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@ARTICLE{Zhang:274467,
author = {G.-J. Zhang and X.-J. Ma$^*$ and Y.-P. Zhang and L.-F. Hao
and L. Wang and J.-T. Zhang and Z. Wu and D. Li},
title = {{S}urgical management and outcome of primary intracranial
{R}osai-{D}orfman disease: a single-institute experience and
pooled analysis of individual patient data.},
journal = {Neurosurgical review},
volume = {46},
number = {1},
issn = {0344-5607},
address = {New York},
publisher = {Springer},
reportid = {DKFZ-2023-00612},
pages = {76},
year = {2023},
note = {DKFZ-ZMBH Alliance},
abstract = {Primary intracranial Rosai-Dorfman disease (PIRDD) is
considered a nonmalignant nonneoplastic entity, and the
outcome is unclear due to its rarity. The study aimed to
elaborate the clinic-radiological features, treatment
strategies, and progression-free survival (PFS) in patients
with PIRDD. Patients with pathologically confirmed PIRDD in
our institute were reviewed. Literature of PIRDD, updated
until December 2019, was systematically searched in 7
databases (Embase, PubMed, Cochrane database, Web of
Science, Wanfang Data Knowledge Service Platform, the VIP
Chinese Science and Technology Periodical Database (VIP),
and the China National Knowledge Infrastructure (CNKI)).
These prior publication data were processed and used
according to the Preferred Reporting Items for Systematic
Reviews and Meta-Analyses (PRISMA) guidelines.
Clinical-radiological characteristics and adverse factors
for PFS were evaluated in the pooled cohort. The pooled
cohort of 124 cases (81 male and 43 female), with a mean age
of 39.7 years, included 11 cases from our cohort and 113
cases from 80 prior studies. Twenty-nine patients $(23.4\%)$
had multiple lesions. Seventy-four patients $(59.7\%)$
experienced gross total resection (GTR), 50 patients
$(40.3\%)$ had non-GTR, 15 patients $(12.1\%)$ received
postoperative adjuvant radiation, and 23 patients $(18.5\%)$
received postoperative steroids. A multivariate Cox
regression revealed that GTR (HR = 4.52; $95\%$ CI
1.21-16.86; p = 0.025) significantly improved PFS, and
multiple lesions (p = 0.060) tended to increase the hazard
of recurrence. Neither radiation (p = 0.258) nor steroids (p
= 0.386) were associated with PFS. The overall PFS at 3, 5,
and 10 years in the pooled cohort was $88.4\%,$ $79.4\%,$
and $70.6\%,$ respectively. The PFS at 5 and 10 years in
patients with GTR was $85.4\%$ and $85.4\%,$ respectively,
which was $71.5\%$ and $35.8\%,$ respectively, in patients
without GTR. Gross total resection significantly improved
PFS and was recommended for PIRDD. Radiation and steroids
were sometimes empirically administered for residual,
multiple, or recurrent PIRDD, but the effectiveness remained
arguable and required further investigation.Systematic
review registration number: CRD42020151294.},
keywords = {Primary intracranial Rosai-Dorfman disease (Other) /
Radiotherapy (Other) / Steroid therapy (Other) / Surgical
resection (Other)},
cin = {A240},
ddc = {610},
cid = {I:(DE-He78)A240-20160331},
pnm = {311 - Zellbiologie und Tumorbiologie (POF4-311)},
pid = {G:(DE-HGF)POF4-311},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:36967440},
doi = {10.1007/s10143-023-01983-9},
url = {https://inrepo02.dkfz.de/record/274467},
}