Hostname: page-component-cd9895bd7-gxg78 Total loading time: 0 Render date: 2024-12-22T09:14:50.507Z Has data issue: false hasContentIssue false

Inflammatory myofibroblastic tumours of the respiratory tract: paediatric case series with varying clinical presentations

Published online by Cambridge University Press:  12 April 2011

M Brodlie*
Affiliation:
Department of Respiratory Paediatrics, Newcastle upon Tyne Hospitals NHS Foundation Trust, Freeman Hospital, Newcastle upon Tyne, UK Institute of Cellular Medicine, Newcastle University, Newcastle upon Tyne, UK
S C Barwick
Affiliation:
Department of Respiratory Paediatrics, Newcastle upon Tyne Hospitals NHS Foundation Trust, Freeman Hospital, Newcastle upon Tyne, UK
K M Wood
Affiliation:
Department of Cellular Pathology, Newcastle upon Tyne Hospitals NHS Foundation Trust, Royal Victoria Infirmary, Newcastle upon Tyne, UK
M C McKean
Affiliation:
Department of Respiratory Paediatrics, Newcastle upon Tyne Hospitals NHS Foundation Trust, Freeman Hospital, Newcastle upon Tyne, UK
A Welch
Affiliation:
Department of Paediatric ENT, Newcastle upon Tyne Hospitals NHS Foundation Trust, Freeman Hospital, Newcastle upon Tyne, UK
*
Address for correspondence: Dr Malcolm Brodlie, SpR in Respiratory Paediatrics, c/o Paediatric Respiratory Secretaries, Old Children's Outpatients, Royal Victoria Infirmary, Queen Victoria Road, Newcastle upon Tyne NE7 7DN, UK Fax: +44 191 233 1417 E-mail: m.j.brodlie@ncl.ac.uk

Abstract

Objectives:

To highlight the clinical importance of inflammatory myofibroblastic tumours of the respiratory tract in children, and to present a case series of three children which illustrates this tumour's variable clinical presentation.

Case history:

The series includes: a nine-year-old girl with a diagnosis of juvenile idiopathic arthritis, who presented with finger clubbing and was found to have an inflammatory myofibroblastic tumour in her right upper lobe; a 15-year-old adolescent with a left main stem bronchial inflammatory myofibroblastic tumour, who presented with breathlessness and chest pain; and a 12-year-old girl with a tracheal inflammatory myofibroblastic tumour who presented with stridor. In each case, the tumour was resected surgically.

Conclusion:

Inflammatory myofibroblastic tumour are a rare but clinically important and pathologically distinct lesion of the respiratory tract in children. The cases in this series highlight some of the varied clinical presentations of inflammatory myofibroblastic tumours, and illustrate some of this tumour's different anatomical locations within the paediatric respiratory tract.

Type
Clinical Records
Copyright
Copyright © JLO (1984) Limited 2011

Access options

Get access to the full version of this content by using one of the access options below. (Log in options will check for institutional or personal access. Content may require purchase if you do not have access.)

References

1Gleason, BC, Hornick, JL. Inflammatory myofibroblastic tumours: where are we now? J Clin Pathol 2008;61:428–37CrossRefGoogle ScholarPubMed
2Dehner, LP. Inflammatory myofibroblastic tumor: the continued definition of one type of so-called inflammatory pseudotumor. Am J Surg Pathol 2004;28:1652–4CrossRefGoogle ScholarPubMed
3Coffin, CM, Dehner, LP, Meis-Kindblom, JM. Inflammatory myofibroblastic tumor, inflammatory fibrosarcoma, and related lesions: an historical review with differential diagnostic considerations. Semin Diagn Pathol 1998;15:102–10Google ScholarPubMed
4Kovach, SJ, Fischer, AC, Katzman, PJ, Salloum, RM, Ettinghausen, SE, Madeb, R et al. Inflammatory myofibroblastic tumors. J Surg Oncol 2006;94:385–91CrossRefGoogle ScholarPubMed
5Dishop, MK, Kuruvilla, S. Primary and metastatic lung tumors in the pediatric population: a review and 25-year experience at a large children's hospital. Arch Pathol Lab Med 2008;132:1079–103Google Scholar
6Karnak, I, Senocak, ME, Ciftci, AO, Caglar, M, Bingol-Kologlu, M, Tanyel, FC et al. Inflammatory myofibroblastic tumor in children: diagnosis and treatment. J Pediatr Surg 2001;36:908–12Google Scholar
7Janik, JS, Janik, JP, Lovell, MA, Hendrickson, RJ, Bensard, DD, Greffe, BS. Recurrent inflammatory pseudotumors in children. J Pediatr Surg 2003;38:1491–5Google Scholar
8Venizelos, I, Papathomas, T, Anagnostou, E, Tsanakas, J, Kirvassilis, F, Kontzoglou, G. Pediatric inflammatory myofibroblastic tumor of the trachea: a case report and review of the literature. Pediatr Pulmonol 2008;43:831–5Google Scholar
9Ochs, K, Hoksch, B, Frey, U, Schmid, RA. Inflammatory myofibroblastic tumor of the lung in a five-year-old girl. Interact Cardiovasc Thorac Surg 2010;10:805–6CrossRefGoogle Scholar
10Rohrlich, P, Peuchmaur, M, Cocci, SN, Gasselin, ID, Garel, C, Aigrain, Y et al. Interleukin-6 and interleukin-1 beta production in a pediatric plasma cell granuloma of the lung. Am J Surg Pathol 1995;19:590–5Google Scholar
11Breen, DP, Dubus, JC, Chetaille, B, Payan, MJ, Dutau, H. A rare cause of an endobronchial tumour in children: the role of interventional bronchoscopy in the diagnosis and treatment of tumours while preserving anatomy and lung function. Respiration 2008;76:444–8Google Scholar
12Pichler, G, Eber, E, Thalhammer, G, Muntean, W, Zach, MS. Arthralgia and digital clubbing in a child: hypertrophic osteoarthropathy with inflammatory pseudotumour of the lung. Scand J Rheumatol 2004;33:189–91Google Scholar
13Mergan, F, Jaubert, F, Sauvat, F, Hartmann, O, Lortat-Jacob, S, Revillon, Y et al. Inflammatory myofibroblastic tumor in children: clinical review with anaplastic lymphoma kinase, Epstein-Barr virus, and human herpesvirus 8 detection analysis. J Pediatr Surg 2005;40:1581–6CrossRefGoogle ScholarPubMed
14Coffin, CM, Patel, A, Perkins, S, Elenitoba-Johnson, KS, Perlman, E, Griffin, CA. ALK1 and p80 expression and chromosomal rearrangements involving 2p23 in inflammatory myofibroblastic tumor. Mod Pathol 2001;14:569–76Google Scholar
15Hoseok, I, Joungho, H, Ahn, KM, Lee, SI, Jhingook, K. Complete surgical resection of inflammatory myofibroblastic tumor with carinal reconstruction in a 4-year-old boy. J Pediatr Surg 2005;40:e23–5Google Scholar
16Przkora, R, Bolder, U, Schwarz, S, Jauch, KW, Spes, J, Andreesen, R et al. Regression of nonresectable inflammatory myofibroblastic tumours after treatment with nonsteroidal anti-inflammatory drugs. Eur J Clin Invest 2004;34:320–1CrossRefGoogle ScholarPubMed
17Sacco, O, Gambini, C, Gardella, C, Toma, P, Rossi, UG, Jasonni, V et al. “Atypical steroid response” in a pulmonary inflammatory myofibroblastic tumor. Pediatr Pulmonol 2010;45:721–6Google Scholar
18Morotti, RA, Legman, MD, Kerkar, N, Pawel, BR, Sanger, WG, Coffin, CM. Pediatric inflammatory myofibroblastic tumor with late metastasis to the lung: case report and review of the literature. Pediatr Dev Pathol 2005;8:224–9Google Scholar