A 53-year-old Man with Idiopathic Bilateral Chylothorax Refractory to Lymphaticovenular Anastomosis: A Case Report
A 53-year-old man with chronic dyspnea and bilateral pleural effusion was subsequently diagnosed with idiopathic chylothorax. Lymphatic scintigraphy confirmed lymphatic fluid leakage at the left venous angle, prompting management with lymphaticovenular anastomosis (LVA). Although the left pleural ef...
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| Published in | Internal Medicine p. 3805-24 |
|---|---|
| Main Authors | , , , , , , , , , |
| Format | Journal Article |
| Language | English |
| Published |
The Japanese Society of Internal Medicine
01.02.2025
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| Subjects | |
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| ISSN | 0918-2918 1349-7235 |
| DOI | 10.2169/internalmedicine.3805-24 |
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| Abstract | A 53-year-old man with chronic dyspnea and bilateral pleural effusion was subsequently diagnosed with idiopathic chylothorax. Lymphatic scintigraphy confirmed lymphatic fluid leakage at the left venous angle, prompting management with lymphaticovenular anastomosis (LVA). Although the left pleural effusion was controlled, the right pleural effusion continued to increase, resulting in bilateral leg lymphedema that was refractory to LVA. Approximately three years and three months after the presentation, the patient succumbed to CO2 narcosis and renal failure. It is crucial to study additional cases in order to uncover new causes and develop pathology-based treatments for this condition. |
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| AbstractList | A 53-year-old man with chronic dyspnea and bilateral pleural effusion was subsequently diagnosed with idiopathic chylothorax. Lymphatic scintigraphy confirmed lymphatic fluid leakage at the left venous angle, prompting management with lymphaticovenular anastomosis (LVA). Although the left pleural effusion was controlled, the right pleural effusion continued to increase, resulting in bilateral leg lymphedema that was refractory to LVA. Approximately three years and three months after the presentation, the patient succumbed to CO2 narcosis and renal failure. It is crucial to study additional cases in order to uncover new causes and develop pathology-based treatments for this condition. |
| Author | Matsuno, Kei Seyama, Kuniaki Hotta, Saori Ayame, Nana Yamada, Tomoko Mihara, Makoto Hara, Hisako Takahashi, Kazuhisa Sugitani, Honami Sugano, Koji |
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Sakakibara T, Seki S, Yaguchi T, Harada A, Nakao A. A case of idiopathic chylothorax. Nihon Rinsho Geka Gakkai Zasshi (Journal of Japanese Surgical Association) 67: 316-319, 2006 (in Japanese). 22. Cha E, Sirjintakarn P. Anatomic variation of the thoracic duct and visualization of mediastinal lymph nodes a lymphographic study. Radiology 119: 45-48, 1976. 24. Yeom S, Cohen B, Clifford R Weiss, et al. Genetic testing in the evaluation of individuals with clinical diagnosis of atypical Sturge-Weber syndrome. Am J Med Genet A 191: 983-994, 2023. |
| References_xml | – reference: 24. Yeom S, Cohen B, Clifford R Weiss, et al. Genetic testing in the evaluation of individuals with clinical diagnosis of atypical Sturge-Weber syndrome. Am J Med Genet A 191: 983-994, 2023. – reference: 26. Mihara M, Hara H, Furniss D, et al. Lymphaticovenular anastomosis to prevent cellulitis associated with lymphoedema. Br J Surg 101: 1391-1396, 2014. – reference: 4. Hirokawa M, Oshita Y, Kitajima T, Koga T, Rikimaru T, Aizawa H. A case of spontaneous bilateral chylothorax. Nihon Kokyuki Gakkai Zasshi (Journal of Japanese Respiratory Society) 42: 362-365, 2004 (in Japanese). – reference: 5. Sakakibara T, Seki S, Yaguchi T, Harada A, Nakao A. A case of idiopathic chylothorax. Nihon Rinsho Geka Gakkai Zasshi (Journal of Japanese Surgical Association) 67: 316-319, 2006 (in Japanese). – reference: 16. Ueno H, Nakashima K, Kadomatsu Y, Okasaka T, Mori S. A case of refractory idiopathic chylothorax involving an adult. Nihon Kokyuki Geka Gakkai Zasshi (The Journal of the Japanese Association for Chest Surgery) 29: 155-159, 2015 (in Japanese). – reference: 13. Nishida Y, Nishi S, Misawa K, Mishima O, Kitano M. A case of idiopathic chylothorax treated with thoracoscopic ligation of thoracic ducts and resection of the cisterna chyli. Nihon Kokyuki Geka Gakkai Zasshi (The Journal of the Japanese Associtation for Chest Surgery) 25: 58-63, 2011 (in Japanese). – reference: 19. Srinivasa RN, Chick JFB, Hage AN, Gemmete JJ, Murrey DC, Srinivasa RN. Endolymphatic thoracic duct stent-graft reconstruction for chylothorax approach, technical success, safety, and short-term outcomes. Ann Vasc Surg 48: 97-103, 2018. – reference: 17. Chick JFB, Reddy SN, Murrey DA, et al. Single-session endolymphatic thoracic duct stent-graft placement for recurrent idiopathic chylothorax. J Vasc Interv Radiol 28: 1063-1067, 2017. – reference: 23. Okuda I, Udagawa H, Takahashi J, Yamase H, Kohno T, Nakajima Y. Magnetic resonance-thoracic ductography: imaging aid for thoracic surgery and thoracic duct depiction based on embryological considerations. Gen Thorac Cardiovasc Surg 57: 640-646, 2009. – reference: 6. Chen-Scarabelli C, Ama R, Abboud J, Jabarren M, Saravolatz L, Scarabelli TM. Use of octreotide, a long-acting somatostatin analogue, in the treatment of chylothorax of idiopathic etiology. Minerva Chir 61: 155-158, 2006. – reference: 9. Xu ZH, Wang CF, Zhang YQ, Shen HH, Xu F. A case of chylothorax treated curatively with Sapylin, a streptococcus preparation. J Zhejiang Univ Sci B 8: 885-887, 2007. – reference: 21. Koyanagi A, Nonomura R, Sasaki T, et al. Idiopathic bilateral chylothorax which presented difficulty in treatment. Kyobu Geka (The Japanese journal of thoracic surgery) 72: 371-374, 2019 (in Japanese). – reference: 7. Kato E, Ishii A, Yamada N, Sugiura T. A case of idiopathic chylothorax detected by the mass examination of chest X-ray. Nihon Kokyuki Gakkai Zasshi (Journal of Japanese Respiratory Society) 44: 1002-1005, 2006 (in Japanese). – reference: 14. Motoishi M, Okamoto K, Kataoka Y, Sawai S, Oshio M, Hanaoka J. Successful treatment of bilateral idiopathic chylothorax with ligation of the thoracic duct and octreotide. Nihon Kokyuki Geka Gakkai Zasshi (The Journal of the Japanese Association for Chest Surgery) 28: 85-90, 2014 (in Japanese). – reference: 18. Rajdev K, Avula A, Sharma D, et al. A Case of Transudative Chylothorax: A diagnostic dilemma. Cureus 10: e2247, 2018. – reference: 22. Cha E, Sirjintakarn P. Anatomic variation of the thoracic duct and visualization of mediastinal lymph nodes a lymphographic study. Radiology 119: 45-48, 1976. – reference: 3. Kotani K, Umemori Y, Makihara S. A case of idiopathic chylothorax. Nihon Rinsho Geka Gakkai Zasshi (Journal of Japanese Surgical Association) 62: 907-910, 2001 (in Japanese). – reference: 1. Nair SK, Petko M, Hayward MP. Aetiology and management of chylothorax in adults. Eur J Cardiothorac Surg 32: 362-369, 2007. – reference: 27. Lin CH. Supermicrosurgical lymphovenous anastomosis for the treatment of recurrent cellulitis-associated lymphedema in the lower limb. J Vasc Surg Cases Innov Tech 7: 790-793, 2021. – reference: 2. Doerr CH, Allen MS, Nichols FC 3rd, Ryu JH. Etiology of chylothorax in 203 patients. Mayo Clin Proc 80: 867-870, 2005. – reference: 20. Sivakumar P, Ahmed L. Use of an alpha-1 adrenoreceptor agonist in the management of recurrent refractory idiopathic chylothorax. Chest 154: e1-e4, 2018. – reference: 11. Koshiko S, Ozawa K, Kimura F, Tashiro Y. Spontaneous bilateral chylothorax. Nihon Rinsho Geka Gakkai Zasshi (Journal of Japanese Surgical Association) 69: 2510-2513, 2008 (in Japanese). – reference: 10. Inomoto Y, Hirose H, Fukahori S, et al. Case of idiopathic chylothorax successfully treated with surgical ligation of thoracic ducts. Nihon Kokyuki Gakkai Zasshi (Journal of Japanese Respiratory Society) 45: 804-807, 2007 (in Japanese). – reference: 25. Hara H, Mihara M, Seki Y, Koshima I. Lymphoedema caused by idiopathic lymphatic thrombus. J Plast Reconstr Aesthet Surg 66: 1780-1783, 2013. – reference: 15. Abouda M, Ferdaous Y, Triki M, Charfi M, Charfi MR. Case report: management of idiopathic chylothorax. F1000Research 4: 1049, 2015. – reference: 8. Kobayashi K, Tachikawa S, Horiguchi T, et al. A case of adult idiopathic chylothorax with transudative ascites. Nihon Kokyuki Gakkai Zasshi (Journal of Japanese Respiratory Society) 44: 990-992, 2006 (in Japanese). – reference: 12. Nakamura Y, Matsumura A, Katsura H, Sakaguchi M, Ito N, Kitahara N. A case of idiopathic chylothorax treated with ligation of the thoracic duct and pleurodesis. Nihon Rinsho Geka Gakkai Zasshi (Journal of Japanese Surgical Association) 70: 697-701, 2009 (in Japanese). |
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| Title | A 53-year-old Man with Idiopathic Bilateral Chylothorax Refractory to Lymphaticovenular Anastomosis: A Case Report |
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