Osteoporosis in Multiple Endocrine Neoplasia Type 1

Burgess, John; David, Ruben; Greenaway, TM; Parameswaran, V.; Shepherd, J. J.

doi:10.1001/archsurg.134.10.1119

Cited by 102 publications

(68 citation statements)

References 23 publications

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“…This specific pattern of bone demineralization at the three bone sites in HPT/MEN1, emphasizing the absence of protection of trabecular bone could be, in part, due to hypogonadism secondary to the combined occurrence of prolactinoma/MEN1 in some cases; early onset of HPT interfering with the normal peak of bone mass; environmental factors, such as vitamin D deficiency; direct action of MENIN deficiency in the development of bone tissue; or associated factors and others (31,47). Environmental factors such as vitamin D deficiency could be excluded in our cases.…”

Section: Hpt and Bmdmentioning

confidence: 98%

“…In familial forms of HPT, such as MEN1, BMD has rarely been reported. Burgess addressed this topic by studying T-and Z-scores in two bone sites, the lumbar spine and the FN, in a group of 20 uncontrolled and nine controlled HPT/MEN1 cases from Tasman 1 family (31).…”

Section: Hpt and Bmdmentioning

confidence: 99%

“…Furthermore, 12 founding MEN1 mutations were reported in MEN1 kindreds from Europe (Finland, France and Sweden), Oceania (Australia), and North America (Canada and USA) (15,(30)(31)(32)(33)(34)(35). Notably, all five previously reported very large MEN1 genealogies were associated with founder chromosomes (12,13,25,26).…”

Section: Introductionmentioning

confidence: 99%

“…Recently, Burgess et al (1999) evaluated bone mineral density (BMD) in MEN1 by analyzing two bone sites: the lumbar spine and femoral neck (FN) (31). The proximal one-third of the distal radius (DR), however, which is enriched in compact bone and is preferentially affected in sporadic HPT, has not been studied so far in MEN1.…”

Section: Introductionmentioning

confidence: 99%

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Multiple endocrine neoplasia type 1 in Brazil: MEN1 founding mutation, clinical features, and bone mineral density profile

Lourenço¹,

Toledo²,

Mackowiak³

et al. 2008

European Journal of Endocrinology

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Objective: Only few large families with multiple endocrine neoplasia type 1 (MEN1) have been documented. Here, we aimed to investigate the clinical features of a seven-generation Brazilian pedigree, which included 715 at-risk family members. Design: Genealogical and geographic analysis was used to identify the MEN1 pedigree. Clinical and genetic approach was applied to characterize the phenotypic and genotypic features of the family members. Results: Our genetic data indicated that a founding mutation in the MEN1 gene has occurred in this extended Brazilian family. Fifty family members were diagnosed with MEN1. Very high frequencies of functioning and non-functioning MEN1-related tumors were documented and the prevalence of prolactinoma (29.6%) was similar to that previously described in prolactinoma-variant Burin (32%). In addition, bone mineral density analysis revealed severe osteoporosis (T, K2.87G0.32) of compact bone (distal radius) in hyperparathyroidism (HPT)/MEN1 patients, while marked bone mineral loss in the lumbar spine (T, K1.95G0.39), with most cancellous bone, and femoral neck (mixed composition; T, K1.48G0.27) were also present. Conclusions: In this study, we described clinically and genetically the fifth largest MEN1 family in the literature. Our data confirm previous findings suggesting that prevalence of MEN1-related tumors in large families may differ from reports combining cumulative data of small families. Furthermore, we were able to evaluate the bone status in HPT/MEN1 cases, a subject that has been incompletely approached in the literature. We discussed the bone loss pattern found in our MEN1 patients comparing with that of patients with sporadic primary HPT.European Journal of Endocrinology 159 259-274

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Section: Hpt and Bmdmentioning

confidence: 98%

Section: Hpt and Bmdmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Multiple endocrine neoplasia type 1 in Brazil: MEN1 founding mutation, clinical features, and bone mineral density profile

Lourenço¹,

Toledo²,

Mackowiak³

et al. 2008

European Journal of Endocrinology

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“…HPT is usually the first clinical expression of MEN1, with a typical age of onset of 20 -25 yr (1-5); this is 30 yr earlier than that from sporadic parathyroid adenoma. Bone mass in women with MEN1 and HPT is already low at age 35 yr, presumably due to early and thus long-standing HPT (18). MEN1 often causes simultaneous HPT and ZES.…”

Section: Parathyroid Tumors In Men1mentioning

confidence: 99%

CONSENSUS: Guidelines for Diagnosis and Therapy of MEN Type 1 and Type 2

Brandi¹,

Gagel²,

Angeli³

et al. 2001

The Journal of Clinical Endocrinology & Metabolism

1,455

445

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Parathyroidectomy Can Improve Bone Mineral Density in Patients With Symptomatic Secondary Hyperparathyroidism

Chou¹

2001

Arch Surg

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Hypothesis:The recovery of osteoporosis or bone mineral density (BMD) after parathyroidectomy and autotransplantation can be improved in patients with symptomatic secondary hyperparathyroidism.Design: Case series.Setting: Tertiary referral center.Patients: Forty-five patients with symptomatic secondary hyperparathyroidism who underwent total parathyroidectomy and autotransplantation were included. They were divided into an osteoporotic group (n = 20) and a nonosteoporotic group (n = 25) according to preoperative T scores less than −2.5 at either the lumbar spine (L1-L4) or the femoral neck (FN).Interventions: Serum levels of calcium, phosphorus, alkaline phosphatase, and intact parathyroid hormone were checked before surgery and 1 day, 1 week, and 3 months after surgery. The BMDs of the FN and L1-L4 were measured using dual-energy x-ray absorptiometry before surgery and 6 months after surgery.Results: Patients with osteoporosis were older (mean±SD, 50.2 ± 14.0 years) than those without osteo-

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Osteoporosis in Multiple Endocrine Neoplasia Type 1

Cited by 102 publications

References 23 publications

Multiple endocrine neoplasia type 1 in Brazil: MEN1 founding mutation, clinical features, and bone mineral density profile

Multiple endocrine neoplasia type 1 in Brazil: MEN1 founding mutation, clinical features, and bone mineral density profile

CONSENSUS: Guidelines for Diagnosis and Therapy of MEN Type 1 and Type 2

Parathyroidectomy Can Improve Bone Mineral Density in Patients With Symptomatic Secondary Hyperparathyroidism

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