Correspondence

Pamidronate for Bone Pain from Osteolytic Lesions in Langerhans'-Cell Histiocytosis

N Engl J Med 2001; 345:225July 19, 2001

Article

To the Editor:

A 23-year-old woman with Langerhans'-cell histiocytosis presented with severe pain in the right iliac crest and right shoulder of three months' duration. The patient had undergone excision and radiation of a skeletal lesion of the left hip at the age of 10; diabetes insipidus had developed when she was 15; and she had been treated with mercaptopurine and prednisone at the age of 19. Skeletal-survey radiographs showed osteolytic lesions in the frontal bone of the skull and the right femoral head. Sustained-release morphine sulfate (60 mg twice daily) was prescribed, and the patient had partial relief of the pain.

The presence of osteolytic lesions and severe bone pain prompted a trial of pamidronate. Immediately after the intravenous administration of the first dose of 90 mg, the patient's pain decreased from a level of 9 to a level of 6, as assessed on a 10-point visual-analogue scale. Treatment was continued on a monthly schedule, and after four infusions, the patient had resolution of pain to a level of 2 on the visual-analogue scale. The sustained-release morphine sulfate was gradually discontinued. Currently, the patient is free of bone pain and has stable osteolytic lesions while receiving pamidronate on a monthly schedule. She has now started chemotherapy with a combination of mercaptopurine, prednisone, and vinblastine because of Langerhans'-cell histiocytosis involving the gastrointestinal tract.

Pamidronate, a second-generation amino bisphosphonate, is a potent inhibitor of osteoclastic bone resorption.1 Experience with this medication in patients with multiple myeloma and those with metastatic breast cancer has shown it to be effective in reducing the extent of osteolytic lesions and the incidence of vertebral and long-bone fractures.2-4 In addition, it has been associated with relief of the pain associated with bone metastases and improvement in the quality of life in 50 percent of treated patients.5

In view of the remarkable relief of pain in our patient, we suggest a trial of pamidronate therapy for bone pain in patients with osteolytic lesions due to Langerhans'-cell histiocytosis.

Karo Arzoo, M.D.
Saeed Sadeghi, M.D.
Vinod Pullarkat, M.D.
Los Angeles County–University of Southern California Medical Center, Los Angeles, CA 90033
ssadeghi@hsc.usc.edu

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   Shanthi Sivendran, Harold Harvey, Allan Lipton, Joseph Drabick. (2011) Treatment of Langerhans cell histiocytosis bone lesions with zoledronic acid: a case series. International Journal of Hematology 93:6, 782-786
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   Milen Minkov. (2011) Multisystem Langerhans Cell Histiocytosis in Children. Pediatric Drugs 13:2, 75-86
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   Athanasios D. Anastasilakis, Gregory A. Kaltsas, Georgios Delimpasis, Ludwig Wilkens, George Kanakis, Polyzois Makras. (2011) Distinctive growth pattern in a patient with a delayed diagnosis of Langerhans’ cell histiocytosis. Pituitary
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   Akira Morimoto, Yoko Shioda, Toshihiko Imamura, Hirokazu Kanegane, Takashi Sato, Kazuko Kudo, Shinichiro Nakagawa, Hisaya Nakadate, Hisamichi Tauchi, Asahito Hama, Masahiro Yasui, Yoshihisa Nagatoshi, Akitoshi Kinoshita, Ryosuke Miyaji, Tadashi Anan, Miharu Yabe, Junji Kamizono. (2011) Nationwide survey of bisphosphonate therapy for children with reactivated Langerhans cell histiocytosis in Japan. Pediatric Blood & Cancer 56:1, 110-115
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   Liliana Montella, Carmela Merola, Geraldina Merola, Luigi Petillo, Giovannella Palmieri. (2009) Zoledronic acid in treatment of bone lesions by Langerhans cell histiocytosis. Journal of Bone and Mineral Metabolism 27:1, 110-113
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   Joan Manel Gasent Blesa, Vicente Alberola Candel, Carlos Solano Vercet, Juan Laforga Canales, Christof Semler, M. Rosa Pérez Antolí, Carlos Rodríguez-Galindo. (2008) Langerhans cell histiocytosis. Clinical and Translational Oncology 10:11, 688-696
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   Marcus Stockschlaeder, Christoph Sucker. (2006) Adult Langerhans cell histiocytosis. European Journal of Haematology 76:5, 363-368
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   DIEGO A. ROSSO, JENNY KARIS, JORGE L. BRAIER, JAN-INGE HENTER, BENGT FADEEL. (2006) Elevated Serum Levels of the Decoy Receptor Osteoprotegerin in Children with Langerhans Cell Histiocytosis. Pediatric Research 59:2, 281-286
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   Marco Pappagallo, Brenda Breuer, Aaron Schneider, Kevin Sperber. (2003) Treatment of chronic mechanical spinal pain with intravenous pamidronate: a review of medical records. Journal of Pain and Symptom Management 26:1, 678-683
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    Junji Kamizono, Yosuke Okada, Akira Shirahata, Yoshiya Tanaka. (2002) Bisphosphonate Induces Remission of Refractory Osteolysis in Langerhans Cell Histiocytosis. Journal of Bone and Mineral Research 17:11, 1926-1928
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    (2001) More on Pamidronate in Langerhans'-Cell Histiocytosis. New England Journal of Medicine 345:20, 1502-1503
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