Case Report: Gorham-Stoute Syndrome with Involvement of Majority of Mandible, and Partial Maxillary, Temporal and Zygomatic Bones

Evrenos, Mustafa Kürşat; Özkaya, Merve; Yaman, Murat; Proff, Levent Yoleri

doi:10.1007/s12663-015-0869-z

Cited by 9 publications

(1 citation statement)

References 14 publications

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“…Severe complications of GSD including paraplegia or death in cases of osteolysis with vertebral involvement [4,7]; respiratory failure, hemothorax, or chylothorax in the cases of chest injury; or bacterial meningitis in cases of osteomeningeal breccia [2,12] are rare. In 16% of cases, this disease is fatal [13].…”

Section: Discussionmentioning

confidence: 99%

Ectopic teeth in a patient with Gorham-Stout disease previously treated by biphosphonates: a case report

Donne

Jaby

Farge

et al. 2020

J Oral Med Oral Surg

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Introduction: Gorham-Stout disease is a rare idiopathic condition of the bone that is characterized by a massive and spontaneous osteolysis, with a vascular or lymphatic proliferation in bone, which is then replaced by fibrous tissue. Observation: a 16-year-old patient was referred to the maxillofacial surgery department to remove ectopic teeth bilaterally in the ramus. He had Gorham-Stout disease, managed for many years in orthopedic surgery department for a lower limb lesion and in neurosurgery department for a breach of the meninges. He was treated for 4 years with bisphosphonates. The removal of the ectopic teeth went well, with a simple postoperative course. Discussion: Gorham-Stout disease physiopathology remains unknown. Facial bones are often involved, especially the mandible. There are many possible treatments, but, due to the rarity of the disease, no therapeutic consensus exists. Bisphosphonates seem to be a good way to control this condition. So far, no case of bisphosphonates related osteonecrosis of the jaw has been reported in children. Conclusion: Gorham-Stout disease can involve the mandible and may lead to ectopic teeth.

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Section: Discussionmentioning

confidence: 99%

Ectopic teeth in a patient with Gorham-Stout disease previously treated by biphosphonates: a case report

Donne

Jaby

Farge

et al. 2020

J Oral Med Oral Surg

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Gorham-Stout disease of skull base leading to cranial settling and rhinorrhea: a case-based review

et al. 2022

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Gorham-Stout Disease: a Clinical Case Report and Immunological Mechanisms in Bone Erosion

Franco-Barrera

Zavala-Cerna

Aguilar-Portillo

et al. 2016

Clinic Rev Allerg Immunol

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Gorham-Stout disease (GSD) is a rare condition of osteolysis with excessive lymphangiogenesis within bone tissue. The etiology of this condition remains unknown but seems to affect mainly children and young adults of both genders all over the world. Unfortunately, there is no standardized method for diagnosis; however, histopathology remains as the gold standard. This condition is often misdiagnosed due to its varying clinical presentations from case-to-case. Here, we report the case of an 8-year-old girl who presented with chronic mandibular pain during mastication and received multiple antibiotic treatment due to infectious origin suspicion. After integrating information from clinical manifestations, radiographic, laboratory, and histopathology information, she was diagnosed with GSD. Additionally, due to the lack of literature with respect to insights into biological mechanisms and standardized treatment for this condition, we underwent a literature revision to provide information related to activation of cells from the immune system, such as macrophages, T-cells, and dendritic cells, and their contribution to the lymphangiogenesis, angiogenesis, and osteoclastogenic process in GSD. It is important to consider these mechanisms in patients with GSD, especially since new studies performed in earlier stages are required to confirm their use as novel diagnostic tools and find new possibilities for treatment.

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Case Report: Gorham-Stoute Syndrome with Involvement of Majority of Mandible, and Partial Maxillary, Temporal and Zygomatic Bones

Cited by 9 publications

References 14 publications

Ectopic teeth in a patient with Gorham-Stout disease previously treated by biphosphonates: a case report

Ectopic teeth in a patient with Gorham-Stout disease previously treated by biphosphonates: a case report

Gorham-Stout disease of skull base leading to cranial settling and rhinorrhea: a case-based review

Gorham-Stout Disease: a Clinical Case Report and Immunological Mechanisms in Bone Erosion

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