Martina Tavarelli1, Julie Sarfati1, Christian De Gennes2, Julien Haroche3, Camille Buffet1, Cécile Ghander1, Jean Marc Simon4, Fabrice Ménégaux5, Laurence Leenhardt1. 1. Unité Thyroïde Tumeurs Endocrines, Groupe Hospitalier et Faculté de Médecine Pitié-Salpêtrière, Institut du Cancer, Université Pierre et Marie Curie, Paris, France. 2. Service de Médecine Interne Immuno-clinique, Groupe Hospitalier et Faculté de Médecine Pitié-Salpêtrière, Institut du Cancer, Université Pierre et Marie Curie, Paris, France. 3. Service de Médecine Interne 2, Groupe Hospitalier et Faculté de Médecine Pitié-Salpêtrière, Institut du Cancer, Université Pierre et Marie Curie, Paris, France. 4. Service de Radiothérapie, Groupe Hospitalier et Faculté de Médecine Pitié-Salpêtrière, Institut du Cancer, Université Pierre et Marie Curie, Paris, France. 5. Service de Chirurgie Générale et Digestive, Groupe Hospitalier et Faculté de Médecine Pitié-Salpêtrière, Institut du Cancer, Université Pierre et Marie Curie, Paris, France.
Abstract
BACKGROUND: Hypertrophic osteoarthropathy (HOA) is a rare condition characterized by bone and joint pain and digital clubbing usually associated with bronchopulmonary diseases. Primary HOA is rare and the pathogenesis remains unclear. OBJECTIVES: Cases of HOA as a paraneoplastic syndrome associated with thyroid carcinoma are very rare - only 2 cases have been described in the literature. RESULTS: We present the first case of a 40-year-old patient affected by HOA associated with invasive differentiated follicular thyroid carcinoma operated in 2 stages. Both operations were followed by radioiodine ablation, and then a rapid unresectable local recurrence developed requiring cervical radiotherapy (70 Gy). A second treatment with 100 mCi of (131)I confirmed it was a refractory thyroid cancer. Further surgery confirmed a poorly differentiated follicular cancer and 12 cycles of chemotherapy by gemcitabine and oxaliplatin followed. During the 8 years of follow-up, cervical recurrence was stable, but severe episodes of hemoptysis occurred requiring iterative embolization of the bronchial and tracheal arteries. Other lung diseases were excluded. Digital clubbing appeared, which was associated with arthritis, bone pain and inflammatory syndrome. X-rays and magnetic resonance imaging found periosteal apposition in the long bones; bone scintigraphy confirmed the HOA diagnosis. Other causes of arthritis were eliminated. She was treated with colchicine, corticosteroids and nonsteroidal anti-inflammatory drugs, but only the combination of methotrexate and hydroxychloroquine reduced the morphine requirements. CONCLUSION: HOA is exceptionally associated with thyroid cancer and we raised the hypothesis of the secretion of a circulating factor in a patient with invasive and recurrent follicular thyroid cancer, refractory to radioiodine.
BACKGROUND:Hypertrophic osteoarthropathy (HOA) is a rare condition characterized by bone and joint pain and digital clubbing usually associated with bronchopulmonary diseases. Primary HOA is rare and the pathogenesis remains unclear. OBJECTIVES: Cases of HOA as a paraneoplastic syndrome associated with thyroid carcinoma are very rare - only 2 cases have been described in the literature. RESULTS: We present the first case of a 40-year-old patient affected by HOA associated with invasive differentiated follicular thyroid carcinoma operated in 2 stages. Both operations were followed by radioiodine ablation, and then a rapid unresectable local recurrence developed requiring cervical radiotherapy (70 Gy). A second treatment with 100 mCi of (131)I confirmed it was a refractory thyroid cancer. Further surgery confirmed a poorly differentiated follicular cancer and 12 cycles of chemotherapy by gemcitabine and oxaliplatin followed. During the 8 years of follow-up, cervical recurrence was stable, but severe episodes of hemoptysis occurred requiring iterative embolization of the bronchial and tracheal arteries. Other lung diseases were excluded. Digital clubbing appeared, which was associated with arthritis, bone pain and inflammatory syndrome. X-rays and magnetic resonance imaging found periosteal apposition in the long bones; bone scintigraphy confirmed the HOA diagnosis. Other causes of arthritis were eliminated. She was treated with colchicine, corticosteroids and nonsteroidal anti-inflammatory drugs, but only the combination of methotrexate and hydroxychloroquine reduced the morphine requirements. CONCLUSION: HOA is exceptionally associated with thyroid cancer and we raised the hypothesis of the secretion of a circulating factor in a patient with invasive and recurrent follicular thyroid cancer, refractory to radioiodine.
Authors: Sandeep Uppal; Christine P Diggle; Ian M Carr; Colin W G Fishwick; Mushtaq Ahmed; Gamal H Ibrahim; Philip S Helliwell; Anna Latos-Bieleńska; Simon E V Phillips; Alexander F Markham; Christopher P Bennett; David T Bonthron Journal: Nat Genet Date: 2008-05-25 Impact factor: 38.330