552072
research-article2014
TAJ0010.1177/2040622314552072Therapeutic Advances in Chronic DiseaseJ. B. Morjaria et al.
Therapeutic Advances in Chronic Disease
Review
Bilateral chylothorax: an unusual complication of cervical rib resection Jaymin B. Morjaria, Imran Aslam, Brian Johnson, Michael A. Greenstone and Jack A. Kastelik
Ther Adv Chronic Dis 2015, Vol. 6(1) 29–33 DOI: 10.1177/ 2040622314552072 © The Author(s), 2014. Reprints and permissions: http://www.sagepub.co.uk/ journalsPermissions.nav
Abstract: Bilateral chylothorax is a rare cause of pleural effusions. Here we report an unusual acute presentation of bilateral chylothorax following thoracic outlet surgery. Unique to this case was the disparate characteristics of pleural fluid analyses with an exudate on the left and a transudate on the right. This report describes the recognition and management of bilateral chylothoraces, an uncommon but potentially serious complication of this frequently performed surgical procedure.
Keywords: bilateral chylothorax, cervical rib, exudative effusion, pleural effusion, transudative effusion Introduction Chylothorax is characterised by a milky white pleural aspirate and is an uncommon cause of pleural effusion with a reported incidence in adults of around 3% [Mcgrath et al. 2010]. The causes of chylothorax are numerous and may be broadly classified into traumatic including iatrogenic injuries, medical or oncological, and miscellaneous [Sassoon and Light, 1985]. Very rarely, chylothorax may be bilateral. Here we report an unusual case of bilateral chylothorax secondary to thoracic duct damage following surgery of a symptomatic cervical rib and conduct a literature review on the topic. Written informed consent was obtained from the patient. Case report An 18-year-old female with no past medical history of note presented with few months’ history of symptoms of non-posture-dependent blueness and numbness of the left forearm. Clinical examination revealed a firm swelling in the left supraclavicular fossa with a prominent subclavian pulse and normal radial pulses bilaterally. Adson’s and Roos’ test were positive indicating the possibility of thoracic outlet syndrome secondary to a cervical rib. The chest and cervical spine radiographs were both unremarkable. A duplex scan confirmed stressing of the subclavian artery at the shoulder joint, indicating thoracic outlet obstruction. Conservative management with
physiotherapy and shoulder girdle strengthening exercises were conducted with little benefit. Hence, following review of the case by the multidisciplinary team of vascular surgeons and radiologists who felt that there was a definite left cervical rib surgical resection via a cervical approach was performed. Although the surgery was uneventful, 12 hours postoperatively the patient developed crushing chest pain and respiratory distress. Clinical examination revealed that the patient was diaphoretic, tachypnoeic with a respiratory rate of 34/minute and oxygen saturations of 91% on room air. Thoracic examination revealed dullness to percussion and decreased air entry at the left base. The electrocardiogram demonstrated sinus tachycardia with no evidence of right heart strain or myocardial ischaemia. Chest radiograph confirmed a moderate leftsided pleural effusion initially, and when repeated due to progressive symptoms demonstrated bilateral pleural effusions (left larger than right) (Figure 1A). A computed tomography (CT) pulmonary angiogram reconfirmed the presence of bilateral pleural effusions but no pulmonary emboli (Figure 1B). Bilateral intercostal chest drains were inserted under ultrasound guidance and drainage of milky pleural fluid was observed. Analysis of the pleural effusions confirmed chylothoraces; however, the left was an exudate and the right a transudate (Table 1). Within 24 hours 600 and 500 ml of chylous fluid were drained on the left and right sides, respectively. The patient’s
Correspondence to: Jaymin B. Morjaria, MBBS, MD Department of Respiratory Medicine, Hull & East Yorkshire Hospitals NHS Trust, Castle Hill Hospital, Cottingham, UK jaymin.morjaria@hey. nhs.uk Imran Aslam, MBBS Department of Respiratory Medicine, Hull & East Yorkshire Hospitals NHS Trust, Castle Hill Hospital, Cottingham, UK Brian Johnson, MBBS Department of Vascular Surgery, Hull & East Yorkshire Hospitals NHS Trust, Hull Royal Infirmary, Hull, UK Michael A. Greenstone, MBChB, MD Department of Respiratory Medicine, Hull & East Yorkshire Hospitals NHS Trust, Castle Hill Hospital, Cottingham, UK Jack A. Kastelik, MBBS, MD Department of Respiratory Medicine, Hull & East Yorkshire Hospitals NHS Trust, Castle Hill Hospital, Cottingham, UK
http://taj.sagepub.com 29
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Therapeutic Advances in Chronic Disease 6(1) spontaneously therefore not requiring further intervention. The predischarge chest radiograph and that performed at 4-week review demonstrated no evidence of recurrence of the pleural effusions. Moreover, the patient reported resolution of her thoracic outlet obstruction and absence of respiratory symptoms.
Figure 1. (A) Chest radiograph with bilateral effusions in our patient. (B) A computed tomography scan demonstrating bilateral pleural effusions in our patient. The arrows indicate the position of the intercostal chest drains.
Discussion Here we present a unique case with two interesting aspects. First, we describe a rare complication of thoracic outlet surgery in the form of bilateral chylothoraces and secondly, an unusual discordance between the biochemical characteristics of the two effusions. Chylothorax is defined as the presence of chyle within the thoracic cavity, and usually occurs as a result of damage to the thoracic duct or one of its tributaries and needs distinguishing from empyema and pseudochylothorax (Table 1) [Sassoon and Light, 1985; Mcgrath et al. 2010]. Malignant infiltration of the thoracic duct, particularly by lymphoma, may account for up to 50% of non-trauma-related cases; whilst the diverse group of other recognised causes, tuberculosis, sarcoidosis, cardiac failure, amyloidosis, superior vena cava obstruction, lymphangioleiomyomatosis and congenital abnormalities, are characterised by a propensity to compress or distend the lymphatic system [Sassoon and Light, 1985]. Traumatic causes of chylothorax are usually iatrogenic as a complication of thoracic or head and neck surgery, or noniatrogenic which can be secondary to knife or gunshot wounds, or
Table 1. Differences between pseudochylothorax and chylothorax, alongside the biochemistry of the pleural fluid from our patient bilaterally. Pleural fluid
Pseudochylothorax
Chylothorax
Left-side pleural fluid
Right-side pleural fluid
Protein (g/l) LDH (iu/l) Triglyceride (mmol/l) Cholesterol (mmol/l)
Usually >29 Usually >260 29 Usually 1.24
>5.18
29 g/l and LDH >300 iu/l. LDH, lactate dehydrogenase.
symptoms improved and after 3 days fluid ceased draining and both intercostal chest drains were removed. For this reason no further intervention such as octeotride, diet therapy or further radiological investigations or surgery were undertaken. In our opinion, the most likely explanation for this would be that the leak settled down
raised intrathoracic pressure during childbirth, forceful emesis or coughing. In our patient, there was no history of marked coughing or vomiting in the postoperative period, therefore the most likely aetiology for her chylothoraces would be iatrogenic trauma complicating her thoracic outlet surgery.
30 http://taj.sagepub.com
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JB Morjaria, I Aslam et al. Vascular or nerve damage are the most frequently reported complications of thoracic outlet surgery and chylothorax is exceptional [Urschel and Kourliset, 2007]. A literature review of three large observational cohorts involving outcomes of 1938 thoracic outlet operations reported only three cases of postoperative chyle leakages which were all unilateral and resulted from accidental traumatic damage of an anatomical variant of the thoracic duct or its tributaries [Sanders and Pearce,1989; Hempel et al. 1996; Leffert, 2004]. In one of the patients the chylothorax resolved spontaneously and in the other two surgical intervention was necessary. The thoracic duct originates as the cisterna chyli in the abdomen, runs for approximately 38–40 cm entering the thorax through the diaphragmatic aortic opening and ends at the left jugulo–subclavian junction [Sassoon and Light, 1985]. Anatomical variations occur in approximately 35% of cases, thus unsurprisingly its damage during surgery. Damage to the thoracic duct below the fifth or sixth thoracic vertebrae (50% of cases) manifests as a rightsided chylothorax, whilst damage above this level (one-third of cases) results in a left-sided chylothorax [Bessone et al. 1971]. The rest may be bilateral, which occurs when the thoracic duct crosses the midline at the level of the fifth or sixth thoracic vertebrae. Cervical rib resection involves removing fibro-osseous structures attached to the scalene tubercle above the first rib and taking structures as close as possible to the transverse process of the seventh cervical vertebra. If the suprapleural membrane behind the first rib (Sibson’s Fascia) is breached then chyle can enter the pleural space directly. In our patient it is likely that the damage occurred above the fourth thoracic vertebra, hence the chylothorax should have been left-sided. Although we did not confirm through radiological testing, it is possible that an anatomical variation of the thoracic duct or unusual tributaries resulted in bilateral chylothoraces in our patient. Chylothoraces are typically exudative, but transudative characteristics have been reported with liver cirrhosis, lymphoproliferative disorders, radiation, nephrotic syndrome, amyloidosis, and obstruction of the superior vena cava [Maldonado et al. 2009; Mcgrath et al. 2010]. In a review of 74 patients with chylothoraces, 10 effusions had characteristics of a transudate, of which 2 were attributed to cardiothoracic or oesophageal surgical procedures but none were attributable to thoracic outlet surgery [Maldonado et al. 2009]. A
literature review of all cases of transudative chylothorax until 2005 and a large observational study describing a 5-year experience of 876 pleural fluid aspirates, described 13 and 7 cases of transudative chylothoraces respectively, of which the commonest causes aetiology were liver cirrhosis and cardiac failure although none were attributed to thoracic outlet surgery [Diaz-Guzman et al. 2005; Agrawal et al. 2008]. Therefore, our case is unique in that it describes postsurgical bilateral chylothoraces with the right-side pleural fluid analysis having the characteristics of a transudate. Essentially the protein concentration in chyle is between 20– 30 g/l, hence chylous pleural effusions would be expected to be transudative; however, it is proposed that chylous pleural effusions become exudative as protein levels increase due to fluid reabsorption from the pleural cavity to vascular space [Agrawal et al. 2008]. In our patient the transudate, although unequivocal, may have been an evanescent phenomenon as the rapidity of sampling of the evolving effusion may not have allowed sufficient time for the postulated reabsorption of water and small solutes (with resultant relative increase in protein concentrations) to occur. Following confirmation of a chylothorax, locating the point of leakage within the thoracic duct is pivotal. Should the diagnosis be missed and chyle leakage persist, this may lead to complications such as malnutrition and immunosuppression [Mcgrath et al. 2010]. Treatment of chylothorax depends on its aetiology and may be conservative, minimally invasive or surgical (Figure 2) [Sassoon and Light, 1985]. A conservative approach may resolve approximately 50% of cases of chylothoraces and entails a low-fat diet or octreotide and somatostatin [Rimensberger et al. 1998; Fernandez Alvarez et al. 1999; Al-Zubairy and Al-Jazairi, 2003]. In symptomatic chylothoraces, drainage of the fluid using an intercostal drain may be required as in our case [Weissberg and Ben-Zeev, 1993]. Other minimally invasive techniques include medical thoracoscopy, thoracic duct embolisation or disruption [Mares and Mathur, 1998; Boffa et al. 2008]. Surgery may be the only therapeutic option in cases when fluid losses of >1.5 l/day, more than 1 l/day for 5 days or continuous chyle leak for more than 2 weeks occur [Selle et al. 1973; Marts et al. 1992; Graham et al. 1994]. In our patient bilateral chylothorax was managed by the insertion of intercostals drains bilaterally and the chylous fluid accumulation ceased rapidly suggesting a trivial leak.
http://taj.sagepub.com 31
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Therapeutic Advances in Chronic Disease 6(1) Clinical suspicion of chylothorax
Chest radiograph and/or Thoracic Ultrasound
Ultrasound-guided thoracocentesis
Pleural fluid analysis for pH, protein, LDH, triglycerides, cholesterol, chylomicrons, cytology and microbiology Consider CT thorax and abdomen, lymphangiography or lymphangioscintigraphy Chylothorax confirmed Treat underlying cause, e.g. congestive cardiac failure, sarcoidosis or malignancy
No Yes
Management options
Conservative
Low-fat diet octreotide or somatostatin
Minimally invasive
Chest drain insertion Medical thoracoscopy Thoracic duct embolization or disruption
Surgical
Thoracic duct ligation ± pleurodesis
Figure 2. Flow diagram to aid in the assessment of patients with suspected chylothorax.
Conclusions Bilateral chylothorax is a rare complication of thoracic outlet syndrome surgery and is most likely due to damage to anatomical variants or tributaries of the thoracic duct. The early recognition was important in our case as it allowed for timely intervention and treatment. This case therefore would be of great interest to many specialists as it highlights a rare but potentially serious complication of this common surgical procedure.
Funding This research received no specific grant from any funding agency in the public, commercial, or notfor-profit sectors.
Conflict of interest statement The authors have no conflicts of interest to declare.
Al-Zubairy, S. and Al-Jazairi, A. (2003) Octreotide as a therapeutic option for management of chylothorax. Ann Pharmacother 37: 679–682.
References Agrawal, V., Doelken, P. and Sahn, S. (2008) Pleural fluid analysis in chylous pleural effusion. Chest 133: 1436–1441.
32 http://taj.sagepub.com
Downloaded from taj.sagepub.com at HOWARD UNIV UNDERGRAD LIBRARY on February 12, 2015
JB Morjaria, I Aslam et al. Bessone, L., Ferguson, T. and Burford, T. (1971) Chylothorax. Ann Thorac Surg 12: 527–550. Boffa, D., Sands, M., Rice, T., Murthy, S., Mason, D., Geisinger, M. et al. (2008) A critical evaluation of a percutaneous diagnostic and treatment strategy for chylothorax after thoracic surgery. Eur J Cardiothorac Surg 33: 435–439.
Mares, D. and Mathur, P. (1998) Medical thoracoscopic talc pleurodesis for chylothorax due to lymphoma: a case series. Chest 114: 731–735. Marts, B., Naunheim, K., Fiore, A. and Pennington, D. (1992) Conservative versus surgical management of chylothorax. Am J Surg 164: 532–534; discussion 534–535.
Diaz-Guzman, E., Culver, D. and Stoller, J. (2005) Transudative chylothorax: report of two cases and review of the literature. Lung 183: 169–175.
Mcgrath, E., Blades, Z. and Anderson, P. (2010) Chylothorax: aetiology, diagnosis and therapeutic options. Respir Med 104: 1–8.
Fernandez Alvarez, J., Kalache, K. and Grauel, E. (1999) management of spontaneous congenital chylothorax: oral medium-chain triglycerides versus total parenteral nutrition. Am J Perinatol 16: 415–420.
Rimensberger, P., Muller-Schenker, B., Kalangos, A. and Beghetti, M. (1998) Treatment of a persistent postoperative chylothorax with somatostatin. Ann Thorac Surg 66: 253–254.
Graham, D., Mcgahren, E., Tribble, C., Daniel, T. and Rodgers, B. (1994) Use of video-assisted thoracic surgery in the treatment of chylothorax. Ann Thorac Surg 57: 1507–1511; discussion 1511–1502. Hempel, G., Shutze, W., Anderson, J. and Bukhari, H. (1996) 770 consecutive supraclavicular first rib resections for thoracic outlet syndrome. Ann Vasc Surg 10: 456–463. Leffert, R. (2004) Complications of surgery for thoracic outlet syndrome. Hand Clin 20: 91–98. Maldonado, F., Hawkins, F., Daniels, C., Doerr, C., Decker, P. and Ryu, J.H. (2009) Pleural fluid characteristics of chylothorax. Mayo Clin Proc 84: 129–133.
Sanders, R. and Pearce, W. (1989) The treatment of thoracic outlet syndrome: a comparison of different operations. J Vasc Surg 10: 626–634. Sassoon, C. and Light, R. (1985) Chylothorax and pseudochylothorax. Clin Chest Med 6: 163–171. Selle, J., Snyder, W., III and Schreiber, J. (1973) Chylothorax: indications for surgery. Ann Surg 177: 245–249. Urschel, H. and Kourlis, H. (2007) Thoracic outlet syndrome: a 50-year experience at Baylor University Medical Center. Proc (Bayl Univ Med Cent) 20: 125–135. Weissberg, D. and Ben-Zeev, I. (1993) Talc pleurodesis. experience with 360 patients. J Thorac Cardiovasc Surg 106: 689–695.
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research-article2014
TAJ0010.1177/2040622314552072Therapeutic Advances in Chronic DiseaseJ. B. Morjaria et al.
Therapeutic Advances in Chronic Disease
Review
Bilateral chylothorax: an unusual complication of cervical rib resection Jaymin B. Morjaria, Imran Aslam, Brian Johnson, Michael A. Greenstone and Jack A. Kastelik
Ther Adv Chronic Dis 2015, Vol. 6(1) 29–33 DOI: 10.1177/ 2040622314552072 © The Author(s), 2014. Reprints and permissions: http://www.sagepub.co.uk/ journalsPermissions.nav
Abstract: Bilateral chylothorax is a rare cause of pleural effusions. Here we report an unusual acute presentation of bilateral chylothorax following thoracic outlet surgery. Unique to this case was the disparate characteristics of pleural fluid analyses with an exudate on the left and a transudate on the right. This report describes the recognition and management of bilateral chylothoraces, an uncommon but potentially serious complication of this frequently performed surgical procedure.
Keywords: bilateral chylothorax, cervical rib, exudative effusion, pleural effusion, transudative effusion Introduction Chylothorax is characterised by a milky white pleural aspirate and is an uncommon cause of pleural effusion with a reported incidence in adults of around 3% [Mcgrath et al. 2010]. The causes of chylothorax are numerous and may be broadly classified into traumatic including iatrogenic injuries, medical or oncological, and miscellaneous [Sassoon and Light, 1985]. Very rarely, chylothorax may be bilateral. Here we report an unusual case of bilateral chylothorax secondary to thoracic duct damage following surgery of a symptomatic cervical rib and conduct a literature review on the topic. Written informed consent was obtained from the patient. Case report An 18-year-old female with no past medical history of note presented with few months’ history of symptoms of non-posture-dependent blueness and numbness of the left forearm. Clinical examination revealed a firm swelling in the left supraclavicular fossa with a prominent subclavian pulse and normal radial pulses bilaterally. Adson’s and Roos’ test were positive indicating the possibility of thoracic outlet syndrome secondary to a cervical rib. The chest and cervical spine radiographs were both unremarkable. A duplex scan confirmed stressing of the subclavian artery at the shoulder joint, indicating thoracic outlet obstruction. Conservative management with
physiotherapy and shoulder girdle strengthening exercises were conducted with little benefit. Hence, following review of the case by the multidisciplinary team of vascular surgeons and radiologists who felt that there was a definite left cervical rib surgical resection via a cervical approach was performed. Although the surgery was uneventful, 12 hours postoperatively the patient developed crushing chest pain and respiratory distress. Clinical examination revealed that the patient was diaphoretic, tachypnoeic with a respiratory rate of 34/minute and oxygen saturations of 91% on room air. Thoracic examination revealed dullness to percussion and decreased air entry at the left base. The electrocardiogram demonstrated sinus tachycardia with no evidence of right heart strain or myocardial ischaemia. Chest radiograph confirmed a moderate leftsided pleural effusion initially, and when repeated due to progressive symptoms demonstrated bilateral pleural effusions (left larger than right) (Figure 1A). A computed tomography (CT) pulmonary angiogram reconfirmed the presence of bilateral pleural effusions but no pulmonary emboli (Figure 1B). Bilateral intercostal chest drains were inserted under ultrasound guidance and drainage of milky pleural fluid was observed. Analysis of the pleural effusions confirmed chylothoraces; however, the left was an exudate and the right a transudate (Table 1). Within 24 hours 600 and 500 ml of chylous fluid were drained on the left and right sides, respectively. The patient’s
Correspondence to: Jaymin B. Morjaria, MBBS, MD Department of Respiratory Medicine, Hull & East Yorkshire Hospitals NHS Trust, Castle Hill Hospital, Cottingham, UK jaymin.morjaria@hey. nhs.uk Imran Aslam, MBBS Department of Respiratory Medicine, Hull & East Yorkshire Hospitals NHS Trust, Castle Hill Hospital, Cottingham, UK Brian Johnson, MBBS Department of Vascular Surgery, Hull & East Yorkshire Hospitals NHS Trust, Hull Royal Infirmary, Hull, UK Michael A. Greenstone, MBChB, MD Department of Respiratory Medicine, Hull & East Yorkshire Hospitals NHS Trust, Castle Hill Hospital, Cottingham, UK Jack A. Kastelik, MBBS, MD Department of Respiratory Medicine, Hull & East Yorkshire Hospitals NHS Trust, Castle Hill Hospital, Cottingham, UK
http://taj.sagepub.com 29
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Therapeutic Advances in Chronic Disease 6(1) spontaneously therefore not requiring further intervention. The predischarge chest radiograph and that performed at 4-week review demonstrated no evidence of recurrence of the pleural effusions. Moreover, the patient reported resolution of her thoracic outlet obstruction and absence of respiratory symptoms.
Figure 1. (A) Chest radiograph with bilateral effusions in our patient. (B) A computed tomography scan demonstrating bilateral pleural effusions in our patient. The arrows indicate the position of the intercostal chest drains.
Discussion Here we present a unique case with two interesting aspects. First, we describe a rare complication of thoracic outlet surgery in the form of bilateral chylothoraces and secondly, an unusual discordance between the biochemical characteristics of the two effusions. Chylothorax is defined as the presence of chyle within the thoracic cavity, and usually occurs as a result of damage to the thoracic duct or one of its tributaries and needs distinguishing from empyema and pseudochylothorax (Table 1) [Sassoon and Light, 1985; Mcgrath et al. 2010]. Malignant infiltration of the thoracic duct, particularly by lymphoma, may account for up to 50% of non-trauma-related cases; whilst the diverse group of other recognised causes, tuberculosis, sarcoidosis, cardiac failure, amyloidosis, superior vena cava obstruction, lymphangioleiomyomatosis and congenital abnormalities, are characterised by a propensity to compress or distend the lymphatic system [Sassoon and Light, 1985]. Traumatic causes of chylothorax are usually iatrogenic as a complication of thoracic or head and neck surgery, or noniatrogenic which can be secondary to knife or gunshot wounds, or
Table 1. Differences between pseudochylothorax and chylothorax, alongside the biochemistry of the pleural fluid from our patient bilaterally. Pleural fluid
Pseudochylothorax
Chylothorax
Left-side pleural fluid
Right-side pleural fluid
Protein (g/l) LDH (iu/l) Triglyceride (mmol/l) Cholesterol (mmol/l)
Usually >29 Usually >260 29 Usually 1.24
>5.18
29 g/l and LDH >300 iu/l. LDH, lactate dehydrogenase.
symptoms improved and after 3 days fluid ceased draining and both intercostal chest drains were removed. For this reason no further intervention such as octeotride, diet therapy or further radiological investigations or surgery were undertaken. In our opinion, the most likely explanation for this would be that the leak settled down
raised intrathoracic pressure during childbirth, forceful emesis or coughing. In our patient, there was no history of marked coughing or vomiting in the postoperative period, therefore the most likely aetiology for her chylothoraces would be iatrogenic trauma complicating her thoracic outlet surgery.
30 http://taj.sagepub.com
Downloaded from taj.sagepub.com at HOWARD UNIV UNDERGRAD LIBRARY on February 12, 2015
JB Morjaria, I Aslam et al. Vascular or nerve damage are the most frequently reported complications of thoracic outlet surgery and chylothorax is exceptional [Urschel and Kourliset, 2007]. A literature review of three large observational cohorts involving outcomes of 1938 thoracic outlet operations reported only three cases of postoperative chyle leakages which were all unilateral and resulted from accidental traumatic damage of an anatomical variant of the thoracic duct or its tributaries [Sanders and Pearce,1989; Hempel et al. 1996; Leffert, 2004]. In one of the patients the chylothorax resolved spontaneously and in the other two surgical intervention was necessary. The thoracic duct originates as the cisterna chyli in the abdomen, runs for approximately 38–40 cm entering the thorax through the diaphragmatic aortic opening and ends at the left jugulo–subclavian junction [Sassoon and Light, 1985]. Anatomical variations occur in approximately 35% of cases, thus unsurprisingly its damage during surgery. Damage to the thoracic duct below the fifth or sixth thoracic vertebrae (50% of cases) manifests as a rightsided chylothorax, whilst damage above this level (one-third of cases) results in a left-sided chylothorax [Bessone et al. 1971]. The rest may be bilateral, which occurs when the thoracic duct crosses the midline at the level of the fifth or sixth thoracic vertebrae. Cervical rib resection involves removing fibro-osseous structures attached to the scalene tubercle above the first rib and taking structures as close as possible to the transverse process of the seventh cervical vertebra. If the suprapleural membrane behind the first rib (Sibson’s Fascia) is breached then chyle can enter the pleural space directly. In our patient it is likely that the damage occurred above the fourth thoracic vertebra, hence the chylothorax should have been left-sided. Although we did not confirm through radiological testing, it is possible that an anatomical variation of the thoracic duct or unusual tributaries resulted in bilateral chylothoraces in our patient. Chylothoraces are typically exudative, but transudative characteristics have been reported with liver cirrhosis, lymphoproliferative disorders, radiation, nephrotic syndrome, amyloidosis, and obstruction of the superior vena cava [Maldonado et al. 2009; Mcgrath et al. 2010]. In a review of 74 patients with chylothoraces, 10 effusions had characteristics of a transudate, of which 2 were attributed to cardiothoracic or oesophageal surgical procedures but none were attributable to thoracic outlet surgery [Maldonado et al. 2009]. A
literature review of all cases of transudative chylothorax until 2005 and a large observational study describing a 5-year experience of 876 pleural fluid aspirates, described 13 and 7 cases of transudative chylothoraces respectively, of which the commonest causes aetiology were liver cirrhosis and cardiac failure although none were attributed to thoracic outlet surgery [Diaz-Guzman et al. 2005; Agrawal et al. 2008]. Therefore, our case is unique in that it describes postsurgical bilateral chylothoraces with the right-side pleural fluid analysis having the characteristics of a transudate. Essentially the protein concentration in chyle is between 20– 30 g/l, hence chylous pleural effusions would be expected to be transudative; however, it is proposed that chylous pleural effusions become exudative as protein levels increase due to fluid reabsorption from the pleural cavity to vascular space [Agrawal et al. 2008]. In our patient the transudate, although unequivocal, may have been an evanescent phenomenon as the rapidity of sampling of the evolving effusion may not have allowed sufficient time for the postulated reabsorption of water and small solutes (with resultant relative increase in protein concentrations) to occur. Following confirmation of a chylothorax, locating the point of leakage within the thoracic duct is pivotal. Should the diagnosis be missed and chyle leakage persist, this may lead to complications such as malnutrition and immunosuppression [Mcgrath et al. 2010]. Treatment of chylothorax depends on its aetiology and may be conservative, minimally invasive or surgical (Figure 2) [Sassoon and Light, 1985]. A conservative approach may resolve approximately 50% of cases of chylothoraces and entails a low-fat diet or octreotide and somatostatin [Rimensberger et al. 1998; Fernandez Alvarez et al. 1999; Al-Zubairy and Al-Jazairi, 2003]. In symptomatic chylothoraces, drainage of the fluid using an intercostal drain may be required as in our case [Weissberg and Ben-Zeev, 1993]. Other minimally invasive techniques include medical thoracoscopy, thoracic duct embolisation or disruption [Mares and Mathur, 1998; Boffa et al. 2008]. Surgery may be the only therapeutic option in cases when fluid losses of >1.5 l/day, more than 1 l/day for 5 days or continuous chyle leak for more than 2 weeks occur [Selle et al. 1973; Marts et al. 1992; Graham et al. 1994]. In our patient bilateral chylothorax was managed by the insertion of intercostals drains bilaterally and the chylous fluid accumulation ceased rapidly suggesting a trivial leak.
http://taj.sagepub.com 31
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Therapeutic Advances in Chronic Disease 6(1) Clinical suspicion of chylothorax
Chest radiograph and/or Thoracic Ultrasound
Ultrasound-guided thoracocentesis
Pleural fluid analysis for pH, protein, LDH, triglycerides, cholesterol, chylomicrons, cytology and microbiology Consider CT thorax and abdomen, lymphangiography or lymphangioscintigraphy Chylothorax confirmed Treat underlying cause, e.g. congestive cardiac failure, sarcoidosis or malignancy
No Yes
Management options
Conservative
Low-fat diet octreotide or somatostatin
Minimally invasive
Chest drain insertion Medical thoracoscopy Thoracic duct embolization or disruption
Surgical
Thoracic duct ligation ± pleurodesis
Figure 2. Flow diagram to aid in the assessment of patients with suspected chylothorax.
Conclusions Bilateral chylothorax is a rare complication of thoracic outlet syndrome surgery and is most likely due to damage to anatomical variants or tributaries of the thoracic duct. The early recognition was important in our case as it allowed for timely intervention and treatment. This case therefore would be of great interest to many specialists as it highlights a rare but potentially serious complication of this common surgical procedure.
Funding This research received no specific grant from any funding agency in the public, commercial, or notfor-profit sectors.
Conflict of interest statement The authors have no conflicts of interest to declare.
Al-Zubairy, S. and Al-Jazairi, A. (2003) Octreotide as a therapeutic option for management of chylothorax. Ann Pharmacother 37: 679–682.
References Agrawal, V., Doelken, P. and Sahn, S. (2008) Pleural fluid analysis in chylous pleural effusion. Chest 133: 1436–1441.
32 http://taj.sagepub.com
Downloaded from taj.sagepub.com at HOWARD UNIV UNDERGRAD LIBRARY on February 12, 2015
JB Morjaria, I Aslam et al. Bessone, L., Ferguson, T. and Burford, T. (1971) Chylothorax. Ann Thorac Surg 12: 527–550. Boffa, D., Sands, M., Rice, T., Murthy, S., Mason, D., Geisinger, M. et al. (2008) A critical evaluation of a percutaneous diagnostic and treatment strategy for chylothorax after thoracic surgery. Eur J Cardiothorac Surg 33: 435–439.
Mares, D. and Mathur, P. (1998) Medical thoracoscopic talc pleurodesis for chylothorax due to lymphoma: a case series. Chest 114: 731–735. Marts, B., Naunheim, K., Fiore, A. and Pennington, D. (1992) Conservative versus surgical management of chylothorax. Am J Surg 164: 532–534; discussion 534–535.
Diaz-Guzman, E., Culver, D. and Stoller, J. (2005) Transudative chylothorax: report of two cases and review of the literature. Lung 183: 169–175.
Mcgrath, E., Blades, Z. and Anderson, P. (2010) Chylothorax: aetiology, diagnosis and therapeutic options. Respir Med 104: 1–8.
Fernandez Alvarez, J., Kalache, K. and Grauel, E. (1999) management of spontaneous congenital chylothorax: oral medium-chain triglycerides versus total parenteral nutrition. Am J Perinatol 16: 415–420.
Rimensberger, P., Muller-Schenker, B., Kalangos, A. and Beghetti, M. (1998) Treatment of a persistent postoperative chylothorax with somatostatin. Ann Thorac Surg 66: 253–254.
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