Literature DB >> 33558959

Bone Status According to Neurofibromatosis Type 1 Phenotype: A Descriptive Study of 60 Women in France.

Maud Jalabert1, Salah Ferkal2, Jean-Claude Souberbielle3, Emilie Sbidian4, Arthur Mageau5, Florent Eymard6, Philippe Le Corvoisier2, Laurence Allanore4, Xavier Chevalier6, Pierre Wolkenstein4, Sandra Guignard6.   

Abstract

There is an increased risk of osteoporosis and an abnormal bone turn over in neurofibromatosis 1 (NF1). Our objective is to evaluate bone status in NF1 and to look for associations with cutaneous phenotype. We conducted a descriptive, monocentric study. We included 60 NF1 women, 18-51 years old, non-menopausal, divided in 2 groups: «at risk phenotype» (ARP) composed by 30 patients with at least 2 subcutaneous neurofibromas (SC-NF) and «classical phenotype» (CP) composed by 30 patients with none or 1 SC-NF. We evaluated low bone mineral density (BMD) risk factors and measured BMD, calcium and phosphorus homeostasis and bone turnover markers. Before 50 years old, Z-score has to be used to assess BMD. Z-score < - 2 is below expected range and represents 2.5% of the population. There was no difference between the two groups. Overall, Z-scores were low and 5 patients had a Z-score < - 2 (8.3%), which is 3 times general population low BMD frequency. 10 fragility fractures occurred in 8 patients, among which 2 were vertebral fractures. 85% had low calcium intake. 12 patients had hypophosphoremia, 25 elevated PTH. Vitamin D levels were low for 86.4%. 41 patients (69.5%) had at least one abnormal bone turnover markers. Low BMD is 3.3 times more frequent in NF1 than in general population, with high fracture risk, regardless of the skin phenotype, classical or at risk, because of high bone turn over and secondary hyperparathyroidism due to vitamin D deficiency and poor calcium intake.

Entities:  

Keywords:  Bone turnover; Fracture; Neurofibromatosis; Osteoporosis; Von Recklinghausen disease

Year:  2021        PMID: 33558959     DOI: 10.1007/s00223-021-00807-6

Source DB:  PubMed          Journal:  Calcif Tissue Int        ISSN: 0171-967X            Impact factor:   4.333


  30 in total

1.  Incidence and mortality of neurofibromatosis: a total population study in Finland.

Authors:  Elina Uusitalo; Jussi Leppävirta; Anna Koffert; Sakari Suominen; Jussi Vahtera; Tero Vahlberg; Minna Pöyhönen; Juha Peltonen; Sirkku Peltonen
Journal:  J Invest Dermatol       Date:  2014-10-29       Impact factor: 8.551

Review 2.  [Neurofibromatosis type 1 or Von Recklinghausen's disease].

Authors:  S Pinson; P Wolkenstein
Journal:  Rev Med Interne       Date:  2005-03       Impact factor: 0.728

3.  Bone mineral density in children and adolescents with neurofibromatosis type 1.

Authors:  David A Stevenson; Laurie J Moyer-Mileur; Mary Murray; Hillarie Slater; Xiaoming Sheng; John C Carey; Bukhosi Dube; David H Viskochil
Journal:  J Pediatr       Date:  2007-01       Impact factor: 4.406

4.  Clinical risk factors for mortality in patients with neurofibromatosis 1: a cohort study of 378 patients.

Authors:  Kiarash Khosrotehrani; Sylvie Bastuji-Garin; Jacques Zeller; Jean Revuz; Pierre Wolkenstein
Journal:  Arch Dermatol       Date:  2003-02

5.  Bone mineral density and bone metabolic markers' status in children with neurofibromatosis type 1.

Authors:  Hatice Gamze Poyrazoğlu; Veysel Nijat Baş; Alev Arslan; Funda Bastug; Mehmet Canpolat; Hüseyin Per; Hakan Gümüs; Sefer Kumandas
Journal:  J Pediatr Endocrinol Metab       Date:  2017-02-01       Impact factor: 1.634

6.  A controlled register-based study of 460 neurofibromatosis 1 patients: increased fracture risk in children and adults over 41 years of age.

Authors:  Eetu Heervä; Anna Koffert; Elina Jokinen; Tommi Kuorilehto; Sirkku Peltonen; Hannu T Aro; Juha Peltonen
Journal:  J Bone Miner Res       Date:  2012-11       Impact factor: 6.741

Review 7.  Neurofibromatosis type 1: a multidisciplinary approach to care.

Authors:  Angela C Hirbe; David H Gutmann
Journal:  Lancet Neurol       Date:  2014-08       Impact factor: 44.182

8.  Decreased bone mineral density in neurofibromatosis type 1: results from a pediatric cohort.

Authors:  Sukhdeep Dulai; Julie Briody; Aaron Schindeler; Kathryn N North; Christopher T Cowell; David G Little
Journal:  J Pediatr Orthop       Date:  2007-06       Impact factor: 2.324

9.  Bone health and fracture rate in individuals with neurofibromatosis 1 (NF1).

Authors:  T Tucker; C Schnabel; M Hartmann; R E Friedrich; I Frieling; H-P Kruse; V-F Mautner; J M Friedman
Journal:  J Med Genet       Date:  2008-12-09       Impact factor: 6.318

10.  Malignant peripheral nerve sheath tumors: clinicopathological aspects, expression of p53 and survival.

Authors:  Karin S G Cunha; Anabela C Caruso; Paulo A S de Faria; Licínio E da Silva; Andréa R C Pires; Mauro Geller; Vânia S Lopes; Rodrigo S de Moura-Neto
Journal:  Clinics (Sao Paulo)       Date:  2012-08       Impact factor: 2.365

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  2 in total

1.  Hypophosphatemic osteomalacia in neurofibromatosis 1 associated with intracranial gliomas and congenital renal agenesis: A rare case report and review of the literature.

Authors:  Angelos Kaspiris; Elias Vasiliadis; Dimitra Melissaridou; Ilias D Iliopoulos; Panayiotis J Papagelopoulos; Olga D Savvidou
Journal:  J Orthop Case Rep       Date:  2022-02

Review 2.  Current Aspects on the Pathophysiology of Bone Metabolic Defects during Progression of Scoliosis in Neurofibromatosis Type 1.

Authors:  Angelos Kaspiris; Olga D Savvidou; Elias S Vasiliadis; Argyris C Hadjimichael; Dimitra Melissaridou; Stella Iliopoulou-Kosmadaki; Ilias D Iliopoulos; Evangelia Papadimitriou; Efstathios Chronopoulos
Journal:  J Clin Med       Date:  2022-01-15       Impact factor: 4.241

  2 in total

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