% IMPORTANT: The following is UTF-8 encoded. This means that in the presence % of non-ASCII characters, it will not work with BibTeX 0.99 or older. % Instead, you should use an up-to-date BibTeX implementation like “bibtex8” or % “biber”. @ARTICLE{SparberSauer:177013, author = {M. Sparber-Sauer and C. Vokuhl and G. Seitz and B. Sorg and M. Tobias$^*$ and T. von Kalle and M. Münter and S. S. Bielack and R. Ladenstein and G. Ljungman and F. Niggli and M. Frühwald and S. Loff and T. Klingebiel and E. Koscielniak}, title = {{I}nfantile myofibromatosis: {E}xcellent prognosis but also rare fatal progressive disease. {T}reatment results of five {C}ooperative {W}eichteilsarkom {S}tudiengruppe ({CWS}) trials and one registry.}, journal = {Pediatric blood $\&$ cancer}, volume = {69}, number = {3}, issn = {1545-5009}, address = {New York, NY}, publisher = {Wiley}, reportid = {DKFZ-2021-02243}, pages = {e29403}, year = {2022}, note = {Volume69, Issue3, March 2022, e29403}, abstract = {Infantile myofibromatosis (IM) is a rare benign soft tissue tumor and often a self-limiting disease but rarely includes life-threatening complications. Little is known about optimal treatment of primary localized (LD) and multifocal disease (MFD).Treatment and outcome of 95 children with IM registered within five Cooperative Weichteilsarkom Studiengruppe (CWS) trials and one registry (1981-2016) were evaluated.LD was diagnosed in 71 patients at a median age of 0.4 years (range 0.0-17.7). MFD was present in 24 patients. The mainstay of treatment was watch-and-wait strategy $(w\&w)$ after initial biopsy or resection. Low-dose chemotherapy (CHT) was administered to 16/71 $(23\%)$ patients with LD and eight of 24 $(33\%)$ patients with MFD, imatinib was added in two. A delayed resection was possible in eight of 71 $(11\%)$ and five of 24 $(21\%)$ patients with LD and MFD, respectively. Overall, patients were alive in complete remission (n = 77) and partial remission (n = 10) at a median follow-up time of 3.4 years after diagnosis (range 0.01-19.4); no data available (n = 5). Three patients died of progressive disease (PD) despite CHT. Gender, tumor size, and location correlated with a favorable event-free survival (EFS) in patients with LD. The 5-year EFS and overall survival of patients with LD were $73\%$ (±12, confidence interval [CI] $95\%)$ and $95\%$ (±6, CI $95\%),$ respectively; for MFD $51\%$ (±22, CI $95\%)$ and $95\%$ (±10, CI $95\%).Prognosis$ is excellent in patients with LD and MFD. Targeted treatment needs to be evaluated for rare fatal PD.}, keywords = {CWS Group (Other) / infantile myofibromatosis (Other) / infants and children (Other) / localized and multifocal disease (Other)}, cin = {C070}, ddc = {610}, cid = {I:(DE-He78)C070-20160331}, pnm = {313 - Krebsrisikofaktoren und Prävention (POF4-313)}, pid = {G:(DE-HGF)POF4-313}, typ = {PUB:(DE-HGF)16}, pubmed = {pmid:34636137}, doi = {10.1002/pbc.29403}, url = {https://inrepo02.dkfz.de/record/177013}, }