Metastatic NUT Midline Carcinoma Treated With Aggressive Neoadjuvant Chemotherapy, Radiation, and Resection: A Case Report and Review of the Literature.


Journal

Journal of pediatric hematology/oncology
ISSN: 1536-3678
Titre abrégé: J Pediatr Hematol Oncol
Pays: United States
ID NLM: 9505928

Informations de publication

Date de publication:
01 2021
Historique:
pubmed: 20 6 2020
medline: 7 4 2021
entrez: 20 6 2020
Statut: ppublish

Résumé

NUT midline carcinoma, characterized by the rearrangement of the nuclear protein in testis (NUTM1) gene, is a rare and aggressive subtype of squamous cell carcinoma. This disease is rarely cured and there have been no reports of cure in patients with distant metastatic disease. In fact, patients typically succumb to NUT midline carcinoma within 6 to 12 months from diagnosis. The authors report on a single patient who presented widely metastatic disease who has now been in remission for 37 months after multimodal therapy with compressed cycles of vincristine, cyclophosphamide, and doxorubicin alternating with ifosfamide and etoposide, high-dose radiation, and postchemotherapy resection.

Identifiants

pubmed: 32555032
doi: 10.1097/MPH.0000000000001860
pii: 00043426-202101000-00026
doi:

Substances chimiques

NUTM1 protein, human 0
Neoplasm Proteins 0
Nuclear Proteins 0

Types de publication

Case Reports Journal Article Review

Langues

eng

Sous-ensembles de citation

IM

Pagination

e73-e75

Références

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Bauer DE, Mitchell CM, Strait KM, et al. Clinicopathologic features and long-term outcomes of NUT midline carcinoma. Clin Cancer Res. 2012;18:5773–5779.
Chau NG, Hurwitz S, Mitchell CM, et al. Intensive treatment and survival outcomes in NUT midline carcinoma of the head and neck. Cancer. 2016;122:3632–3640.
Lemelle L, Pierron G, Fréneaux P, et al. NUT carcinoma in children and adults: a multicenter retrospective study. Pediatr Blood Cancer. 2017. Doi: 10.1002/pbc.26693.
doi: 10.1002/pbc.26693
Giridhar P, Mallick S, Kashyap L, et al. Patterns of care and impact of prognostic factors in the outcome of NUT midline carcinoma: a systematic review and individual patient data analysis of 119 cases. Eur Arch Otorhinolaryngol. 2018;275:815–821.
Mertens F, Wiebe T, Adlercreutz C, et al. Successful treatment of a child with t(15;19)-positive tumor. Pediatr Blood Cancer. 2007;49:1015–1017.
Storck S, Kennedy AL, Marcus KJ, et al. Pediatric NUT-midline carcinoma: therapeutic success employing a sarcoma based multimodal approach. Pediatr Hematol Oncol. 2017;34:231–237.
Womer RB, West DC, Krailo MD, et al. Randomized controlled trial of interval-compressed chemotherapy for the treatment of localized Ewing sarcoma: a report from the Children’s Oncology Group. J Clin Oncol. 2012;30:4148–4154.
Filippakopoulos P, Qi J, Picaud S, et al. Selective inhibition of BET bromodomains. Nature. 2010;468:1067–1073.
Schwartz BE, Hofer MD, Lemieux ME, et al. Differentiation of NUT midline carcinoma by epigenomic reprogramming. Cancer Res. 2011;71:2686–2696.
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O’Dwyer PJ, Piha-Paul SA, French C, et al. Abstract CT014: GSK525762, a selective bromodomain (BRD) and extra terminal protein (BET) inhibitor: results from part 1 of a phase I/II open-label single-agent study in patients with NUT midline carcinoma (NMC) and other cancers. Cancer Res . 2016;76(suppl 14):CT014.
Haack H, Johnson LA, Fry CJ, et al. Diagnosis of NUT midline carcinoma using a NUT-specific monoclonal antibody. Am J Surg Pathol. 2009;33:984–991.
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French CA. NUT midline carcinoma. Cancer Genet Cytogenet. 2010;203:16–20.
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Le Loarer F, Pissaloux D, Watson S, et al. Clinicopathologic features of CIC-NUTM1 sarcomas, a new molecular variant of the family of CIC-fused sarcomas. Am J Surg Pathol. 2019;43:268–276.

Auteurs

Jenna Sopfe (J)

Department of Pediatrics, Center for Cancer and Blood Disorders.

Brian Greffe (B)

Department of Pediatrics, Center for Cancer and Blood Disorders.

Amy L Treece (AL)

Department of Pathology, University of Colorado School of Medicine, Aurora, CO.

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