Exp Clin Endocrinol Diabetes 2001; 109(6): 350-354
DOI: 10.1055/s-2001-17398
Case Report

© Johann Ambrosius Barth

Rapid increase in bone mineral density in a child with osteoporosis and autoimmune hypoparathyroidism treated with PTH 1-34[*]

C. A. Koch
  • Pediatric and Reproductive Endocrinology Branch, National Institute of Child Health and Human Development; National Institutes of Health, Bethesda, Maryland, U.S.A.
Further Information

Publication History

Publication Date:
19 September 2001 (online)

Summary:

We describe a 16-year-old girl with autoimmune polyglandular syndrome type 1 including hypoparathyroidism, who had osteoporosis that improved rapidly with parathyroid hormone replacement therapy. Patients with hypoparathyroidism usually have high bone mass. Our patient developed vertebral compression fractures at age 10, shortly after hypoparathyroidism was diagnosed. She continued to have low lumbar bone mass until age 16, when a dual energy x-ray absorptiometry (DEXA) revealed a Z score of - 2.2 SD. Several factors including decreased physical activity, total body magnesium depletion, and intermittent ketoconazole and short-term prednisone treatment, may have contributed to the development and progression of osteoporosis. Therapy with synthetic human parathyroid hormone (PTH) 1-34 rapidly normalized lumbar bone mass, as assessed by DEXA.

1 * Presented as poster at the 81st Meeting of the Endocrine Society in San Diego, June 13 1999 (abstract p2-686)

References

  • 1 Abugassa S, Nordenstrom J, Eriksson S, Sjoden G. Bone mineral density in patients with chronic hypoparathyroidism.  J Clin Endocrinol Metab. 6 1617-1621 1993; 
  • 2 Ahonen P, Myllarniemi S, Sipila I, Perheentupa J. Clinical variation of autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED) in a series of 68 patients.  N Engl J Med. 322 1829-1836 1990; 
  • 3 Albright F. Hypoparathyroidism as a cause of osteomalacia.  J Clin Endocrinol Metab. 16 419 1956; 
  • 4 Betterle C, Greggio N A, Volpato M. Autoimmune polyglandular syndrome type 1.  J Clin Endocrinol Metab. 83 1049-1055 1998; 
  • 5 Dempster D W, Cosman F, Parisien M, Shen V, Lindsay R. Anabolic actions of PTH on bone.  Endocr Rev. 14 690-709 1993; 
  • 6 Dempster D W, Cosman F, Parisien M, Shen V, Lindsay R. Anabolic actions of parathyroid hormone on bone. Update 1995.  Endocr Rev. 4 247-250 1995; 
  • 7 Dimich A, Bedrossian P R, Wallach S. Hypoparathyroidism: Clinical observations in 34 patients.  Arch Intern Med. 120 449-458 1967; 
  • 8 Duan Y, de Luca V, Seeman E. Parathyroid hormone deficiency and excess: Similar effects on trabecular bone but differing effects on cortical bone.  J Clin Endocrinol Metab. 84 718-722 1999; 
  • 9 Drezner M K, Neelon F A, Jowsey J, Lebovitz H E. Hypoparathyroidism: A possible cause of osteomalacia.  J Clin Endocrinol Metab. 45 114 1977; 
  • 10 Emersson K, Walsh F B, Howard J E. Idiopathic hypoparathyroidism: A report of two cases.  Ann Intern Med. 14 1256 1941; 
  • 11 Feldkamp J, Becker A, Witte O W, Scharff D, Scherbaum W A. Long-term anticonvulsant therapy leads to low bone mineral density - evidence for direct drug effects of phenytoin and carbamazepine on human osteoblast-like cells.  Exp Clin Endocrinol Diabetes. 108 37-43 2000; 
  • 12 Fujiyama K, Kiriyama T, Ito M. Attenuation of postmenopausal high turnover bone loss in patients with hypoparathyroidism.  J Clin Endocrinol Metabol. 80 2135-2138 1995; 
  • 13 Glass A R, Eil C. Ketoconazole-induced reduction in serum 1,25-dihydroxy-vitamin D and total serum calcium in hypercalcemic patients.  J Clin Endocrinol Metab. 66 934-938 1988; 
  • 14 Hahn T J, Hendin B A, Scharp C R, Haddad J G. Effect of chronic anticonvulsant therapy on serum 25-hydroxycalciferol levels in adults.  N Engl J Med. 287 900-904 1972; 
  • 15 Harris M, Goldhaber P. Root abnormality in epileptics and the inhibition of parathyroid hormone-induced bone resoption by diphenylhydantoin in tissue culture.  Arch Oral Biol. 19 981 1974; 
  • 16 Koch C A, Heiss J D, Krakoff J, Pacak K, Winer K K, Wasserman E M. Chiari malformation type 1 and osteoporosis.  Neurosurg Rev. 23 171-172 2000; 
  • 17 Kurland E S, Cosman F, McMahon D J, Rosen C J, Lindsay R, Bilezikian J P. Parathyroid hormone as a therapy for idiopathic osteoporosis in men: Effects on bone mineral density and bone markers.  J Clin Endocrinol Metab. 85 3069-3076 2000; 
  • 18 Langdahl B L, Mortensen L, Vesterby A, Eriksen E F, Charles P. Bone histomorphometry in hypoparathyroid patients treated with vitamin D.  Bone. 18 103-108 1996; 
  • 19 Lindsay R, Nieves J, Formica C, Henneman E, Woelfert L, Shen V, Dempster D, Cosman F. Randomized controlled study of effect of parathyroid hormone on vertebral-bone mass and fracture incidence among postmenopausal women on oestrogen with osteoporosis.  Lancet. 350 550-555 1997; 
  • 20 Mithal A, Menon P SN, Ammini A C, Karmarkar M G, Ahuja M MS. Spontaneous hypoparathyroidism: Clinical, biochemical and radiological features.  Indian J Pediatr. 56 267-272 1989; 
  • 21 Oliveri M B, Cassinelli H, Bergada C, Mautalen C A. Bone mineral density of the spine and radius shaft in children with X-linked hypophosphatemic rickets (XLH).  Bone Miner. 12 91-100 1991; 
  • 22 Orr-Walker B, Harris R, Holdaway I M, Foote G, Reid I R. High peripheral and axial bone densities in a postmenopausal woman with untreated hypoparathyroidism.  Postgrad Med J. 66 1061-1063 1990; 
  • 23 Reid I R, Murphy W A, Hardy D C, Teitelbaum S L, Bergfeld M A, Whyte M P. X-linked hypophosphatemia: skeletal mass in adults assessed by histomorphometry, computed tomography, and absorptiometry.  Am J Med. 90 63-69 1991; 
  • 24 Rude R K, Kirchen M E, Gruber H E, Myer M H, Luck J S, Crawford D L. Magnesium deficiency-induced osteoporosis in the rat: Uncoupling of bone formation and bone resorption.  Magnes Res. 12 257-267 1999; 
  • 25 Rude R K, Kirchen M E, Gruber H E, Stasky A A, Meyer M H. Magnesium deficiency induces bone loss in the rat.  Miner Electrolye Metab. 24 314-320 1998; 
  • 26 Schulman J L, Ratner H. Idiopathic hypoparathyroidism with bone demineralization and cardiac decompensation.  Pediatrics. 16 848 1955; 
  • 27 Schutt-Aine J C, Young M A, Pescovitz O H, Chrousos G P, Marx S J. Hypoparathyroidism: A possible cause of rickets.  J Pediat. 106 255-259 1982; 
  • 28 Seeman E, Wahner H W, Offord K P, Kumar R, Johnson W J, Riggs B L. Differential effects of endocrine dysfunction on the axial and the appendicular skeleton.  J Clin Invest. 69 1302-1309 1982; 
  • 29 Selye H. On the stimulation of new bone formation with parathyroid extract and irradiated ergosterol.  Endocrinology. 16 547-558 1932; 
  • 30 Smith R. Idiopathic juvenile osteoporosis: Experience of twenty-one patients.  Br J Rheumatol. 34 68-77 1995; 
  • 31 Steinberg H, Waldron B R. Idiopathic hypoparathyroidism: An analysis of 52 cases including the report of a new case.  Medicine. 31 133-153 1952; 
  • 32 Touliatos J S, Sebes J I, Hinton A, McCommon D, Karas J G, Palmieri G MA. Hypoparathyroidism counteracts risk factors for osteoporosis.  Am J Med Sci. 310 56-60 1995; 
  • 33 Whitfield J F, Morley P, Ross V, Isaacs R J, Rixon R H. Restoration of severly depleted femoral trabecular bone in ovariectomized rats by parathyroid hormone (1-34).  Calcif Tissue Int. 56 227-231 1995; 
  • 34 Winer K K, Yanovski J A, Cutler G B. Synthetic human parathyroid hormone 1-34 vs calcitriol and calcium in the treatment of hypoparathyroidism.  JAMA. 276 631-636 1996; 
  • 35 Winer K K, Yanovski J A, Sarani B, Cutler G B. A randomized, cross-over trial of once-daily versus twice-daily parathyroid hormone 1-34 for treatment of hypoparathyroidism.  J Clin Endocrinol Metab. 83(10) 3480-3486 1998; 

1 * Presented as poster at the 81st Meeting of the Endocrine Society in San Diego, June 13 1999 (abstract p2-686)

Dr. Christian A. Koch

F.A.C.E., C.N.S.

National Institutes of Health

NICHD, PREB

Building 10, Rm 9D42

Bethesda, MD 20892

U.S.A.

Phone: +301 402 4620

Fax: +301 402 0180

Email: Kochc@exchange.nih.gov

    >