PMID- 32543116
OWN - NLM
STAT- MEDLINE
DCOM- 20200907
LR  - 20210825
IS  - 1545-5017 (Electronic)
IS  - 1545-5009 (Print)
IS  - 1545-5009 (Linking)
VI  - 67
IP  - 8
DP  - 2020 Aug
TI  - Treatment and revaccination of children with paraneoplastic 
      opsoclonus-myoclonus-ataxia syndrome and neuroblastoma: The Memorial Sloan 
      Kettering experience.
PG  - e28319
LID - 10.1002/pbc.28319 [doi]
AB  - OBJECTIVE: To review the treatment and revaccination of neuroblastoma-associated 
      opsoclonus-myoclonus-ataxia syndrome (OMAS) patients at Memorial Sloan Kettering 
      Cancer Center (MSK). PROCEDURE: Institutional Review Board approval was obtained 
      for this retrospective study of patients with neuroblastoma-associated OMAS 
      followed at MSK from 2000 to 2016. RESULTS: Fourteen patients (nine female) were 
      9-21 (median 17) months old at diagnosis of neuroblastoma and OMAS syndrome. They 
      had stage 1 (n = 12), stage 2B, or intermediate-risk stage 4. Tumor histology was 
      favorable in 11 patients, unfavorable in two, and unknown in one patient. No 
      patient had amplified MYCN. All patients underwent tumor resection at diagnosis. 
      Anti-neuroblastoma treatment was limited to chemotherapy in one patient. Overall 
      survival is 100% at 3-16 (median 10) years. For OMAS, 13 patients received 
      intravenous immune globulin (IVIg), adrenocorticotropic hormone (ACTH), and 
      rituximab, and one received ACTH and IVIg. Seven patients experienced OMAS 
      relapse. For these relapses, five patients received low-dose cyclophosphamide and 
      two received rituximab. The mean total OMAS treatment was 20-96 (median 48) 
      months. Seven patients started rituximab </=3 months from diagnosis and did not 
      relapse. The other six experienced OMAS relapse. To date, six patients have been 
      revaccinated at a minimum of 2 years after completion of OMAS therapy without 
      OMAS recurrence. CONCLUSIONS: Patients with neuroblastoma-associated OMAS had 
      excellent overall survival. Early initiation of rituximab, IVIg, and ACTH may 
      reduce risks of OMAS relapse. Revaccination can be resumed without exacerbation 
      of OMAS. Further investigation with a larger cohort of patients is needed.
CI  - (c) 2020 Wiley Periodicals, Inc.
FAU - Patel, Ami
AU  - Patel A
AUID- ORCID: 0000-0001-9928-8662
AD  - New York University School of Medicine, New York, New York.
FAU - Fischer, Cheryl
AU  - Fischer C
AD  - Department of Pediatrics, Memorial Sloan Kettering Cancer Center, New York, New 
      York.
FAU - Lin, Yi-Chih
AU  - Lin YC
AD  - Department of Pediatrics, Memorial Sloan Kettering Cancer Center, New York, New 
      York.
FAU - Basu, Ellen M
AU  - Basu EM
AD  - Department of Pediatrics, Memorial Sloan Kettering Cancer Center, New York, New 
      York.
FAU - Kushner, Brian H
AU  - Kushner BH
AD  - Department of Pediatrics, Memorial Sloan Kettering Cancer Center, New York, New 
      York.
FAU - De Braganca, Kevin
AU  - De Braganca K
AD  - Department of Pediatrics, Memorial Sloan Kettering Cancer Center, New York, New 
      York.
AD  - Department of Neurology, Memorial Sloan Kettering Cancer Center, New York, New 
      York.
FAU - Khakoo, Yasmin
AU  - Khakoo Y
AD  - Department of Pediatrics, Memorial Sloan Kettering Cancer Center, New York, New 
      York.
AD  - Department of Neurology, Memorial Sloan Kettering Cancer Center, New York, New 
      York.
AD  - Department of Pediatrics, Weill Medical College of Cornell University, New York, 
      New York.
LA  - eng
GR  - P30 CA008748/CA/NCI NIH HHS/United States
GR  - R25 CA020449/CA/NCI NIH HHS/United States
GR  - R25CA020449/NH/NIH HHS/United States
PT  - Journal Article
PT  - Research Support, N.I.H., Extramural
PT  - Research Support, Non-U.S. Gov't
DEP - 20200615
PL  - United States
TA  - Pediatr Blood Cancer
JT  - Pediatric blood & cancer
JID - 101186624
RN  - 0 (Immunoglobulins, Intravenous)
RN  - 4F4X42SYQ6 (Rituximab)
RN  - 8N3DW7272P (Cyclophosphamide)
RN  - 9002-60-2 (Adrenocorticotropic Hormone)
SB  - IM
MH  - Adrenocorticotropic Hormone/administration & dosage
MH  - Antineoplastic Combined Chemotherapy Protocols/*administration & dosage
MH  - Cyclophosphamide/administration & dosage
MH  - Disease-Free Survival
MH  - Female
MH  - Humans
MH  - Immunoglobulins, Intravenous/administration & dosage
MH  - Infant
MH  - Male
MH  - Neoplasm Staging
MH  - *Neuroblastoma/diagnosis/mortality/therapy
MH  - *Opsoclonus-Myoclonus Syndrome/diagnosis/mortality/therapy
MH  - Retrospective Studies
MH  - Rituximab/administration & dosage
MH  - Survival Rate
PMC - PMC8382509
MID - NIHMS1724686
OTO - NOTNLM
OT  - neuroblastoma
OT  - opsoclonus-myoclonus syndrome
OT  - revaccination
COIS- Conflict of Interest: None
EDAT- 2020/06/17 06:00
MHDA- 2020/09/08 06:00
PMCR- 2021/08/23
CRDT- 2020/06/17 06:00
PHST- 2019/12/09 00:00 [received]
PHST- 2020/03/23 00:00 [revised]
PHST- 2020/03/24 00:00 [accepted]
PHST- 2020/06/17 06:00 [pubmed]
PHST- 2020/09/08 06:00 [medline]
PHST- 2020/06/17 06:00 [entrez]
PHST- 2021/08/23 00:00 [pmc-release]
AID - 10.1002/pbc.28319 [doi]
PST - ppublish
SO  - Pediatr Blood Cancer. 2020 Aug;67(8):e28319. doi: 10.1002/pbc.28319. Epub 2020 
      Jun 15.