A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report

被引:8
作者
Wang, Peng [1 ]
Liao, Wenbo [1 ]
Cao, Guangru [1 ]
Jiang, Yongyan [2 ]
机构
[1] Zunyi Med Univ, Affiliated Hosp, Zunyi, Peoples R China
[2] Zunyi Med Univ, Affiliated Hosp 3, Zunyi, Peoples R China
关键词
Gorham-stout syndrome; Thoracic spine; Chylothorax; LYMPHATIC MALFORMATIONS; CERVICAL-SPINE; DISEASE; MANAGEMENT;
D O I
10.1186/s12891-019-2542-z
中图分类号
R826.8 [整形外科学]; R782.2 [口腔颌面部整形外科学]; R726.2 [小儿整形外科学]; R62 [整形外科学(修复外科学)];
学科分类号
摘要
Background: Gorham-Stout syndrome (GSS) with spinal involvement is extremely rare, and bilateral chylothorax as a complication is usually fatal. In our case, pleural effusion appeared in the left hemithorax after ligating the right thoracic duct. Case presentation: A 14-year-old patient presented with GSS affecting the thoracic spine with bilateral chylothorax. The case was successfully managed using combined conservative and surgical treatments. At the 2-year follow-up visit, the amount of pleural fluid was reduced, the patient's respiratory function had improved, and the deformity on the thoracic spine had gradually stabilized. Conclusions: GSS is a rare disorder of the musculoskeletal system that responds poorly to therapies and exhibits very high morbidity and mortality. Chylothorax is a common complication when lesions involve the thoracic spine, and physicians should be vigilant for possible serious pulmonary complications.
引用
收藏
页数:6
相关论文
共 22 条
[1]   A novel treatment approach for paediatric Gorham-Stout syndrome with chylothorax [J].
Brodszki, Nicholas ;
Lansberg, John-Kalle ;
Dictor, Michael ;
Gyllstedt, Erik ;
Ewers, Sven-Borje ;
Larsson, Marcus K. ;
Eklund, Erik A. .
ACTA PAEDIATRICA, 2011, 100 (11) :1448-1453
[2]   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
[3]   Gorham-Stout disease of costo-vertebral localization: Radiographic, scintigraphic, computed tomography, and magnetic resonance imaging findings [J].
Ceroni, D ;
De Coulon, G ;
Regusci, M ;
Kaelin, A .
ACTA RADIOLOGICA, 2004, 45 (04) :464-468
[4]   Viewpoints on vessels and vanishing bones in Gorham-Stout disease [J].
Dellinger, Michael T. ;
Garg, Nupur ;
Olsen, Bjorn R. .
BONE, 2014, 63 :47-52
[5]   Gorham-Stout Syndrome with Chylothorax in a Six-Year-Old Boy [J].
Deveci, Murat ;
Inan, Nagihan ;
Corapcioglu, Funda ;
Ekingen, Gulsen .
INDIAN JOURNAL OF PEDIATRICS, 2011, 78 (06) :737-739
[6]   Lymphatic malformations: Diagnosis and management [J].
Elluru, Ravindhra G. ;
Balakrishnan, Karthik ;
Padua, Horacio M. .
SEMINARS IN PEDIATRIC SURGERY, 2014, 23 (04) :178-185
[7]   DISAPPEARING BONES - A RARE FORM OF MASSIVE OSTEOLYSIS - REPORT OF 2 CASES, ONE WITH AUTOPSY FINDINGS [J].
GORHAM, LW ;
WRIGHT, AW ;
SHULTZ, HH ;
MAXON, FC .
AMERICAN JOURNAL OF MEDICINE, 1954, 17 (05) :674-682
[8]   PERSPECTIVES ON MASSIVE OSTEOLYSIS - REPORT OF A CASE AND REVIEW OF THE LITERATURE [J].
HEFFEZ, L ;
DOKU, HC ;
CARTER, BL ;
FEENEY, JE .
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY ORAL RADIOLOGY AND ENDODONTICS, 1983, 55 (04) :331-343
[9]  
HELLYER J, 2004, AJP, V6, pE355, DOI DOI 10.1055/S-0036-1593443
[10]   PTEN hamartoma tumor syndrome and Gorham-Stout phenomenon [J].
Hopman, Saskia M. J. ;
Van Rijn, Rick R. ;
Eng, Charis ;
Bras, Johannes ;
Alders, Marielle ;
van der Horst, Chantal M. ;
Hennekam, Raoul C. M. ;
Merks, Johannes H. M. .
AMERICAN JOURNAL OF MEDICAL GENETICS PART A, 2012, 158A (07) :1719-1723