Literature DB >> 20635362

Living with Gaucher disease: Emotional health, psychosocial needs and concerns of individuals with Gaucher disease.

Wendy Packman1, Teresa Wilson Crosbie, Monic Behnken, Kerry Eudy, Seymour Packman.   

Abstract

Gaucher disease (GD) is the most common hereditary lysosomal storage disorder. Of the three variants of GD, type 1 accounts for 90% of cases. Patients with GD suffer from multiple medical symptoms and conditions. Clinical features of type 1 GD include hepatosplenomegaly; hematologic complications such as anemia and thrombocytopenia; and skeletal disease leading to avascular necrosis, osteopenia, and osteosclerosis. GD has unique features as a chronic illness: the disorder often presents with mild symptoms, and is frequently diagnosed in later childhood or adulthood. The treatment, enzyme replacement therapy (ERT), is efficacious. However, that same effective treatment is intrusive, expensive, and requires that patients restructure their work and personal schedules. Since the age of presentation can be anywhere between infancy and the eighth decade, the diagnostic process can be prolonged and stressful. The success of ERT notwithstanding, GD patients show distinct psychological complications [Packman et al. (2006); J Inherit Metab Dis 29:99-105]. In the present study, we describe the concerns, needs, challenges and positive effects of GD from the patients' perspective using in depth interviews of 28 individual affected by GD. Five core themes emerge: (1) difficulty coping with the diagnosis; (2) impact of pain on work, career, and recreational activities; (3) insurance concerns; (4) psychological distress (e.g., mood changes and anxiety); and (5) positive effects-strengthened family and social relationships and positive outlook. Our results highlight and expand awareness of the psychological and social needs of GD patients. This study calls for a collaborative, multidisciplinary effort in treating these patients and their families.

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Year:  2010        PMID: 20635362     DOI: 10.1002/ajmg.a.33527

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  8 in total

1.  Development and use of new therapeutics for rare diseases: views from patients, caregivers, and advocates.

Authors:  Aaron S Kesselheim; Sarah McGraw; Lauren Thompson; Kelly O'Keefe; Joshua J Gagne
Journal:  Patient       Date:  2015-02       Impact factor: 3.883

2.  Receiving enzyme replacement therapy for a lysosomal storage disorder: a preliminary exploration of the experiences of young patients and their families.

Authors:  R Freedman; M Sahhar; L Curnow; J Lee; H Peters
Journal:  J Genet Couns       Date:  2013-03-28       Impact factor: 2.537

Review 3.  Left to themselves: Time to target chronic pain in childhood rare diseases.

Authors:  Christine B Sieberg; Alyssa Lebel; Erin Silliman; Scott Holmes; David Borsook; Igor Elman
Journal:  Neurosci Biobehav Rev       Date:  2021-03-24       Impact factor: 8.989

4.  Child and family experiences with inborn errors of metabolism: a qualitative interview study with representatives of patient groups.

Authors:  Sara D Khangura; Kylie Tingley; Pranesh Chakraborty; Doug Coyle; Jonathan B Kronick; Anne-Marie Laberge; Julian Little; Fiona A Miller; John J Mitchell; Chitra Prasad; Shabnaz Siddiq; Komudi Siriwardena; Rebecca Sparkes; Kathy N Speechley; Sylvia Stockler; Yannis Trakadis; Brenda J Wilson; Kumanan Wilson; Beth K Potter
Journal:  J Inherit Metab Dis       Date:  2015-07-25       Impact factor: 4.982

5.  Development and validation of Gaucher disease type 1 (GD1)-specific patient-reported outcome measures (PROMs) for clinical monitoring and for clinical trials.

Authors:  Deborah Elstein; Nadia Belmatoug; Patrick Deegan; Özlem Göker-Alpan; Derralynn A Hughes; Ida Vanessa D Schwartz; Neal Weinreb; Nicola Bonner; Charlotte Panter; Donna Fountain; Andrew Lenny; Louise Longworth; Rachael Miller; Koonal Shah; Jörn Schenk; Rohini Sen; Ari Zimran
Journal:  Orphanet J Rare Dis       Date:  2022-01-06       Impact factor: 4.123

6.  Qualitative analysis of patient interviews on the burden of neuronopathic Gaucher disease in Japan.

Authors:  Yuta Koto; Aya Narita; Shinichi Noto; Midori Ono; Anna Lissa Hamada; Norio Sakai
Journal:  Orphanet J Rare Dis       Date:  2022-07-19       Impact factor: 4.303

7.  The European Gaucher Alliance: a survey of member patient organisations' activities, healthcare environments and concerns.

Authors:  Irena Žnidar; Tanya Collin-Histed; Pascal Niemeyer; Johanna Parkkinen; Anne-Grethe Lauridsen; Sandra Zariņa; Yossi Cohen; Jeremy Manuel
Journal:  Orphanet J Rare Dis       Date:  2014-09-02       Impact factor: 4.123

8.  Health-related quality of life in children and adolescents living with Gaucher disease and their parents.

Authors:  Eduardo Remor; Antonio Baldellou
Journal:  Health Psychol Behav Med       Date:  2018-04-12
  8 in total

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