Literature DB >> 12807509

Increased fracture rates in Turner's syndrome: a nationwide questionnaire survey.

Claus Højbjerg Gravholt1, Peter Vestergaard, Anne Pernille Hermann, Leif Mosekilde, Kim Brixen, Jens Sandahl Christiansen.   

Abstract

BACKGROUND AND OBJECTIVES: Reduced bone mineral content (BMC) and bone mineral density (BMD) have previously been reported in Turner's syndrome, although appropriate GH treatment and early induction of puberty seem to permit normal bone mass accumulation. Furthermore, an increased risk of fractures and osteoporosis have been reported in a registry study. The aim of the present study was to further characterize the risk of fractures in TS and to explore risk factors, in a historical follow-up survey based on a self-administered questionnaire. STUDY GROUPS: The questionnaire was issued to all females with TS (n = 632) in Denmark and to 1888 randomly selected controls (C) matched for age and geographical region. A total of 322 patients (51%) and 1169 controls (62%) returned the questionnaire.
RESULTS: TS women were younger than C (30 years, range: 1-73 years vs. 34 years, range 2-82 years, P < 0.0005), smoked less often (17%vs. 27%, P < 0.0005), and had less frequent spontaneous menstruation (18%vs. 86%, P < 0.0005). In contrast, they used hormonal replacement therapy (HRT) more often (71%vs. 7%, P < 0.0005). The median age at start of HRT was 16 years (range 8-59 years) in TS vs. 42 years (range 12-53 years) in C (P < 0.0005). Above the age of 15 years, 83% of TS and 8% of C used HRT. GH had been used by 37% of TS but only 0.2% of C. Both type 1 and 2 diabetes were increased sevenfold among TS. Altogether, 77 individuals with TS had 109 fractures. The fracture risk was increased in TS [hazard ratio (HR, status) 1.35, confidence interval (CI) 1.04-1.75, P = 0.025]. Time to first fracture was reduced in TS (53 +/- 2 vs. 63 +/- 1; log-rank P = 0.03). Spontaneous menstruation was protective in females above 13 years of age (HR: 0.70, CI 0.54-0.93, P = 0.012). A history of parental fractures increased the risk (HR 1.92, CI 1.62-2.27, P < 0.001). Fractures of the forearm was more frequent among TS (P = 0.02).
CONCLUSION: The present nationwide survey, based on questionnaires, confirms an increased risk of early fractures in TS, especially in those without ovarian function and with a positive family history of fracture and osteoporosis. It thereby emphasizes the need for being vigilant with respect to BMD measurements in these patients.

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Year:  2003        PMID: 12807509     DOI: 10.1046/j.1365-2265.2003.01807.x

Source DB:  PubMed          Journal:  Clin Endocrinol (Oxf)        ISSN: 0300-0664            Impact factor:   3.478


  14 in total

Review 1.  Sex hormone replacement in Turner syndrome.

Authors:  Christian Trolle; Britta Hjerrild; Line Cleemann; Kristian H Mortensen; Claus H Gravholt
Journal:  Endocrine       Date:  2011-12-07       Impact factor: 3.633

Review 2.  Effects of hypogonadism on bone metabolism in female adolescents and young adults.

Authors:  Madhusmita Misra
Journal:  Nat Rev Endocrinol       Date:  2012-01-24       Impact factor: 43.330

3.  Reduced cortical bone density with normal trabecular bone density in girls with Turner syndrome.

Authors:  C R Holroyd; J H Davies; P Taylor; K Jameson; C Rivett; C Cooper; E M Dennison
Journal:  Osteoporos Int       Date:  2010-02-05       Impact factor: 4.507

4.  Artificially low cortical bone mineral density in Turner syndrome is due to the partial volume effect.

Authors:  O Soucek; E Schönau; J Lebl; Z Sumnik
Journal:  Osteoporos Int       Date:  2014-10-07       Impact factor: 4.507

Review 5.  Effect of estrogen replacement therapy on bone and cardiovascular outcomes in women with turner syndrome: a systematic review and meta-analysis.

Authors:  Dahima Cintron; Rene Rodriguez-Gutierrez; Valentina Serrano; Paula Latortue-Albino; Patricia J Erwin; Mohammad Hassan Murad
Journal:  Endocrine       Date:  2016-07-29       Impact factor: 3.633

6.  Bone size and density measurements in prepubertal children with Turner syndrome prior to growth hormone therapy.

Authors:  P Pitukcheewanont; N Numbenjapon; D Safani; S Rossmiller; V Gilsanz; G Costin
Journal:  Osteoporos Int       Date:  2010-09-09       Impact factor: 4.507

7.  Turner syndrome: French National Diagnosis and Care Protocol (NDCP; National Diagnosis and Care Protocol).

Authors:  Elodie Fiot; Bertille Alauze; Bruno Donadille; Dinane Samara-Boustani; Muriel Houang; Gianpaolo De Filippo; Anne Bachelot; Clemence Delcour; Constance Beyler; Emilie Bois; Emmanuelle Bourrat; Emmanuel Bui Quoc; Nathalie Bourcigaux; Catherine Chaussain; Ariel Cohen; Martine Cohen-Solal; Sabrina Da Costa; Claire Dossier; Stephane Ederhy; Monique Elmaleh; Laurence Iserin; Hélène Lengliné; Armelle Poujol-Robert; Dominique Roulot; Jerome Viala; Frederique Albarel; Elise Bismuth; Valérie Bernard; Claire Bouvattier; Aude Brac; Patricia Bretones; Nathalie Chabbert-Buffet; Philippe Chanson; Regis Coutant; Marguerite de Warren; Béatrice Demaret; Lise Duranteau; Florence Eustache; Lydie Gautheret; Georges Gelwane; Claire Gourbesville; Mickaël Grynberg; Karinne Gueniche; Carina Jorgensen; Veronique Kerlan; Charlotte Lebrun; Christine Lefevre; Françoise Lorenzini; Sylvie Manouvrier; Catherine Pienkowski; Rachel Reynaud; Yves Reznik; Jean-Pierre Siffroi; Anne-Claude Tabet; Maithé Tauber; Vanessa Vautier; Igor Tauveron; Sebastien Wambre; Delphine Zenaty; Irène Netchine; Michel Polak; Philippe Touraine; Jean-Claude Carel; Sophie Christin-Maitre; Juliane Léger
Journal:  Orphanet J Rare Dis       Date:  2022-07-12       Impact factor: 4.303

8.  Official positions of the International Society for Clinical Densitometry (ISCD) on DXA evaluation in children and adolescents.

Authors:  Maria Luisa Bianchi; Sanford Baim; Nick J Bishop; Catherine M Gordon; Didier B Hans; Craig B Langman; Mary B Leonard; Heidi J Kalkwarf
Journal:  Pediatr Nephrol       Date:  2009-07-15       Impact factor: 3.714

Review 9.  Fracture risk and bone mineral density in Turner syndrome.

Authors:  Vladimir K Bakalov; Carolyn A Bondy
Journal:  Rev Endocr Metab Disord       Date:  2008-04-15       Impact factor: 6.514

Review 10.  Type 1 diabetes and osteoporosis: A review of literature.

Authors:  Pooja Dhaon; Viral N Shah
Journal:  Indian J Endocrinol Metab       Date:  2014-03
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