Melanotic Neuroectodermal Tumor of Infancy Successfully Treated with Metformin: A Case Report

Overview

Journal Medicine (Baltimore)

Specialty General Medicine

Date 2020 Nov 7

PMID 33157911

Citations 1

Authors

Yu Liang

Ruicheng Tian

Jing Wang

Yuhua Shan

Hongxiang Gao

Chenjie Xie

Jingjing Li

Lei Zhang

Min Xu

Song Gu

Affiliations

Soon will be listed here.

Abstract

Rationale: Melanotic neuroectodermal tumor of infancy (MNTI) is a rare tumor originated from neural crest cells with the potential for recurrence and metastasis. The peak age for the disease is during the first year after birth. The current therapy is primarily surgery. The patient reported here is the first case of MNTI treated with metformin.

Patient Concerns: A case of a 4-month-old infant with a history of swelling in the mouth for 1 month.

Diagnosis: The tumor was diagnosed using radiology, pathology, and immunohistochemistry, and it was performed with complete surgical resection. Unfortunately, the tumor recurred 3 months after surgery.

Interventions: We prescribed metformin for the infant.

Outcomes: Currently, after 9 months of treatment, the tumor is well controlled without apparent side effects.

Lessons: The case presented suggested that metformin may be an underlying therapy for MNTI.

Citing Articles

Inflammatory myofibroblastic tumor successfully treated with metformin: A case report and review of literature.

Liang Y, Gao H, Tian R, Wang J, Shan Y, Zhang L World J Clin Cases. 2021; 9(2):429-435.

PMID: 33521112 PMC: 7812890. DOI: 10.12998/wjcc.v9.i2.429.

References

Pernicova I, Korbonits M . Metformin--mode of action and clinical implications for diabetes and cancer. Nat Rev Endocrinol. 2014; 10(3):143-56. DOI: 10.1038/nrendo.2013.256. View

Amin S, Lux A, OCallaghan F . The journey of metformin from glycaemic control to mTOR inhibition and the suppression of tumour growth. Br J Clin Pharmacol. 2018; 85(1):37-46. PMC: 6303203. DOI: 10.1111/bcp.13780. View

BORELLO E, GORLIN R . Melanotic neuroectodermal tumor of infancy--a neoplasm of neural crese origin. Report of a case associated with high urinary excretion of vanilmandelic acid. Cancer. 1966; 19(2):196-206. DOI: 10.1002/1097-0142(196602)19:2<196::aid-cncr2820190210>3.0.co;2-6. View

Tiwari A, Yadav M . Melanotic Neuroectodermal Tumor of Infancy: A Rare Case Report. Cureus. 2020; 11(12):e6521. PMC: 6991144. DOI: 10.7759/cureus.6521. View

Abdelmonsif D, Sultan A, El-Hadidy W, Abdallah D . Targeting AMPK, mTOR and β-Catenin by Combined Metformin and Aspirin Therapy in HCC: An Appraisal in Egyptian HCC Patients. Mol Diagn Ther. 2017; 22(1):115-127. DOI: 10.1007/s40291-017-0307-7. View

Morales D, Morris A . Metformin in cancer treatment and prevention. Annu Rev Med. 2014; 66:17-29. DOI: 10.1146/annurev-med-062613-093128. View

Ren Q, Chen H, Wang Y, Xu J . Melanotic Neuroectodermal Tumor of Infancy Arising in the Skull and Brain: A Systematic Review. World Neurosurg. 2019; 130:170-178. DOI: 10.1016/j.wneu.2019.07.017. View

Emmerling M, York T, Caccamese J . Melanotic Neuroectodermal Tumor of Infancy: Case Report and Review of Management. J Oral Maxillofac Surg. 2018; 77(2):315-320. DOI: 10.1016/j.joms.2018.09.033. View

Kalcheim C . Neural crest emigration: From start to stop. Genesis. 2018; 56(6-7):e23090. DOI: 10.1002/dvg.23090. View

10.

Wu L, Zhou B, Oshiro-Rapley N, Li M, Paulo J, Webster C . An Ancient, Unified Mechanism for Metformin Growth Inhibition in C. elegans and Cancer. Cell. 2016; 167(7):1705-1718.e13. PMC: 5390486. DOI: 10.1016/j.cell.2016.11.055. View

11.

Wu X, Yeerna H, Goto Y, Ando T, Wu V, Zhang X . Metformin Inhibits Progression of Head and Neck Squamous Cell Carcinoma by Acting Directly on Carcinoma-Initiating Cells. Cancer Res. 2019; 79(17):4360-4370. PMC: 6726568. DOI: 10.1158/0008-5472.CAN-18-3525. View

12.

Gupta R, Gupta R, Kumar S, Saxena S . Melanotic neuroectodermal tumor of infancy: review of literature, report of a case and follow up at 7 years. J Plast Reconstr Aesthet Surg. 2015; 68(3):e53-4. DOI: 10.1016/j.bjps.2014.12.014. View

13.

Pontes F, de Souza L, Uchoa D, Joaquim A, Nascimento L, Rezende D . Melanotic neuroectodermal tumor of infancy of the jaw bones: Update on the factors influencing survival and recurrence. Head Neck. 2018; 40(12):2749-2756. DOI: 10.1002/hed.25514. View

14.

Kheirandish M, Mahboobi H, Yazdanparast M, Kamal W, Kamal M . Anti-cancer Effects of Metformin: Recent Evidences for its Role in Prevention and Treatment of Cancer. Curr Drug Metab. 2018; 19(9):793-797. DOI: 10.2174/1389200219666180416161846. View

15.

Kruse-Losler B, Gaertner C, Burger H, Seper L, Joos U, Kleinheinz J . Melanotic neuroectodermal tumor of infancy: systematic review of the literature and presentation of a case. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2006; 102(2):204-16. DOI: 10.1016/j.tripleo.2005.08.010. View

16.

Rachidi S, Sood A, Patel K, Nguyen S, Hamilton H, Neville B . Melanotic Neuroectodermal Tumor of Infancy: A Systematic Review. J Oral Maxillofac Surg. 2015; 73(10):1946-56. DOI: 10.1016/j.joms.2015.03.061. View

17.

Unsal H, Yalcin M . Melanotic Neuroectodermal Tumor of Infancy in the Maxilla. J Craniofac Surg. 2017; 29(1):e28-e30. DOI: 10.1097/SCS.0000000000003993. View

18.

Coyle C, Cafferty F, Vale C, Langley R . Metformin as an adjuvant treatment for cancer: a systematic review and meta-analysis. Ann Oncol. 2016; 27(12):2184-2195. PMC: 5178140. DOI: 10.1093/annonc/mdw410. View

19.

Saladini S, Aventaggiato M, Barreca F, Morgante E, Sansone L, Russo M . Metformin Impairs Glutamine Metabolism and Autophagy in Tumour Cells. Cells. 2019; 8(1). PMC: 6356289. DOI: 10.3390/cells8010049. View

20.

Ebel F, Thieringer F, Kunz C, Klein-Franke A, Scheinemann K, Guzman R . Melanotic neuroectodermal tumor of infancy to the skull: case-based review. Childs Nerv Syst. 2020; 36(4):679-688. DOI: 10.1007/s00381-020-04509-6. View