Improvement of Disseminated Lymphangiomatosis with Recombinant Interferon Therapy

Overview

Journal Pediatr Pulmonol

Specialties Pediatrics
Pulmonary Medicine

Date 2000 Mar 29

PMID 10738021

Citations 16

Authors

C Laverdiere

M David

J Dubois

P Russo

L Hershon

J G Lapierre

Affiliations

Soon will be listed here.

Abstract

Disseminated lymphangiomatosis is a rare disorder with a poor prognosis. We present a case involving a 3-year-old boy who presented with pulmonary infiltrates, multiple lytic lesions of the ribcage, and small cystic lesions in the spleen. Open-lung and bone biopsies revealed disseminated lymphangiomatosis. Significant clinical and radiologic improvement were observed and persisted after 28 months of treatment with recombinant interferon alpha-2b (IFN alpha-2b). No significant toxicity has been observed.

Citing Articles

Pathologic Fracture as Presentation of Generalized Lymphangiomatosis in an Eight-Year-Old: A Case Report and Literature Review.

Ghorishi A, Levin N, Nomula K, Green J, Eisner E Cureus. 2022; 14(4):e24380.

PMID: 35619846 PMC: 9126478. DOI: 10.7759/cureus.24380.

Generalized lymphatic anomalies and review of the current management landscape: a case report and review of the literature.

Liu T, Basseri S, Mussari B, Dabreo D, SenGupta S, Villalobos D J Med Case Rep. 2021; 15(1):398.

PMID: 34372919 PMC: 8353871. DOI: 10.1186/s13256-021-02953-9.

A gene missense mutation in diffuse pulmonary lymphangiomatosis with thrombocytopenia: A case report.

Zheng G, Tang H, Su R, Liang Y, He Z, Zhang J Medicine (Baltimore). 2020; 99(39):e21941.

PMID: 32991402 PMC: 7523817. DOI: 10.1097/MD.0000000000021941.

Unusual case of chylothorax with unilateral limb swelling.

Jha V, Jha A BMJ Case Rep. 2020; 13(2).

PMID: 32047079 PMC: 7021149. DOI: 10.1136/bcr-2019-230379.

Somatic activating mutations in cause generalized lymphatic anomaly.

Rodriguez-Laguna L, Agra N, Ibanez K, Oliva-Molina G, Gordo G, Khurana N J Exp Med. 2018; 216(2):407-418.

PMID: 30591517 PMC: 6363432. DOI: 10.1084/jem.20181353.