Etiology analysis and computed tomography imaging of a tonsillar inflammatory myofibroblastic tumor: report of an immunocompetent patient and brief review
© Luo et al; licensee BioMed Central Ltd. 2012
Received: 4 February 2012
Accepted: 9 March 2012
Published: 9 March 2012
The etiology of Inflammatory myofibroblastic tumor(IMT) is contentious. In this study, we used computed tomography (CT) to examine tonsillar IMT and further analyzed the etiology of this entity.
We presented CT features of left tonsillar IMT and reviewed the English-language literature published between 1984 and 2011.
To our knowledge, there are only six published cases of tonsillar IMT including the present case. Two patients were asymptomatic at initial presentation. Two patients were taking immunosuppressants, and one was pregnant and in an immunomodulated state. CT of our patient revealed a 2.6 × 1.8 cm irregular soft tissue mass between the left tonsil and the base of the tongue. It did not invade surrounding structures and was not enhanced on contrast-enhanced imaging.
Tonsillar IMT may be a benign tumor. We suggest that preoperative recognition of tonsillar IMT by CT may be important to avoid unnecessary expanded surgery.
KeywordsInflammatory myofibroblastic tumor Tonsil Etiology Computed tomography Treatment
Inflammatory myofibroblastic tumor (IMT) is a rare mesenchymal tumor. The etiology of IMT is contentious, and debate exists as to whether IMT is benign or malignant[1–3]. Some IMTs are found to be associated with systemic inflammatory disorders[1, 4], autoimmune diseases, or neoplasms. Its prognosis and behavior are controversial. Most patients with IMT have a good prognosis and can be cured by resection. Recent evidence shows that IMTs may have different etiologies and clinicopathologic features at different sites[3, 7, 8].
Tonsillar IMT is rare; we reviewed the English-literature regarding tonsillar IMT and only five previous reports covered this topic[9–13]. Two of the five patients were taking immunosuppressants, and one was pregnant and in an immunomodulated state. We herein report the sixth case of tonsillar IMT in the English-language literature in an immunocompetent patient and first describe the computed tomography (CT) imaging features of tonsillar IMT.
Clinical Features of IMT Cases in the tonsil
no past medical history of note
A progressive enlarged mass in the left tonsil
2.6 × 1.8
3 m, NED
et al (2011)12
no past medical history of note
pain in the neck region, cough with vomiting, dyspnoea and dyslexia
5.2 × 2.4 × 1.5
(lesion in the left tosil)
et al (2010)13
sore throat, fever and malaise
13 m, NED
Gangopadhyay et al (1997)14
chronic renal failure received a cadaveric renal transplant, HCV+, prednisone, azathioprine, enalapril cyclosporine since renal transplant.
lump in the throat, odynophagia
2 × 2 × 2
10 m, NED
Newman et al
Asthma, retroperitoneal fibrosis,
3.5 × 2.5 × 2
16 m, NED
et al (1984)16
no past medical history of note
an enlarged right tonsil
2.5 × 1.5 × 1
Although debate exists as to whether IMT is a pseudotumor or a neoplasm, the concept of IMT as a neoplasm was recently solidified  with the discovery of cytogenetic aberrations and the subsequent recognition of ALK gene rearrangements on the short arm of the chromosome as a recurrent aberration. However, the etiology, benign or malignant nature, and prognosis of IMT remain unknown and controversial. Our previous reports and other studies revealed that IMTs originating in different organs may lead to diverse etiologies and clinicopathologic features[2, 3, 7, 8]. Etiologies at the other sites may be infectious, traumatic, or autoimmune syndromic. Viruses that are suspected to be involved in the development of IMTs include HIV, HHV-8, and EBV. Some investigators consider it to be an immunologic response to an infectious agent[16, 17]. For tonsillar IMT, two patients were in an immunodepressive state[11, 12] and one was a pregnant woman. The patient with IMT of the tonsil, reported by Newman and Shinn (1995), had been on long-term prednisone for asthma and retroperitoneal fibrosis. Gangopadhyay et al. reported that the patient had received long-term prednisone and other immunosuppressants following cadaveric renal transplant 9 years previously. Magill et al. reported a pregnant woman with IMT in the right tonsil. They suggested that pregnancy is an immunomodulated state, even during the earliest gestational period. The present patient was immunocompetent.
Magill et al. mentioned that CT confirmed the presence of an enlarged right tonsil along with right-sided nodes, but did not supply any CT imaging. Here, we first describe CT imaging of tonsillar IMT. CT revealed a 2.6 × 1.8 cm irregular soft tissue mass between the left tonsil and the base of the tongue that did not invade surrounding structures and was not enhanced on contrast-enhanced imaging. These findings suggested that the mass might have been a benign tumor. The results were confirmed by surgery and pathology. The surgical specimen showed a smooth surface with some small nodules, which lead to the appearance of an irregular soft tissue mass on CT imaging. Thus, preoperative recognition of this benign lesion on CT is important to avoid unnecessary radical resection. Our imaging findings were not similar to those of other IMTs in the head and neck. Most IMTs on contrast-enhanced images show early enhancement, and sinonasal IMTs have a more aggressive appearance. On CT, a moderately enhanced mass is usually seen in orbital IMTs, accompanied by fat infiltration or edema. Our case showed no enhancement on contrast-enhanced imaging and did not involve surrounding tissue.
IMTs arising in mesenteric, omental, peritoneal, pelvic, and retroperitoneal sites, and the maxillary sinus tend to recur, with a potential for metastatic spread in rare instances[2, 3, 20]. In the six tonsillar IMTs reported, there were no recurrences or metastases. This suggests that tonsillar IMTs may be benign and have a good outcome. However, the prognosis, recurrence, and metastatic rate have not been confirmed in a larger series, and there is a lack of long-term follow-up.
The optimal treatment for IMTs at other sites is still controversial, but excision of the tonsils with IMT, either by tonsillectomy or excision with a CO2 laser, seems to be an effective treatment based on the outcome of four tonsillar IMTs[10–12]. Treatment experience will continue to accumulate along with increases in the number of tonsillar IMTs.
Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
Inflammatory myofibroblastic tumor
Human immunodeficiency virus
Polymerase chain reaction., The English in this document has been checked by at least two professional editors both native speakers of English. For a certificate, please see: http://www.textcheck.com/certificate/nSTlES
The immunohistochemical analysis for this studies was supported by Department of Science and Technology of Zhejiang Provincial (contract grant number: 2009 C33026) and Health Bureaue of Zhejiang Provincial (contract grant number:2010KYA062)
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