1996
DOI: 10.1001/archderm.132.11.1390
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Juvenile xanthogranuloma, neurofibromatosis 1, and juvenile chronic myeloid leukemia

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Cited by 32 publications
(5 citation statements)
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“…In children with JXG and NF1, the risk of developing JMML may be as high as 1 in 62 to 250, equivalent to approximately 0.4% to 1.6% . These numbers must be interpreted carefully since they are based on small numbers from different studies, leading to statistical uncertainties, although these results correlate well with those from an earlier study by Gutmann et al , who found an incidence of 3.6 per 1,000 children with NF1 and JXG. Furthermore, this association may be underestimated because patients with JMML may die at an age at which children do not manifest sufficient clinical signs to make a diagnosis of NF1 .…”
Section: Discussionsupporting
confidence: 62%
“…In children with JXG and NF1, the risk of developing JMML may be as high as 1 in 62 to 250, equivalent to approximately 0.4% to 1.6% . These numbers must be interpreted carefully since they are based on small numbers from different studies, leading to statistical uncertainties, although these results correlate well with those from an earlier study by Gutmann et al , who found an incidence of 3.6 per 1,000 children with NF1 and JXG. Furthermore, this association may be underestimated because patients with JMML may die at an age at which children do not manifest sufficient clinical signs to make a diagnosis of NF1 .…”
Section: Discussionsupporting
confidence: 62%
“…Unfortunately, most, if not all, MPNST (either from NF1 or non-NF1 patients) are rarely diagnosed at the level of the precursor pNF lesion due to their deep anatomical location, precluding anyone from conclusively answering this critical question. In other words, our model predicts that the incidence of MPNST, and breast cancer 46 50 as well as juvenile myelomonocytic leukemia (JMML) 51 53 that are associated with NF1, would be much higher in NF1 patients if it were not due to the repressive role of the NF1 +/− microenvironment.…”
Section: Discussionmentioning
confidence: 97%
“…They estimated that children with NF and JXG had a 20‐ to 32‐fold higher risk for JCML than do patients with NF without JXG. The following year, in a letter to the editor, Gutmann et al (22) contended that methods used in the former analysis were incorrect, and that these methods had led to an overestimation of the frequency of the triple association and of the risk of JCML. They concluded that the incidence of JCML in patients with NF1 and JXG would still be very rare, even if overestimated, and believed it premature to routinely monitor children for the development of signs and symptoms of JCML.…”
Section: Discussionmentioning
confidence: 99%