Characteristics of Receipt Rate of Financial Aid for Intractable Disease Patients in Gifu Prefecture , Japan

The data on intractable disease patients who applied for official financial aid for medical treatment in Gifu Prefecture, Japan, in fiscal year 1997 were analyzed. In addition, to assess the nonapplied patients, a questionnaire on intractable disease patients with disabilities needing assistance was sent to all medical institutions in the prefecture. The results suggest that the low receipt rate of financial aid in Gifu Prefecture results from both nonapplication by patients and underdiagnosis at medical institutions. Percent completion of medical treatment for intractable disease patients in a single secondary medical service area was low. Although the percentage of patients with disabilities authorized to receive financial aid was 4.6%, the estimated ratio of patients with disabilities needing assistance exceeded this value. Therefore, the dissemination of information on official services for patients and medical care facilities must be strengthened. J Epidemiol, 2000 ; 10 : 94-102


INTRODUCTION
In Japan, measures against intractable diseases have been carried out based on the Outline of Measures against Intractable Diseases (1972) 1).In this outline, intractable diseases are defined as: (1) those that are of unknown cause, for which there is no established treatment, and that are likely to cause aftereffects; or (2) chronic diseases that, in addition to economic issues, place a serious burden on the family because they require labor-intensive care for such things as personal care and diseases that create a serious psychological burden.
The countermeasures against these intractable diseases consist of five principles: (1) promotion of investigation and research; (2) provision of medical care facilities concerned; (3) reduction of the copayment for medical costs; (4) improvement and coordination of health care, medical care, and welfare services at the community level; and (5) promotion of welfare service measures aimed at improving quality of life of patients and their families 2).
As one of the above mentioned countermeasures, a financial aid system is provided for patients with 43 types (in fiscal year 1998) of designated intractable diseases.To receive this financial aid, every year a patient with an intractable disease must submit an application accompanied with a medical certificate to the prefectural government through a public health center governing his or her residence.In each prefecture, the applications are evaluated by the Committee on Measures for Intractable Diseases, which consists of specialists.As a result, payments of medical expenditure (a certain portion of patient copayments that is not covered by the social insurance system) are reduced for authorized intractable disease patients.These payments are waived especially for patients with disabilities needing physical assistance due to the designated intractable diseases and for patients with subuacute myelo-optico-neuropathy (SMON), Creutzfeldt-Jakob disease (CJD), fulminant hepatitis, or severe acute pancreatitis.In Gifu Prefecture, the rates of total authorized patients per population in fiscal years 1984 3), 1988, 1992 4) and 1997 5) were very low as compared with those in Japan.
This study intended to clarify the reasons of the lower receipt rate of financial aid in Gifu Prefecture, to estimate the number of patients with disabilities needing assistance due to intractable diseases, and to assess the nonapplied patients.In addition to the analysis of application forms (medical certificates) for financial aid, a questionnaire on intractable disease patients with disabilities was sent to all medical institutions in Gifu Prefecture.

METHODS
Thirty-nine types of diseases were covered by the financial aid system for intractable disease patients in fiscal year 1997 (from April 1997 to March 1998; Table 1).Until April 1998, all patients' copayments for these diseases had been waived, but a partial fixed copayment was introduced in May 1998.This copayment is waived for patients who have been proven to have disabilities due to the designated intractable diseases by an additional medical certificate or by a physically handicapped person's handbook (handicap class 1 or 2) as well as for patients with SMON, CJD, fulminant hepatitis, or severe acute pancreatitis.
First, the receipt rate of financial aid for each designated intractable disease at the end of fiscal year 1997 was compared between Gifu Prefecture and Japan.The rate was calculated by dividing the number of patients receiving aid on March 31 , 1998 5) by the total population on October 1, 1997 .
In fiscal year 1997, the Committee on Measures for Intractable Diseases in Gifu Prefecture accepted applications from 4986 patients.The committee rejected 35 applications and made inquiries to the physicians in charge for additional clinical data for 253 applications.The percentages of rejected and inquired applications were compared for each disease to evaluate the difficulty of acceptance.Periods between application and acceptance for financial aid also were calculated for each disease.
The author excluded the 320 repetitive patients applying for the following fiscal year of the 4986 patients; thus, the analysis was based on applied medical certificates of 4666 patients .Mean age and sex ratio of patients , percentage of newly applied patients, and mean period of suffering from the disease were calculated for each disease.Percent completion of medical treatment in each secondary medical service area were compared.The receipt rate of financial aid for each disease was also compared for each secondary medical service area .Authorized patients with disabilities needing physical assistance due to the designated intractable diseases during the period from May to December 1998 were linked to 4666 patients , and the percentage of patients with disabilities for each disease was calculated.
Next, a questionnaire on intractable disease patients with disabilities needing physical assistance was sent to all medical institutions (119 hospitals and 1339 clinics) in Gifu Prefecture during September and October 1997.The list and diagnostic criteria for designated intractable diseases 6) were distributed to all physicians via medical institutions.Then , data on the code of the disease, age, sex, place of residence, and possession of the physically handicapped person's handbook of the individual patients were collected.
Chi-square statistics were used to compare categorical variables, and analysis of variance and t-test were used to compare continuous variables.All tests were two-tailed.

RESULTS
In Gifu Prefecture, the total receipt rate of financial aid was the second lowest (following Yamanashi Prefecture) of 47 prefectures in Japan at the end of fiscal year 1997.The receipt rates of financial aid for sarcoidosis and systemic sclerosis, dermatomyositis, and polymyositis were significantly higher in Gifu Prefecture than in Japan, but were lower for 24 types of diseases (Table 1).
The decisions of the Committee on Measures for Intractable Diseases in Gifu Prefecture against the applications are listed in Table 2.The total percentage of rejected applications was 0.7%.Idiopathic interstitial pneumonia (lip), ossification of posterior longitudinal ligament (OPLL), primary immunodeficiency syndrome, and malignant rheumatoid arthritis (MRA) were rejected at significantly higher rates than the other diseases.Inquiries were made on 4.6% of the applications, with fulminant hepatitis, pigmentary degeneration of the retina , MRA, IIP, idiopathic dilated cardiomyopathy, and occlusive disease in circle of Willis inquired at significantly higher rates than the other diseases.Mean period between application and acceptance was significantly longer for pigmentary degeneration of the retina.
Table 3 presents the characteristics of intractable disease patients receiving financial aid.The total female:male ratio was 1.68.Mean age of intractable disease patients ranged from 40 to 60 years for 27 types of diseases, exceeded 60 years for SMON, Parkinson's disease, amyloidosis, OPLL , Shy-Drager syndrome, IIP, and CID, but was lower than 40 years for Crohn's disease, occlusive disease in circle of Willis , epidermolysis bullosa, and primary immunodeficiency syndrome .Percentages of newly accepted patients were significantly higher for fulminant hepatitis , IIP, severe acute pancreatitis, amyotrophic lateral sclerosis (ALS), idiopathic osteonecrosis of the femoral head (ION), periarthritis nodosa (PN) , OPLL, Parkinson's disease, and idiopathic dilated cardiomyopathy , and lower for systemic lupus erythematosus (SLE) , aortitis syndrome, myasthenia gravis, occlusive disease in circle of Willis, Behcet's disease, and systemic sclerosis , dermatomyositis, and polymyositis.Mean periods that patients suffered the intractable diseases exceeded 10 years for 11 types of diseases .
Fifty-seven percent of the authorized intractable disease patients consulted public hospitals, compared with 18% for university hospitals , 17% for private hospitals, and 9% for clinics (data not shown).The percentage of patients with primary biliary cirrhosis who consulted public hospitals (84 .3%)and those with pigmentary degeneration of the retina who consulted clinics (33.0%) were relatively higher.Medical treatment of 3541 (75.9%) intractable disease patients was completed in each secondary medical service area, 4095 (87.8%) within the prefecture, and 571 (12.2%) were treated in medical institutions outside Gifu Prefecture.The percent completion of med-ical treatment in a secondary medical service area was significantly lower in the Chuno area (46.7%) as compared with those in the Gifu area (86.6%),Seino area (72.0%),Tono area (79.4%), and Hida area (88.6%).Rates of treatment in medical institutions outside the prefecture were significantly higher for ION, aplastic anemia, multiple sclerosis (MS), myasthenia The receipt rate of financial aid for each secondary medical service area is listed in Table 4.The total receipt rate was significantly higher in the Hida area.Differences in receipt rates by area were significant for sarcoidosis, systemic sclerosis , dermatomyositis, and polymyositis, idiopathic thrombocytopenic purpura (ITP), spino-cerebellar degeneration , Crohn's disease, occlusive disease in circle of Willis , idiopathic dilated cardiomyopathy, ION , and pigmentary degeneration of the retina.
The total ratio of authorized patients with disabilities needing physical assistance due to intractable diseases was 4 .6%(Table 5).The percentages were significantly higher for Huntington's chorea, ALS , spino-cerebellar degeneration, MRA, MS, Parkinson's disease , and OPLL (excluding SMON, CJD, fulminant hepatitis, or severe acute pancreatitis) .(3) fewer applications from patients due to ignorance of the financial aid system; (4) fewer application to this financial aid system because it is judged to have fewer merits than other aid systems , such as those for the handicapped and for the aged; and (5) harsh judgement against the applied medical certificates for the diagnostic criteria .Low rejection rates imply easiness of application acceptance .However, both the number and rate of inquiries into applications of pigmentary degeneration of the retina were high .lar parkinsonism, which are commonly associated with parkinsonian features.Inquiries into applications of fulminant hepatitis, occlusive disease in circle of Willis, idiopathic dilated cardiomyopathy, and IIP were due to lack of clinical recording or laboratory data needed for the medical certificates.Relatively higher rates of inquiry and rejection for MRA resulted from difficulties in diagnosis.Rejection against applications of primary immunodeficiency syndrome and lIP were due to higher serum immunoglobulin concentration and higher arterial blood oxygen pressure than the authorization criteria, respectively.The reason for rejection of 15 patients of OPLL was relative mildness of the disability according to the authorization criteria for financial aid.The receipt rates for financial aid were broadly classified into: (1) generally lower receipt rates in all secondary medical service areas (for example, Behcet's disease, MRA and OPLL); and (2) lower receipt rates in particular areas (for example, idiopathic dilated cardiomyopathy and pigmentary degeneration of the retina).Underdiagnosis at medical institutions may cause inequality in receipt rates at secondary medical service areas.
Low receipt rates for orthopedic intractable diseases such as MRA, OPLL, and spinal canal stenosis may be due to a lower ratio of orthopedists per population (9.4 per 100,000 in Gifu Prefecture vs. 13.0 in Japan; P < 0.01) 7).These patients may be opting for alternative therapies such as bonesetting and chiropractic therapy 8).However, diagnosis rates of systemic sclerosis, dermatomyositis, and polymyositis was higher in Gifu Prefecture because the university school of medicine in Gifu Prefecture specializes on these diseases 9.10) The total percentage of new patients accepted for financial aid was higher (P < 0.01) than that in Japan in fiscal year 1992 11).These results suggest fewer continuous applications for financial aid.To reduce nonapplication due to patients' ignorance of the financial aid system, the prefectural government asked all physicians in medical institutions to urge patients to apply.However, because similar insurance systems are provided for handicapped persons, for persons aged 70 years or older and persons aged 65 years or older who are bedridden etc., and for persons receiving livelihood assistance, some of the eligible patients tend not to apply 0. The low percentage of patients aged 70 years or older suggest nonapplication by the aged 13).The discrepancy between the number of patients accepted for financial aid and that of patients with disabilities due to intractable diseases (as reported on the questionnaire) arises from these intentional nonapplications for the financial aid.
Treatment of intractable diseases is not always completed in the secondary medical service area because many patients require specialized medical treatment which is supplied at the prefecture level 14).Many intractable disease patients, especially those with disabilities, wish to receive medical care near home, for example, by visiting consultation at a certain hospital in the secondary medical service area 15).
The number of designated intractable diseases eligible for financial aid has been increasing year by year, and thus the number of patients receiving the financial aid has been increasing l).In Japan, measures against intractable diseases are now being reevaluated.Welfare services for intractable disease patients are being strengthened by dispatching home helpers and providing short stays according to the Fundamental Law for People with Disabilities (1995).Also these welfare measures begin when the patients apply to the services; therefore, the patients should be informed about various measures against intractable diseases via: (1) public information, including the Internet; (2) medical institutions in charge; and (3) patients' association.Cooperation among public health, medical, and welfare service institutions is essential for providing a continu-ous and comprehensive assistance system for intractable disease patients and their families.In addition, for evaluation of these services, the author advocates linking and analyzing these services, self-rated quality-of-life measures from patients, and death certificates 16,17).

Table 1 .
Receipt rate of financial aid for designated intractable disease patients at the end of fiscal year 1997.P < 0 .05;+P < 0.01 vs. Japan by chi-square test. *

Table 2 .
Judgement of the committee against the applications for financial aid in fiscal year 1997.

Table 3 .
Characteristics of the intractable disease patients authorized for financial aid in fiscal year 1997.

Table 5 also
presents the results of the questionnaire completed by medical institutions.Among the responding 86 hospitals (response rate 72.3%) and 517 clinics (response rate 38.6%), 61 hospitals (70.9%) and 195 clinics (37.7%) reported 1164 patients of intractable disease patients with disabilities needing physical assistance for the 39 types of applicable diseases.Patients with fulminant hepatitis, severe acute pancreatitis, or CJD were not reported.The number of reported patients with disabilities needing assistance exceeded that of authorized patients with disabilities by from twofold to fivefold.Figure1illustrates the age distribution of all patients authorized for financial aid and the patients with disabilities reported on the questionnaire.

Table 4 .
Receipt rate (per 100,000 population) of financial aid for each intractable disease by secondary medical care area in fiscal year 1997.
DISCUSSIONSeveral possible causes of lower receipt rate of financial aid are: (1) lower incidence of intractable diseases in Gifu Prefecture; (2) underdiagnosis of intractable diseases in medical institutions;

Table 5 .
Number of authorized patients with disability due to intractable diseases in fiscal year 1997 and the number of intractable disease patients with disabilities needing assistance as reported by Questionnaire.Age distribution of all patients authorized for financial aid for intractable diseases in fiscal year 1997 and the intractable disease patients with disabilities reported on the questionnaire.
These resulted from lack of fluorescein angiography examination, copies of electroretinography, or measures of visual field in the medical certificates 6) (data not shown).Inquiries into applications of ulcerative colitis (UC) were chiefly (37/39) caused by lack of differential diagnosis by stool examination.Inquiries into Parkinson's disease resulted from the difficulty of distinguishing the disease from other syndromes of parkinsonism (18/23), such as those due to multiple-system atrophy, progressive supranuclear palsy, cortical-basal ganglionic (corticobasal) degeneration, dementia with Lewy bodies, and vascu-