DOI: https://dx.doi.org/10.18203/2349-3291.ijcp20223070
Published: 2022-11-24

Inflammatory myofibroblastic tumour of an unusual site in an infant

Rahul Chidurala, Latha M. Sneha, Balaji Thiruvengadam Kothandan, Ravikumar Aruncahalam, Shivapriya Jeyabalakrishnan, Vasugi Gramani Arumugam, Gokulakrishnan Periyakaruppan

Abstract


Inflammatory myofibroblastic tumors are rare mesenchymal tumors of intermediate malignant potential, usually seen in the first or second decades of life. They have a high local recurrence rate but rarely metastasize. The involvement of paranasal sinuses or the nasal cavity is relatively uncommon, especially in infants. In children, there is a predilection for the abdominal cavity and it is rare in infants. Complete surgical excision is the treatment of choice. We report a case of a 10-month-old girl with an inflammatory myofibroblastic tumor of the left nasal cavity and sinuses who was initially treated with chemotherapy in view of the complicated surgical process and later underwent near-total resection as she was not responding to chemotherapy. Knowledge of such tumors among physicians and ENT surgeons will improve the prognosis, as completely excised tumors have a very low recurrence rate and adjuvant therapy may not be needed.


Keywords


Infant, Mesenchymal tumor, Sinus cavity

Full Text:

PDF

References


Allen CE, Kamdar KY, Bollard CM. Malignant Non-Hodgkin Lymphomas in Children. In: Pizzo PA, Poplack DG, eds. Principles and Practice of Pediatric Oncology. 7th ed. Philadelphia Pa: Lippincott Williams and Wilkins. 2016.

Lopez-Nunez O, John I, Panasiti RN. Infantile inflammatory myofibroblastic tumors: clinicopathological and molecular characterization of 12 cases. Mod Pathol. 2020;33:57690.

Childhood Soft Tissue Sarcoma Treatment (PDQ®)–Health Professional Version. Available at: https://www.cancer.gov/types/soft-tissue-sarcoma/hp/child-soft-tissue-treatment-pdq. Accessed on 25 October, 2022.

Casanova M, Brennan B, Alaggio R. Inflammatory myofibroblastic tumor: The experience of the European pediatric Soft Tissue Sarcoma Study Group (EpSSG). Eur J Cancer. 2020;127:123-9.

Alzate Amaya F, Invencio da Costa LF, Martinez Moran A. Unusual Nasal Insufficiency in an Infant: What's Behind the Nostril? Ann Otol Rhinol Laryngol. 2019;128:1078-80.

Holsinger FC, Hafemeister AC, Hicks MJ. Differential diagnosis of pediatric tumors of the nasal cavity and paranasal sinuses: a 45-year multi-institutional review. Ear Nose Throat J. 2010;89:534-40.

Lazim, NM, Abdullah, B. Multidisciplinary approach to children with sinonasal tumors: A review. Pediatr Invest. 2019;3:173-9.

Van Wyhe RD, Chamata ES, Hollier LH. Midline Craniofacial Masses in Children. Semin Plast Surg. 2016;30:176-80.

Lai LM, McCarville MB, Kirby P. Shedding light on inflammatory pseudotumor in children: spotlight on inflammatory myofibroblastic tumor. Pediatr Radiol. 2015;45:1738-52.

Kube S, Vokuhl C, Dantonello T. Inflammatory myofibroblastic tumors-A retrospective analysis of the Cooperative Weichteilsarkom Studien gruppe. Pediatr Blood Cancer. 2018;65:e27012.

Chun-Yan H, Dong GH, Yang DM. Inflammatory myofibroblastic tumors of the nasal cavity and paranasal sinus: a clinicopathologic study of 25 cases and review of the literature. Eur Arch Otorhinolaryngol. 2015;272:789-97.

Cho Sung-Il, Choi J Y, Do NY. An inflammatory myofibroblastic tumor of the nasal dorsum. J of Pediatr Surg. 2008;43;e35-7.

Mahajan P, Casanova M, Ferrari A. Inflammatory myofibroblastic tumor: molecular landscape, targeted therapeutics, and remaining challenges. Curr Probl Cancer. 2021;45:100768.