Mass screening in Japan increased the detection of infants with neuroblastoma without a decrease in cases in older children

Bessho, Fumio; Hashizume, K.; Nakajo, Toshio; Kamoshita, Shigehiko

doi:10.1016/s0022-3476(05)80733-4

Cited by 101 publications

(47 citation statements)

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“…Several reports have shown that the relative frequencies of the tumor failed to decrease in older children following screening (Bessho et al, 1991) or have suggested that a large number of the tumors found by screening might otherwise have spontaneously regressed (Nishi et al, 1995). We have estimated the age-specific incidence of neuroblastoma, using data from the Japan Children's Cancer Registry, to determine whether screening has resulted in any changes in the age-specific annual incidence rate.…”

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Effects of mass screening on age-specific incidence of neuroblastoma

Bessho

1996

Int. J. Cancer

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Using data from the Japan Children's Cancer Registry, we estimated the age-specific incidence rates of neuroblastoma. Before the neuroblastoma screening program started in 1985, the age-standardized incidence rates of neuroblastoma ranged from 7.5 to 9. I x for children under I5 years of age. After the introduction of the screening program, the annual incidence rate rose to 19.5 X The annual incidence rate for neuroblastoma in children under I year of age was 150.60 X I OW6 in the years I989 to 1992. whereas the incidence rates only varied between 23.6 and 34. I 3 X I 0-6 in the 3 preceding 5-year periods. This increase in incidence for infants was accompanied by a minor decrease in incidence for children 2 to 3 years of age.However, this decrease may only partly explain the large increase in incidence for infants. Therefore, we suggest that screening may result in the detection of otherwise spontaneously regressing tumors. It is urgent to determine the contribution of screening to decreasing mortality before deciding whether this screening program should be continued.o I994 Wiley-Liss, Inc.Since its inception, there have been controversies regarding the effectiveness of neuroblastoma screening for 6-month-old infants (Woods and Tuchman, 1987; Mauer, 1988;Tuchman et al., 1990;Murphy et al., 1991). Several reports have shown that the relative frequencies of the tumor failed to decrease in older children following screening (Bessho et al., 1991) or have suggested that a large number of the tumors found by screening might otherwise have spontaneously regressed (Nishi et al., 1995). We have estimated the age-specific incidence of neuroblastoma, using data from the Japan Children's Cancer Registry, to determine whether screening has resulted in any changes in the age-specific annual incidence rate. MATERIAL AND METHODSNumbers of neuroblastoma cases registered with the Japan Children's Cancer Registry (JCCR) between 1969 and 1992 were obtained from 3 volumes of 5-year reports (JCCR Committee, 1975, 1983 and from annual reports of the JCCR (JCCR Committee, 1987. Neuroblastoma, ganglioneuroblastoma and ganglioneuroma were collectively included as "neuroblastoma". Ganglioneuroma was included because neuroblastoma and ganglioneuroblastoma could mature to ganglioneuroma in vivo when they were left untreated (Haas et al., 1988).Sizes of the population in each age group were calculated by year from vital statistics published by the Statistics and Information Department, Minister's Secretariat, Ministry of Health and Welfare of Japan. The population of infants under 1 year of age was the number of births. The population of a particular age group in a particular year was calculated by subtracting the number of deaths from the population data of the I-year-younger age group in the previous year.The results of screening are reported annually by the Neuroblastoma Screening Committee. Estimation of registration rate of JCCRThe JCCR is not population-based and its exact registration rate is not known. Therefore, an estimation of ...

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Effects of mass screening on age-specific incidence of neuroblastoma

Bessho

1996

Int. J. Cancer

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“…A simple non-invasive test exists (Tuchman et al, 1987;Mathieu et al, 1993), and a mass screening programme was started in Japan in 1985 after several pilot studies. After more than 7 years of operation, however, there is still little evidence that screening for neuroblastoma is effective (Hanawa et al, 1990;Nishi et al, 1992), and several researchers have expressed doubts about the appropriateness of the procedure set up in Japan, suggesting in particular that screening at 6 months is too early (Cole and Parker, 1990;Bessho et al, 1991;Parker et al, 1991). Screening efficacy for neuroblastoma is still an open question, and before embarking on large evaluation trials it is necessary to review the epidemiological and biological evidence to assess whether the evaluation of screening at an age older than 6 months is feasible, or even if it is needed at all, given the progress recently made on understanding the biology of the disease which suggests that neuroblastoma is not a single disease entity.…”

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Is neuroblastoma screening evaluation needed and feasible?

Estève

Parker²,

Roy³

et al. 1995

Br J Cancer

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Despite the five million children who have been screened for neuroblastoma in Japan through detection of catecholamine metabolites, it is still uncertain whether screening for this disease is beneficial. The Japanese study has clearly indicated that screening at 6 months or earlier leads to heavy overdiagnosis. It is shown in this paper that screening at a later age may give the same reduction in mortality with possibly less overdiagnosis. However, it is estimated that, even with two screens at 12 and 18 months, the reduction in mortality would not greatly exceed 25%, under realistic hypotheses on the length of the preclinical phase of the disease. The evaluation of the efficacy of this screening strategy would need the recruitment of half a million children per year over 5-7 years and the follow-up of an equal number of controls. Such a trial would improve our knowledge of the natural history of the disease and might help to answer some questions raised recently regarding its biological heterogeneity.

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“…6 Since introduction of the screening, the incidence of neuroblastomas in infants has increased, as well as the number of stage 1, 2, and 4s neuroblastomas, as can be seen in Fig 4. However, there has been no significant change in the number of stage 3 or 4 diseases diagnosed in older children. 7 According to the Japan Children's Cancer Registry, there seems to be some difference in the site of origin of neuroblastomas detected by mass screening compared with those presenting clinically. Tumors of retroperitoneal and pelvic origin are more frequent in mass screening positive cases (Fig 5).…”

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Neuroblastoma of the Urinary Bladder, Preclinically Detected by Mass Screening

Yokoyama¹,

Hirakawa²,

Ueno³

et al. 1999

Pediatrics

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ABSTRACT. Background. Since the introduction of mass screening of infants for neuroblastoma, the incidence of neuroblastoma has increased in Japan. The reason for this increased incidence is the possible inclusion of many neuroblastomas that would have regressed spontaneously and would never have been detected clinically. An extremely rare tumor at the dome of the urinary bladder in a 7-month-old infant was detected by the mass screening.Methods. A case of neuroblastoma of the urinary bladder is reported with a review of the literature. The data in the Japan Children's Cancer Registry are also reviewed to analyze the incidence and the site of origin of neuroblastoma for evaluation of mass screening.Results. A 7-month-old female infant was referred because of a positive urine vanillylmanderic acid screening test. Ultrasonography showed a solid mass attached to the urinary bladder. At laparotomy a 35 ؋ 30 ؋ 25-mm egg-shaped tumor was found at the dome of the bladder, and a partial cystectomy was performed. During the operation no changes in blood pressure occurred when the tumor was manipulated. Histologic diagnosis was rosette-fibrillary neuroblastoma originating in the bladder wall, with a favorable Shimada histopathologic classification. N-myc was not amplified, which predicted a favorable prognosis, and no postoperative chemotherapy was given. The patient was free of symptoms and tumor after a follow-up period of 16 months. Literature review revealed that this was the second case of neuroblastoma of the urinary bladder ever reported in the world, although several cases of pheochromocytoma originating in the bladder wall had been reported. Both neuroblastoma and pheochromocytoma derive from the neural crest. The sympathogonia from the neural crest, a common stem cell, differentiates into a ganglion cell or into a secretory cell known as a chromaffin cell, able to manufacture catecholamines. The first case in the world that was reported as neuroblastoma of the urinary bladder was in a 4-month-old infant who was noted to have a 4-cm lower abdominal mass on routine physical examination. A ganglioneuroblastoma of the dome of the bladder was excised and the patient was doing well. On reviewing the Japan Children's Cancer Registry, the incidence of neuroblastomas in infants has increased as well as the number of stage 1, 2, and 4s (stage 4 special) neuroblastomas since the introduction of mass screening. However, there has been no significant change in the number of stage 3 or 4 diseases diagnosed in older children. According to the Japan Children's Cancer Registry, pelvic origin neuroblastoma, which has been noted to have spontaneous regression, was more frequent in the primary tumors detected by mass screening when compared with those presenting clinically. During preparation of this manuscript another case of bladder dome neuroblastoma was detected by urinary vanillylmanderic acid screening of 6-month-old infants for neuroblastoma in Japan.Conclusion. These extremely rare cases of neuroblastoma of the urinary blad...

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Mass screening in Japan increased the detection of infants with neuroblastoma without a decrease in cases in older children

Cited by 101 publications

References 24 publications

Effects of mass screening on age-specific incidence of neuroblastoma

Effects of mass screening on age-specific incidence of neuroblastoma

Is neuroblastoma screening evaluation needed and feasible?

Neuroblastoma of the Urinary Bladder, Preclinically Detected by Mass Screening

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