Midodrine
2013; Elsevier BV; Volume: 144; Issue: 3 Linguagem: Inglês
10.1378/chest.12-3081
ISSN1931-3543
AutoresDouglas Z. Liou, Heather Warren, Dermot P. Maher, Harmik J. Soukiasian, Nicolas Melo, Alí Salim, Eric J. Ley,
Tópico(s)Vascular Malformations and Hemangiomas
ResumoThoracic duct injury is a rare but serious complication following surgery of the neck or chest that leads to uncontrolled chyle leak. Conventional management includes drainage, nutritional modification, or aggressive surgical interventions such as thoracic duct ligation, flap coverage, fibrin glue, or talc pleurodesis; few successful medical therapeutics are available. We report a case of a high-output chylothorax refractory to aggressive medical and surgical interventions. Chyle output decreased substantially after initiating midodrine, an α1-adrenergic agonist that causes vasoconstriction of the lymph system, reducing chyle flow. This case report suggests that midodrine may be a novel therapeutic for refractory chyle leaks. Thoracic duct injury is a rare but serious complication following surgery of the neck or chest that leads to uncontrolled chyle leak. Conventional management includes drainage, nutritional modification, or aggressive surgical interventions such as thoracic duct ligation, flap coverage, fibrin glue, or talc pleurodesis; few successful medical therapeutics are available. We report a case of a high-output chylothorax refractory to aggressive medical and surgical interventions. Chyle output decreased substantially after initiating midodrine, an α1-adrenergic agonist that causes vasoconstriction of the lymph system, reducing chyle flow. This case report suggests that midodrine may be a novel therapeutic for refractory chyle leaks. total parenteral nutrition Thoracic duct injury is a rare but morbid complication following surgery of the neck and thorax.1Nussenbaum B Liu JH Sinard RJ Systematic management of chyle fistula: the Southwestern experience and review of the literature.Otolaryngol Head Neck Surg. 2000; 122: 31-38Crossref PubMed Scopus (81) Google Scholar, 2Valentine CN Barresi R Prinz RA Somatostatin analog treatment of a cervical thoracic duct fistula.Head Neck. 2002; 24: 810-813Crossref PubMed Scopus (51) Google Scholar, 3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 4Zhengjiang L Sabesan T Pingzhang T Ilankovan V Omohyoid muscle flap in prevention of chyle fistula.J Oral Maxillofac Surg. 2007; 65: 1430-1432Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 5Ilczyszyn A Ridha H Durrani AJ Management of chyle leak post neck dissection: a case report and literature review.J Plast Reconstr Aesthet Surg. 2011; 64: e223-e230Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar Chylous drainage from a duct injury may lead to wound infections, skin flap necrosis, chylothorax, and, in severe instances, carotid exposure and rupture.3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 4Zhengjiang L Sabesan T Pingzhang T Ilankovan V Omohyoid muscle flap in prevention of chyle fistula.J Oral Maxillofac Surg. 2007; 65: 1430-1432Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 5Ilczyszyn A Ridha H Durrani AJ Management of chyle leak post neck dissection: a case report and literature review.J Plast Reconstr Aesthet Surg. 2011; 64: e223-e230Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar High-output chylous drainage can cause severe metabolic, nutritional, and immunologic derangements. Mortality from postesophagectomy chylothorax has been reported to be as high as 50%.1Nussenbaum B Liu JH Sinard RJ Systematic management of chyle fistula: the Southwestern experience and review of the literature.Otolaryngol Head Neck Surg. 2000; 122: 31-38Crossref PubMed Scopus (81) Google Scholar, 3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 4Zhengjiang L Sabesan T Pingzhang T Ilankovan V Omohyoid muscle flap in prevention of chyle fistula.J Oral Maxillofac Surg. 2007; 65: 1430-1432Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 5Ilczyszyn A Ridha H Durrani AJ Management of chyle leak post neck dissection: a case report and literature review.J Plast Reconstr Aesthet Surg. 2011; 64: e223-e230Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar, 6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google ScholarManagement with closed drainage of the thoracic cavity and nutritional modification with a medium-chain triglyceride diet or total parenteral nutrition (TPN) is typically the initial strategy.1Nussenbaum B Liu JH Sinard RJ Systematic management of chyle fistula: the Southwestern experience and review of the literature.Otolaryngol Head Neck Surg. 2000; 122: 31-38Crossref PubMed Scopus (81) Google Scholar, 4Zhengjiang L Sabesan T Pingzhang T Ilankovan V Omohyoid muscle flap in prevention of chyle fistula.J Oral Maxillofac Surg. 2007; 65: 1430-1432Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 7de Gier HH Balm AJ Bruning PF Gregor RT Hilgers FJ Systematic approach to the treatment of chylous leakage after neck dissection.Head Neck. 1996; 18: 347-351Crossref PubMed Google Scholar Successful treatment with the addition of pharmacologic interventions, such as octreotide, orlistat, and etilefrine, has been reported.6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar, 8Belloso A Saravanan K de Carpentier J The community management of chylous fistula using a pancreatic lipase inhibitor (orlistat).Laryngoscope. 2006; 116: 1934-1935Crossref PubMed Scopus (11) Google Scholar, 9Nyquist GG Hagr A Sobol SE Hier MP Black MJ Octreotide in the medical management of chyle fistula.Otolaryngol Head Neck Surg. 2003; 128: 910-911Crossref PubMed Scopus (19) Google Scholar, 10Barili F Polvani G Topkara VK et al.Administration of octreotide for management of postoperative high-flow chylothorax.Ann Vasc Surg. 2007; 21: 90-92Abstract Full Text Full Text PDF PubMed Scopus (20) Google Scholar, 11Kelly RF Shumway SJ Conservative management of post-operative chylothorax using somatostatin.Ann Thorac Surg. 2000; 69: 1944-1945Abstract Full Text Full Text PDF PubMed Scopus (96) Google Scholar, 12Oguz E Apaydin AZ Ayik F et al.Chylothorax after thoracoabdominal aneurysm repair: efficacy of somatostatin.Ann Vasc Surg. 2011; 25: 267.e11-267.e13Abstract Full Text Full Text PDF Scopus (1) Google Scholar, 13Rimensberger PC Müller-Schenker B Kalangos A Beghetti M Treatment of a persistent postoperative chylothorax with somatostatin.Ann Thorac Surg. 1998; 66: 253-254Abstract Full Text Full Text PDF PubMed Scopus (171) Google Scholar For high-output chyle leaks following esophagectomy, early operative therapy is strongly advocated.14Merigliano S Molena D Ruol A et al.Chylothorax complicating esophagectomy for cancer: a plea for early thoracic duct ligation.J Thorac Cardiovasc Surg. 2000; 119: 453-457Abstract Full Text Full Text PDF PubMed Scopus (120) Google Scholar, 15Lagarde SM Omloo JM de Jong K Busch OR Obertop H van Lanschot JJ Incidence and management of chyle leakage after esophagectomy.Ann Thorac Surg. 2005; 80: 449-454Abstract Full Text Full Text PDF PubMed Scopus (68) Google Scholar Persistent chylothorax despite optimal medical management also requires surgery. Possible interventions include direct thoracic duct ligation (cervical, thoracic, or abdominal approach), muscle flap coverage (pectoralis, sternocleidomastoid, or scalene), fibrin glue, or talc pleurodesis.4Zhengjiang L Sabesan T Pingzhang T Ilankovan V Omohyoid muscle flap in prevention of chyle fistula.J Oral Maxillofac Surg. 2007; 65: 1430-1432Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar, 7de Gier HH Balm AJ Bruning PF Gregor RT Hilgers FJ Systematic approach to the treatment of chylous leakage after neck dissection.Head Neck. 1996; 18: 347-351Crossref PubMed Google Scholar, 16Brennan PA Blythe JN Herd MK Habib A Anand R The contemporary management of chyle leak following cervical thoracic duct damage.Br J Oral Maxillofac Surg. 2012; 50: 197-201Abstract Full Text Full Text PDF PubMed Scopus (37) Google Scholar, 17Vaiman M Eviatar E Lymphatic fistulae after neck dissection: the fibrin sealant treatment.J Surg Oncol. 2008; 98: 467-471Crossref PubMed Scopus (6) Google Scholar Thoracic duct ligation at the level of the diaphragm has been reported as the most effective surgical approach, with a success rate of 80%.11Kelly RF Shumway SJ Conservative management of post-operative chylothorax using somatostatin.Ann Thorac Surg. 2000; 69: 1944-1945Abstract Full Text Full Text PDF PubMed Scopus (96) Google ScholarLimited literature exists for the successful management of refractory chylothorax. We report a case of recurrent high-output chylothorax that was treated successfully with the addition of midodrine, an α1-agonist, to the standard TPN and octreotide therapy.Case ReportA 64-year-old woman underwent an uneventful Ivor Lewis esophagogastrectomy for esophageal adenocarcinoma of the gastroesophageal junction. On postoperative day 1, high chylous output was noted from the left chest tube (Fig 1). Enteral feeds were held, and the patient was started on TPN and octreotide. Because of persistent high-output chylothorax (1-2 L/d), the patient returned to the operating room on postoperative day 9 for reexploration, high thoracic duct ligation, and talc pleurodesis. The patient was managed with TPN and octreotide until postoperative day 28, when she went back to the operating room for repeat ligation of the thoracic duct at the level of the diaphragm.Following thoracic duct ligation, a substantial decrease in chylous output was noted (5 mL/d). Initiation of enteral feeds 7 days after ligation, however, led to a recurrent high-volume chyle leak (2-3 L/d), which persisted despite reinstituting TPN and octreotide. At this juncture, after attempts at medical management and two surgical interventions, alternative treatments were entertained. α1-Agonists had been reported as treatment of postoperative chyle leaks6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar and, therefore, midodrine was added as a potential therapeutic on postoperative day 52. Within 24 h of midodrine initiation, chyle drainage decreased, and complete resolution was achieved after 4 days. The chyle leak remained at zero after resumption of enteral nutrition and through discharge.DiscussionPostoperative chylothorax is usually the result of iatrogenic injury to the thoracic duct or surrounding collateral lymphatic ducts during surgery. The length of the duct, anatomic variations, and inherent delicacy all contribute to its susceptibility to injury during dissections of the thorax and neck.3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 18Greenfield J Gottlieb MI Variations in the terminal portion of the human thoracic cuct.Arch Surg. 1956; 73: 955-959Crossref Scopus (85) Google ScholarThe optimal management of postoperative chylothorax is varied. Nutritional and pharmacologic interventions are generally implemented first, except in cases of high-output chyle leaks (> 2 L/d), when an early operation is warranted.15Lagarde SM Omloo JM de Jong K Busch OR Obertop H van Lanschot JJ Incidence and management of chyle leakage after esophagectomy.Ann Thorac Surg. 2005; 80: 449-454Abstract Full Text Full Text PDF PubMed Scopus (68) Google Scholar Ultimately, surgery may be required for chyle leaks that fail medical management. Compromise of skin flaps, exposed great vessels, and significant metabolic, nutritional, and immunologic derangements may also necessitate reoperation.1Nussenbaum B Liu JH Sinard RJ Systematic management of chyle fistula: the Southwestern experience and review of the literature.Otolaryngol Head Neck Surg. 2000; 122: 31-38Crossref PubMed Scopus (81) Google Scholar, 3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 10Barili F Polvani G Topkara VK et al.Administration of octreotide for management of postoperative high-flow chylothorax.Ann Vasc Surg. 2007; 21: 90-92Abstract Full Text Full Text PDF PubMed Scopus (20) Google Scholar There are currently no recommendations for the management of fistulas refractory to optimal medical and surgical interventions.Case reports suggest etilefrine as an adjunct treatment of postoperative chyle leaks.6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar Etilefrine is a sympathomimetic drug with α and β adrenergic properties used to treat postural hypotension. The theorized benefit is centered on the drug's α1-adrenergic properties, which cause smooth-muscle contraction, resulting in reduced diameter of the lymphatic vessels. This α agonism may lead to decreased chyle flow and augment spontaneous closure of the chyle leaks.6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar Etilefrine, however, is not commercially available in the United States.Midodrine is a readily available, oral, selective α adrenergic drug used in treating orthostatic and hemodialysis-induced hypotension.19Ward CR Gray JC Gilroy JJ Kenny RA Midodrine: a role in the management of neurocardiogenic syncope.Heart. 1998; 79: 45-49Crossref PubMed Scopus (208) Google Scholar, 20Prakash S Garg AX Heidenheim AP House AA Midodrine appears to be safe and effective for dialysis-induced hypotension: a systematic review.Nephrol Dial Transplant. 2004; 19: 2553-2558Crossref PubMed Scopus (99) Google Scholar Although no previous reports describe using midodrine to treat chylothorax, its isolated α adrenergic properties suggest activity on lymphatic vessels, which may result in contraction and decreased chyle flow. In this case, midodrine was instituted after 7 weeks of standard-of-care treatment. Use of TPN, octreotide, and nil per os in addition to midodrine may have contributed to the resolution of the chyle leak. Although additional studies are required to delineate the efficacy and side effects of midodrine, this case report demonstrates that it may be a useful adjunct in the management of chylous fistulas refractory to surgical intervention. Thoracic duct injury is a rare but morbid complication following surgery of the neck and thorax.1Nussenbaum B Liu JH Sinard RJ Systematic management of chyle fistula: the Southwestern experience and review of the literature.Otolaryngol Head Neck Surg. 2000; 122: 31-38Crossref PubMed Scopus (81) Google Scholar, 2Valentine CN Barresi R Prinz RA Somatostatin analog treatment of a cervical thoracic duct fistula.Head Neck. 2002; 24: 810-813Crossref PubMed Scopus (51) Google Scholar, 3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 4Zhengjiang L Sabesan T Pingzhang T Ilankovan V Omohyoid muscle flap in prevention of chyle fistula.J Oral Maxillofac Surg. 2007; 65: 1430-1432Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 5Ilczyszyn A Ridha H Durrani AJ Management of chyle leak post neck dissection: a case report and literature review.J Plast Reconstr Aesthet Surg. 2011; 64: e223-e230Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar Chylous drainage from a duct injury may lead to wound infections, skin flap necrosis, chylothorax, and, in severe instances, carotid exposure and rupture.3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 4Zhengjiang L Sabesan T Pingzhang T Ilankovan V Omohyoid muscle flap in prevention of chyle fistula.J Oral Maxillofac Surg. 2007; 65: 1430-1432Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 5Ilczyszyn A Ridha H Durrani AJ Management of chyle leak post neck dissection: a case report and literature review.J Plast Reconstr Aesthet Surg. 2011; 64: e223-e230Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar High-output chylous drainage can cause severe metabolic, nutritional, and immunologic derangements. Mortality from postesophagectomy chylothorax has been reported to be as high as 50%.1Nussenbaum B Liu JH Sinard RJ Systematic management of chyle fistula: the Southwestern experience and review of the literature.Otolaryngol Head Neck Surg. 2000; 122: 31-38Crossref PubMed Scopus (81) Google Scholar, 3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 4Zhengjiang L Sabesan T Pingzhang T Ilankovan V Omohyoid muscle flap in prevention of chyle fistula.J Oral Maxillofac Surg. 2007; 65: 1430-1432Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 5Ilczyszyn A Ridha H Durrani AJ Management of chyle leak post neck dissection: a case report and literature review.J Plast Reconstr Aesthet Surg. 2011; 64: e223-e230Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar, 6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar Management with closed drainage of the thoracic cavity and nutritional modification with a medium-chain triglyceride diet or total parenteral nutrition (TPN) is typically the initial strategy.1Nussenbaum B Liu JH Sinard RJ Systematic management of chyle fistula: the Southwestern experience and review of the literature.Otolaryngol Head Neck Surg. 2000; 122: 31-38Crossref PubMed Scopus (81) Google Scholar, 4Zhengjiang L Sabesan T Pingzhang T Ilankovan V Omohyoid muscle flap in prevention of chyle fistula.J Oral Maxillofac Surg. 2007; 65: 1430-1432Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 7de Gier HH Balm AJ Bruning PF Gregor RT Hilgers FJ Systematic approach to the treatment of chylous leakage after neck dissection.Head Neck. 1996; 18: 347-351Crossref PubMed Google Scholar Successful treatment with the addition of pharmacologic interventions, such as octreotide, orlistat, and etilefrine, has been reported.6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar, 8Belloso A Saravanan K de Carpentier J The community management of chylous fistula using a pancreatic lipase inhibitor (orlistat).Laryngoscope. 2006; 116: 1934-1935Crossref PubMed Scopus (11) Google Scholar, 9Nyquist GG Hagr A Sobol SE Hier MP Black MJ Octreotide in the medical management of chyle fistula.Otolaryngol Head Neck Surg. 2003; 128: 910-911Crossref PubMed Scopus (19) Google Scholar, 10Barili F Polvani G Topkara VK et al.Administration of octreotide for management of postoperative high-flow chylothorax.Ann Vasc Surg. 2007; 21: 90-92Abstract Full Text Full Text PDF PubMed Scopus (20) Google Scholar, 11Kelly RF Shumway SJ Conservative management of post-operative chylothorax using somatostatin.Ann Thorac Surg. 2000; 69: 1944-1945Abstract Full Text Full Text PDF PubMed Scopus (96) Google Scholar, 12Oguz E Apaydin AZ Ayik F et al.Chylothorax after thoracoabdominal aneurysm repair: efficacy of somatostatin.Ann Vasc Surg. 2011; 25: 267.e11-267.e13Abstract Full Text Full Text PDF Scopus (1) Google Scholar, 13Rimensberger PC Müller-Schenker B Kalangos A Beghetti M Treatment of a persistent postoperative chylothorax with somatostatin.Ann Thorac Surg. 1998; 66: 253-254Abstract Full Text Full Text PDF PubMed Scopus (171) Google Scholar For high-output chyle leaks following esophagectomy, early operative therapy is strongly advocated.14Merigliano S Molena D Ruol A et al.Chylothorax complicating esophagectomy for cancer: a plea for early thoracic duct ligation.J Thorac Cardiovasc Surg. 2000; 119: 453-457Abstract Full Text Full Text PDF PubMed Scopus (120) Google Scholar, 15Lagarde SM Omloo JM de Jong K Busch OR Obertop H van Lanschot JJ Incidence and management of chyle leakage after esophagectomy.Ann Thorac Surg. 2005; 80: 449-454Abstract Full Text Full Text PDF PubMed Scopus (68) Google Scholar Persistent chylothorax despite optimal medical management also requires surgery. Possible interventions include direct thoracic duct ligation (cervical, thoracic, or abdominal approach), muscle flap coverage (pectoralis, sternocleidomastoid, or scalene), fibrin glue, or talc pleurodesis.4Zhengjiang L Sabesan T Pingzhang T Ilankovan V Omohyoid muscle flap in prevention of chyle fistula.J Oral Maxillofac Surg. 2007; 65: 1430-1432Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar, 7de Gier HH Balm AJ Bruning PF Gregor RT Hilgers FJ Systematic approach to the treatment of chylous leakage after neck dissection.Head Neck. 1996; 18: 347-351Crossref PubMed Google Scholar, 16Brennan PA Blythe JN Herd MK Habib A Anand R The contemporary management of chyle leak following cervical thoracic duct damage.Br J Oral Maxillofac Surg. 2012; 50: 197-201Abstract Full Text Full Text PDF PubMed Scopus (37) Google Scholar, 17Vaiman M Eviatar E Lymphatic fistulae after neck dissection: the fibrin sealant treatment.J Surg Oncol. 2008; 98: 467-471Crossref PubMed Scopus (6) Google Scholar Thoracic duct ligation at the level of the diaphragm has been reported as the most effective surgical approach, with a success rate of 80%.11Kelly RF Shumway SJ Conservative management of post-operative chylothorax using somatostatin.Ann Thorac Surg. 2000; 69: 1944-1945Abstract Full Text Full Text PDF PubMed Scopus (96) Google Scholar Limited literature exists for the successful management of refractory chylothorax. We report a case of recurrent high-output chylothorax that was treated successfully with the addition of midodrine, an α1-agonist, to the standard TPN and octreotide therapy. Case ReportA 64-year-old woman underwent an uneventful Ivor Lewis esophagogastrectomy for esophageal adenocarcinoma of the gastroesophageal junction. On postoperative day 1, high chylous output was noted from the left chest tube (Fig 1). Enteral feeds were held, and the patient was started on TPN and octreotide. Because of persistent high-output chylothorax (1-2 L/d), the patient returned to the operating room on postoperative day 9 for reexploration, high thoracic duct ligation, and talc pleurodesis. The patient was managed with TPN and octreotide until postoperative day 28, when she went back to the operating room for repeat ligation of the thoracic duct at the level of the diaphragm.Following thoracic duct ligation, a substantial decrease in chylous output was noted (5 mL/d). Initiation of enteral feeds 7 days after ligation, however, led to a recurrent high-volume chyle leak (2-3 L/d), which persisted despite reinstituting TPN and octreotide. At this juncture, after attempts at medical management and two surgical interventions, alternative treatments were entertained. α1-Agonists had been reported as treatment of postoperative chyle leaks6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar and, therefore, midodrine was added as a potential therapeutic on postoperative day 52. Within 24 h of midodrine initiation, chyle drainage decreased, and complete resolution was achieved after 4 days. The chyle leak remained at zero after resumption of enteral nutrition and through discharge. A 64-year-old woman underwent an uneventful Ivor Lewis esophagogastrectomy for esophageal adenocarcinoma of the gastroesophageal junction. On postoperative day 1, high chylous output was noted from the left chest tube (Fig 1). Enteral feeds were held, and the patient was started on TPN and octreotide. Because of persistent high-output chylothorax (1-2 L/d), the patient returned to the operating room on postoperative day 9 for reexploration, high thoracic duct ligation, and talc pleurodesis. The patient was managed with TPN and octreotide until postoperative day 28, when she went back to the operating room for repeat ligation of the thoracic duct at the level of the diaphragm. Following thoracic duct ligation, a substantial decrease in chylous output was noted (5 mL/d). Initiation of enteral feeds 7 days after ligation, however, led to a recurrent high-volume chyle leak (2-3 L/d), which persisted despite reinstituting TPN and octreotide. At this juncture, after attempts at medical management and two surgical interventions, alternative treatments were entertained. α1-Agonists had been reported as treatment of postoperative chyle leaks6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar and, therefore, midodrine was added as a potential therapeutic on postoperative day 52. Within 24 h of midodrine initiation, chyle drainage decreased, and complete resolution was achieved after 4 days. The chyle leak remained at zero after resumption of enteral nutrition and through discharge. DiscussionPostoperative chylothorax is usually the result of iatrogenic injury to the thoracic duct or surrounding collateral lymphatic ducts during surgery. The length of the duct, anatomic variations, and inherent delicacy all contribute to its susceptibility to injury during dissections of the thorax and neck.3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 18Greenfield J Gottlieb MI Variations in the terminal portion of the human thoracic cuct.Arch Surg. 1956; 73: 955-959Crossref Scopus (85) Google ScholarThe optimal management of postoperative chylothorax is varied. Nutritional and pharmacologic interventions are generally implemented first, except in cases of high-output chyle leaks (> 2 L/d), when an early operation is warranted.15Lagarde SM Omloo JM de Jong K Busch OR Obertop H van Lanschot JJ Incidence and management of chyle leakage after esophagectomy.Ann Thorac Surg. 2005; 80: 449-454Abstract Full Text Full Text PDF PubMed Scopus (68) Google Scholar Ultimately, surgery may be required for chyle leaks that fail medical management. Compromise of skin flaps, exposed great vessels, and significant metabolic, nutritional, and immunologic derangements may also necessitate reoperation.1Nussenbaum B Liu JH Sinard RJ Systematic management of chyle fistula: the Southwestern experience and review of the literature.Otolaryngol Head Neck Surg. 2000; 122: 31-38Crossref PubMed Scopus (81) Google Scholar, 3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 10Barili F Polvani G Topkara VK et al.Administration of octreotide for management of postoperative high-flow chylothorax.Ann Vasc Surg. 2007; 21: 90-92Abstract Full Text Full Text PDF PubMed Scopus (20) Google Scholar There are currently no recommendations for the management of fistulas refractory to optimal medical and surgical interventions.Case reports suggest etilefrine as an adjunct treatment of postoperative chyle leaks.6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar Etilefrine is a sympathomimetic drug with α and β adrenergic properties used to treat postural hypotension. The theorized benefit is centered on the drug's α1-adrenergic properties, which cause smooth-muscle contraction, resulting in reduced diameter of the lymphatic vessels. This α agonism may lead to decreased chyle flow and augment spontaneous closure of the chyle leaks.6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar Etilefrine, however, is not commercially available in the United States.Midodrine is a readily available, oral, selective α adrenergic drug used in treating orthostatic and hemodialysis-induced hypotension.19Ward CR Gray JC Gilroy JJ Kenny RA Midodrine: a role in the management of neurocardiogenic syncope.Heart. 1998; 79: 45-49Crossref PubMed Scopus (208) Google Scholar, 20Prakash S Garg AX Heidenheim AP House AA Midodrine appears to be safe and effective for dialysis-induced hypotension: a systematic review.Nephrol Dial Transplant. 2004; 19: 2553-2558Crossref PubMed Scopus (99) Google Scholar Although no previous reports describe using midodrine to treat chylothorax, its isolated α adrenergic properties suggest activity on lymphatic vessels, which may result in contraction and decreased chyle flow. In this case, midodrine was instituted after 7 weeks of standard-of-care treatment. Use of TPN, octreotide, and nil per os in addition to midodrine may have contributed to the resolution of the chyle leak. Although additional studies are required to delineate the efficacy and side effects of midodrine, this case report demonstrates that it may be a useful adjunct in the management of chylous fistulas refractory to surgical intervention. Postoperative chylothorax is usually the result of iatrogenic injury to the thoracic duct or surrounding collateral lymphatic ducts during surgery. The length of the duct, anatomic variations, and inherent delicacy all contribute to its susceptibility to injury during dissections of the thorax and neck.3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 18Greenfield J Gottlieb MI Variations in the terminal portion of the human thoracic cuct.Arch Surg. 1956; 73: 955-959Crossref Scopus (85) Google Scholar The optimal management of postoperative chylothorax is varied. Nutritional and pharmacologic interventions are generally implemented first, except in cases of high-output chyle leaks (> 2 L/d), when an early operation is warranted.15Lagarde SM Omloo JM de Jong K Busch OR Obertop H van Lanschot JJ Incidence and management of chyle leakage after esophagectomy.Ann Thorac Surg. 2005; 80: 449-454Abstract Full Text Full Text PDF PubMed Scopus (68) Google Scholar Ultimately, surgery may be required for chyle leaks that fail medical management. Compromise of skin flaps, exposed great vessels, and significant metabolic, nutritional, and immunologic derangements may also necessitate reoperation.1Nussenbaum B Liu JH Sinard RJ Systematic management of chyle fistula: the Southwestern experience and review of the literature.Otolaryngol Head Neck Surg. 2000; 122: 31-38Crossref PubMed Scopus (81) Google Scholar, 3Gregor RT Management of chyle fistulization in association with neck dissection.Otolaryngol Head Neck Surg. 2000; 122: 434-439Crossref PubMed Google Scholar, 10Barili F Polvani G Topkara VK et al.Administration of octreotide for management of postoperative high-flow chylothorax.Ann Vasc Surg. 2007; 21: 90-92Abstract Full Text Full Text PDF PubMed Scopus (20) Google Scholar There are currently no recommendations for the management of fistulas refractory to optimal medical and surgical interventions. Case reports suggest etilefrine as an adjunct treatment of postoperative chyle leaks.6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar Etilefrine is a sympathomimetic drug with α and β adrenergic properties used to treat postural hypotension. The theorized benefit is centered on the drug's α1-adrenergic properties, which cause smooth-muscle contraction, resulting in reduced diameter of the lymphatic vessels. This α agonism may lead to decreased chyle flow and augment spontaneous closure of the chyle leaks.6Guillem P Papachristos I Peillon C Triboulet JP Etilefrine use in the management of post-operative chyle leaks in thoracic surgery.Interact Cardiovasc Thorac Surg. 2004; 3: 156-160Crossref PubMed Scopus (38) Google Scholar Etilefrine, however, is not commercially available in the United States. Midodrine is a readily available, oral, selective α adrenergic drug used in treating orthostatic and hemodialysis-induced hypotension.19Ward CR Gray JC Gilroy JJ Kenny RA Midodrine: a role in the management of neurocardiogenic syncope.Heart. 1998; 79: 45-49Crossref PubMed Scopus (208) Google Scholar, 20Prakash S Garg AX Heidenheim AP House AA Midodrine appears to be safe and effective for dialysis-induced hypotension: a systematic review.Nephrol Dial Transplant. 2004; 19: 2553-2558Crossref PubMed Scopus (99) Google Scholar Although no previous reports describe using midodrine to treat chylothorax, its isolated α adrenergic properties suggest activity on lymphatic vessels, which may result in contraction and decreased chyle flow. In this case, midodrine was instituted after 7 weeks of standard-of-care treatment. Use of TPN, octreotide, and nil per os in addition to midodrine may have contributed to the resolution of the chyle leak. Although additional studies are required to delineate the efficacy and side effects of midodrine, this case report demonstrates that it may be a useful adjunct in the management of chylous fistulas refractory to surgical intervention. Financial/nonfinancial disclosures: The authors have reported to CHEST that no potential conflicts of interest exist with any companies/organizations whose products or services may be discussed in this article. Other contributions: CHEST worked with the authors to ensure that the Journal policies on patient consent to report information were met.
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