Mass Screening for Neuroblastoma in Infants in Japan

Sawada, Tadashi; Nakata, Takao; Takasugi, Nobuo; Maeda, Kunihiro; Hanawa, Y; Shimizu, Koichiro; Hirayama, Motoaki; Takeda, Takeo; Mori, Takeshi; Koide, R; Tsunoda, Akio; Nagahara, Noboru; Yamamoto, Keiko

doi:10.1016/s0140-6736(84)90311-8

Cited by 145 publications

(49 citation statements)

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“…It has been generally recognized that stage I, II, and IVS (special stage IV with favorable outcome) patients have good prognoses and stage III, and IV (so called "advanced NB") have poor prognoses. [1][2][3]25) Regarding gender and age at diagnosis, 8 cases were males and 14 cases females, with a mean age of 2.9 years (range 0.4 to 10.6 years). It has been reported that patients younger than 1 year old have a much more favorable outcome than older patients.…”

Section: Resultsmentioning

confidence: 99%

“…[1][2][3]25) The organs of origin were the retroperitoneum in 6, the adrenal gland in 14 and the mediastinum in 2 cases (in general, NBs of adrenal gland origin have poor prognoses). [1][2][3]25) MYCN gene amplification is very much related to patient prognosis and was recognized only each of the stage II and stage IV patients. As for patient outcome, all patients at stages I, II, III, and VIS are alive, on the contrary, 5 of the 11 stage IV patients are alive and 6 have died.…”

Section: Resultsmentioning

confidence: 99%

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Analyses of Novel Prognostic Factors in Neuroblastoma Patients

Ootsuka

Asami

Sasaki

et al. 2007

Biological & Pharmaceutical Bulletin

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Analyses of Novel Prognostic Factors in Neuroblastoma Patients

Ootsuka

Asami

Sasaki

et al. 2007

Biological & Pharmaceutical Bulletin

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“…The disease is notorious for its vagueness of symptoms (Carlsen et al, 1987;Wilson & Draper, 1974) and, as age under one year and stages I-II disease have a crucial effect on prognosis, paediatric oncologists emphasise the importance of a good abdominal examination whenever a child is seen by a physician, and mass screening programmes in infants have been proposed (Sawada et al, 1984a(Sawada et al, , 1987aScriver et al, 1987;Woods & Tuchman, 1987). Pastore et al (1984).…”

Section: Patient Populationmentioning

confidence: 99%

“…However, the benefits of a mass screening in infants (Sawada et al, 1984a(Sawada et al, , 1987aScriver et al, 1987;Woods & Tuchman, 1987) has been questioned because it is argued that some cases detected at screening might have subsequently regressed spontaneously (Norman et al, 1987;Pastore et al, 1984).…”

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confidence: 99%

How frequent is spontaneous remission of neuroblastomas? Implications for screening

Carlsen

1990

Br J Cancer

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Summary The 'true' incidence of spontaneous regression of neuroblastomas is uncertain. However, the frequency of spontaneous regression is important when the benefits of screening procedures are considered. In the population-based Danish neuroblastoma survey 1943-80, spontaneous regression was documented in less than 2% of cases. However, the 'true' incidence may be higher. The epidemiological findings of increased incidence and survival rates with an unchanged mortality rate may suggest the inclusion of borderline lesions among 'truly' malignant neuroblastomas in recent decades in Denmark. However, it is more likely to be a result of improved diagnosis, changes in the social composition of the population and possibly unidentified environmental agents. However, if some premalignant lesions in fact had been included, they are most likely to be stages I-II tumours of infancy. In this study we describe cases of spontaneous regression of neuroblastoma from the Danish population-based survey 1943-80. Numerous anecdotal reports of cases of spontaneous remission of neuroblastomas have been published. For example, a Danish patient diagnosed in 1941 has been the subject of several reports (Hansen, 1953;Rosendal, 1942;Visfeldt, 1963). Neuroblastomas undergoing spontaneous regression form 17% of the cases of spontaneous remission in man collected from the literature by Everson and Cole (1966). In a review of cases registered in Children's Cancer Study Group, Evans et al. (1976) estimated the frequency to be 8% of cases (including stage IV-S cases). Pritchard and Kemshead (1983), excluding stage IV-S cases, found the frequency to be only 1-2% in their experience. However, a population-based study has not been carried out so far.Most childhood neuroblastomas are likely to be congenital (Birch et al., 1980;Carlsen, 1988a;Carlsen et al., 1986a;Rubin, 1968;Sutow, 1958;Wilson & Draper, 1974), and the prognosis is poor for most patients over the age of 2 years. However, the benefits of a mass screening in infants (Sawada et al., 1984a(Sawada et al., , 1987aScriver et al., 1987;Woods & Tuchman, 1987) has been questioned because it is argued that some cases detected at screening might have subsequently regressed spontaneously (Norman et al., 1987;Pastore et al., 1984).The Danish population-based survey 1943 -80 (Carlsen, 1986, 1988bCarlsen et al., 1986a Carlsen et al., , b, 1987) offers a unique opportunity to estimate the frequency of spontaneous regression from clinical overt disease in an unselected population of neuroblastomas of childhood. This study reports and discusses cases of confirmed regression and also the questionable cases. The study also considers age, stage and fate of cases found incidentally by abdominal examination or chest X-rays, as these cases are suggested to be equivalent to the cases found by screening (Kosloske et al., 1987;Sawada et al., 1988). As data from the Danish survey indicate a zerotime shift (Bailar & Smith, 1986;Feinstein et al., 1985) in the study period (Carlsen, 1986;Carlsen et al., 1...

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“…In 1973, the Mass Screening Program for Neuroblastoma (MSPN) was commenced for the early detection of NB in children living in Kyoto, Japan. A nationwide MSPN for 6-month-old infants began in 1985 (Sawada et al, 1984). The latter MSPN revealed incidences of infantile NB in the early stages and good biological prognostic factors of tumours increased (Hachitanda et al, 1994;Sawada et al, 1998).…”

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confidence: 99%

MYCN gene amplification is a powerful prognostic factor even in infantile neuroblastoma detected by mass screening

et al. 2006

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MYCN is the most powerful prognostic factor in cases of older children. However, how MYCN is related to the prognosis of infantile cases is not clear. A mass screening program was carried out by measuring urinary catecholamine metabolites (VMA and HVA) from 6-month-old infants. Of 2084 cases detected by the screening program, MYCN amplification (MNA) was examined by Southern blot analyses in 1533 cases from 1987 to 2000. Of the 1533 cases examined, 1500 (97.8%) showed no MNA, 20 cases (1.3%) showed MNA from three to nine copies, and 13 (0.8%) cases showed more than 10 copies. The 4-year overall survival rates of these three groups (99, 89 and 53%, respectively) were significantly different (Po0.001), indicating that MYCN copy number correlates with the prognosis. Cases with MNA more than 10 copies were more advanced than those without amplification (stage III, IV vs I, II, IVs; Po0.001). Patients with MNA more than 10 copies had significantly higher serum levels of neuron-specific-enolase (NSE) and ferritin than non-amplified patients (P ¼ 0.049, P ¼ 0.025, respectively). MYCN amplification was strongly correlated with a poor prognosis in infantile neuroblastoma cases. Therefore, for the selection of appropriate treatment, an accurate determination of MNA is indispensable.

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Mass Screening for Neuroblastoma in Infants in Japan

Cited by 145 publications

References 5 publications

Analyses of Novel Prognostic Factors in Neuroblastoma Patients

Analyses of Novel Prognostic Factors in Neuroblastoma Patients

How frequent is spontaneous remission of neuroblastomas? Implications for screening

MYCN gene amplification is a powerful prognostic factor even in infantile neuroblastoma detected by mass screening

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