Burden of illness in not adequately controlled chronic hypoparathyroidism: Findings from a 13-country patient and caregiver survey

Heide Siggelkow; Bart L Clarke; John Germak; Claudio Marelli; Kristina Chen; Helen Dahl-Hansen; Elizabeth Glenister; Nawal Bent-Ennakhil; Davneet Judge; Katie Mycock; Jens Bollerslev

doi:10.1111/cen.14128

Burden of illness in not adequately controlled chronic hypoparathyroidism: Findings from a 13-country patient and caregiver survey

Clin Endocrinol (Oxf). 2020 Feb;92(2):159-168. doi: 10.1111/cen.14128. Epub 2019 Dec 11.

Authors

Affiliations

¹ Clinic of Gastroenterology and Endocrinology, University of Göttingen, Göttingen, Germany.
² Mayo Clinic Division of Endocrinology, Diabetes, Metabolism, and Nutrition, Rochester, MN, USA.
³ Shire International GmbH, a member of the Takeda group of companies, Zug, Switzerland.
⁴ Shire Human Genetic Therapies, Inc., a member of the Takeda group of companies, Cambridge, MA, USA.
⁵ hypoPARA NORGE, Vettre, Norway.
⁶ Hypopara UK, East Grinstead, UK.
⁷ Adelphi Real-World, Adelphi Mill, Bollington, UK.
⁸ Section of Specialized Endocrinology, Oslo University Hospital, Oslo, Norway.
⁹ Faculty of Medicine, University of Oslo, Oslo, Norway.

Abstract

Objective: To address knowledge gaps regarding burdens associated with not adequately controlled chronic hypoparathyroidism.

Design: Global patient and caregiver survey.

Study populations: Patients with chronic hypoparathyroidism not adequately controlled on conventional therapy and their caregivers.

Measurements: Health-related quality of life (HRQoL) and health status were evaluated using the 36-item Short Form version 2 (SF-36 v2.0) and Five-Level EuroQoL 5 Dimensions (EQ-5D-5L) instruments, respectively. Hypoparathyroidism-associated symptoms were assessed by a disease-specific Hypoparathyroidism Symptom Diary and caregiver burden via the Modified Caregiver Strain Index (MCSI).

Results: Data were obtained from 398 patients and 207 caregivers. Patients' self-rated hypoparathyroidism-related symptom severity was none (3%), mild (32%), moderate (53%) or severe (12%). Per the Hypoparathyroidism Symptom Diary, patients reported moderate, severe or very severe symptoms of physical fatigue (73%), muscle cramps (55%), heaviness in limbs (55%) and tingling (51%) over a 7-day recall period. Impacts (rated 'somewhat' or 'very much') were reported by 84% of patients for ability to exercise, 78% for sleep, 75% for ability to work and 63% for family relationships. Inverse relationships were observed between patient self-rated overall symptom severity and HRQoL and health status assessment scores-the greater the symptom severity, the lower the SF-36 and EQ-5D-5L scores. Caregiver burden increased with patient self-rated symptom severity: none, 1.7 MCSI; mild, 5.4 MCSI; moderate, 9.5 MCSI; and severe, 12.5 MCSI.

Conclusion: Patients with not adequately controlled hypoparathyroidism reported substantial symptoms and impacts. Greater patient symptom severity was associated with decreased patient HRQoL and health status assessments and increased caregiver burden.

Keywords: caregiver; health status; hypoparathyroidism; parathyroid hormone; patient; quality of life; surveys and questionnaires.

Publication types

Multicenter Study
Research Support, Non-U.S. Gov't

MeSH terms

Adult
Aged
Caregiver Burden / epidemiology
Caregiver Burden / etiology
Caregivers* / psychology
Caregivers* / statistics & numerical data
Chronic Disease
Cost of Illness*
Female
Health Status
Humans
Hypoparathyroidism / blood
Hypoparathyroidism / epidemiology*
Hypoparathyroidism / psychology
Hypoparathyroidism / therapy*
Male
Middle Aged
Parathyroid Hormone / blood
Quality of Life*
Stress, Psychological / epidemiology
Stress, Psychological / etiology
Surveys and Questionnaires

Substances

PTH protein, human
Parathyroid Hormone