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Introduction
Infammatory myofbroblastic tumour (IMT) is a rare pathology of
unknown etiology. It was called infammatory pseudotumor (IPT) until
1998, when the term infammatory myofbroblastic tumour (IMT) was
proposed as being a more descriptive name.
1
IMT and IPT terminologies
are most confusing and interchangeably used due to very numerous
similarities in histological presentations. But despite numerous
similarities in histology/subtle differences such as marked spindle cell
proliferation in IMT and prominent lymphoplasmacytic infltration in
IPT help to histologically differentiate them. Immunohistochemical
markers are invaluable in differentiating both of them. Though there is
no signifcant difference in the clinical standpoint and are considered
synonymous.
2,3
Now it is belong to the group of soft tissue tumours and
have been known by various synonyms; infammatory pseudotumor,
plasma cell granuloma, fbrous histiocytoma and sometimes low
grade sarcoma or infammatory fbrosarcoma.
4−15
It was referred to by
several different terms until the World Health Organization (WHO)
classifed IMT as a distinct entity.
6
The diverse nomenclature is mostly
descriptive and refects the uncertainty regarding its true biologic
nature of these lesions.
7
Therefore IMT is clinico-pathologically
distinctive but biologically controversial entity, which was originally
described as true neoplastic lesion.
5−8
Exact etio-pathogenesis is not
known, though it is considered to be an exaggerated infammatory
reaction to tissue injury of unknown cause.
9
Recently the -concept of
this lesion being reactive has been challenged based on the clinical
demonstration of recurrences and metastasis and further cytogenetic
evidence of acquired clonal chromosomal abnormalities.
10
Moreover
the presence of human herpes virus-8 DNA sequences and the over
expression of interleukin 6 and cyclin D1 have been reported in
IMTs.
11
Then conception of infammatory origin was refuted by some
researchers. Later it was described may arise as an immunologic host
reaction to stimuli such as microorganisms (e.g Epstein –Barr Virus,
Human Herpes Virus-8), foreign bodies or neoplastic tissues, chronic
infammation and may even trauma.
12
Though in some studies found
an association between trauma and IMT that may lead to reactive
infammation has been suggested but causation cannot be proven
completely.
10−12
Therefore whatever the cause a localized derangement
in the immune response after the initial insult may be an underlying
mechanism.
13
IMT is characterized by solitary, well demarcated mass
with fbroblastic or myofbroblastic spindle cell proliferation with
varying degrees of infammatory cell infltration.
1−14
There are three
basic histologic patterns are recognized:
a. Myxoid, vascular, and infammatory areas resembling nodular
fasciitis;
b. Compact spindle cells with intermingled infammatory cells
(lymphocytes, plasma cells, and eosinophils) resembling
fbrous histiocytoma; and Dense plate-like collagen resembling
a desmoid or scar.
These three patterns could overlap, or priority is given to (a)
or (b), and this pathological classifcation method is universally
accepted presently.
12
The differential diagnosis of the head and neck
IMTs should include various benign and malignant spindle cell
proliferations which may pose considerable histological overlap with
IMTs. The main benign entities include nodular fasciitis, fbromatosis,
myofbroma, myofbromatosis, solitary fbrous tumour, benign
fbrous histiocytoma, Wegener’s granulomatosis, neural and smooth
muscle lesions; the malignant differential diagnostic entities include
fbrosarcoma, myofbrosarcoma, low-grade myofbroblastic sarcoma,
malignant fbrous histiocytoma, spindle cell carcinoma, sarcomatoid
carcinoma, leiomyosarcoma, rhabdomyosarcoma, and malignant
peripheral nerve sheath tumour (MPNST). If there was a predominant
J Dent Health Oral Disord Ther. 2017;8(2):476‒480. 476
©2017 Sultana et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which
permits unrestricted use, distribution, and build upon your work non-commercially.
Infammatory myofbroblastic tumour, an unusual
presentation in maxilla and paranasal sinuses: review
of literature and a case report
Volume 8 Issue 2 - 2017
Jachmen Sultana,
1
Abul Bashar,
2
Mohammed
Kamal,
3
Sobhan Morol
1
1
Department of Oral and Maxillofacial Surgery, Dhaka Dental
College and Hospital, Bangladesh
2
Department of Paediatrics, Comilla Medical College and
Hospital, Bangladesh
3
Department of Pathology, Bangabandhu Sheikh Mujib Medical
University, Bangladesh
Correspondence: Jachmen sultana, Department of Oral
and Maxillofacial Surgery, Dhaka Dental College and Hospital,
Bangladesh, Email
Received: June 09, 2017 | Published: September 13, 2017
Abstract
Infammatory myofbroblastic tumours (IMT) are uncommon lesions, rarely affect the head
and neck region. Its etio-pathogenesis and biological behaviour is still uncertain. IMT is just
a reactive process or a true neoplasia is of great controversy. Because of its unpredictable
biological behaviour and histopathological presentation clinically and radiologically it
may simulate a malignant tumour. Though most of the IMT of head and neck shows a
benign course but IMT of paranasal sinuses are highly aggressive in behavior with poor
response to surgery, radiotherapy and chemotherapy, showing multiple recurrences and
fatal outcome. Therefore management of IMT of maxillofacial region is challenging as
there is no established protocol of treatment, but effort on individual IMT demonstrates its
well response on combination regimen of prednisolone, methotrexate and celecoxib acid
(cyclooxygenase 2 inhibitor) responds well and there is virtually total regression of the
lesion. We hereby report a case of IMT in maxilla and paranasal sinuses and a review of its
infammatory versus neoplastic behavior.
Keywords: cyclooxygenase 2 inhibitor, infammatory myofbroblastic tumour, maxillary
swelling, methotrexate, prednisolone, prognosis, treatment
Journal of Dental Health Oral Disorders & Terapy
Case Report
Open Access