Patients and loved ones

As a part of the ROADMAP project participation, we asked the rare disease nonprofits to forward the survey to their US-based patient, loved one network and invite them to take the survey. Rare disease patients and their loved ones received the same set of questions, focused on the patient’s experience with off-label drugs for their rare disease, including roadblocks in access, as well as their general attitude towards and comfort level with drug repurposing. We had participation from 340 patients and 170 loved ones from 64 organizations.

*Note that most of the survey questions were not required, so the total number of responses is noted for each graph, as they vary.

Involvement in organization’s activities

Patients are central to a rare disease nonprofit organization. They may also be involved in various activities. In our data, the top selected choices were contributing data through surveys, helping raise awareness about their rare disease, and providing support to other patients through various community resources and events.

Note: respondents were able to select multiple answer choices.

n (patients): 429

Prescription drugs used for rare disease treatment in the last 5 years

We were also interested in what drugs patients were being prescribed for their rare disease. Among the many drugs listed, specifically 1,291, the most common one by far was prednisone, with 129 reports. It is a glucocorticoid medication used to suppress the immune system and decrease inflammation in various diseases.

*Note: the set of questions regarding drugs merges data from 1-5 drug(s) listed from each respondent. Where applicable, both patients and drug totals are provided. These numbers vary by how many patients answered each question and how many drugs they listed

n (drugs): 1291
n (patients): 348

Prescription drugs: approval status

We also asked patients to state whether the drugs they listed are FDA-approved for their rare disease, off-label or if they weren’t sure. Curiously, the data showed almost an even split between the three categories, illustrating that the discussion of whether the treatments are FDA-approved or not may not be part of the discussions between doctor and patient.

Also, from our data we know that there are 64 unique rare diseases represented in the patient data. It is unlikely that such a high percentage of them are FDA-approved for the patient’s rare disease(s).

n (drugs): 1275
n (patients): 347

Prescription drugs: duration of use

We asked patients to list the drugs they have been prescribed in the last 5 years, but we also wanted to know how much time they have been taking them in order to better understand the later questions about their effectiveness on various aspects of their rare disease(s). From our data, most of the drugs have been utilized for at least a few years and thus long enough to have a good measure of effectiveness.

n (drugs): 460
n (patients): 121

Prescription drugs: comfort level

In our data, most of the patients listed quite a high level of comfort with the drugs they have prescribed and utilized in the past 5 years: 165 drugs were listed as associated with a “very comfortable” level.

n (drugs): 463
n (patients): 121

Prescription drugs: symptom reduction effectiveness

Overall, over 300 drugs were reported as somewhat or very effective at reducing the overall symptoms associated with the rare disease(s), regardless of their approval status.

n (drugs): 460
n (patients): = 121

Prescription drugs: preventing disease worsening

In regards to preventing worsening of the rare disease symptoms, there are still 232 drugs reported as very or somewhat effective, but also a high number of drugs listed as not effective - 91 drugs.

n (drugs): 459
n (patients): 120

Prescription drugs: addressing all challenges

When asked about whether the currently prescribed drugs address all challenges related to a patient’s rare disease, we see quite a high degree of drugs that patients report to be not at all effective - 124 (27%). Though a significant amount - 172 (37%) are reported to be very or somewhat effective as well, illustrating that even though existing drugs are helping a portion of the patients, there is still a need which can be potentially filled via additional drug repurposing research.

n (drugs): 460
n (patients): 121

Prescription drugs: roadblocks to access

One of the important things we wanted to identify is whether existing drugs are generally freely available to the patients, or whether there are roadblocks in access. Mostly, patients reported having no roadblocks in access to 301 drugs, though some drugs are still difficult to get due to insurance issues, availability, prescription, and drug costs.

Note: respondents were able to select multiple answer choices.

n (drugs): 464
n (patients): 122


Awareness of the term “off-label drugs”

In our data, the majority of patients (322) reported being familiar with the term “off-label drugs” prior to this survey.

n (patients): 420

Off-label drugs: Requesting prescription

Surprisingly, despite the majority of patients being aware of the term off-label drug use in general, the majority (288 patients) have not directly asked their doctor to try an off-label drug for their rare disease before.

n (patients): 418

Off-label drugs: awareness of other options

One of the potential reasons for why patients may not directly request to try an off-label drug for their rare disease is a lack of awareness of specific drugs that can potentially be helpful for their rare disease.

We asked patients to list which off-label drugs they are aware of that they have been given to other patients with their rare disease, but they haven’t received themselves. Among all these drugs - 246 drugs specifically - sirolimus was the most common (24 patients listed it), with infliximab coming up as the second most common (17 patients listed it).

n (patients): 418

n (drugs): 246
n (patients): 129

Off-label drugs: willingness to take

Another aspect of the rare disease patients’ experience that we really wanted to find out about is whether they had reservations of utilizing drugs which are not FDA-approved for their rare disease. In our data, this doesn’t seem to be the case, with the 48% of patients (196) reporting being very willing to follow their doctor’s recommendations to take an off-label drug.

n (patients): 409

Off-label drugs: factors

Among the reasons for the factors which might influence their decision as to taking an off-label drug or not, the top 3 in our data were there being enough data on the drug’s safety and effectiveness, and trust in their doctor’s expertise.

Note: respondents were able to select multiple answer choices.

n (patients): 413


Drug Repurposing

For purposes of this project, we utilized a broad definition of drug repurposing, as a process of research to identify potential treatments that are already FDA-approved or in development for one disease, for use in another disease by gathering data and analyzing efficacy in order to improve treatment guidelines and access.

Drug repurposing: level of familiarity

Generally, patients in our data were not very familiar with the term “drug repurposing” or what it entailed prior to this survey. 119 (30%) patients reported being not familiar at all, 167 (42%) were slightly to moderately familiar.

Note: as a part of the survey, we did provide a definition to help familiarize patients with our definition of drug repurposing.

n (patients): 399

Drug repurposing: level of interest

Despite a low level of familiarity with drug repurposing prior to this survey, a lot of patients (135, 34%) were extremely interested in what drug repurposing can offer for their rare disease.

n (patients): 397

Drug repurposing: awareness

Most patients (238) in our data were not aware of any examples of drug repurposing being done for their rare disease. Interestingly, this also includes patients from the organizations which are currently supporting drug repurposing projects.

n (patients): 396

Drug repurposing: most impactful aspects

Among the aspects that patients selected as ones that drug repurposing could offer the most impact on their rare disease experience, the top three were having more options for treatments, including off-label drugs, with sufficient safety and efficacy data.

Note: respondents were able to select multiple answer choices.

n (patients): 394

Drug repurposing: safety, importance and necessity assessment

The patients in our data stated that they feel that drug repurposing is generally safe, important and necessary for their rare disease.

To exclude certain answer choices and view a subset of the data, please click the answer choices in the chart legend.

n (patients): 358