Fourteen years follow-up of massive pelvic girdle osteolysis caused by lymphatic malformation (Gorham-Stout Disease)

Ballas, Richard; Caduc, Madeline; Ovigue, Jordan; Elkienbaum, Philippe; Sauvat, Frédérique

doi:10.1016/j.jbspin.2016.12.009

Cited by 3 publications

(5 citation statements)

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“…To identify previously reported cases of GSS in spine, PubMed was searched using the keywords,“Spine,” and, “Gorham-Stout syndrome,” which yielded 9 results of GSS of the lumbar and sacral spine. [ 6 – 14 ] References from these articles were reviewed but yielded no additional results. As we present a rare case of GSS located in the lumbar and sacral spine, an effort was made to search for similar cases published ever (Table 1 ).…”

Section: Discussionmentioning

confidence: 99%

“…As we present a rare case of GSS located in the lumbar and sacral spine, an effort was made to search for similar cases published ever (Table 1 ). [ 6 – 14 ]…”

Section: Discussionmentioning

confidence: 99%

“…In patients with GSS in the spine, surgical treatment are recommended if severe low back pain, pathologic fracture, paresthesia, functional impairment and paralysis occur. [ 6 – 14 ] To date, surgical management of GSS of spine has remained under evaluation, with no standard criteria. We described the case of GSS in lumbar and sacral spine that underwent vertebroplasty with cement augmentation.…”

Section: Discussionmentioning

confidence: 99%

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Successful treatment of Gorham–Stout syndrome in the spine by vertebroplasty with cement augmentation

Liu¹,

Zhou²,

Song

et al. 2018

Medicine

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Rationale:Gorham-Stout syndrome in the spine is extremely rare, and there is no standard curative management thus far. The objective of this article is to report a very rare case of Gorham-Stout syndrome of the lumbar and sacral spine with chylothorax and chyloperitoneum successfully treated by combination of vertebroplasty with cement augmentation and medication treatment. We described the clinical characteristics and postoperative therapy of the patient, and reviewed all of the published cases of Gorham-Stout syndrome of the lumbar and sacral spine.Patient concerns:A 31-year-old man presented with increasingly serious abdominal distention and back pain. MRI showed massive bony destruction of the spine and pelvis. CT and ultrasonography demonstrated massive ascites and mild hydrothorax.Diagnoses:We believe this is the first report of a case of Gorham-Stout syndrome with both chylothorax and chyloperitoneum.Interventions:Chest and abdominal cavity puncture was performed for symptomatic relief and the test results confirmed chylothorax and chyloperitoneum. Tissue biopsy and percutaneous vertebroplasty at L5 were performed and the postoperative pathology together with symptoms and examinations were reported to be consistent with Gorham-Stout syndrome. Subsequently, we administered combination medical treatment consisting of interferon-α-2b, zoledronic acid and calcitriol.Outcomes:At the 1-year and 2-year follow-up visit, he had nearly full complete remission and reported palliative back pain. Moreover, the amount of pleural and peritoneal fluid was successfully reduced gradually.Lessons:Vertebroplasty by cement augmentation may be a treatment option for patients with Gorham-Stout Syndrome in the spine who cannot undergo appropriate surgery or decline open surgery. This represents a safe and minimally invasive approach to sustainably relieve pain and stabilize vertebral bodies with Gorham-Stout syndrome in the spine.

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Section: Discussionmentioning

confidence: 99%

“…As we present a rare case of GSS located in the lumbar and sacral spine, an effort was made to search for similar cases published ever (Table 1 ). [ 6 – 14 ]…”

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Successful treatment of Gorham–Stout syndrome in the spine by vertebroplasty with cement augmentation

Liu¹,

Zhou²,

Song

et al. 2018

Medicine

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“…Certaines images sont très impressionnantes comme la disparition complète de l'hémibassin droit d'une femme au cours de 14 ans de suivi rapportée par Ballas [15]. Les parties molles adjacentes peuvent être infiltrées par la lésion.…”

Section: Imagerieunclassified

Perspectives dans la maladie de Gorham–Stout

Estublier

Chavassieux

Vignot

et al. 2019

Revue du Rhumatisme Monographies

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“…Gorham-Stout disease was initially described by Jackson in 1838 and later classified by Gorham and Stout in 1955 [3, 4]. Worldwide, considering all the potential affected regions only 200 cases have been reported.…”

Section: Introductionmentioning

confidence: 99%

A report of two children with Gorham-Stout disease

et al. 2019

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Background Gorham-Stout disease is a rare condition characterized by unifocal and massive type IV osteolysis (variant of idiopathic nonhereditary osteolytic disease) with a slow progression, which is self-limiting for some years. It is characterized by recurrent vascular tumors with disruption of the anatomical architecture and intraosseous proliferation of vascular channels that leads to the destruction and resorption of the bone matrix. The aim of this study is to present the clinical features of this disease, as well as the importance of prompt diagnosis and treatment, with a review of the reported cases. Case reports We describe two cases of Gorham-Stout disease between 2013 and 2017 with surgical interventions, follow-up and results. Case one involves an 11-year-old male with involvement of the left iliac bone, with adequate evolution after a surgical procedure with a lyophilized cadaveric tricortical bone allograft. Case two involves a 6-year-old male with cervical spine C1-C3 repercussion; in the protocol for surgical treatment, he presented with signs of spinal cord compression and died. Conclusion Diagnosis of Gorham-Stout disease is made by exclusion, and its clinical presentation varies widely, from spontaneous remission to a fatal outcome. Electronic supplementary material The online version of this article (10.1186/s12887-019-1561-0) contains supplementary material, which is available to authorized users.

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Fourteen years follow-up of massive pelvic girdle osteolysis caused by lymphatic malformation (Gorham-Stout Disease)

Cited by 3 publications

References 1 publication

Successful treatment of Gorham–Stout syndrome in the spine by vertebroplasty with cement augmentation

Successful treatment of Gorham–Stout syndrome in the spine by vertebroplasty with cement augmentation

Perspectives dans la maladie de Gorham–Stout

A report of two children with Gorham-Stout disease

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