Gorham-Stout Syndrome with Chylothorax in a Six-Year-Old Boy

被引:23
作者
Deveci, Murat [1 ]
Inan, Nagihan [2 ]
Corapcioglu, Funda [3 ]
Ekingen, Gulsen [4 ]
机构
[1] Ege Univ, Fac Med, Dept Pediat Cardiol, TR-35040 Bornova, Turkey
[2] Kocaeli Univ, Dept Radiol, Fac Med, Kocaeli, Turkey
[3] Kocaeli Univ, Dept Pediat Oncol, Fac Med, Kocaeli, Turkey
[4] Kocaeli Univ, Dept Pediat Surg, Fac Med, Kocaeli, Turkey
关键词
Gorham-Stout syndrome; Chylothorax; Osteolysis; MASSIVE OSTEOLYSIS; DISEASE;
D O I
10.1007/s12098-010-0328-2
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Gorham-Stout syndrome, also called "disappearing bone disease, diffuse cystic angiomatosis of bone, disseminated lymphangiomatosis, Gorham's vanishing bone disease, phantom bone disease or idiopathic massive osteolysis, is a rare disease of unknown etiology and pathogenesis. It is characterized by rapidly progressive localized massive osteolysis associated with proliferation of vascular structures of benign origin in which the absence of new bone formation is representative. When it is complicated by chylothorax, the prognosis is poor. The authors report a 6-year-old boy with Gorham-Stout syndrome who presented with pleural effusion showing features of chylothorax, who responsed poorly to currently available therapeutic modalities.
引用
收藏
页码:737 / 739
页数:3
相关论文
共 10 条
[1]   Cutaneous lymphatic malformations in disappearing bone (Gorham-Stout) disease: A novel clue to the pathogenesis of a rare syndrome [J].
Bruch-Gerharz, Daniela ;
Gerharz, Claus-Dieter ;
Stege, Helger ;
Krutmann, Jean ;
Pohl, Michael ;
Koester, Rainer ;
Ruzicka, Thomas .
JOURNAL OF THE AMERICAN ACADEMY OF DERMATOLOGY, 2007, 56 (02) :S21-S25
[2]   MASSIVE OSTEOLYSIS (ACUTE SPONTANEOUS ABSORPTION OF BONE, PHANTOM BONE, DISAPPEARING BONE) - ITS RELATION TO HEMANGIOMATOSIS [J].
GORHAM, LW ;
STOUT, AP .
JOURNAL OF BONE AND JOINT SURGERY-AMERICAN VOLUME, 1955, 37 (05) :985-1004
[3]   α-2b interferon and oral clodronate for Gorham's disease [J].
Hagberg, F ;
Lamberg, K ;
Åström, G .
LANCET, 1997, 350 (9094) :1822-1823
[4]   Gorham-Stout disease - Stabilization during bisphosphonate treatment [J].
Hammer, F ;
Kenn, W ;
Wesselmann, U ;
Hofbauer, LC ;
Delling, G ;
Allolio, B ;
Arlt, W .
JOURNAL OF BONE AND MINERAL RESEARCH, 2005, 20 (02) :350-353
[5]   Gorham disease: correlation of MR findings with histopathologic changes [J].
Manisali, M ;
Ozaksoy, D .
EUROPEAN RADIOLOGY, 1998, 8 (09) :1647-1650
[6]   The Gorham-Stout syndrome (Gorham's massive osteolysis) -: A report of six cases with histopathological findings [J].
Möller, G ;
Priemel, M ;
Amling, M ;
Werner, M ;
Kuhlmey, AS ;
Delling, G .
JOURNAL OF BONE AND JOINT SURGERY-BRITISH VOLUME, 1999, 81B (03) :501-506
[7]   Gorham's Disease or Massive Osteolysis [J].
Patel, Dipak V. .
CLINICAL MEDICINE & RESEARCH, 2005, 3 (02) :65-74
[8]   Bilateral chylothorax complicating massive osteolysis (Gorham's syndrome) [J].
Riantawan, P ;
Tansupasawasdikul, S ;
Subhannachart, P .
THORAX, 1996, 51 (12) :1277-1278
[9]   Remission induced by interferon alfa in a patient with massive osteolysis and extension of lymph-hemangiomatosis: a severe case of Gorham-Stout syndrome [J].
Takahashi, A ;
Ogawa, C ;
Kanazawa, T ;
Watanabe, H ;
Suzuki, M ;
Suzuki, N ;
Tsuchida, Y ;
Morikawa, A ;
Kuwano, H .
JOURNAL OF PEDIATRIC SURGERY, 2005, 40 (03) :E47-E50
[10]   CHYLOTHORAX IN GORHAMS SYNDROME - A COMMON COMPLICATION OF A RARE DISEASE [J].
TIE, MLH ;
POLAND, GA ;
ROSENOW, EC .
CHEST, 1994, 105 (01) :208-213