Purpose: This study was aimed to investigate the clinical features, imaging examination, and treatment of the patients with Gorham-Stout disease (GSD) in maxillofacial region, so as to improve the understanding of GSD.
Methods: The medical records of the patients with GSD who were referred to Shanghai Ninth People's Hospital from January 2010 to May 2016 were reviewed. Their ages, lesion location, imaging results, laboratory examination results, treatment, and therapeutic effects were analyzed.
Results: A total of 4 cases were included (males 2, females 2). The average onset age was 40 years. GSD attacked the mandible in 2 cases; mandible and temporal bone in 1 case; and mandible, temporal bone, and zygoma in 1 case. All cases were examined by computed tomography (CT), which showed bone resorption and atrophy of soft tissue in involved region. Four patients were given alendronate for treatment. All of them had no significant signs of progress after treatment.
Conclusions: GSD can affect one single bone or multiple bones in maxillofacial region. The diagnosis mainly depends on the imaging examinations. Enhanced CT or magnetic resonance imaging is advocated for differential diagnosis of this disease. Alendronate was used with apparent good effect in these patients.
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http://dx.doi.org/10.1097/SCS.0000000000004188 | DOI Listing |
Pediatr Radiol
January 2025
Department of Radiology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Complex lymphatic anomalies are unique diseases marked by abnormal lymphatic vessel development and growth. Imaging is crucial in the evaluation and management of complex lymphatic anomalies, with dynamic contrast-enhanced MR lymphangiography emerging as a valuable modality for visualizing abnormal lymphatic structures and informing treatment decisions. This article gives an overview of complex lymphatic anomalies and their management strategies, focusing specifically on generalized lymphatic anomaly, Gorham-Stout disease, Kaposiform lymphangiomatosis, and central conducting lymphatic anomalies.
View Article and Find Full Text PDFAnn Thorac Surg Short Rep
December 2024
Thoracic Surgery Department, Nottingham University Hospitals NHS Trust, UK.
Gorham-Scout disease (GSD) is a rare skeletal disorder of unknown etiology characterized by progressive osteolysis and excessive lymphovascular proliferation. Chylothorax is a life-threatening complication. A teenager presented with a left pleural effusion on a background of chronic flank collection secondary to lymphovascular malformation.
View Article and Find Full Text PDFBMC Musculoskelet Disord
December 2024
Department of Orthopaedic Surgery, Peking Union Medical College Hospital, Peking Union Medical College, Chinese Academy of Medical Science, Beijing, People's Republic of China.
Background: Gorham-Stout disease (GSD) is a rare disease characterized by osteolysis and lymphatic malformations. GSD involving the spine is exceptionally rare and lacks a standard cure. The aim of this article was to report a case of GSD with scoliosis treated via corrective surgery and medication.
View Article and Find Full Text PDFJBJS Case Connect
October 2024
Department of Orthopaedics, Texas Scottish Rite Hospital for Children, Dallas, Texas.
Case: This is a case of a 6-year-old patient diagnosed with Gorham-Stout disease (GSD), a rare lymphangiogenic skeletal disorder, localized to the left femur. Initial nonoperative treatment with pharmaceuticals and bracing was unsuccessful. We describe a definitive operative treatment with radical femoral resection and a modified rotationplasty technique through a tibiopelvic rotational hip arthroplasty.
View Article and Find Full Text PDFJ Inflamm Res
November 2024
Department of orthopedics, the Affiliated Changzhou Second People's Hospital of Nanjing Medical University, Changzhou, People's Republic of China.
Purpose: The polarization of macrophages towards the pro-inflammatory M1 phenotype and osteoclast overactivation play a significant role in the pathogenesis of aseptic loosening of orthopedic implants. This study sought to examine the expression and activation of macrophages and osteoclasts in implant biopsies with respect to epidermal growth factor receptor (EGFR) signaling and to assess the potential of EGFR inhibition in mitigating titanium particle-induced bone resorption in a cranial resorption murine model.
Methods: Bone marrow-derived macrophages (BMDMs) were stimulated with Tumor Necrosis Factor-alpha (TNF-α) and Interferon-gamma (IFN-γ) initially.
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