Inflammatory myofibroblastic tumor of the lacrimal gland: case report of an exceptional location.
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ABSTRACT:
SUBMITTER: Boudhas A
PROVIDER: S-EPMC5561625 | biostudies-other | 2017
REPOSITORIES: biostudies-other
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Project description:•This is the first case report of inflammatory myofibroblastic tumor in the literature to present with extrauterine disease.•A prompt work-up of symptoms may have precluded a tumor debulking procedure.
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Project description:IntroductionInflammatory myofibroblastic tumor is a benign entity that may present as a locally aggressive malignancy, predominantly in the lung. 500 cases have been reported in the literature, and an estimated prevalence ranges from 0.04% to 0.7%.Case presentationAn eighteen-year old male presented to the surgical clinic with abdominal pain. The pain was recurring despite trials of analgesics and remained undiagnosed. Radiological imaging demonstrated a lesion in the spleen. An infectious cause was presumed due to their endemicity in South Asia, however pathology showed an inflammatory myofibroblastic tumor.DiscussionPre-operative imaging is yet to develop a set criterion that may identify this lesion, though clinicians may be clued in by the benign appearance despite the large size. Usual diagnosis is on pathology after complete surgical excision, which is the mainstay treatment advocated.ConclusionReporting of the tumor in unusual sites such as the spleen is scarce, increase of which may help establish guidelines, understand tumor behavior and guide clinicians that may encounter it in surgical practice.
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Project description:IntroductionInflammatory pseudotumor encompasses a broad range of non-neoplastic and neoplastic entities, including inflammatory myofibroblastic tumors (IMTs). Because it is a rare mesenchymal tumor of unknown etiology and pathogenesis, and its clinical symptoms and radiologic features are not distinctive, intracranial IMT could be misdiagnosed as other extra-axial tumors. Here, we present a case of intracranial IMT suspected to be a brain abscess.Case presentationIn this case, a 73-year-old woman presented headaches, nausea, and vertigo. Brain computed tomography (CT) and magnetic resonance imaging showed 4 × 3 cm sized oval rim-enhanced lesion on the left cerebellopontine angle. Considering the patient's history of otitis media and CT findings, we hypothesized that this lesion was a chronic brain abscess. The initial burr hole drain surgery was unsuccessful because there was no abscess, leading to a second radical excision surgery. Histopathological and immunohistochemical analyses eventually revealed a final diagnosis of intracranial IMT.ConclusionIntracranial IMT is a rare disease with unknown pathogenesis. Diagnosis primarily depends on histopathological and immunohistochemistry analyses. As observed in our case, this disease may be mistaken for meningiomas, solitary fibrous tumors, or chronic abscesses due to its rare occurrence.
| S-EPMC11250673 | biostudies-literature
Project description:IntroductionAn Inflammatory myofibroblastic tumor, a neoplasm of intermediate biological potential, of the small bowel mesentery, is a rare tumor, most commonly reported in but not confined to the pediatric age group.Case presentationThis case report underlines a case of a (small bowel) mesentery IMT in an adult female presenting with recurrent symptoms similar to acute appendicitis.DiscussionIt can present with symptoms similar to acute appendicitis necessitating a high index of suspicion for its prompt diagnosis. Treatment primarily includes surgical resection with recent advances in targeted therapy with tyrosine kinase inhibitors showing promising results.ConclusionIMTs can present as clinical as well as histopathological mimickers of a variety of diseases especially in the abdomen. Prompt diagnosis requires both imaging and histopathological examination.
| S-EPMC9486733 | biostudies-literature
Project description:BackgroundInflammatory myofibroblastic tumor (IMT) is a rare central nervous system (CNS) tumor. We first report a rare case of IMT in the lateral ventricle and describe the magnetic resonance imaging (MRI) findings of the tumor with an emphasis on the advanced MRI features.Case presentationA 49-year-old female patient with headaches and blurred vision for 2 months. Brain MRI revealed a well-circumscribed, lobulated mass occupying the left lateral ventricle trigone, with marked perilesional brain edema. The tumor showed heterogeneous significant hyperintensity on T2-weighted imaging (T2WI) and hypointensity on T1-weighted imaging (T1WI). After the administration of gadolinium, the mass exhibited marked contrast enhancement and the halo sign was observed. On advanced MRI, the lesion showed decreased perfusion on perfusion MRI and reduced diffusion on diffusion-weighted imaging (DWI). On susceptibility-weighted imaging (SWI), there was a punctate low signal intensity in the tumor. The patient underwent surgical resection of the mass and a pathological examination confirmed the lesion to be an inflammatory myofibroblastic tumor with negative expression of anaplastic lymphoma kinase (ALK). This patient had remained healthy without evidence of recurrence during a 20-month follow-up.ConclusionsOn MRI, marked perilesional brain edema, significant hyperintensity on T2WI, hypoperfusion on perfusion MRI but with an obvious enhancement, no diffusion restriction on DWI, and halo sign may be the characteristic findings of intraventricular IMT. The advanced MRI characteristics could provide abundant information to reflect the histological features and physiological metabolic characteristics of the tumor.
| S-EPMC9717528 | biostudies-literature
Project description:The term inflammatory myofibroblastic tumor more commonly referred to as "pseudostumor ", denotes a pseudosarcomatous inflammatory lesion that contains spindle cells, myofibroblasts, plasma cells, lymphocytes and histiocytes. It exhibits a variable biological behavior that ranges from frequently benign lesions to more aggressive variants. Inflammatory myofibroblastic tumor (IMT) of the stomach is extremely rare and its prognosis is unpredictable. We present a 45-year-old diabetic man with a gastric Inflammatory myofibroblastic tumor. The histopathological and immunohistochemical analysis was the key to reach diagnosis.
| S-EPMC3964230 | biostudies-literature
Project description:IntroductionInflammatory myofibroblastic tumor (IMT) is a rare entity, classified within soft tissue sarcomas. It is an intermediate malignancy tumor, which seldom presents as metastatic disease. The treatment of choice is surgery, except in cases where surgery is not possible due to localization or if it presents with metastatic disease. Approximately 50% of IMTs will exhibit ALK translocation, providing a therapeutic target for these patients.Case presentationA case is presented of a patient with metastatic IMT in complete response to treatment with alectinib, maintained for over 4 years.ConclusionThis case showed a long time complete response in patient with IMT treated with alectinib.
| S-EPMC11324221 | biostudies-literature
Project description:IntroductionInflammatory myofibroblastic tumor (IMT) of the trachea is rare tumor mostly found in children and young adults.Case reportWe report a case of a 28 year-old woman who presented chronic isolated coughing. Chest CT scan showed a tracheal tumor. Rigid bronchoscopy allowed the complete removal of the tumor, and histology confirmed the diagnosis of IMT. 12 months follow-up found no recurrence.DiscussionIMT is a rare tumor exhibiting both benign and aggressive behaviour. The endoscopic approach of tracheal should be considered when there is a minimal tracheal wall invasion.ConclusionThrough this case, we want to emphasise the role of rigid bronchoscopy in the complete removal of endotracheal IMT.
| S-EPMC8767228 | biostudies-literature
Project description:BackgroundInflammatory myofibroblastic tumor (IMT) is a rare disease that mainly involves the lung and the abdomen. The gold standard of the IMT treatment is radical surgery, while chemotherapy and radiotherapy are represented usually for unresectable lesions. Anaplastic lymphoma kinase (ALK) rearrangements are present in approximately 50% of IMT patients, and several clinical trials of ALK tyrosine kinase inhibitors (TKIs) in the treatment of ALK-positive IMT patients are underway.Case descriptionWe reported a case of IMT in the right pelvic cavity. Initially, the patient underwent resection of multiple lesions. Unfortunately, the patient's tumor recurred half a year later, and enhanced computerized tomography (CT) of the whole abdomen revealed multiple low-density masses. Then the patient underwent resection of the recurrent tumors. Immunohistochemical staining exhibited the expression of ALK in the tumor cells, and next-generation sequencing (NGS) technology revealed two novel ALK fusions, ALK-ribosome binding protein 1 (RRBP1) and hydroxyacid oxidase 1 (HAO1)-ALK fusions. These fusions were able to be transcribed and captured by RNA level. And the two fusions have not been reported in the IMTs.ConclusionsThis case expanded the range of ALK fusion types and provided a promising molecular-targeted treatment strategy. In addition, the two novel ALK fusions may be the recurrent oncogenic mechanism in clinically aggressive IMT.
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Project description:The present study reported on the histomorphological observations and immunohistochemical features of five cases of gastric inflammatory myofibroblastic tumor (IMT). Loosely arranged fat fusiform myofibroblast-fibroblasts and diffusely or patchily distributed inflammatory cells, which formed a diverse morphological structure, were observed. In the mucous vascular structure, mucoid or collagenous areas, fibromatosis- or scar-like lesions were generally <10 mm in size and both had diffuse or patchy plasma cells, lymphocytes and other inflammatory-cell infiltration backgrounds. The immunophenotype was vimentin- and smooth muscle actin-positive with pan-cytokeratin, desmin and calponin expression and CD34-positive foci; furthermore, three cases were positive for anaplastic lymphoma kinase expression. Gastric IMT is rare, with unique histopathological changes and corrosion-like invasion of the smooth muscle of the stomach wall, blood vessels, nerves and adipose tissue. It should be differentiated from a variety of spindle cell tumor types and tumor-like lesions.
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