Lymphocytic panhypophysitis in a young man with involvement of the cavernous sinus and clivus

Lymphocytic hypophysitis is an unusual inflammatory lesion that is caused by autoimmune destruction of the pituitary gland. We report a case of 42-year-old man who presented with a 6-month history of severe headache, blurred vision in the right eye, hearing loss, polyuria, polydipsia, and impotence. Medical history showed that he and his mother had osteopetrosis. The results of the physical examination and laboratory tests showed that secondary hypothyroidism, hypogonadism, and hypocortisolism had developed. Central diabetes insipidus was diagnosed by water deprivation test. MRI of the sella showed pituitary enlargement with symmetrical suprasellar expansion, compression of the chiasma, thickened infundibulum, and involvement of both bilateral cavernous sinuses and clivus. Hormonal substitution with hydrocortisone, levothyroxine, and DDAVP resulted in rapid improvement of all symptoms and signs. Transsphenoidal biopsy was diagnostic of lymphocytic hypophysitis. In spite of extensive literature reviewing, we have not been aware of any case of lymphocytic hypophysitis with clivus involvement. The present case represents a variant of lymphocytic hypophysitis which has progressed to involve bilateral cavernous sinuses and the clivus

Calcitriol treatment in patients with low vitamin D levels

The aim of the the study is to compare the effects of cholecalciferol and calcitriol on bone mineral metabolism in women with vitamin D deficiency. Calcitriol was associated with a significant increase in bone mineral density at the lumbar spine in patients with low vitamin D levels.Purpose/introductionActive vitamin D analogs may have larger impact in decreasing bone loss and fracture rate compared to cholecalciferol in osteoporosis. However, their effects in the treatment of vitamin D deficiency compared to cholecalciferol are not clear. The aim of the present study is to compare the effects of cholecalciferol and calcitriol on bone mineral metabolism and bone mineral density in pre- and postmenopausal women with vitamin D deficiency.MethodsThis was a 6-month prospective, open-label, controlled clinical trial. Eligible 120 participants were pre- and postmenopausal women diagnosed with vitamin D deficiency. Forty-three subjects (group 1) received 1000IU of cholecalciferol and 1g of calcium daily. The other 77 subjects (group 2) received 0.5g calcitriol in addition to 400IU of cholecalciferol and 1g of calcium daily.ResultsOral vitamin D supplementation did not increase bone mineral density after 6months of intervention in group 1. On the other hand, bone mineral density at the lumbar spine increased from 0.8090.172 to 0.848 +/- 0.161g/cm(2) in group 2 patients (p<0.017 vs baseline).ConclusionsOral daily calcitriol was associated with a significant increase in bone mineral density at the lumbar spine in patients with low vitamin D, elevated PTH, and osteoporosis

Vitamin D receptor alleles, bone mineral density and turnover in postmenopausal osteoporotic and healthy women

Objective: Vitamin D receptor (VDR) gene polymorphisms and bone metabolic markers were investigated as potential genetic markers for osteoporosis in postmenopausal Turkish women. The relationship between their VDR gene polymorphisms and bone states was determined. Materials and Methods: Restriction fragment length polymorphisms at the VDR gene locus (i.e., for BsmI, ApaI, and TaqI) was investigated in 75 postmenopausal osteoporotic (53.16+/-1.31 years) and 66 healthy (52.62+/-1.69 years) Turkish women and the genotypes were related to bone mineral density (BMD) at femoral neck (FN), lumbar spine (L1-4), trochanter, Ward's triangle (Ward's) and metabolic parameters of bone turnover. Results: In osteoporotic women, TaqI genotype-related differences of the VDR gene were found to be significant at all BMD sites; TT genotype had higher L1-4 BMD values than Tt and tt (p<0.05); tt genotype had significantly lower BMD at FN (p<0.05), trochanter (p<0.01), and Ward's (p<0.05) compared to TT genotype. The tt genotype was found to be associated with higher (p<0.05) serum osteocalcin levels compared to Tt and TT genotypes in the osteoporotic women, whereas no such association was found for the healthy women. Conclusion: Our data showed an association between VDR TaqI genotype and BMD at the FN, L1-4, trochanter and Ward's triangle in nonobese postmenopausal osteoporotic women. Thus the VDR gene TaqI polymorphism modulates differences in BMD in the postmenopausal osteoporotic women. Copyright (C) 2004 S. Karger AG, Basel

Propylthiouracil-Induced Thrombotic Leukocytoclastic Vasculitis

Antithyroid drug therapy with Propylthiouracil (PTU) is commonly used in Graves' disease. One of the most serious complications of this drug is PTU-induced leukocytoclastic vasculitis. We report a 23 year-old woman who developed leukocytoclastic vasculitis which is a rare and serious side effect during PTU therapy for Graves' disease. The observation of cutaneous vasculitis in this patient during administration of PTU indicates that reguler follow-up of patients on antithyroid drug regimen is mandatory

Lack of association between vitamin D receptor gene polymorphism (BsmI) and osteomalacia

Vitamin D receptor (VDR) gene polymorphism has been reported to be a determinant of bone formation and intestinal calcium absorption. We carried out this study to assess the role of VDR gene polymorphism in the pathogenesis of osteomalacia. We investigated BsmI polymorphisms in the gene encoding the 1,25 dihydroxyvitamin D receptor in 38 patients with osteomalacia and 31 healthy controls, along with examination of serum calcium, phosphorus, alkaline phosphatase, parathyroid hormone, and 25 hydroxyvitamin D levels. VDR allelic variants were: BB, 31.6%; Bb, 44.7%; and bb, 23.7% in the osteomalacia patients and BB, 19.4%; Bb, 61.3%; and bb, 19.4% in the controls. Although heterozygotes (Bb) were more frequent than other genotypes in both groups, the BB genotype was found to be more prevalent in osteomalacia than in controls. There was no statistical relationship between VDR genotype and osteomalacia. It is concluded that, in this small group of patients, there was no relationship between VDR allelic polymorphisms and osteomalacia

A case of late presented with autoimmune polyglandular syndrome type 1

Autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy is also known as autoimmune polyglandular syndrome type I (APS-I). We report a case of late presented APS-I with non-endocrine manifestations. A 41-year-old male presented with chronic mucocutaneous candidiasis as the first manifestation of the disease, followed by adrenal insufficiency. Other associated diseases that developed in time included gonadal insufficiency, alopecia, vitiligo, chronic urticaria. chronic active hepatitis and malabsorption. Although our patient presented late with APS-I and non-endocrine manifestations, APS-I usually occurs in childhood and is an autosomal recessive disorder characterized by a combination of autoimmune manifestations affecting endocrine and non-endocrine organs. In conclusion, patients with autoimmune endocrine disease should be followed on a regular basis