Improvement of disseminated lymphangiomatosis with recombinant interferon therapy.
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.
Laverdière
C
C
Section of Hematology/Oncology, Division of Pediatrics, Ste-Justine Hospital, University of Montreal, Montreal, Canada.
David
M
M
Dubois
J
J
Russo
P
P
Hershon
L
L
Lapierre
J G
JG
D020533:Angiogenesis Inhibitors / Q000627:therapeutic use*; D000970:Antineoplastic Agents / Q000627:therapeutic use*; D002675:Child, Preschool; D006801:Humans; D000077190:Interferon alpha-2; D016898:Interferon-alpha / Q000627:therapeutic use*; D008175:Lung Neoplasms / Q000000981:diagnostic imaging / Q000188:drug therapy*; D018192:Lymphangioleiomyomatosis / Q000000981:diagnostic imaging / Q000188:drug therapy*; D008297:Male; D011994:Recombinant Proteins; D014057:Tomography, X-Ray Computed
D020533:Angiogenesis Inhibitors; D000970:Antineoplastic Agents; D000077190:Interferon alpha-2; D016898:Interferon-alpha; D011994:Recombinant Proteins