Inoperable inflammatory myofibroblastic tumour of the para-nasal sinuses and orbit with recurrence responding to methotrexate and prednisolone: a case report

Navinan, Mitrakrishnan Rayno; Liyanage, Isurujith; Herath, Sandamalee; Yudhishdran, Jevon; Shivanthan, Chrishan; Beneragama, Dulani; Kulatunga, Aruna

doi:10.1186/s13104-015-0993-3

Cited by 6 publications

(9 citation statements)

References 26 publications

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“…Any local recurrence or aggressive behavior of IMT is attributable to an incomplete resection of the mass. 3,7,15 In the present case, it was determined that surgical excision would prove to be too risky to attempt. In such non-resectable cases corticosteroid therapy and chemotherapy have been suggested as a good alternative.…”

Section: Discussionmentioning

confidence: 75%

“…In HPE of our patient and in other case reports, the prominent cell type has been spindle cells. 3 This tumor also invariably shows positive immunoreactivity for SMA, vimentin, and calponin. 2,3,7,14 Surgical excision of the tumor is the treatment of choice of IMT.…”

Section: Discussionmentioning

confidence: 87%

“…3 This tumor also invariably shows positive immunoreactivity for SMA, vimentin, and calponin. 2,3,7,14 Surgical excision of the tumor is the treatment of choice of IMT. In orbital tumors it may not be possible to completely excise the tumor.…”

Section: Discussionmentioning

confidence: 87%

“…2 The World Health Organization has defined IMT as "a tumor composed of differentiated myofibroblastic spindle cells usually accompanied by numerous plasma cells and/or lymphocytes." [1][2][3][4][5] It commonly occurs in children and young adults, with no sexual or racial predilection. 6,7 Due to its prevalence in pediatric patients, a differential diagnosis of rhabdomyosarcoma should be kept in mind.…”

Section: Discussionmentioning

confidence: 99%

“…[2][3][4]12 Radiotherapy is used for recurrence or unresectable tumors. Usually high doses are required which may prove a limiting factor.…”

Section: Discussionmentioning

confidence: 99%

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Inflammatory Myofibroblastic Tumor—A Case Report

Lau¹,

Ahmad²,

Farhad³

et al. 2016

US Ophthalmic Review

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Inflammatory myofibroblastic tumors (IMTs) are rarely reported, benign tumors. They are uncommon in the ocular structures. A four-year-old girl presented to us with bilateral proptosis. Subsequent radiologic and histopathologic investigations proved it to be bilateral IMT. This case report is being presented because of its rarity and some of the unique features of the condition seen in our patient. Case ReportA four-year-old, apparently healthy girl presented to Queen Elizabeth Hospital, Kota Kinabalu two years ago, with a gradually progressive proptosis, lagophthalmos, and watering from her right eye for the past eight months, as noted by her mother. There was no blurring of vision, diplopia, pain, or redness noted. There was no history of trauma, nor symptoms suggestive of malignancy, thyroid abnormalities, connective tissue disease, or infection. She was well and active at home. There was also no family history of malignancy reported.Examination of the child revealed an active girl with no features of dysmorphism. The visual acuities were 6/6 in both eyes. The conjunctivae in both eyes were mildly injected. The rest of the anterior, as well as posterior, segments were normal bilaterally. She was noted to have bilateral non-axial proptosis, which was worse on the right eye. The right eye was deviated downwards and inwards. Exophthalmometer measurements at 95 mm were 18 mm on the right eye and 16 mm on the left eye. No lagophthalmos or exposure keratopathy were present.Her extraocular movements were normal. There was no relative afferent pupillary defect noted (Figures 1 and 2). AbstractThis case report highlights a 4-year old child with recurrent bilateral proptosis. Investigations revealed the presence of inflammatory myofibroblastic tumors in both eyes. These are rarely reported benign tumors. Our case showed unusual features such as orbital involvement, bony erosion and response to corticosteroids. KeywordsGranuloma, plasma cell, orbital diseases, orbital neoplasms Disclosure: Lindfay Laura Lau, Syed Shoeb Ahmad, Anis Farhad, and Shuaibah Abdul Ghani have nothing to declare in relation to this article. No funding was received in the publication of this article.Compliance with Ethics: All procedures were followed in accordance with the responsible committee on human experimentation and with the Helsinki Declaration of 1975 and subsequent revisions, and informed consent was received from the parent/guardian of the patient involved in this case study.

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Section: Discussionmentioning

confidence: 75%

Section: Discussionmentioning

confidence: 87%

Section: Discussionmentioning

confidence: 87%

Section: Discussionmentioning

confidence: 99%

“…[2][3][4]12 Radiotherapy is used for recurrence or unresectable tumors. Usually high doses are required which may prove a limiting factor.…”

Section: Discussionmentioning

confidence: 99%

See 3 more Smart Citations

Inflammatory Myofibroblastic Tumor—A Case Report

Lau¹,

Ahmad²,

Farhad³

et al. 2016

US Ophthalmic Review

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Inflammatory Myofibroblastic Tumors in Paranasal Sinus and Nasopharynx: A Clinical Retrospective Study of 13 Cases

Zhu

Zha

Wang

et al. 2018

BioMed Research International

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Background Inflammatory myofibroblastic tumor (IMT), as a mesenchymal tumor, is common in the lung and abdomen but rare in the paranasal sinus and nasopharynx. Objective This study aimed to summarize the clinical characteristics of IMT in the paranasal sinus and nasopharynx and analyze the relationship between the treatment and the overall survival (OS). Method The clinical features, treatment, and follow-up data of patients diagnosed with IMT of the paranasal sinus or nasopharynx from 2006 to 2017 were retrospectively analyzed, and the previous literature was reviewed. Results IMT often presents as an ill-defined soft-tissue mass with bone destruction and invasion of surrounding structures. The treatment methods used in this study were different combinations of surgery, prednisone, radiotherapy, and chemotherapy or observation alone. Three of the 13 patients were lost and the follow-up time of the remaining 10 cases ranged from 2 to 87 months (median, 39 months). Two patients died of the disease; the other eight patients were stable. The 5-year survival rate was 72%. Among the four methods of treatment, only treatment with prednisone was significantly correlated with better OS (P = 0.046). Conclusions IMT is an intermediate tumor that often mimics malignancy. We are not sure if IMTs in the nasal cavity are more aggressive because of the biology or if the location and local therapy in the head region is more complicated. Radiologic findings help know the extent of the lesion. For unresectable nasal IMT, combined therapy with glucocorticoids, chemotherapy, and radiotherapy is sometimes a better choice. Glucocorticoids are especially recommended as a basic part of the integrated therapy. However, the standard treatment needs further research.

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Inflammatory myofibroblastic tumor directly invading the right first rib treated with oral steroids: a case report

Watanabe,

Ano,

Kikuchi

et al. 2024

BMC Pulm Med

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Background We present a case of an inflammatory myofibroblastic tumor cured with a short period of steroid administration, a treatment previously unreported for such cases. Case presentation A 49-year-old man had a chief complaint of chest pain for more than 3 days. Computed tomography (CT) revealed a tumoral lesion suspected to have infiltrated into the right first rib and intercostal muscles, with changes in lung parenchymal density around the lesion. The maximal standardized uptake value on 18 F-fluorodeoxyglucose positron emission tomography/computed tomography was high (16.73), consistent with tumor presence. CT-guided biopsy revealed an inflammatory myofibroblastic tumor with no distant metastases. Surgery was indicated based on the disease course. However, he had received an oral steroid before the preoperative contrast-enhanced CT scan due to a history of bronchial asthma, and subsequent CT showed that the tumor shrank in size after administration; he has been recurrence-free for more than a year. Conclusions Surgery is still the first choice for inflammatory myofibroblastic tumors, as the disease can metastasize and relapse; however, this condition can also be cured with a short period of steroid therapy.

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Inoperable inflammatory myofibroblastic tumour of the para-nasal sinuses and orbit with recurrence responding to methotrexate and prednisolone: a case report

Cited by 6 publications

References 26 publications

Inflammatory Myofibroblastic Tumor—A Case Report

Inflammatory Myofibroblastic Tumor—A Case Report

Inflammatory Myofibroblastic Tumors in Paranasal Sinus and Nasopharynx: A Clinical Retrospective Study of 13 Cases

Inflammatory myofibroblastic tumor directly invading the right first rib treated with oral steroids: a case report

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