CLINICAL IMPROVEMENT OF DIFFUSE LYMPHANGIOMATOSIS WITH PEGYLATED INTERFERON ALFA-2B THERAPY: Case Report and Review of the Literature

Ozeki, Michio; Funato, Michinori; Kanda, Kaori; Ito, Masako; Teramoto, Tamio; Kaneko, Hideo; Fukao, Toshiyuki; Kondo, Naomi

doi:10.1080/08880010701533603

Cited by 48 publications

(34 citation statements)

References 22 publications

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“…Disease manifestation in bone and surrounding soft tissue is called Gorham’s disease or vanishing bone disease 4 – 6. Other localisations are liver, spleen and mediastinum 7 – 14…”

Section: Discussionmentioning

confidence: 99%

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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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“…Disease manifestation in bone and surrounding soft tissue is called Gorham’s disease or vanishing bone disease 4 – 6. Other localisations are liver, spleen and mediastinum 7 – 14…”

Section: Discussionmentioning

confidence: 99%

“…Interferon α, a cytokine with antiangiogenic activity, appears to be beneficial and has been used for the treatment of lymphangiomatosis for almost 20 years. Clinical improvement has been reported in the majority of cases, although the optimal treatment duration remains unclear and ranges from 3 weeks to several years 7 , 21 , 24 , 26 , 27…”

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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“…Interferon, which has mild side-effects, seems to stop or reduce lesion growth, but at this time, no data confirm symptom resolution after discontinuation of therapy [45,78,79].…”

Section: Therapymentioning

confidence: 94%

Thoracic involvement in generalised lymphatic anomaly (or lymphangiomatosis)

2016

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Generalised lymphatic anomaly (GLA), also known as lymphangiomatosis, is a rare disease caused by congenital abnormalities of lymphatic development. It usually presents in childhood but can also be diagnosed in adults. GLA encompasses a wide spectrum of clinical manifestations ranging from singleorgan involvement to generalised disease. Given the rarity of the disease, most of the information regarding it comes from case reports. To date, no clinical trials concerning treatment are available. This review focuses on thoracic GLA and summarises possible diagnostic and therapeutic approaches. @ERSpublications Possible diagnostic and therapeutic approaches to generalised lymphatic anomaly (lymphangiomatosis)

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“…116 Lymphangiomas are dilated endothelial-lined lymph channels with thick walls containing smooth muscle. 118 Hemangiomas and lymphangiomas classically are malformations or low-grade and benign neoplasms with no capacity for metastasis. 118 Hemangiomas and lymphangiomas classically are malformations or low-grade and benign neoplasms with no capacity for metastasis.…”

Section: Pathology and Pathways Of Spreadmentioning

confidence: 99%