L. W. Gorham and A. P. Stout, "Massive osteolysis (Acute Spontaneous Absorption of Bone, Phantom Bone, Disappearing Bone); its relation to hemangiomatosis," J. Bone Joint Surg. Am., vol. 37-A, pp. 985-1004, 1955.
V. S. Nikolaou, D. Chytas, D. Korres, and N. Efstathopoulos, "Vanishing bone disease (Gorham-Stout Syndrome): A review of a rare entity," World J. Orthop., vol. 5, pp. 694-698, 10.5312/wjo.v5.i5.694. eCollection 2014.3. Jackson JB. A boneless arm. Boston Med Surg J. 1838; 18:368-369, 2014.
J. Patrick, "Massive osteolysis complicated by chylothorax successfully treated by pleurodesis," J. Bone Joint Surg. Br., vol. 58, pp. 347-349, 1976.
D. Feigl, L. Seidel, and A. Marmor, "Gorham's disease of the clavicle with bilateral pleural effusions," Chest, vol. 79, pp. 242-244, 1981.
D. Feigl and A. Marmor, "Gorham's disease of the clavicle with bilateral pleural effusions. Eight years later," Chest, vol. 92, p. 189b-189, 1987.
L. R. Brown, H. M. Reiman, E. C. Rosenow, P. M. Gloviczki, and M. B. Divertie, "Intrathoracic lymphangioma," Mayo Clin. Proc., vol. 61, pp. 882-892, 1986.
N. Hejgaard and P. R. Olsen, "Massive Gorham osteolysis of the right hemipelvis complicated by chylothorax: Report of a case in a 9-year-old boy successfully treated by pleurodesis," J. Pediatr. Orthop., vol. 7, pp. 96-99, 1987.
C. S. Mitchell, M. T. Parisi, and R. E. Osborn, "Gorham's disease involving the thoracic skeleton. Plain films and CT in two cases," Pediatr Radiol., vol. 23, pp. 543-544, 1993.
M. L. Tie, G. A. Poland, and E. C. Rosenow, "Pleural effusion: A complication of Gorham's disease," Pediatr Radiol., vol. 24, p. 542-542, 1994.
S. E. Ng and Y. T. Wang, "Gorham's syndrome with pleural effusion and colonic carcinoma," Singapore Med. J., vol. 36, pp. 102-104, 1995.
K. D. McNeil, K. M. Fong, Q. J. Walker, P. Jessup, and P. V. Zimmerman, "Gorham's syndrome: A usually fatal cause of pleural effusion treated successfully with radiotherapy," Thorax, vol. 51, pp. 1275-1286, 1996.
P. Riantawan, S. Tansupasawasdikul, and P. Subhannachart, "Bilateral chylothorax complicating massive osteolysis (Gorham's Syndrome)," Thorax, vol. 51, pp. 1277-1278, 1996.
M. Aoki, F. Kato, H. Saito, K. Mimatsu, and H. Iwata, "Successful treatment of chylothorax by bleomycin for Gorham's disease," Clin. Orthop. Relat Res., vol. 330, pp. 193-197, 1996.
H. Hagberg, K. Lamberg, and G. Aström, "Alpha-2b interferon and oral clodronate for Gorham's disease," Lancet., vol. 350, pp. 1822-1823, 1997.
G. Podevin, G. Levard, M. Larroquet, and M. Gruner, "Pleuroperitoneal shunt in the management of chylothorax caused by thoracic lymphatic dysplasia," J. Pediatr Surg., vol. 34, pp. 1420-1422, 1999.
N. Chavanis, P. Chaffanjon, G. Frey, G. Vottero, and P. Y. Brichon, "Chylothorax complicating Gorham's disease," Ann. Thorac. Surg., vol. 72, pp. 937-939, 2001.
G. G. Miller, "Treatment of chylothorax in Gorham's disease: Case report and literature review," Can J. Surg., vol. 45, pp. 381-382, 2002.
K. Fujiu, R. Kanno, H. Suzuki, N. Nakamura, and M. Gotoh, "Chylothorax associated with massive osteolysis Gorham's syndrome," Ann. Thorac. Surg., vol. 73, pp. 1956-1957, 2002.
S. Y. Yoo, J. M. Goo, and J. G. Im, "Mediastinal lymphangioma and chylothorax: Thoracic involvement of Gorham's disease," Korean J. Radiol., vol. 3, pp. 130-132, 2002.
W. S. Lee, S. H. Kim, I. Kim, H. K. Kim, K. S. Lee, S. Y. Lee, D. S. Heo, B. S. Jang, Y. J. Bang, and N. K. Kim, "Chylothorax in Gorham's disease," J. Korean Med. Sci., vol. 17, pp. 826-829, 2002.
J. Fontanesi, "Radiation therapy in the treatment of Gorham disease," J. Pediatr. Hematol. Oncol., vol. 25, pp. 816-817, 2003.
M. R. Swelstad, C. Frumiento, A. Garry-McCoy, R. Agni, and T. L. Weigel, "Chylotamponade: An unusual presentation of Gorham's syndrome," Ann. Thorac. Surg., vol. 75, pp. 1650-1652, 2003.
L. Kren, P. Rotterova, M. Hermanova, Z. Krenova, J. Sterba, K. Dvorak, V. Goncharuk, G. D. Wilner, and B. J. McKenna, "Chylothorax as a possible diagnostic pitfall: A report of 2 cases with cytologic findings," Acta. Cytol., vol. 49, pp. 441-444, 2005.
A. Takahashi, C. Ogawa, T. Kanazawa, H. Watanabe, M. Suzuki, N. Suzuki, Y. Tsuchida, A. Morikawa, and H. R. Kuwano, "Remission induced by interferon alfa in a patient with massive osteolysis and extension of lymph-hemangiomatosis: A severe case of Gorham-Stout syndrome," J. Pediatr Surg., vol. 40, pp. E47-50, 2005.
B. M. Duffy, R. Manon, R. R. Patel, and J. S. Welsh, "A case of Gorham's disease with chylothorax treated curatively with radiation therapy," Clin. Med. Res., vol. 3, pp. 83-86, 2005.
Pfleger, W. Schwinger, A. Maier, J. Tauss, H. H. Popper, and M. S. Zach, "Gorham-Stout syndrome in a male adolescent-case report and review of the literature," J. Pediatr. Hematol. Oncol., vol. 28, pp. 231-233, 2006.
J. Underwood, J. Buckley, and B. Manning, "Gorham disease: An intraoperative case study," Aana. J., vol. 74, pp. 45-48, 2006.
J. Hagendoorn, T. P. Padera, T. I. Yock, G. P. Nielsen, E. Di Tomaso, D. G. Duda, T. F. Delaney, H. A. Gaissert, J. Pearce, A. E. Rosenberg, R. K. Jain, and D. H. Ebb, "Platelet-derived growth factor receptor-beta in Gorham's disease," Nat. Clin. Pract. Oncol., vol. 3, pp. 693-697, 2006.
V. J. Vigorita, S. Magitsky, and E. Bryk, "Gorham's disease: An autopsy report," Clin. Orthop. Relat. Res., vol. 451, pp. 267-273, 2006.
M. J. Boyle, P. Alison, G. Taylor, and B. A. Lightbourne, "A case of Gorham's disease complicated by bilateral chylothorax," Heart Lung Circ., vol. 17, pp. 64-66, 2008.
M. Kose, S. Pekcan, D. Dogru, C. Akyuz, U. Ozcelik, Y. Ozsurekci, B. Gulhan, M. Demircin, and N. Kiper, "Gorham-Stout syndrome with chylothorax: Successful remission by interferon alpha-2b," Pediatr Pulmonol., vol. 44, pp. 613-615, 2009.
T. S. Yildiz, A. Kus, M. Solak, and K. Toker, "The Gorham-Stout syndrome: One lung ventilation with a bronchial blocker. A case of Gorham's disease with chylothorax," Paediatr Anaesth., vol. 19, pp. 190-191, 2009.
D. K. Kuriyama, S. C. McElligott, D. W. Glaser, and K. Thompson, "Treatment of Gorham-Stout disease with zoledronic acid and interferon-?: A case report and literature review," J. Pediatr Hematol. Oncol., vol. 32, pp. 579-584, 2010.
Y. K. Seok, S. Cho, and E. Lee, "Early surgical management of chylothorax complicated by Gorham's disease," Thorac. Cardiovasc Surg., vol. 58, pp. 492-493, 2010.
K. De Smet, M. De Maeseneer, E. Huijssen-Huisman, V. Van Gorp, S. Hachimi-Idrissi, and C. Ernst, "A rare cause of dyspnea due to chylothorax," Emerg Radiol., vol. 17, pp. 503-505, 2010.
M. Deveci, N. Inan, F. Corapç?o?lu, and G. Ekingen, "Gorham-Stout syndrome with chylothorax in a six-year-old boy," Indian J. Pediatr., vol. 78, pp. 737-739, 2011.
N. Brodszki, J. K. Länsberg, M. Dictor, E. Gyllstedt, S. B. Ewers, M. K. Larsson, and E. A. Eklund, "A novel treatment approach for paediatric Gorham-Stout syndrome with chylothorax," Acta. Paediatr., vol. 100, pp. 1448-1453, 2011.
M. W. Zheng, M. Yang, J. X. Qiu, X. P. Nan, L. Y. Huang, W. D. Zhang, L. Gong, and Z. Z. Huang, "Gorham-Stout syndrome presenting in a 5-year-old girl with a successful bisphosphonate therapeutic effect," Exp. Ther. Med., vol. 4, pp. 449-451, 2012.
R. Kotecha, L. Mascarenhas, H. A. Jackson, and R. Venkatramani, "Radiological features of Gorham's disease," Clin. Radiol., vol. 67, pp. 782-788, 2012.
S. M. Hopman, R. R. Van Rijn, C. Eng, J. Bras, M. Alders, C. M. Van Der Horst, R. C. Hennekam, and J. H. Merks, "PTEN hamartoma tumor syndrome and Gorham-Stout phenomenon," Am. J. Med. Genet. A., vol. 158A, pp. 1719-1723, 2012.
M. Noda, C. Endo, Y. Hoshikawa, N. Ishibashi, T. Suzuki, Y. Okada, and T. Kondo, "Successful management of intractable chylothorax in Gorham-Stout disease by awake thoracoscopic surgery," Gen. Thorac. Cardiovasc. Surg., vol. 61, pp. 356-358, 2013.
A. Barman, R. Bhide, A. Viswanathan, J. George, R. Thomas, and G. Tharion, "Gorham's disease of the spine," NeuroRehabilitation, vol. 33, pp. 121-126, 2013.
B. Chen, X. Lv, J. Wu , X. Zhang, X. Jiao, J. Zhao, Q. Cheng, and C. Cui, "Bone loss in Gorham's disease: A case study," Exp. Ther. Med., vol. 5, pp. 1017-1018, 2013.
C. S. Choi, S. J. Hong, M. A. Chu, S. J. Lee, J. M. Lee, H. I. Bae, and B. Choe, "Gastrointestinal tract involvement of Gorham's disease with expression of D2-40 in Duodenum," Pediatr Gastroenterol Hepatol. Nutr., vol. 17, pp. 52-56, 2014.
R. Ozcan, A. Alptekin, S. Emre, S. Kuruo?lu, M. Inan, and G. Tekant, "An unusual cause of recurrent chylothorax: Gorham syndrome," APSP Journal of Case Reports 5.2, 2014.
A. Daneshvar Kakhaki, K. Khodadad, S. Pejhan, S. Karimi, M. Arab, R. Saghebi, M. Behgam Shadmehr, and R. Farzanegan, "Gorham's disease With chest wall involvement: A case report and a review of the literature," Iran Red. Crescent Med. J., vol. 16, p. e12180. Doi:10.5812/ircmj.12180, 2014.
B. Szabo and W. Habre, "Gorham syndrome: Anaesthetic management," Anaesthesia, vol. 55, pp. 157-159, 2000.
Kitami, T. Suzuki, S. Suzuki, R. Usuda, Y. Kamio, and M. Kadokura, "Gorham's disease complicated by chyloma of the chest wall," Jpn. J. Thorac Cardiovasc Surg., vol. 54, pp. 311-313, 2006.
F. Hammer, W. Kenn, U. Wesselmann, L. C. Hofbauer, G. Delling, B. Allolio, and W. Arlt, "Gorham-Stout disease stabilization during bisphosphonate treatment," J. Bone Miner Res., vol. 20, pp. 350-353, 2005.
M. Schoppet, K. T. Preissner, and L. C. Hofbauer, "RANK ligand and osteoprotegerin: Paracrine regulators of Bone metabolism and vascular function," Arterioscler Thromb. Vasc. Biol., vol. 22, pp. 549-553, 2002.
R. D. Devlin, H. G. Bone, and G. D. Roodman, "Interleukin-6: A potential mediator of the massive osteolysis in patients with Gorham-Stout disease," J. Clin. Endocrinol Metab., vol. 81, pp. 1893-1897, 1996.
E. M. Cassidy, D. Manning, S. Byrne, E. Bolger, F. Murray, N. Sharifi, E. Wallace, M. Keogan, and V. O'Keane, "Acute effects of low-dose interferon-alpha on serum cortisol and plasma interleukin-6," J. Psychopharmacol., vol. 16, pp. 230-234, 2002.
J. Evans, M. F. Clark, L. Mincher, and V. A. Varney, "Chylous effusions complicating lymphoma: A serious event with octreotide as a treatment option," Hematol Oncol., vol. 21, pp. 77-81, 2003.
Valsecchi Alberto , Arondi Sabrina , Marchetti Giampietro (2015). Pleural Effusion in Gorham-Stout Syndrome: A Case Report and a Literature Review. Cancers Review, 2(2): 11-22. DOI: 10.18488/journal.95/2015.2.2/220.127.116.11
Gorham-Stout syndrome (GSS) is an uncommon disease characterized by massive osteolysis due to bone resorption associated with proliferation of blood or lymphatic vessels. Pleural effusion is an uncommon manifestation. We present a case report of GSS in a 46 year old patient admitted for bilateral serohematic pleural effusion and a review of the literature regarding GSS associated with pleural effusion. We analyzed 48 clinical casesfounded in English language literature concerning pleural effusion secondary to GSS. Patients affected by GSS manifesting pleural effusion are prevalently male with a large range of age. Pleural effusion is more frequently chylous (86%), but it can be also serohematic, transudative or bloody. Direct pleural involvement by the disease has been assessed only in some cases of chylothorax, otherwise the pathological process underlying the liquid production remains unknown. Pleural effusion is most of time bilateral. More frequently it conditions ingravescent dyspnea and when it is chylous it can determine malnutrition. No standard therapy has been established. In patients with relapsing pleural effusion several therapeutic approaches have been used: radiation therapy on mediastinum, pleurodesis, thoracic duct ligation, octreotide administration. Radiation therapy seems to be effective even when applied only on the bone lesions. Bisphosphonates and alpha-2b Interferon are useful to improve the multiple sites of disease, thanks to their systemic action. The papers considered report a short follow up time, during which almost 50% of the treated patients had no recurrences, but more or less 20% of the patients died.
This study contributes to the existing literature by presenting the first case of bilateral serohematic pleural effusion in Gorham-Stout syndrome that was investigated with chest ultrasound and medical thoracoscopy bilaterally. Furthermore, this study provides the first review of clinical cases reporting pleural effusion secondary to Gorham-Stout syndrome.