Rationale and efficacy of CD52 targeting in HTLV-1-associated myositis.
We retrospectively analysed two selected patients, referred to our Haematology Department for refractory HTLV-1 associated myositis with circulating pathologic T-cell population with ATL phenotype. They respectively presented also HTLV-1 associated Crohn-like disease and myelopathy. Muscle biopsy of both patients was analysed to determine the pathologic infiltrate. Alemtuzumab was proposed as salvage therapy. Targeting CD52 with alemtuzumab showed good efficacy on myopathy of both patients for respectively 11 and 10 months. Interestingly, this treatment showed also efficacy on circulating pathologic T-cell population and on concomitant digestive and neurological diseases. The double infected cells ablation and immunosuppressive propriety of alemtuzumab probably explains its interest in this infectious and dysimmunitary disorder. Even though alemtuzumab probably remains a suspensive treatment, its place should be assessed in controlled trial in this difficult to treat rare disease.
Service d'Hématologie Adultes, Université Paris Descartes, Paris Sorbonne Cité, Institut Imagine, Faculté de Médecine, AP-HP, Necker-Enfants-Malades, 149, rue des Sèvres, 75743 Paris cedex 15, France. Electronic address: delphine_cochereau@hotmail.fr.
Delphine
D
Cochereau
Service de Médecine Interne, Hôpital Tenon, Université Pierre-et-Marie-Curie, AP-HP, 4, rue de la Chine, 75020 Paris, France.
Sophie
S
Georgin-Lavialle
Laboratoire de Neuropathologie, Hôpital Pitié-Salpêtrière, Université Pierre-et-Marie-Curie, 47, boulevard de l'Hôpital, 75651 Paris cedex 13, France.
Thierry
T
Maisonobe
Laboratoire de Neuropathologie, Hôpital Pitié-Salpêtrière, Université Pierre-et-Marie-Curie, 47, boulevard de l'Hôpital, 75651 Paris cedex 13, France.
Odile
O
Dubourg
Service de Médecine Interne, Hôpital Jean-Verdier, avenue du 14-Juillet, 93140 Bondy, France.
Sara
S
Melboucy-Belkhir
Service d'Hématologie Adultes, Université Paris Descartes, Paris Sorbonne Cité, Institut Imagine, Faculté de Médecine, AP-HP, Necker-Enfants-Malades, 149, rue des Sèvres, 75743 Paris cedex 15, France.
Olivier
O
Hermine
Service d'Hématologie Adultes, Université Paris Descartes, Paris Sorbonne Cité, Institut Imagine, Faculté de Médecine, AP-HP, Necker-Enfants-Malades, 149, rue des Sèvres, 75743 Paris cedex 15, France; Service de Médecine Interne, Hôpital Antoine-Béclère, Université Paris XI, AP-HP, 157, rue de la Porte-de-Trivaux, 92140 Clamart, France.
Achille
A
Aouba
D000074323:Alemtuzumab; D061067:Antibodies, Monoclonal, Humanized / Q000627:therapeutic use*; D015703:Antigens, CD; D000951:Antigens, Neoplasm; D001706:Biopsy; D000074301:CD52 Antigen; D005260:Female; D006023:Glycoproteins / Q000037:antagonists & inhibitors*; D015368:Human T-lymphotropic virus 1; D006801:Humans; D007166:Immunosuppressive Agents / Q000627:therapeutic use*; D008875:Middle Aged; D009132:Muscles / Q000473:pathology; D009220:Myositis / Q000188:drug therapy* / Q000473:pathology / Q000821:virology
D061067:Antibodies, Monoclonal, Humanized; D015703:Antigens, CD; D000951:Antigens, Neoplasm; D000074301:CD52 Antigen; C000613862:CD52 protein, human; D006023:Glycoproteins; D007166:Immunosuppressive Agents; D000074323:Alemtuzumab
Alemtuzumab; Anti-CD52; Crohn-like disease; HTLV-1; Myelopathy/tropical spastic paraparesis; Myositis