Gorham disease of the craniocervical junction: X-ray, computed tomography, and magnetic resonance imaging findings

Kılıçoğlu, Zeynep Gamze; Kış, Naciye; Aker, Fügen; Berkman, Mehmet Zafer; Şimşek, Mikdat

doi:10.1016/j.spinee.2013.01.039

Cited by 8 publications

(5 citation statements)

References 8 publications

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“…As reported both in our cases and in the literature (9,16,(23)(24)(25)(26)(27)(28)(29), and summarized in Table 5, spinal GSS can occur at any age and at any level from the cervical spine to the sacrum; however, the cervical and thoracic vertebrae are the most frequently involved regions. The early clinical manifestations of GSS are often subtle and nonspecific.…”

Section: Discussionsupporting

confidence: 71%

Spinal Gorham-Stout syndrome: radiological changes and spinal deformities

Du¹,

Li²,

Xu³

et al. 2019

Quant. Imaging Med. Surg

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Background: Gorham-Stout syndrome (GSS) involving the spine is a rare clinical entity, and there is a paucity of comprehensive study on its radiological features. We aimed to present the radiological changes and spinal deformities in patients with spinal GSS. Methods: From January 2005 to December 2017, 11 consecutive GSS patients with spinal deformity were identified. Their medical records and imaging features were retrospectively reviewed. Computed tomography (CT) and magnetic resonance imaging (MRI) were used for a precise evaluation of spinal involvement. Posteroanterior and lateral standing radiographs were used to evaluate the spinal deformity. Results: CT showed multi-level generalized osteolytic lesions, with ill-defined fluid attenuation adjacent to the osseous changes. MRI demonstrated hyperintense signals on both T1-and T2-weighted images, while the unaffected segments showed normal signal intensity. Seven patients (63.6%) had a dominant feature of kyphosis, and 4 (36.4%) had scoliosis when spinal GSS was diagnosed. Kyphosis variably spanned from C7 to L1, averaged 94° (range, 53° to 158°), and was associated with sagittal imbalance in 4 cases. In our series, the apex of kyphosis and scoliosis coincided within the most seriously osteolytic segment. All patients received medication for GSS. Two adolescents taking Boston braces showed a relatively stable deformity. Four patients received long posterior spinal fusion, but two had fusion failure. Conclusions: CT and MRI investigations are important in the initial diagnosis and continued management for spinal GSS. A typical spinal deformity secondary to GSS presents as kyphosis or kyphoscoliosis, which is usually highly variable but highly concordant with osteolysis in terms of span and apex.

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Section: Discussionsupporting

confidence: 71%

Spinal Gorham-Stout syndrome: radiological changes and spinal deformities

Du¹,

Li²,

Xu³

et al. 2019

Quant. Imaging Med. Surg

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“…Clinical manifestations vary depending on the site and the extent of involvement. Some patients may present with abrupt pain due to pathologic fracture [3].…”

Section: Discussionmentioning

confidence: 99%

Clavicle pain

Bai

Zhang

2018

Skeletal Radiol

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“… 6 To the best of our knowledge, 13 cases of GD of spine have been reported after 2005. 8 20 21 22 23 24 25 26 27 28 29 30 31 A brief review of these cases with treatment and outcome has been shown in Table 2 . Out of the total 41 reported cases of GD of spine, cervical spine is the most commonly affected with 11 cases involving only cervical spine and additional 6 cases involving cervicothoracic spine.…”

Section: Discussionmentioning

confidence: 99%

Vanishing bone disease of chest wall and spine with kyphoscoliosis and neurological deficit: A case report and review of literature

Srivastava¹,

Aggarwal²,

Nemade³

et al. 2017

IJOO

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Vanishing bone disease is an extremely rare disorder of unknown etiology characterized by idiopathic osteolysis of bone. We describe a case of vanishing bone disease of chest wall and spine with kyphoscoliosis and neurological deficit. A 17-year-old male presented with gradually progressive deformity of back and dorsal compressive myelopathy with nonambulatory power in lower limbs. Radiographs revealed absent 4th–7th ribs on the right side with dorsal kyphoscoliosis and severe canal narrowing at the apex. The patient was given localized radiotherapy and started on a monthly infusion of 4 mg zoledronic acid. Posterior instrumented fusion with anterior reconstruction via posterolateral approach was performed. The patient had a complete neurological recovery at 5 weeks following surgery. At 1 year, anterior nonunion was noted for which transthoracic tricortical bone grafting was done. Bone graft from the patient's mother was used both times. At 7 months following anterior grafting, the alignment was maintained and the patient was asymptomatic; however, fusion at graft-host interface was not achieved. Bisphosphonates and radiotherapy were successful in halting the progress of osteolysis.

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Gorham disease of the craniocervical junction: X-ray, computed tomography, and magnetic resonance imaging findings

Cited by 8 publications

References 8 publications

Spinal Gorham-Stout syndrome: radiological changes and spinal deformities

Spinal Gorham-Stout syndrome: radiological changes and spinal deformities

Clavicle pain

Vanishing bone disease of chest wall and spine with kyphoscoliosis and neurological deficit: A case report and review of literature

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