Table 3 Summary of major changes made to the CIQ
Type of change | Preliminary MLD-CIQ (25 items) Concepts present in preliminary draft | MLD-CIQ final draft (33 items) Concepts added or removed | MPS II-CIQ (28 items) Concepts added or removed | MPS IIIA-CIQ (30 items) Concepts added or removed | |||
|---|---|---|---|---|---|---|---|
Revision | Rationale | Revision | Rationale | Revision | Rationale | ||
Social/family life/leisure/relationships | Difficulty in: relaxing; participating in social activities; leaving home; completing household tasks; seeing family and friends; making long-term plans; paying attention to other family members or causing conflict between family members | Concepts added: Effort to go out in public with patient | Descriptions of difficulty taking child out of the house, not related to social stigma | Concepts added: Difficulty due to the patient’s disruptive behavior Effects of disruptive behavior on: (1) enjoying time with family or friends; (2) doing daily activities; (3) caring for the patient; (4) social activities; (5) managing the patient’s behavior | Overarching concept raised by caregivers, reflecting the behavioral symptoms common in severe MPS II | No new concepts added or removed | |
Effort to participate in leisure activities | Descriptions by caregivers of leisure activities being impacted | ||||||
Effort to communicate with patient | Suggestion to focus on the frequency of communication difficulties, because not all patients are unable to communicate | ||||||
Emotional/psychological functioning | Worried; unhappy; overwhelmed; helpless; angry; discouraged; disappointed that you cannot communicate with your child | Concepts added: Easily impatient or irritable; discouraged about limited treatment options; feel stressed | Frequent reports of general stress and specific stressful events | Concepts added: Frustrated that you could not communicate with the patient | Raised by caregivers as an important concept not covered by existing questions Removed as an approved treatment is available for MPS II Deemed not specific enough | Concepts added: Anxious about the patient’s future; grieving about the patient’s illness; being hit, kicked, or bitten by the child | All raised by caregivers as important concepts that were not covered by existing questions |
Concepts removed: Discouraged about limited treatment options Worried | |||||||
Physical functioning | Tired; physical strain; do not have enough physical strength to care for your child; feel exhausted; sleep disturbance | Concepts added: Unable to take care of own health | Reports of delayed caregiver treatment or neglect of own health due to prioritizing caregiving | Concepts added: Physically exhausted | Concepts removed Difficulty dealing with disruptive behavior | Deemed redundant (covered by other questions) | |
Daily activities | Families giving up things they usually do; personal time reduced | Concepts added: Unable to participate in activities because of limited time | Range of activities ‘given up’ by caregivers (e.g. seeing family, exercising) | No new concepts added or removed | No new concepts added or removed | ||
Financial/productivity | Financial strain on family; days missed from work | Concepts added: Can’t participate in activities because of limited finances Concepts removed: Days missed from work | Focus shifted to the effect of financial strain on the caregiver rather than the whole family | Concepts added: Worry about finances Concepts removed: Can’t participate in activities because of limited finances | Endorsed concepts of ‘worry’ and ‘financial difficulties’ | No new concepts added or removed | |