Hypercalcemia in Langerhans' cell granulomatosis with elevated 1,25 dihydroxyvitamin D (calcitriol) level

H. Al-Ali, A. Abou Yabis, E. Issa, Z. Salem, A. Tawil, N. Khoury, G. H.El Hajj Fuleihan

Research output: Contribution to journalArticle

18 Scopus citations

Abstract

A 48-year-old woman presented with a history of premature menopause, polyuria, polydipsia, fever, and diffuse bony tenderness. Her evaluation revealed central diabetes insipidus, hypothalamic amenorrhea, an elevated free calcium on multiple occasions with an elevated 1,25 dihydroxyvitamin D level, and osteoporosis by densitometry. Skeletal series revealed multiple lytic lesions involving the long bones. The diagnosis of Langerhans' cell granulomatosis was made. She was treated with hormone replacement therapy, radiotherapy, and vinblastine, with a dramatic improvement in her pain and a near normalization of her free calcium. Whereas hypercalcemia has been described in several granulomatous disorders and is secondary to unregulated extrarenal production of 1,25 dihydroxyvitamin D, it is, however, extremely rare in Langerhans' cell granulomatosis. This is the first case report of Langerhans' cell granulomatosis with hypercalcemia and documented elevated increased 1,25 dihydroxyvitamin D level that responded to the treatment of her primary disease.

Original languageEnglish (US)
Pages (from-to)331-334
Number of pages4
JournalBone
Volume30
Issue number1
DOIs
StatePublished - 2002

Keywords

  • 1,25(OH)D
  • Hypercalcemia
  • Langerhans'
  • cell granulomatosis

ASJC Scopus subject areas

  • Endocrinology, Diabetes and Metabolism
  • Physiology
  • Histology

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    Al-Ali, H., Yabis, A. A., Issa, E., Salem, Z., Tawil, A., Khoury, N., & Fuleihan, G. H. E. H. (2002). Hypercalcemia in Langerhans' cell granulomatosis with elevated 1,25 dihydroxyvitamin D (calcitriol) level. Bone, 30(1), 331-334. https://doi.org/10.1016/S8756-3282(01)00651-2