Bringing Hidden Conditions to the Fore: Factors Influencing Funding of Research and Services

Sharon L. Tennstedt PhD, RN
New England Research Institutes
Watertown, MA 02472

In a rational world, the level of funding for research and services for a health condition would be related to objective criteria regarding the burden of that condition.  The fact that it is not is often at the very least puzzling and more often distressing to persons with that condition.  Decisions about funding of research are influenced by more than objective measures of a disease or health problem.

In the late 1990s, the Institute of Medicare proposed that the amount of disease-specific research funding by the National Institutes of Health (NIH) be compared with the burden of disease for society¹.  NIH had previously identified five major criteria for the allocation of research funds: public health needs, the scientific quality of research, the probability of success, the maintenance of a diverse research portfolio, and the maintenance of an adequate scientific infrastructure^2.   There should be little disagreement with most of these factors.  First, only scientifically rigorous research with a high probability of success should be funded. Second, NIH’s role in building and maintaining the scientific infrastructure for health research in this country is part of its federal mandate.  Further, it would seem that the objective of a diverse research portfolio would ensure attention to important health issues.  However, it all comes down to how “public health needs” are defined and measured.

A 1999 study by Gross and colleagues³ investigated the relationship between NIH funding on research of specific diseases with the burden of these diseases.  Six measures of disease burden were used: 1) the prevalence or the total number of people with a disease; 2) the incidence or frequency of new cases; 3) total mortality; 4) years of life lost (or life expectancy minus age at death); 5) use of resources and cost, measured by the number of hospital days; and 6) disability-adjusted life-years (years of healthy life lost because of either disability or death).  Their analysis of NIH funding in 1996 found that the level of funding was not related to the number of people affected by a disease (prevalence or incidence) or to the resources used and costs of a disease.  Disease-related number of deaths and years of life lost were weakly related to research funding for a disease.  However, the number of disability-adjusted life years was more strongly predictive of level of funding (r=0.62, p< .001).

Investigators then tried to predict expected funding for a disease based on mortality, years of life lost, and number of disability-adjusted life years.  A comparison of expected to actual funding revealed some interesting findings.  HIV/AIDS, breast cancer, diabetes, and dementia received relatively generous funding support whereas research on other common or disabling diseases such as peptic ulcers, and chronic pulmonary disease were relatively underfunded.  Urinary incontinence was not included in the 29 conditions included in this investigation.

While the study findings that NIH funding levels are related to disease burden are reassuring, the findings also question the appropriateness of funding amounts across various diseases with similarly high levels of disease burden.  The results suggest that there are factors other than disease burden that influence how much money is directed at research on various health conditions.  These factors might include the amount of research supported by other federal agencies such as Veterans Affairs or the Department of Defense, or by private industry.  Industry funds at least twice as much biomedical research as does the NIH⁴.  However, even this shared commitment does not ensure that all health conditions receive needed attention.

What other factors might influence funding of research – and services – directed at specific diseases or health problems?  In thinking about this, these factors introduce a level of subjectivity and challenge the objective criteria that seem desirable in funding decisions.

Why did the Gross et al³ study not find any relationship between funding and prevalence of a health problem?  One obvious answer might be that prevalence rates are underestimated.  This is not likely if the mortality rate for a health condition or disease is high.  However, if a health condition is not disabling and, further, is invisible, it might be very well be under-reported.  Many factors can contribute to under-reporting of a health problem, including fear of a diagnosis and/or no perceived chance of a cure; assumption that the problem is part of the aging process and therefore one must “live with it”; perception that the problem is personal and that to talk about it is taboo; and the stigma – real or assumed – associated with the condition.

Health conditions can be visible or invisible.  Visible conditions such as physical deformities, cerebral palsy or other neurological diseases, terminal stages of cancer, and end stages of heart and lung disease are obvious to others and cannot be hidden.  Even if health care is not actively sought, the condition eventually comes to the attention of providers and the incidence and prevalence of the condition are captured.  However, the complete capture of invisible conditions is dependent on reporting by the patient.  For these conditions, the social factors previously mentioned can greatly influence the accuracy of incidence and prevalence rates.

Urinary incontinence is an excellent example of such an invisible condition.  It is often a problem considered too personal to discuss or even to reveal to a physician.  Many women consider it just another part of aging and something to be tolerated.  There is a lack of knowledge of available treatments or the efficacy of these treatments.  Even if revealed, the misperceptions can be reinforced by peers or even by an uninformed health care provider who prescribes no treatment or perhaps a treatment that is not effective.  Take for example, the woman who is told (bit not taught how) to do pelvic floor muscle exercises for urge incontinence or who is prescribed an anticholinergic drug for stress incontinence.  With no appreciable reduction in accidents, she is not likely to seek further treatment, especially if her peers report similar experiences.

Other symptoms that have been similarly under-reported include rectal bleeding that can be a sign of rectal or colon cancer.  Sometimes attributed to hemorrhoids, this bleeding is not a problem conducive to public admission, even to a physician. Mental illness is also laden with values and therefore can remain hidden.  Even when apparent to others, the response can be one of derision rather than support.  Reactions such as “Pull yourself together” or “Snap out of it” suggest that the individual is in control of the symptoms and therefore can willfully change the despondency or sadness of a clinical depression.  Shame can also interfere with recognition and health care seeking.  The symptoms of a more serious psychiatric disorder, even when resulting in inappropriate, disruptive, or dangerous behaviors, can be tolerated and hidden by families because of the shame associated with the presenting problem.  This is particularly true in certain cultural groups and even social classes.

What factors bring a disease or health condition to the fore – to grab the attention of biomedical and public health researchers? The measures of disease burden in the study by Gross and colleagues³ are a good place to start.  Most often it is a confluence of these factors that increases the likelihood of immediate attention and funding.  The case of a highly infectious disease with a high mortality rate is a good example.  HIV/AIDS and SARS are two such examples that offer interesting contrasts.  Critical differences in these two diseases are listed in Table 1.  While generously funded at the current time, research funding for AIDS was slow in coming.  It took years to identify the AIDS virus whereas the bacterial origin of SARS was identified in a matter of months. While there is a large difference in the mortality rates of the two diseases, death from SARS can occur in days whereas a person with HIV can live for several years untreated.  Similarly the incubation rate for SARS is very short whereas an HIV-infected person can live symptom-free for several years.  However, an important distinguishing factor is perceived vulnerability, as influenced by the mode of transmission.  At first, transmission of HIV was thought confined to subgroups of the population – to homosexual communities and to those who had intimate sexual contact with infected persons.  Thus, most individuals felt reassured that they were protected from disease because they were not a member of the at-risk population or they did not engage in at-risk behaviors.  It was not until the disease appeared in other population groups and the transmission via contaminated blood was discovered, along with an increasing death rate, that public alarm and the recognition of the greater risk to the wider population increased and funding for biomedical research and services began to grow at exponential rates.

Table 1
	HIV/AIDS	SARS
Time to discover etiology	>3 years	4-5 months
Mortality Rate	> 60%	2%
Effects of treatment	chronic illness	cure
Mode of transmission	sexual contact	airborne
	contaminated blood
Period of incubation	long: 9 years	short: days

It is only in the past few years that HIV/AIDS has been termed a global epidemic.  Although originating in another country (Africa) and considered an epidemic in both industrial and developing countries, the global nature of the AIDS epidemic took more than a decade to be so recognized and termed as such.

In contrast, take the case of SARS (Severe Acute Respiratory Syndrome).  Similarly, it originated in another country (China), but its perceived threat to not only the U.S. but to the rest of the world was immediate.  With the first cases reported in China in early 2003, the report of cases in other parts of Asia and in Toronto among persons traveling from China led the World Health Organization to issue a global alert on March 12, 2003 and then a global travel alert on April 2, 2003.  With eleven laboratories around the world working on SARS, by mid-April 2003, scientists had identified the coronavirus responsible for SARS and further had mapped its genome.

The economic damage caused by a disease is another factor that can spur a timely response from the research community.  Interestingly, it is the acute contagious diseases rather than the disabling chronic diseases that have this effect.  Economic risks posed by infectious diseases are rising because air travel, international trade, and global communication make the world a smaller place.  SARS has recently been reported as one of the costliest diseases to emerge in the past decade. The costs referred to are not health care costs but the economic costs resulting from trade embargoes and travel restrictions.  The estimated impact of SARS is $50 billion⁵.

Another example of immediate public health response is the monkeypox virus. Unlike SARS, the monkeypox virus was known to the medical community, but the last major outbreak was in the Congo (Africa) in 1996-1997.  Therefore, in this case, the urgent question was the origin of the virus in the U.S.  As we know from recent wide coverage in the press, the infection was spread via Gambian giant rats to prairie dogs that are exotic pets in this country.  While neither highly contagious from person-to-person nor fatal (1-10%), again the general public felt vulnerable.

One could argue that personal vulnerability has been enhanced following the terrorist attacks of 9/11 (2001).  Certainly parallels can be drawn between reactions to the cases of anthrax considered (although not proven) to be terrorist-related and a highly contagious and fatal disease of unknown etiology such as SARS.  However, there are other factors at play here as well.  The symptoms of SARS and monkeypox are socially acceptable to report and ones for which a person is not personally responsible.

An increasing problem still considered to be one of personal responsibility is obesity. Called “a public issue but a private matter” by the media⁶, the high prevalence and serious health consequences are now well recognized for persons of all ages.  A recent survey by the Harvard School of Public Health revealed that nearly 8 of 10 Americans believe that obesity represents a national health crisis, yet at the same time there is a deep ambivalence about whether the problem should be addressed through public initiatives or should remain a private responsibility.  For years, it was thought that eating less and exercising more would take care of this problem.  Certainly this perspective has been profitable for the diet and weight control industry.  Yet obesity continues to flourish for up to one-third of the U.S. population^7-9.  Even more alarming is the recent trend of weight increase among the most or morbidly obese¹⁰. Unlike Richard Simmons who took the diet and exercise route to a reasonable weight, many of these morbidly obese individuals become socially reclusive, literally becoming prisoners of their home when excessive weight interferes with mobility and with finding clothing that fits them.  Even those obese individuals at lower weights who attempt to carry on their lives are not socially accepted.  Think of your own reaction when a very large person sits in the seat next to you on an airplane – or an airline’s requirement that an obese person purchase two seats.  The point here is that the social response to this highly visible problem can contribute to its becoming invisible.  For the morbidly obese, it is not the obesity but rather the associated diseases – diabetes, hypertension, heart disease – that bring them to the attention of physicians.  With the discovery in 2002 of the gene JNK (c-Jun Amino-terminal kinases), also known to researchers as “Junk”, that influences the metabolic pathway for obesity and obesity-induced type 2 diabetes¹¹, the perception of personal responsibility for obesity hopefully will change.

The fact that individual or social reaction to a health condition can interfere with health care seeking is the basis for other increasingly important factors in bringing hidden conditions to the fore.  Those factors are celebrity admission or endorsement and the important role of advocacy groups.  There are numerous examples of celebrity admissions that have influenced both reporting of a problem or have increased funding of research and services.  Consider the following:  Betty Ford’s admission of substance abuse; Bob Dole’s admission not only of prostate cancer but subsequent problems resulting from his surgery; Nancy Reagan’s admission of breast cancer; Mike Wallace’s admission of depression on a television special.  There are many others, but one stands out in its successful efforts to increase timely detection and treatment for colorectal cancer.  In response to her husband’s death at a young age, Katie Couric has led the efforts to bring this second-leading cause of cancer deaths into the public arena – from her highly visible colonoscopy aired live on the Today show to the establishment of a foundation, the National Colorectal Cancer Research Alliance.  Through this foundation, a multimedia CD-Rom has been produced to provide a discreet way to find out more about this condition.  In contrast, and attesting to the increasing public recognition of the problem, there is a traveling exhibit of the GI system that allows a person to walk through a model of the colon and rectum.

Pancreatic cancer has received a similar boost in visibility and research attention since the death in 1999 of a powerful cable television executive, Marc Lustgarten, Vice President of Cablevision.  The President of the company (Charles Dolan) started the Lustgarten Foundation, awarding $3 million in its first year to fund basic research into the disease¹².  Pancreatic cancer, typically eluding detection until end stages, kills 30,000 Americans annually usually within three months of diagnosis. In 2001, National Institutes of Health provided $21.8 million for research; whereas for colorectal cancer that kills just twice that many, NIH provided 10 times that amount ($207.4 million) in funds.  Researchers and clinicians are encouraged that private attention to the problem will spur increased federal support.

And most recently the coverage in the media about Bruce Edwards, the golf caddy for Tom Watson, has brought attention to the devastating and incurable neurological disease ALS, also known as Lou Gehrig’s disease.  Afflicting about 30,000 persons in this country and with no known cure, public attention is directed to this disease primarily through efforts of the ALS Association, an advocacy group.  As with Couric’s efforts on behalf of colorectal cancer, Watson has just announced that he will fund a foundation for research and services for individuals with ALS.

These private foundations and advocacy groups provide an invaluable source to consumers and to the clinical research community.  By lobbying Congress, they can be instrumental in bringing the importance of hidden conditions to the fore of the federal funding arena.  The power of an individual should not be underestimated.  As these few examples show, no one is immune.  The personal experience of a disease or health condition is a powerful motivating force.

REFERENCES

1.      Committee on NIH Research Priority-Setting Process.  “Scientific opportunities and public needs: improving priority setting and public input at the National Institutes of Health.”  National Academy Press 1998.

2.      Working Group on Priority Setting.  “Setting research priorities at the National Institutes of Health”.  National Institutes of Health 1997.

3.      Gross, CP, Anderson, GF, Powe, NR. (1999) “The Relation between Funding by the National Institutes of Health and the Burden of Disease”. The New England Journal of Medicine 340 (1881-1887): 24.

4.      Office of Financial Management.  “NIH Key Facts and history of funding.” National Institutes of Health 1996.

5.      Aoki, N.  “SARS impact could hit $100B”, The Boston Globe, 23 May 2003: C1.

6.      Smith, S.  “Obesity survey sees public issue, and a private matter.”  The Boston Globe, 12 June 2003:A3.

7.      Flegal, KM, Carroll, MD, Ogden, CL, and Johnson, CL. “Prevalence and trends in obesity among US adults, 1999-2000.” Journal of the American Medical Association 288.14 (2002): 1723-7.

8.      Choban, PS, Onyejekwe, J, Burge, JC, and Flancbaum, L. “A health status assessment of the impact of weight loss following Roux-en-Y gastric bypass for clinically severe obesity.”  Journal of the American College of Surgeons 188.5 (1999): 491-7.

9.      Kuczmarski, RJ, Flegal, KM, Campbell, SM, and Johnson, CL. “Increasing prevalence of overweight among US adults.  The National Health and Nutrition Examination Surveys, 1960 – 1991.”  Journal of the American Medical Association 272.3 (1994): 205 – 11.

10.  Kuczmarski, RJ.  “Prevalence of overweight and weight gain in the United States.”  The American Journal of Clinical Nutrition 55.2 (1992): 495S – 502S.

11.  Harvard School of Public Health. “Key Gene Discovered for Obesity and Diabetes.”  Press Release, 11/20/2002, available on www.hsph.harvard.edu/press/releases/press11202002b.html

12.  Mishra, R.  “Pancreatic Cancer gets new focus, funds.”  The Boston Globe, 23 May 2003, A1+.