title: Cystic bone angiomatosis: a case report treated with aminobisphosphonates and review of the literature.
abstract: Cystic angiomatosis (CA) is a rare disease characterized by multifocal hemangiomatous and/or lymphangiomatous lesions of the skeleton with possible visceral organ involvement. The exact pathogenetic mechanism of the disease is still unknown. We describe a patient affected by CA of bone treated with surgical procedures and subsequently with intravenous aminobisphosphonates for 7 years. During the follow-up progression of lesions, the painful symptoms, markers of bone turnover, computed tomographic examination, and bone mineral density were evaluated. Aminobisphosphonate therapy showed an immediate effectiveness in reducing bone pain, with a significant decrease in circulating bone alkaline phosphatase and stable radiological findings during clinical follow-up. In addition, at baseline, high levels of bone biomarkers and cytokines (osteoprotegerin, osteopontin, and interleukin-6) capable of controlling bone metabolism and angiomatosis were identified. Aminobisphosphonate treatment produced a decrease of all these increased markers. Local cell therapy with bone marrow osteoblast precursors did not produce any measurable clinical improvement. Aminobisphosphonate therapy represents an elective treatment for bone angiomatosis syndromes, but further studies are necessary to understand the molecular basis of these disorders and of their pharmacological treatment.
authors.affiliation: Bone and Mineral Metabolic Unit, Department of Internal Medicine, University of Florence Medical School, Largo Palagi 1, 50139, Florence, Italy.
authors.forename: Gemma
authors.initials: G
authors.lastname: Marcucci
authors.forename: Laura
authors.initials: L
authors.lastname: Masi
authors.forename: Anna Maria
authors.initials: AM
authors.lastname: Carossino
authors.forename: Alessandro
authors.initials: A
authors.lastname: Franchi
authors.forename: Rodolfo
authors.initials: R
authors.lastname: Capanna
authors.forename: Luigi
authors.initials: L
authors.lastname: Sinigaglia
authors.forename: Maria Luisa
authors.initials: ML
authors.lastname: Brandi
mesh_terms: D000798:Angiomatosis / Q000150:complications* / Q000188:drug therapy*; D001845:Bone Cysts / Q000150:complications* / Q000188:drug therapy*; D004164:Diphosphonates / Q000627:therapeutic use*; D006801:Humans; D008159:Lumbar Vertebrae; D008297:Male; D013122:Spinal Diseases / Q000150:complications / Q000188:drug therapy; D055815:Young Adult
chemical_list: D004164:Diphosphonates