Literature DB >> 17942587

Rare diseases in disabled children: an epidemiological survey.

P Guillem1, C Cans, E Robert-Gnansia, S Aymé, P S Jouk.   

Abstract

AIM: To estimate the contribution of rare diseases (RD) to severe impairment in 7-year-old children.
METHODS: Data from a morbidity register of childhood impairments in a single French region were used. Impairments were classified as a mental, sensorial, neuromuscular (skeletal or movement-related) impairment (MSN_I) according to the International Classification of Functioning. Details of children born from 1980 to 1994 and resident in the county under study when they were 7 years old were recorded. A rare disease was defined as a prevalence rate of <1 per 2000 general population.
RESULTS: 26% of children with severe MSN_I had a rare disease; in 36% the MSN_I was of unknown origin. The proportion of impairments that were due to a rare disease varied according to the type of impairment: 3.3% for severe psychiatric disorders; 16.0% for intellectual impairment; 37.2% for hearing impairment; 41.2% for neuromuscular, skeletal and movement impairment; and 81.1% for visual impairment. The overall prevalence rate of rare diseases was 2.1 per 1000 (459/218 283), and it increased significantly over time (p = 0.003). The latter increase was not associated with a decrease in the proportion of impairments of unknown origin, indicating an improvement in the survival of the children with a rare disease.
CONCLUSIONS: In this study, a rare disease was at the origin of 26% of cases of severe MSN_I. This proportion remained stable over time, whereas the prevalence rate, as well as the prevalence rate of MSN_I disability, increased over time.

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Year:  2007        PMID: 17942587     DOI: 10.1136/adc.2006.104455

Source DB:  PubMed          Journal:  Arch Dis Child        ISSN: 0003-9888            Impact factor:   3.791


  10 in total

1.  Factors Associated with Parental Adaptation to Children with an Undiagnosed Medical Condition.

Authors:  Tatiane Yanes; Linda Humphreys; Aideen McInerney-Leo; Barbara Biesecker
Journal:  J Genet Couns       Date:  2016-12-30       Impact factor: 2.537

2.  Identification of alkaptonuria in the general population: a United Kingdom experience describing the challenges, possible solutions and persistent barriers.

Authors:  L Ranganath; A M Taylor; A Shenkin; W D Fraser; J Jarvis; J A Gallagher; N Sireau
Journal:  J Inherit Metab Dis       Date:  2011-02-11       Impact factor: 4.982

3.  Awakening Australia to Rare Diseases: symposium report and preliminary outcomes.

Authors:  Hugh J S Dawkins; Caron M Molster; Leanne M Youngs; Peter C O'Leary
Journal:  Orphanet J Rare Dis       Date:  2011-08-18       Impact factor: 4.123

Review 4.  Review: Understanding Rare Genetic Diseases in Low Resource Regions Like Jammu and Kashmir - India.

Authors:  Arshia Angural; Akshi Spolia; Ankit Mahajan; Vijeshwar Verma; Ankush Sharma; Parvinder Kumar; Manoj Kumar Dhar; Kamal Kishore Pandita; Ekta Rai; Swarkar Sharma
Journal:  Front Genet       Date:  2020-04-30       Impact factor: 4.599

5.  Results of a Patient Reported Experience Measure (PREM) to measure the rare disease patients and caregivers experience: a Spanish cross-sectional study.

Authors:  Mercedes Guilabert; Alba Martínez-García; Marina Sala-González; Olga Solas; José Joaquín Mira
Journal:  Orphanet J Rare Dis       Date:  2021-02-05       Impact factor: 4.123

6.  Enhanced rare disease mapping for phenome-wide genetic association in the UK Biobank.

Authors:  Matthew T Patrick; Redina Bardhi; Wei Zhou; James T Elder; Johann E Gudjonsson; Lam C Tsoi
Journal:  Genome Med       Date:  2022-08-09       Impact factor: 15.266

7.  A population-based registry as a source of health indicators for rare diseases: the ten-year experience of the Veneto Region's rare diseases registry.

Authors:  Monica Mazzucato; Laura Visonà Dalla Pozza; Silvia Manea; Cinzia Minichiello; Paola Facchin
Journal:  Orphanet J Rare Dis       Date:  2014-03-19       Impact factor: 4.123

8.  Difficulties in the diagnosis and treatment of rare diseases according to the perceptions of patients, relatives and health care professionals.

Authors:  Marcos Thomazin Lopes; Vera Hermina Koch; Vicente Sarrubbi-Junior; Paulo Rogério Gallo; Magda Carneiro-Sampaio
Journal:  Clinics (Sao Paulo)       Date:  2018-04-05       Impact factor: 2.365

9.  A patient survey on the impact of alkaptonuria symptoms as perceived by the patients and their experiences of receiving diagnosis and care.

Authors:  Mattias Rudebeck; Ciarán Scott; Nicolas Sireau; Lakshminarayan Ranganath
Journal:  JIMD Rep       Date:  2020-03-07

Review 10.  Diagnosis of Rare Diseases: a scoping review of clinical decision support systems.

Authors:  Jannik Schaaf; Martin Sedlmayr; Johanna Schaefer; Holger Storf
Journal:  Orphanet J Rare Dis       Date:  2020-09-24       Impact factor: 4.123

  10 in total

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