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Chylopericardium with symptoms of tamponade on the grounds of extensive neck vein thrombosis

Chylopericardium with symptoms of tamponade on the grounds of extensive neck vein thrombosis Chylopericardium is a recognized complication of thoracic trauma, surgery or malignancy. Idiopathic or primary presenta- tions, however, are rarely encountered in clinical practice. The severity of its presentation varies from the complete absence of symptoms to cardiac tamponade. We present the case of a 23-year-old woman with chylopericardium and extensive neck vein thrombosis that was managed surgically with a pericardial window. INTRODUCTION CASE REPORT Chylopericardium is a rare clinical entity that is characterized A 23-year-old woman was admitted with dyspnoea and hypo- by the presence of chyle in the pericardial cavity [1]. Its associ- tension of recent onset. Her past medical history was signifi- ation with lymphangiomas, cystic hygromas, thoracic and car- cant only for endometriosis. She had been admitted into diac surgery, trauma, radiation and malignancy is well reported hospital 2 weeks earlier due to a community acquired pneumo- [2, 3]. However, primary chylopericardium, in which there is nia with parapneumonic collection, which required drainage absence of known precipitating factors, is rarely encountered in and intravenous antibiotics. Her initial recovery was uneventful clinical practice. and thus the patient was discharged home a few days later. On The severity of clinical manifestations of chylopericardium this admission, her initial work-up with routine blood tests and is varied and may range from the complete absence of symp- biochemistry was unremarkable. toms to cardiac tamponade, and thus a high index of suspicion A large cardiac shadow was seen on the chest radiogram, is required for the prompt recognition of the underlying diag- and pericardial effusion confirmed on computed tomog- nosis and management of its sequalae [2]. raphy (CT) of the thorax. The scan also demonstrated dif- In this report, we present a case of chylopericardium in a fuse lymphadenopathy as well as extensive and multifocal 23-year-old woman, presenting with acute shortness of breath thrombosis of the internal jugular veins bilaterally, the bra- in the background of extensive neck vein and superior vena chiocephalic vein and of the SVC, managed with therapeutic cava (SVC) thrombosis, of unknown aetiology. dose of subcutaneous tinzaparin. Further diagnostic screening Received: November 21, 2016. Accepted: January 1, 2017 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2017. 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 1 2 M. Nardini et al. for rheumatological, auto-immune and infectious causes was unremarkable, whilst lymph node biopsies were negative for malignancy. Aspiration of the pericardial collection was sug- gestive of the presence of chyle, a suspicion that was confirmed on biochemical analysis of the aspirate. Cytology and cultures of the aspiratewerenegative for malignant and infectious causes, respectively (Fig. 1). After pericardiocentesis, the patient had recurrence and was thus taken to theatre, underwent an anterior left mini- thoracotomy, through which a pericardial window was fash- ioned, 600 ml of chyle were collected and an 18 Fr drain was placed in the pleural cavity. Her postoperative course was uneventful, drain removed on Day 3, and she was discharged Figure 1: Pre-operative chest radiograph and axial view of a CT thorax, demon- home as the collection and her symptoms had resolved. She strating a large pericardial collection. remains symptom and collection free at 1 year postoperatively, with no evidence of recurrence on follow-up chest radiographs. The patient remains symptom-free a year postoperatively, and has been put on a surveillance programme with frequent follow-up. DISCUSSION In conclusion, chylopericardium is a clinical entity that is not frequently encountered in the daily clinical practice and The association of chylopericardium with trauma, previous requires a high index of suspicion. Its management needs to thoracic or cardiac surgery, congenital lymphangiomas, radi- address both the prevention of cardiac tamponade and the cor- ation and malignancy is well recognized [2–5]. However, pri- rection of nutritional and metabolic compromise that can mary chylopericardium is a clinical entity that is more rarely ensue. encountered and only diagnosed if all known precipitants have been excluded [6–8]. Its pathophysiology remains controversial, and is thought to REFERENCES be related to either elevated pressure in the thoracic duct, or to abnormal communications between the thoracic duct and the 1. Hasebrock K. Analyse einer chylosen pericardialen lymphatics of the pericardium [3, 6]. Clinical manifestations Flussigkeit (Chylopericardium). Ztschr Physiol Chemie 1888; are varied as it is the age spectrum of patients affected, with 12:289–94. reports of chylopericardium in neonates through to older 2. Chan BB, Murphy MC, Rodgers BM. Management of chylo- adult [8]. pericardium. J Pediatr Surg 1990;25:1185–9. The imaging modalities that can be used to ascertain the 3. Dib C, Tajik AJ, Park S, Kheir ME, Khandieria B, Mookadam F. underlying cause, with varying specificity and sensitivity, Chylopericardium in adults: a literature review over the include lymphangioscintigraphy, lymphangiography, monitor- past decade (1996–2006). J Thorac Cardiovasc Surg 2008;136: ing of chest radioactivity after oral intake of I-triolein and 650–6. observation of the distribution of Sudan III dye in the pericar- 4. Al Jaaly E, Baig K, Patni R, Anderson J, Haskard DO. Surgical dial cavity after ingestion [2, 3]. However, the diagnosis still management of chylopericardium and chylothorax in a relies on the cytology, chemistry and microbiology of pericar- patient with Behcet’s disease. Clin Exp Rheumatol 2011;29: dial aspirate, obtained via pericardiocentesis. S68–70. The management of this condition though controversial has 5. Zhang L, Zu N, Lin B, Wang G. Chylothorax and chyloperi- two main therapeutic aims. The first is the prevention of car- cardium in Behcet’s diseases: case report and literature diac tamponade. This can be achieved via pericardiocentesis or review. Clin Rheumatol 2013;32:1107–11. surgically with the formation of a pericardial window/partial 6. Cho BC, Kang SM, Lee SC, Moon JG, Lee DH, Lim SH. Primary pericardiectomy, which can be combined with ligation of the Idiopathic chylopericardium associated with cervicome- thoracic duct and by conventional open methods or via video- diastinal cystic hygroma. Yonsei Med J 2005;46:439–44. assisted thoracoscopic surgery (VATS). The second aim is the 7. Itkin M, Swe NM, Shapiro SE, Shrager JB. Spontaneous chy- avoidance of metabolic, nutritional and immunological com- lopericardium: delineation of the underlying anatomic promise, resulting from the loss of chyle, via a diet rich in pathology by CT lymphangiography. Ann Thorac Surg 2009; medium chain triglycerides or, if necessary total parenteral 87:1595–7. nutrition [2, 9, 10]. 8. Musemeche CA, Riveron FA, Backer CL, Zales VR, Idriss FS. In this instance, we decided to employ a stepwise Massive primary chylopericardium: a case report. J Pediatr approach to the management of this patient. As pericardio- Surg 1990;25:840–2. centesis is associated with a high incidence of recurrence, we 9. Azizkhan RG, Canfield J, Alford BA, Rodgers BM. opted for the pericardial window, combined with dietetic Pleuroperitoneal shunts in the management of neonatal input to address the patient’s nutritional requirements. The chylothorax. J Pediatr Surg 1983;18:842–50. patient having been informed of her options, opted for the 10. Milsom JW, Kron IL, Rheuban KS, Rodgers BM. Chylothorax: option of conventional surgery in preference to VATS, a meth- an assessment of current surgical management. J Thorac od that can also be safely used for the same purpose [2–4]. Cardiovasc Surg 1985;89:221–7. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Chylopericardium with symptoms of tamponade on the grounds of extensive neck vein thrombosis

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Oxford University Press
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Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2017.
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2042-8812
DOI
10.1093/jscr/rjw242
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Abstract

Chylopericardium is a recognized complication of thoracic trauma, surgery or malignancy. Idiopathic or primary presenta- tions, however, are rarely encountered in clinical practice. The severity of its presentation varies from the complete absence of symptoms to cardiac tamponade. We present the case of a 23-year-old woman with chylopericardium and extensive neck vein thrombosis that was managed surgically with a pericardial window. INTRODUCTION CASE REPORT Chylopericardium is a rare clinical entity that is characterized A 23-year-old woman was admitted with dyspnoea and hypo- by the presence of chyle in the pericardial cavity [1]. Its associ- tension of recent onset. Her past medical history was signifi- ation with lymphangiomas, cystic hygromas, thoracic and car- cant only for endometriosis. She had been admitted into diac surgery, trauma, radiation and malignancy is well reported hospital 2 weeks earlier due to a community acquired pneumo- [2, 3]. However, primary chylopericardium, in which there is nia with parapneumonic collection, which required drainage absence of known precipitating factors, is rarely encountered in and intravenous antibiotics. Her initial recovery was uneventful clinical practice. and thus the patient was discharged home a few days later. On The severity of clinical manifestations of chylopericardium this admission, her initial work-up with routine blood tests and is varied and may range from the complete absence of symp- biochemistry was unremarkable. toms to cardiac tamponade, and thus a high index of suspicion A large cardiac shadow was seen on the chest radiogram, is required for the prompt recognition of the underlying diag- and pericardial effusion confirmed on computed tomog- nosis and management of its sequalae [2]. raphy (CT) of the thorax. The scan also demonstrated dif- In this report, we present a case of chylopericardium in a fuse lymphadenopathy as well as extensive and multifocal 23-year-old woman, presenting with acute shortness of breath thrombosis of the internal jugular veins bilaterally, the bra- in the background of extensive neck vein and superior vena chiocephalic vein and of the SVC, managed with therapeutic cava (SVC) thrombosis, of unknown aetiology. dose of subcutaneous tinzaparin. Further diagnostic screening Received: November 21, 2016. Accepted: January 1, 2017 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2017. 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 1 2 M. Nardini et al. for rheumatological, auto-immune and infectious causes was unremarkable, whilst lymph node biopsies were negative for malignancy. Aspiration of the pericardial collection was sug- gestive of the presence of chyle, a suspicion that was confirmed on biochemical analysis of the aspirate. Cytology and cultures of the aspiratewerenegative for malignant and infectious causes, respectively (Fig. 1). After pericardiocentesis, the patient had recurrence and was thus taken to theatre, underwent an anterior left mini- thoracotomy, through which a pericardial window was fash- ioned, 600 ml of chyle were collected and an 18 Fr drain was placed in the pleural cavity. Her postoperative course was uneventful, drain removed on Day 3, and she was discharged Figure 1: Pre-operative chest radiograph and axial view of a CT thorax, demon- home as the collection and her symptoms had resolved. She strating a large pericardial collection. remains symptom and collection free at 1 year postoperatively, with no evidence of recurrence on follow-up chest radiographs. The patient remains symptom-free a year postoperatively, and has been put on a surveillance programme with frequent follow-up. DISCUSSION In conclusion, chylopericardium is a clinical entity that is not frequently encountered in the daily clinical practice and The association of chylopericardium with trauma, previous requires a high index of suspicion. Its management needs to thoracic or cardiac surgery, congenital lymphangiomas, radi- address both the prevention of cardiac tamponade and the cor- ation and malignancy is well recognized [2–5]. However, pri- rection of nutritional and metabolic compromise that can mary chylopericardium is a clinical entity that is more rarely ensue. encountered and only diagnosed if all known precipitants have been excluded [6–8]. Its pathophysiology remains controversial, and is thought to REFERENCES be related to either elevated pressure in the thoracic duct, or to abnormal communications between the thoracic duct and the 1. Hasebrock K. Analyse einer chylosen pericardialen lymphatics of the pericardium [3, 6]. Clinical manifestations Flussigkeit (Chylopericardium). Ztschr Physiol Chemie 1888; are varied as it is the age spectrum of patients affected, with 12:289–94. reports of chylopericardium in neonates through to older 2. Chan BB, Murphy MC, Rodgers BM. Management of chylo- adult [8]. pericardium. J Pediatr Surg 1990;25:1185–9. The imaging modalities that can be used to ascertain the 3. Dib C, Tajik AJ, Park S, Kheir ME, Khandieria B, Mookadam F. underlying cause, with varying specificity and sensitivity, Chylopericardium in adults: a literature review over the include lymphangioscintigraphy, lymphangiography, monitor- past decade (1996–2006). J Thorac Cardiovasc Surg 2008;136: ing of chest radioactivity after oral intake of I-triolein and 650–6. observation of the distribution of Sudan III dye in the pericar- 4. Al Jaaly E, Baig K, Patni R, Anderson J, Haskard DO. Surgical dial cavity after ingestion [2, 3]. However, the diagnosis still management of chylopericardium and chylothorax in a relies on the cytology, chemistry and microbiology of pericar- patient with Behcet’s disease. Clin Exp Rheumatol 2011;29: dial aspirate, obtained via pericardiocentesis. S68–70. The management of this condition though controversial has 5. Zhang L, Zu N, Lin B, Wang G. Chylothorax and chyloperi- two main therapeutic aims. The first is the prevention of car- cardium in Behcet’s diseases: case report and literature diac tamponade. This can be achieved via pericardiocentesis or review. Clin Rheumatol 2013;32:1107–11. surgically with the formation of a pericardial window/partial 6. Cho BC, Kang SM, Lee SC, Moon JG, Lee DH, Lim SH. Primary pericardiectomy, which can be combined with ligation of the Idiopathic chylopericardium associated with cervicome- thoracic duct and by conventional open methods or via video- diastinal cystic hygroma. Yonsei Med J 2005;46:439–44. assisted thoracoscopic surgery (VATS). The second aim is the 7. Itkin M, Swe NM, Shapiro SE, Shrager JB. Spontaneous chy- avoidance of metabolic, nutritional and immunological com- lopericardium: delineation of the underlying anatomic promise, resulting from the loss of chyle, via a diet rich in pathology by CT lymphangiography. Ann Thorac Surg 2009; medium chain triglycerides or, if necessary total parenteral 87:1595–7. nutrition [2, 9, 10]. 8. Musemeche CA, Riveron FA, Backer CL, Zales VR, Idriss FS. In this instance, we decided to employ a stepwise Massive primary chylopericardium: a case report. J Pediatr approach to the management of this patient. As pericardio- Surg 1990;25:840–2. centesis is associated with a high incidence of recurrence, we 9. Azizkhan RG, Canfield J, Alford BA, Rodgers BM. opted for the pericardial window, combined with dietetic Pleuroperitoneal shunts in the management of neonatal input to address the patient’s nutritional requirements. The chylothorax. J Pediatr Surg 1983;18:842–50. patient having been informed of her options, opted for the 10. Milsom JW, Kron IL, Rheuban KS, Rodgers BM. Chylothorax: option of conventional surgery in preference to VATS, a meth- an assessment of current surgical management. J Thorac od that can also be safely used for the same purpose [2–4]. Cardiovasc Surg 1985;89:221–7.

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Journal of Surgical Case ReportsOxford University Press

Published: Jan 25, 2017

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