ObjectiveTo address knowledge gaps regarding burdens associated with not adequately controlled chronic hypoparathyroidism.DesignGlobal patient and caregiver survey.Study PopulationsPatients with chronic hypoparathyroidism not adequately controlled on conventional therapy and their caregivers.MeasurementsHealth‐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).ResultsData 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.ConclusionPatients 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.
Summary Oral calcium salts are recommended for the treatment of chronic hypoparathyroidism (HypoPT), although dosimetry is variable between individual patients and clinicians. However, patient feedback on calcium salts can be negative, particularly due to gastrointestinal side effects and hypercalciuria‐related complications. We begin with a clinical case of a HypoPT patient taking oral calcium salts following thyroid surgery, who requested support in reducing her dose of these with a view to stopping entirely. To evaluate her request, we first describe the usual treatment of HypoPT according to current guidance and then present data from (a) a case note review of a cohort of 24 HypoPT patients managed with a “no calcium” treatment regimen by single physician (b) a comprehensive online survey of HypoPT patients’ treatment and experiences (n = 330). The case note review found that target range serum calcium levels were successfully achieved in all 24 patients since transitioning to a “no calcium” regimen, without any breakthrough hypocalcaemia‐related symptoms, the development of new renal stones, the occurrence of calcium‐related hospital admissions or the finding of significant hypercalciuria. The online survey identified 36% of HypoPT patients who continued to take activated vitamin D, but had discontinued calcium supplements. HypoPT patients not currently taking calcium reported a significantly lower prevalence of adverse effects and outcomes, both compared with their previous experiences whilst taking calcium and also compared with the 64% of patients who continued to take oral calcium. We conclude that, subject to methodological limitations, there are significant issues of tolerability arising from conventional calcium‐based treatment regimens for patients with chronic HypoPT. For selected patients, it may be reasonable to facilitate a managed therapeutic transition to “no calcium” regimen, and we also propose that calcium‐based regimes be prospectively evaluated against calcium‐free (or calcium‐low) alternatives.
Significant knowledge gaps exist regarding the humanistic effects of hypoparathyroidism (HypoPT), a rare debilitating disorder. We report results from a global survey (13 countries) to characterize the impact of HypoPT on health-related quality of life (HRQoL) from the patient perspective. An anonymous survey was conducted in patients with uncontrolled chronic HypoPT (as determined by patients’ report of persistent symptoms and/or poorly controlled calcium levels as told by their physician) despite receiving conventional therapy. Patients currently or previously treated with parathyroid hormone were excluded. HRQoL and health status were evaluated using 2 validated instruments, 36-item short form health survey (SF-36) and 5-level EuroQoL 5 dimensions (EQ-5D-5L). All analyses were descriptive. Survey was conducted Oct 2017─Mar 2018 in 398 patients who met the study inclusion criteria (mean age, 51.7 years; women, 78%; surgery as main cause of HypoPT, 80%; mean duration of HypoPT, 8.9 years). There was an apparent inverse relationship between HypoPT symptom severity and both HRQoL assessment scores - the greater the severity, the lower the HRQoL and health status scores. Mean EQ-5D-5L utility scores (0-1) were 0.9/0.8/0.7/0.4 for patients reporting no/mild/moderate/severe HypoPT symptoms, respectively. EQ-5D-5L visual analog scale scores (0-100) were 86.9/72.5/57.7/41.1 for patients reporting no/mild/moderate/severe HypoPT symptoms, respectively. Mean SF-36 summary scores (0‒100) were 45.4/44.6/37.1/28.7 (physical component summary) and 53.2/44.9/35.2/31.6 (mental health component summary) for patients reporting no/mild/moderate/severe HypoPT symptoms, respectively. Findings showed that despite receiving conventional treatment, HypoPT was associated with burdensome symptoms that negatively affected HRQoL and health status. The degree of HRQoL reduction reflects the magnitude of symptom severity reported by patients with chronic HypoPT. SF-36 scores (mental and physical) reported for HypoPT patients in this survey were generally lower than scores reported in literature for patients with other disease states, such as heart disease, hematologic disorders, diabetes, or cancer. Funding: Shire
Hypoparathyroidism (HypoPT) is associated with significant symptom burden. We present results from a global survey (13 countries) to characterize HypoPT burden from the patient and caregiver perspectives. An anonymous survey was conducted in patients with uncontrolled chronic HypoPT (as determined by patients’ report of persistent symptoms and/or poorly controlled calcium levels as told by their physician) despite receiving conventional therapy. Patients currently or previously treated with parathyroid hormone were excluded. Levels of employment were evaluated using the Work Productivity and Activity Impairment (WPAI) tool. Caregiver burden was evaluated using the Modified Caregiver Strain Index (MCSI). All analyses were descriptive. Survey was conducted Oct 2017─Mar 2018 in 398 patients who met study inclusion criteria (mean age, 51.7 yrs; women, 78%; surgery as main cause of HypoPT, 80%; mean HypoPT duration, 8.9 yrs) and 207 caregivers (mean age, 52.4 yrs; women, 50%; spouse/partner, 70%; mean time providing care, 5.7 yrs; mean hrs/wk providing care, 24.1). 58% of patients had full-time work before HypoPT diagnosis vs 34% at time of survey. Change of employment as a result of HypoPT was associated with an increase in symptom severity (83%, severe; 67%, moderate; 29%, mild). WPAI scores showed patients working and performing activities at 59% and 58% of their total capacity, respectively. Proportion of patients reporting “a major impact” on relationships increased with symptom severity: spouse/partner (55%, severe; 33%, moderate; 14%, mild), family (61%, severe; 34% moderate; 13% mild), and friends (65%, severe; 45%, moderate; 16%, mild). 51% of caregivers had full-time work before caring for their patient with HypoPT vs 41% at time of survey; 26% of 42 caregivers who changed employment attributed it to HypoPT. WPAI scores showed caregivers working and performing activities at 79% and 67% of their total capacity, respectively. Caregivers reported “a major impact” on their relationship with spouse/partner (28%), family (21%), and friends (21%). Increased caregiver burden was associated with patient symptom severity: severe, 12.5 MSCI; moderate, 9.5 MSCI; mild, 5.4 MSCI. Findings from this global survey demonstrated that HypoPT negatively impacts the ability of patients and caregivers to maintain productive employment and personal relationships; degree of burden increased with symptom severity. These results provide unique insight on the burden associated with chronic HypoPT from both the patient and caregiver perspective. Funding: Shire
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