A Case of Gorham's Disease with Chylothorax Treated Curatively with Radiation Therapy

Duffy, Bethany M.; Mañon, R.; Patel, Rakesh; Welsh, James S.

doi:10.3121/cmr.3.2.83

Cited by 45 publications

(45 citation statements)

References 24 publications

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“…In this issue of Clinical Medicine & Research, Duffy and colleagues 148 report a case of Gorham's disease with chylothorax that was successfully treated with radiation therapy. The authors should be congratulated for bringing such a rare case to the attention of the medical community and helping to expand upon our poor existing knowledge of this ailment.…”

Section: Discussionmentioning

confidence: 99%

Gorham's Disease or Massive Osteolysis

Patel¹

2005

Clinical Medicine & Research

211

275

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Section: Discussionmentioning

confidence: 99%

Gorham's Disease or Massive Osteolysis

Patel¹

2005

Clinical Medicine & Research

211

275

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“…It has a propensity for involvement of the maxilla, shoulder girdle, ribs, spine and pelvis (shoulder and pelvis most common sites). The disease can also affect the skull, mandible, hand and foot (Duffy, Manon, Patel, & Welsh, 2005). Morbidity and mortality are high in those with spinal and/or visceral involvement.…”

Section: Discussionmentioning

confidence: 99%

Gorham's disease of the spine

Barman

Bhide

Viswanathan

et al. 2013

NRE

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BACKGROUND: Gorham's disease of the spine is very rare and usually associated with poor prognosis. Till date very few cases have been reported in the literature. It is characterized by osseous invasion by angiomatous vascular mass without skip areas, eventually causing lysis of affected bone. Morbidity and mortality are high in those with spinal and/or visceral involvement. Neurological complications increase the mortality to 33% whereas with chylothorax, mortality increases to more than 50%. OBJECTIVES: To describe the clinical course and the challenges in planning and implementing effective rehabilitation services for management of patients with Gorham's disease of the spine. METHODS: We report two patients with Gorham's disease who had neurological deficits secondary to spinal cord involvement, and their rehabilitation perspectives. RESULTS: Both patients achieved satisfactory ambulation and functional outcomes. CONCLUSION: Multiple revision surgeries may be required to ensure spinal stability. When working with these patients, one must remain vigilant about spinal stability as well as about possible serious pulmonary complications, and be prepared to make appropriate management decisions when necessary.

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“…Acute adverse effects such as radiation pneumonitis have been described23 , 32 but seldom occur with fractional doses of 20 Gy or less 33. Chronic complications such as secondary malignancies and restrictive lung disease are rare 34…”

Section: Discussionmentioning

confidence: 99%

Serious complications of pulmonary biopsy in a boy with chylopericardium and suspected pulmonary lymphangiomatosis

Harnisch¹,

Sukhai

Oudesluys‐Murphy

2010

Case Reports

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Lymphangiomatosis is a rare disorder. It is commonly localised in the skeletal system and called Gorham's disease, and in thoracic or abdominal organs. Involvement of the pericardium is rare and has been described in less than 20 patients worldwide. The case of a 14-year-old boy presenting with asymptomatic chylopericardium and interstitial lung disease is presented. After lung biopsy, performed to confirm the diagnosis of pulmonary lymphangiomatosis, he developed chylothorax and massive loss of chyle via chest drains. Thoracic duct ligation did not result in clinical improvement. Treatment with interferon α2b was given and because of clinical deterioration radiotherapy was added to the treatment. This resulted in a rapid decrease of chyle production in the patient.

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A Case of Gorham's Disease with Chylothorax Treated Curatively with Radiation Therapy

Cited by 45 publications

References 24 publications

Gorham's Disease or Massive Osteolysis

Gorham's Disease or Massive Osteolysis

Gorham's disease of the spine

Serious complications of pulmonary biopsy in a boy with chylopericardium and suspected pulmonary lymphangiomatosis

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