998 by The Society of Thoracic Surgeons. Print ISSN: 0003-4975; eISSN: 1552-6259.
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Ann Thorac Surg 1998;66:559 – 60
CASE REPORT TOLLENS ET AL INTRAPERICARDIAL TERATOMA
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Fetal Cardiac Tamponade Due to an Intrapericardial Teratoma Tim Tollens, MD, Filip Casselman, MD, Hugo Devlieger, MD, PhD, Marc H. Gewillig, MD, PhD, Kamiel Vandenberghe, MD, Toni E. Lerut, MD, PhD, and Willem J. Daenen, MD Departments of Cardiac Surgery, Thoracic Surgery, Neonatology, and Gynecology and Obstetrics, University Hospitals Gasthuisberg, Leuven, Belgium
A case of an intrapericardial tumor diagnosed in utero at 26 weeks of gestation is presented. The prenatal echocardiographic follow-up of an incipient hydrops fetalis determined the management and the emergency surgical treatment. Histologically, the tumor appeared to be a benign teratoma, grade I. In the postoperative period an unexpected mediastinal tumor was found and removed later. This tumor also appeared to be a benign teratoma, grade 0. Both teratomas were independent and therefore primary. (Ann Thorac Surg 1998;66:559 – 60) © 1998 by The Society of Thoracic Surgeons
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xtrapericardial mediastinal teratomas are not uncommon, but intrapericardial teratomas are rare [1–3]. They may cause nonimmunologic hydrops fetalis and cardiac tamponade [1, 2, 4]. However, as demonstrated in the following case, structured planning guided by ultrasound follow-up can lead to a satisfying outcome. A 26-year-old primigravida, without a previous medical history, presented with polyhydramnios at 26 weeks of gestation. The ultrasound examination showed an intrapericardial mass (3.8 3 2.9 3 3.1 cm), pericardial effusion, and ascites. Hydrops fetalis developed with obstruction of venous return and cardiac tamponade. To improve the fetal survival chances, regular follow-up was planned. Initially the hydrops fetalis remained stable. Lung maturity was stimulated with thyrotropin-releasing hormone and betamethasone (Celestone, Schering Corp, Kenilworth, NJ). At 30 weeks of gestation the intrapericardial mass appeared cystic on ultrasound, suggesting a teratoma [4]. The pericardial effusion had increased. At 32 weeks of gestation there was a considerable increase in hydrops fetalis. Cardiac tamponade developed and fetal cardiac output decreased dramatically. Because of sudden fetal distress a cesarean section was performed. The 1,930-g male newborn had immediate cardiorespiratory distress. Peripheral circulation was poor and cyanosis was obvious. No murmurs were heard on ausculAccepted for publication Feb 27, 1998. Address reprint requests to Prof Daenen, Department of Cardiac Surgery, University Hospital Gasthuisberg, Herestraat 49, 3000 Leuven, Belgium (e-mail:
[email protected]).
Fig 1. Operative picture after sternotomy and opening of the pericardium showing a large cystic tumor compressing the right heart and aorta.
tation. The Apgar score was 5 after 1 minute. Endotracheal intubation was performed immediately. The chest radiograph showed an extremely widened cardiac shadow, and on echocardiography a large intrapericardial cystic tumor was confirmed. A pericardiocentesis yielded 15 mL of serous fluid. Subsequently, a severe respiratory distress syndrome developed, which was treated with high-frequency oscillation and surfactant therapy. Despite stabilization of the respiratory distress syndrome, the hemodynamic status deteriorated rapidly and we decided to operate 14 hours after the infant’s birth. After sternotomy and pericardiotomy, a large intrapericardial cystic tumor was found, compressing the right atrium, right ventricle, and aorta (Fig 1). The tumor was well demarcated and easily removed by dividing a single nutrient artery from the ascending aorta. The hemodynamic status improved immediately. The postoperative chest radiograph was apparently normal, except for a widened upper mediastinum. Histologic examination of the resected specimen yielded a mature benign cystic teratoma, grade I. The immediate postoperative course was uncomplicated. Two weeks after surgical excision, the chest radiograph still showed a widened upper mediastinum. Ultrasonographic examination and computed tomographic scanning demonstrated a small additional extrapericardial cystic tumor (1.4 3 2.1 cm) in the upper left anterior mediastinum. At 10 weeks the patient underwent a left thoracotomy with opening of the mediastinal pleura. A multilobulated cystic mass was found with a nutrient artery arising from the left internal mammary artery. The tumor was excised and histologic examination showed a mature teratoma, grade 0, without signs of malignancy. Because there was no connection between the intrapericardial and medias-
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CASE REPORT MARUI ET AL ISOLATED TRICUSPID REGURGITATION
tinal tumor, it was obvious that we were dealing with two independent primary teratomas.
Comment Cardiac neoplasms are known to originate in any part of the heart, including the pericardium, but are extremely rare [3]. A teratoma usually appears as a cystic mass outside the cardiac cavities and is sometimes calcified [2]. These tumors are voluminous and sometimes larger than the heart [5]. In contrast to our patient, they mostly occur in female patients [3]. The rapid growth and differentiation is somewhere between 20 and 40 weeks of gestation [1, 2, 6]. As in our case, the majority are located anteriorly to the right side, where they may compress the heart, great vessels, and in some cases the trachea [5, 6]. In general, the tumor is attached to the heart or great vessels by a fibrous pedicle [6] and, like in our patient, resection is easy. Multiple intrathoracic localizations are rare; however, our patient demonstrates that a second localization should always be ruled out. Clinical signs usually occur in the newborn period or in early infancy [7]. However, if detected in utero, nonimmunologic hydrops fetalis, characterized by fetal anasarca and pleural or pericardial effusion, is the main clinical feature [2, 4, 6, 7]. In the newborn with pericardial teratoma, ascites, cardiomegaly, and cardiopulmonary distress with dyspnea, hypoxia and acidosis are common [1, 3]. The clinical findings result from an obstructed systemic venous return by the tumor and the pericardial effusion. Consequently, the cardiac output is reduced. Therefore, pericardiocentesis may be lifesaving [4]. In the neonate, diagnosis is based on clinical, radiographic, and echocardiographic findings [3]. Fetal echocardiography may already have shown the cystic intrapericardial mass, pericardial effusion, and ascites. Chest radiography may show a widened mediastinum. Heart catheterization should be avoided, especially in the symptomatic patient, because of the high risk involved [5]. The combination of a large cardiothymic shadow and a pericardial effusion is characteristic of an intrapericardial teratoma and should cause suspicion of this diagnosis [5]. Echocardiography again will confirm the diagnosis. Although these tumors are histologically benign, surgical excision is mandatory because of the hemodynamic repercussion. Preoperative hemodynamic and respiratory stabilization should be achieved as coexisting pulmonary disease, especially in the premature infant, might considerably increase the operative risk. The intrinsic prognosis is generally good because this tumor is well delimited, usually without associated cardiac malformations [2]. With early intrauterine fetal hydrops, the prognosis is certainly worse, and optimal management presents a challenge. In conclusion, this article presents a preterm infant with an intrapericardial teratoma associated with an unexpected and very rare second mediastinal teratoma. Our study is furthermore of clinical interest because prenatal diagnosis improved obstetric management, avoiding hasty decisions and optimizing accurate neona-
Ann Thorac Surg 1998;66:560 –2
tal care. In this case, the decision for surgical excision was taken after hemodynamic and respiratory stabilization. The life-threatening cardiac tamponade was released in time and the final recovery was complete.
References 1. Todros T, Gaglioti P, Presbitero P. Management of a fetus with intrapericardial teratoma diagnosed in utero. J Ultrasound Med 1991;10:287–90. 2. Perez-Aytes A, Sanchis N, Barbal A, et al. Nonimmunological hydrops fetalis and intrapericardial teratoma: case report and review. Prenatal Diagn 1995;15:859– 63. 3. De Geeter B, Kretz J, Nisand I, Eisenmann B, Kieny M, Kieny R. Intrapericardial teratoma in a newborn infant: use of fetal echocardiography. Ann Thorac Surg 1983;35:664– 6. 4. Rheuban K, McDaniel N, Feldman P, Mayes D, Rodgers B. Intrapericardial teratoma causing nonimmune hydrops fetalis and pericardial tamponade: a case report. Pediatr Cardiol 1991;12:54– 6. 5. Sumner T, Crowe J, Klein A, McKone R, Weaver R. Intrapericardial teratoma in infancy. Pediatr Radiol 1980;10:51–3. 6. Guirgis N, Azancot A, Blot P. Unusual evolution of a pericardial mass diagnosed by fetal echocardiography. Pediatr Cardiol 1991;12:57–9. 7. Zerella J, Halpe D. Intrapericardial teratoma—neonatal cardiorespiratory distress amenable to surgery. J Pediatr Surg 1980;15:961–3.
Isolated Tricuspid Regurgitation Caused by a Dilated Tricuspid Annulus Akira Marui, MD, Takaaki Mochizuki, MD, Norimasa Mitsui, MD, Tadaaki Koyama, MD, and Mayumi Horibe, MD Division of Cardiovascular Surgery and Department of Anesthesiology, Akane-Foundation Tsuchiya General Hospital, Hiroshima, Japan
In most of the previously reported cases of isolated tricuspid regurgitation, both tricuspid leaflets and subvalvar tissue have been absent, hypoplastic, or fused. For this reason, tricuspid valvoplasty was difficult and valve replacement was adopted in many cases. In the present case of a 52-year-old man, however, the tricuspid valve showed no abnormalities other than a severely dilated tricuspid annulus. Ring annuloplasty was performed, and this resulted in a subsequent satisfactory course without anticoagulant therapy. (Ann Thorac Surg 1998;66:560 –2) © 1998 by The Society of Thoracic Surgeons
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solated tricuspid regurgitation (TR) is extremely rare. In the present case, functional tricuspid regurgitation was ruled out because there was no pulmonary hyper-
Accepted for publication Feb 28, 1998. Address reprint requests to Dr Marui, Division of Cardiovascular Surgery, Akane-Foundation Tsuchiya General Hospital, 3-30 Nakajima, Naka, Hiroshima 730, Japan (e-mail:
[email protected]).
© 1998 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
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Fetal cardiac tamponade due to an intrapericardial teratoma Tim Tollens, Filip Casselman, Hugo Devlieger, Marc H. Gewillig, Kamiel Vandenberghe, Toni E. Lerut and Willem J. Daenen Ann Thorac Surg 1998;66:559-560
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