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 El-Hajj Fuleihan

doi:10.1016/S8756-3282(01)00651-2

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

Journal article

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Peer reviewed

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 and G El-Hajj Fuleihan

Bone (New York, N.Y.), Vol.30(1), pp.331-334

01/2002

DOI: 10.1016/S8756-3282(01)00651-2

PMID: 11792606

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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.

Antineoplastic Agents, Phytogenic - therapeutic use

Calcitriol - blood

Calcium - blood

Deamino Arginine Vasopressin - therapeutic use

Diabetes Insipidus - complications

Diabetes Insipidus - drug therapy

Estrogen Replacement Therapy

Female

Histiocytosis, Langerhans-Cell - complications

Histiocytosis, Langerhans-Cell - diagnosis

Histiocytosis, Langerhans-Cell - therapy

Humans

Hypercalcemia - complications

Middle Aged

Osteoporosis - complications

Vinblastine - therapeutic use

Details

Title: Subtitle: Hypercalcemia in Langerhans' cell granulomatosis with elevated 1,25 dihydroxyvitamin D (calcitriol) level
Creators: H Al-Ali - American University of Beirut Medical Center
A Abou Yabis
E Issa
Z Salem
A Tawil
N Khoury
G El-Hajj Fuleihan
Resource Type: Journal article
Publication Details: Bone (New York, N.Y.), Vol.30(1), pp.331-334
DOI: 10.1016/S8756-3282(01)00651-2
PMID: 11792606
NLM abbreviation: Bone
ISSN: 8756-3282
eISSN: 1873-2763
Language: English
Date published: 01/2002
Academic Unit: Radiology
Record Identifier: 9984697735402771

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