Estimation of EuroQol 5-Dimensions health status utility values in hereditary angioedema

Figures & data

Table 1 HAE-BOIS-Europe interview results: impact of last attack on usual activities and transfer to EQ-5D response levelsa

HAE-BOIS-Europe survey patient-rated level	0	1	2	3	4	5	6	7	8	9	10
Reason givenby patient(s) for HAE-BOIS-Europe survey patient-rated level		I was able to do everything I wanted. It was a slight discomfort. I was hardly affected.	There still is some emotional stress in connection with the disease. It only affected me a little bit.	I can resume my activities very soon afterwards. I was still able to get out of bed.	There still is a swelling, so some kind of limitation of my activities but not too much. I was uncomfortable but I just didn’t do much.	It is difficult but I still can do most of the things I need to do. In my personal life, if I had to go out to eat with someone, I wouldn’t eat. I would prefer to relax to keep the attack down, so that I do not have to delay things.	There indeed are quite a lot of things that I am unable to do. An attack in my hand would impair my activities about 50%. It hampers me in doing a lot of things.	It prevented me from making food, picking up my house, driving, so I couldn’t travel where I wanted to. It affected everything; I couldn’t eat, drink, or move – I had to lie in bed because I couldn’t move. I had to just rest.	It keeps me from going to work. I can only sit down in my chair and just stare. And then my wife has to do all the work at home on her own, because I don’t take my share. I was able to stay at home and do a few things, but I had to sit down all day. I was able to read, for example.	I couldn’t do anything, I couldn’t work, I had to stay in bed […] I was very afraid. I had to put off doing everything I had planned at home and go to bed. I couldn’t do the things I had planned for my evening and the next day.	I cannot do anything anymore. Dizziness, nausea, diarrhea, general feeling of malaise. It was like I was in a coma, I couldn’t do anything. The pain is so bad that I have to stay in bed or just pace around. I am not able to do anything.

Note:

a 0–10 scale; higher score indicates worse impairment.

Abbreviations: EQ-5D, EuroQol 5-Dimensions; HAE-BOIS-Europe, Hereditary Angioedema Burden of Illness Study in Europe.

Table 2 HAE-BOIS-Europe interview results: level of anxiety about future attacks and transfer to EQ-5D response levelsa

HAE-BOIS-Europe survey patient-rated level	0	1	2	3	4	5	6	7	8	9	10
Reason given by patient(s) for HAE-BOIS-Europe survey patient-ratedlevel	I am not afraid, I have to live with the disease, and my medication is effective. I know how to handle my attacks, so I am not afraid about them coming back. I am not afraid because I know where to get treatment, I have gotten used to it.	[My new medications] help me very much, so I know I will be able to handle the next attack efficiently. I feel safe and confident and have no worries because of this disease […]because I have got a medication that works, that suits me. I am not anxious about having an attack. They come when they come – this is all.	I know that I can help myself, which is reassuring. Also I have kind of gotten used to it. They’re not as intense as before. Also, with the years, it affects me less. Like my age has something to do with how they are less severe now.	The uncertainty about what will be after the pregnancy. Having the baby and all, and how to handle severe abdominal attacks, especially when my husband is away. On the other hand, my mother lives nearby, so I will certainly have support.	I remain anxious, especially when I have sore throats. I say, “mmm, this isn’t going to be a throat attack of HAE, is it?” There is always that nightmare. I know that HAE can cause a lot of discomfort and cancelling a lot of things, but now I also know that I can take my medication.	I generally feel well-treated with my medication, but there is some fear about an eventual attack in the larynx. I am also in contact with my physicians; my boyfriend has been trained how to inject me, should there be an emergency, so generally I feel safer.		I’m preoccupied by it, it’s on my mind all the time. […] I have the uncertainty of the possibility of having a strong attack, like the throat attack I had. The uncertainty that it will be a serious attack and you’d have to respond quickly. Because of the bother of having to go to hospital to get the medicine or leave work during an attack if you’re working.	The only thing that frightens me is not knowing whether they would eventually occur in the larynx (father had a tracheotomy which scares me). I’d be lying if I said that it didn’t worry me […]. But, with time and with outside help, little by little, I am playing it down, trying to, at least.	Worried and anxious. I have anxiety and tension […] I’m not at ease […]It’s like it’s very present, it’s almost like a job.	I am always worried. Especially when I go on a holiday. If I go for a bike ride then I know that the next day I may have swelling in my crotch. Or my hands or feet. So it is always at the back of my mind, what you can plan to do and what you do.

Note:

a 0–10 scale; higher score indicates worse impairment.

Abbreviations: EQ-5D, EuroQol 5-Dimensions; HAE, hereditary angioedema; HAE-BOIS-Europe, Hereditary Angioedema Burden of Illness Study in Europe.

Table 3 Mapping of EQ-5D responses to HAE-BOIS-Europe survey items

EQ-5D domain^Citation26	EQ-5D description^Citation26	EQ-5D disutility (UK)^Citation26		Rating of HAE-BOIS-Europe survey items for EQ-5D utility
				During the latest attack	Between attacks
Pain/discomfort			Question on survey	What was the worst pain you had during your most recent HAE attack?	In general, how would you describe your health?
Level 1	No pain or discomfort	−0.0		No pain	Excellent or very good
Level 2	Moderate pain or discomfort	−0.123		Mild or moderate pain	Good or fair
Level 3	Extreme pain or discomfort	−0.386		Severe pain	Poor
Mobility			Question on survey	How long did the attack stop you from performing daily activities?	N/A
Level 1	No problems in walking about	−0.0		2 h or 2–4 h	Assume HAE does not impact mobility between attacks (ie, EQ-5D disutility =−0.0)
Level 2	Some problems in walking about	−0.069		4–12 h, 12–24 h, or 2 d
Level 3	Confined to bed	−0.314		3 d, 4 d, or >4 d
Self-care			Question on survey	N/A	N/A
Level 1	No problems with self-care	−0.0		Assume HAE attack does not impact self-care (ie, EQ-5D disutility =−0.0)	Assume HAE does not impact self-care between attacks (ie, EQ-5D disutility =−0.0)
Level 2	Some problems washing or dressing myself	−0.104
Level 3	Unable to wash or dress myself	−0.214
Usual activities			Question on survey	During your most recent attack, how much did the HAE attack affect your ability to do your regular daily activities, other than work at a job/be a student?	In the past 6 months, please think how much HAE affected your ability to do your regular daily activities, other than work at a job/be a student, between attacks
Level 1	No problems with performing my usual activities	−0.0		No problems (ie, HAE-BOIS-Europe survey patient-rated level =0.0–1.0)	No problems (ie, HAE-BOIS-Europe survey patient-rated level =0.0–1.0)
Level 2	Some problems with performing my usual activities	−0.036		Some or moderate levels (ie, HAE-BOIS-Europe survey patient-rated level =2.0–6.0)	Some or moderate levels (ie, HAE-BOIS-Europe survey patient-rated level =2.0–6.0)
Level 3	Unable to perform my usual activities	−0.094		Extreme problems (ie, HAE-BOIS-Europe survey patient-rated level =7.0–10.0)	Extreme problems (ie, HAE-BOIS-Europe survey patient-rated level =7.0–10.0)
Anxiety/depression			Question on survey	How anxious are you about having an HAE attack in the future?	Average of How often did you get sudden feelings of panic about HAE symptoms/attacks during the past 6 months? and How distressed are you about your HAE attacks/symptoms now? (1–5 scale each)
Level 1	Not anxious or depressed	−0.0		No problems (ie, HAE-BOIS-Europe survey patient-rated level =0.0–1.0)	HAE-BOIS-Europe survey patient-rated level <2.5 (1–5 scale)
Level 2	Moderately anxious or depressed	−0.071		Some or moderate levels (ie, HAE-BOIS-Europe survey patient-rated level =2.0–6.0)	HAE-BOIS-Europe survey patient-rated level =2.5–4.0
Level 3	Extremely anxious or depressed	−0.236		Extreme problems (ie, HAE-BOIS-Europe survey patient-rated level =7.0–10.0)	HAE-BOIS-Europe survey patient-rated level >4.0 (1–5 scale)

Notes: For EQ-5D disutility (UK), the constant term accounted for any dysfunctional state =−0.081, the constant term N3 accounted for if level 3 occurs within at least one dimension: −0.269, and full health =1. The summation of EQ-5D disutility (UK) values, including these three terms and EQ-5D disutility values for each EQ-5D domain at one of three levels in each domain, gives rise to the estimated value of EQ-5D utility.²⁶

Abbreviations: EQ-5D, EuroQol 5-Dimensions; HAE, hereditary angioedema; HAE-BOIS-Europe, Hereditary Angioedema Burden of Illness Study in Europe; h, hours, d, days; N/A, not applicable.

Table 4 Estimated EQ-5D utilities during and between attacks by country

HAE-related health state	All		Spain		Germany		Denmark		P-value
	n	Mean ± SD	n	Mean ± SD	n	Mean ± SD	n	Mean ± SD
Acute HAE state
Overall	111	0.444±0.30	41	0.412±0.30	40	0.514±0.29	30	0.396±0.32	0.184
By pain severity of last attack:*
No pain or mild	41	0.613±0.26	22	0.538±0.28	12	0.672±0.22	7	0.745±0.22
Moderate	48	0.467±0.27	14	0.358±0.25	19	0.606±0.20	15	0.394±0.27
Severe	22	0.080±0.08	5	0.006±0.01	9	0.110±0.08	8	0.093±0.09
Between HAE attacks	111	0.722±0.23	41	0.705±0.26	40	0.737±0.19	30	0.728±0.24	0.816

Notes:

a Analysis of variance test.

* P<0.001 for comparison of differences in utility values by pain severity levels among overall sample.

Abbreviations: EQ-5D, EuroQol 5-Dimensions; HAE, hereditary angioedema; SD, standard deviation.

Table 5 EQ-5D utilities reported for selected conditions

Reference/country	Condition	Utility
Stafford et al (2012)/UK^Citation31	Migraine: chronic state	0.87
Kind et al (1999)/UK^Citation30	UK general population norm	0.86
Westerhuis et al (2011)/the Netherlands^Citation32	Epilepsy	0.80
Dyer et al (2010)/worldwide^Citation35	Ischemic heart disease with mild angina	0.80
Ismail et al (2010)/England^Citation38	Type I diabetes mellitus	0.79
Terzano et al (2012)/Italy^Citation33	Asthma, partially controlled or uncontrolled	0.72
HAE-BOIS-Europea	HAE: chronic (between attacks)	0.72
Mustur et al (2009)/Montenegro^Citation34	Ankylosing spondylitis	0.69
Marra et al (2004)/Canada^Citation39	Rheumatoid arthritis	0.66
Pickard et al (2004)/Canada^Citation40	6 months after stroke	0.62
HAE-BOIS-Europea	HAE: attack with no pain or mild pain	0.61
Stafford et al (2012)/UK^Citation31	Migraine: moderate severity	0.53
HAE-BOIS-Europea	HAE: attack with moderate pain	0.47
Dyer et al (2010)/worldwide^Citation35	Ischemic heart disease with moderate/severe angina	0.45
Lee et al (2005)/Wales^Citation36	Renal failure on hemodialysis	0.44
HAE-BOIS-Europea	HAE: acute attack (overall)	0.44
Pickard et al (2004)/Canada^Citation40	Acute stroke	0.31
HAE-BOIS-Europea	HAE: attack with severe pain	0.08

Note:

a Refers to current study with data shown in bold.

Abbreviations: EQ-5D, EuroQol 5-Dimensions; HAE, hereditary angioedema; HAE-BOIS-Europe, Hereditary Angioedema Burden of Illness Study in Europe.

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