scholarly journals Unusual Case of Inflammatory Myofibroblastic Tumor in Maxilla

2013 ◽  
Vol 2013 ◽  
pp. 1-4 ◽  
Author(s):  
Jaana Rautava ◽  
Tero Soukka ◽  
Esko Peltonen ◽  
Petri Nurmenniemi ◽  
Markku Kallajoki ◽  
...  
Keyword(s):  
Inflammatory Myofibroblastic Tumor ◽  
Unusual Case ◽  
Root Resorption ◽  
Histopathological Examination ◽  
Smooth Muscle Actin ◽  
Clinical Manifestations ◽  
Anaplastic Lymphoma ◽  
First Case ◽  
Rare Lesion ◽  
Children And Young Adults

Inflammatory myofibroblastic tumor (IMT) is a rare lesion found mostly in children and young adults and originates from the lung, abdominopelvic region, and retroperitoneum. Clinical manifestations of IMT or imaging are nonspecific and diagnosis is based on histopathological and immunohistochemical findings. Minority of all IMTs will metastasize. IMT in the oral cavity is an extreme rarity and this is a first case report of IMT in maxilla causing delayed tooth eruption and multiple cervical root resorption with an 11-year-old child. The IMT reported here was positive for smooth muscle actin, vimentin, and anaplastic lymphoma kinase (ALK1) with immunohistochemistry. Only three IMTs of the jaws have been reported so far and none of them had delayed root eruption and tooth resorption. This unusual case of IMT in a child was also ALK1- positive supporting neoplastic origin of her tumor. The case presented here underscores the importance of histopathological examination of the tissue found in any root resorption especially in the case of multiple resorptions.

scholarly journals Collision Tumor Composed of an Inflammatory Myofibroblastic Tumor and Adenocarcinoma of the Colon: a Rare Entity

2020 ◽  
Vol 29 (3) ◽  
pp. 461-463
Author(s):  
Chiara Cornacchia ◽  
Mariangela Rutigliani ◽  
Fiorenza Belli ◽  
Nicola Romano ◽  
Alberto Serventi ◽  
...  
Keyword(s):  
Inflammatory Myofibroblastic Tumor ◽  
Histopathological Examination ◽  
Clinical Information ◽  
Collision Tumor ◽  
Rare Entity ◽  
Anaplastic Lymphoma ◽  
Tumor Relapse ◽  
Favorable Prognosis ◽  
First Case ◽  
Adenocarcinoma Of The Colon

This report presents the case of an 83-year old man with a collision tumor consisting of an inflammatory myofibroblastic tumor (IMT) and adenocarcinoma of the left colon. As the clinical and radiologic features of IMT are non-specific, only the accurate histopathological examination from the left hemicolectomy specimen was diagnostic. Although the prognosis of a colorectal IMT seemed more favorable than in other sites, four months after surgery the patient developed a tumor relapse. Therefore, malignant behavior of IMT could not be totally excluded. Recent studies have demonstrated that a chromosomal rearrangement involving 2p23, the site of the anaplastic lymphoma kinase (ALK) gene, is present in a subset of these tumors. In our patient, tumor cells did not present ALK-1 perinuclear positivity and it could have indicated a less favorable prognosis. The collision of these different entities is extremely rare and this is the first case reported in literature. Further cases of collision tumors with clinical information including their treatment and prognosis are needed.

scholarly journals Intra-abdominal Solitary Myofibroma in a Child: A Rare Case Report

2021 ◽  
Author(s):  
Smita Singh ◽  
Jyoti Garg ◽  
Kusha Sharma ◽  
Kiran Agarwal
Keyword(s):  
Histopathological Examination ◽  
Smooth Muscle Actin ◽  
Iliac Fossa ◽  
Correct Diagnosis ◽  
Neck Region ◽  
Abdominal Distension ◽  
Malignant Tumours ◽  
Rare Presentation ◽  
Anaplastic Lymphoma ◽  
Abdominal Masses

Intra-abdominal masses in children are usually malignant. Benign tumours at this location are not seen frequently. It is even rarer to find solitary myofibromas intra-abdominally as these tumours are known to have predilection for the head and neck region. We present an unusual case of solitary myofibroma with abdominal localisation in a six-year-old male child who presented with abdominal distension since six months. Computed Tomography (CT) abdomen revealed a complex solid-cystic mass extending from umbilicus to right iliac fossa and measuring 9×6×4 cm. Histopathological examination revealed a tumour displaying biphasic nodular pattern with the presence of lighter staining fascicles of mature myoid cells along with darker staining and more cellular areas of smaller primitive cells. On Immunohistochemistry (IHC), tumour cells were positive for vimentin and Smooth Muscle Actin (SMA) with variable reactivity for desmin while these were negative for CD34 and Anaplastic Lymphoma Kinase 1 (ALK). Based on the histopathological and immunohistochemical findings, final diagnosis of myofibroma was made. Recognition of these lesions is extremely challenging owing to their rare presentation intra-abdominally and also because of their close morphological overlap with other spindle cell tumours commonly found at this site. This case highlights the combined role played by histopathology and IHC in making a clear distinction between different entities. It is imperative for both clinicians and histopathologists to establish the correct diagnosis as excision of the solitary myofibroma is curative in most cases and offers better clinical course than the more commonly found malignant tumours at this site.

scholarly journals Lipoleiomyosarcoma an extremely unusual sarcoma of uterus: A case report

1970 ◽  
Vol 2 (1) ◽  
pp. 67-70 ◽  
Author(s):  
Abhimanyu Jha ◽  
Gita Sayami ◽  
Deepti Adhikari
Keyword(s):  
Smooth Muscle ◽  
Abdominal Pain ◽  
Case Report ◽  
Postmenopausal Woman ◽  
Unusual Case ◽  
Histopathological Examination ◽  
Abdominal Mass ◽  
Lower Abdominal Pain ◽  
First Case ◽  
Well Differentiated

Uterine lipoleiomyosarcoma is a heterologous sarcoma composed of variable proportions of malignant lipoblasts histologically corresponding to well differentiated liposarcoma and malignant smooth muscle cells corresponding to leiomyosarcoma. Finding of benign lipomatous component in a typical leiomyoma (lipoleiomyoma) is not an uncommon, however, lipoleiomyosarcoma is an extremely rare malignant tumor and only very few cases have been reported so far. We report a case of lipoleiomyosarcoma of uterine corpus in a postmenopausal woman presenting with lower abdominal pain and abdominal mass. Diagnosis of lipoleiomyosarcoma was confirmed by histopathological examination of hysterectomy specimen. This is the first case of lipoleiomyosarcoma of uterus reported from Nepal. Keywords: Lipoleiomyosarcoma, uterus, histopathology, unusual case.   doi:10.3126/njog.v2i1.1482    N. J. Obstet. Gynaecol Vol. 2, No. 1, p. 67 - 70 May -June 2007

scholarly journals An Unusual Presentation of Leiomyosarcoma Posing a Diagnostic Dilemma: A Case Report

2020 ◽  
Author(s):  
Smita Singh ◽  
Kusha Sharma ◽  
Vipan Kumar ◽  
Partap Yadav
Keyword(s):  
Smooth Muscle ◽  
Head And Neck ◽  
Histopathological Examination ◽  
Smooth Muscle Actin ◽  
Correct Diagnosis ◽  
Neck Region ◽  
Temporal Region ◽  
Diagnostic Dilemma ◽  
First Case ◽  
Painless Mass

Leiomyosarcoma (LMS) is a malignant tumour of smooth muscle origin commonly seen in genital and gastrointestinal location. However, its presence in the head and neck region in a young child is extremely rare. Authors present a unique case of LMS in a one year and five-month-old child who presented with a painless mass in the right temporal region of the head. Histopathological examination revealed a highly cellular tumour arranged in intersecting fascicles of spindle cells with brisk mitotic activity and interspersed areas of necrosis. On Immunohistochemistry (IHC), the tumour cells were positive for Smooth Muscle Actin (SMA), Desmin and Vimentin and negative for S100 and Myogenin. A diagnosis of LMS was thus, made. Head and neck sarcoma is a broad entity encompassing plethora of differentials with closely overlapping morphological features which renders them diagnostically challenging, this can be resolved by employing various immunohistochemical stains readily available in all laboratories. This case highlights the combined role played by histopathology and immunohistochemistry in arriving at the correct diagnosis. To the best of our knowledge, this is the first case of LMS reported in the temporal region of head and is distinct with respect to its rare incidence, location and age at presentation. LMS at this site may masquerade as deceptively benign painless mass and may not be suspected initially, however one should bear in mind that these are moderate-to-high grade tumours and any delay in management may portend poor prognosis. Timely and aggressive surgical management is thus, the mainstay of treatment and critical to patient survival.

scholarly journals Fatal Tumour Lysis Syndrome Induced by Brigatinib in a Lung Adenocarcinoma Patient Treated With Sequential ALK Inhibitors: A Case Report

2021 ◽  
Vol 12 ◽  
Author(s):  
Yadong Wang ◽  
Tiange Wang ◽  
Jianchao Xue ◽  
Ziqi Jia ◽  
Xinyu Liu ◽  
...  
Keyword(s):  
Lung Adenocarcinoma ◽  
Clinical Manifestations ◽  
Sequential Therapy ◽  
Tumour Lysis Syndrome ◽  
Resistance Mutations ◽  
Laboratory Findings ◽  
Tumour Lysis ◽  
Anaplastic Lymphoma ◽  
First Case ◽  
Days Of Therapy

Tumour lysis syndrome (TLS) represents a group of fatal metabolic derangements resulting from the rapid breakdown of tumour cells. TLS typically occurs soon after the administration of chemotherapy in haematologic malignancies but is rarely observed in solid tumours. Here, we report a case of brigatinib-induced TLS after treatment with sequential anaplastic lymphoma kinase (ALK) inhibitors in a patient with advanced ALK-rearranged lung adenocarcinoma. The patient was treated sequentially with crizotinib, alectinib, and ensartinib. High-throughput molecular profiling after disease progression indicated that brigatinib may overcome ALK resistance mutations, so the patient was administered brigatinib as the fourth-line treatment. After 22 days of therapy, he developed oliguria, fever, and progressive dyspnoea. Clinical manifestations and laboratory findings met the diagnostic criteria for TLS. The significant decrease in the abundance of ALK mutations in plasma indicated a therapeutic response at the molecular level. Consequently, the diagnosis of brigatinib-induced TLS was established. To the best of our knowledge, this is the first case of TLS induced by sequential targeted therapy in non-small cell lung cancer. With the extensive application of sequential therapy with more potent next-generation targeted therapeutic drugs, special attention should be given to this rare but severe complication.

Intraocular Inflammatory Myofibroblastic Tumor With ALK Overexpression

2004 ◽  
Vol 128 (1) ◽  
pp. e5-e7
Author(s):  
Dennis P. O'Malley ◽  
Christopher Poulos ◽  
Magdalena Czader ◽  
Warren G. Sanger ◽  
Attilio Orazi
Keyword(s):  
Tumor Cells ◽  
Inflammatory Myofibroblastic Tumor ◽  
Plasma Cells ◽  
Smooth Muscle Actin ◽  
Chromosome 2 ◽  
Muscle Actin ◽  
Anaplastic Lymphoma ◽  
Spindle Cells ◽  
Multiple Copies

Abstract We report a case of an intraocular inflammatory myofibroblastic tumor nearly filling the vitreous cavity of the eye of a 50-year-old man. The tumor was composed of a mixture of spindle cells and mixed inflammatory elements, including numerous plasma cells. The differential diagnosis included inflammatory pseudotumor and neoplastic mimics of this condition. Further investigation with immunohistochemistry revealed the mass to be composed of myofibroblasts, positive for smooth muscle actin stains and with weak anaplastic lymphoma kinase (ALK) expression in some tumor cells. Evaluation by fluorescence in situ hybridization revealed the tumor cells to have multiple copies of chromosome 2 and ALK but no rearrangement of the ALK gene. The authors propose that multiple copies of the ALK gene may be involved in inflammatory myofibroblastic tumor tumorigenesis, in addition to ALK gene rearrangements.

Anaplastic Lymphoma Kinase‐Positive Inflammatory Myofibroblastic Tumor Mimicking Meningioma: A Case Report

Neurographics ◽  
2021 ◽  
Vol 11 (2) ◽  
pp. 75-79
Author(s):  
J.B. Stone ◽  
R. Stonebridge ◽  
R.D. Bhuta ◽  
J.L. Boxerman
Keyword(s):  
Anaplastic Lymphoma Kinase ◽  
Inflammatory Myofibroblastic Tumor ◽  
Malignant Degeneration ◽  
En Bloc ◽  
Anaplastic Lymphoma ◽  
Intracranial Involvement ◽  
Activating Mutation ◽  
Positive Immunostaining ◽  
Children And Young Adults ◽  
Fibrous Tumor

A 15-year-old girl presented with intermittent nausea and vomiting, headache, and vision changes. MR imaging of the brain revealed an avidly enhancing infratemporal dural-based mass arising from the tentorium, with hyperintensity on T2WI and transdural extension into the posterior cranial fossa. The well-encapsulated fibrous tumor was resected en bloc after cauterization of its rich tentorial arterial supply. Histologic examination demonstrated pleomorphic myofibroblastic cells admixed with an inflammatory infiltrate. Spindle cells showed strong, diffusely positive immunostaining for anaplastic lymphoma kinase, and genomic sequencing uncovered a tropomyosin 3 gene and anaplastic lymphoma kinase fusion and an activating mutation in the Kirsten rat sarcoma oncogene. A diagnosis of inflammatory myofibroblastic tumor was made. Primary intracranial involvement of inflammatory myofibroblastic tumor is exceptionally rare, and few cases that feature an anaplastic lymphoma kinase translocation have been described. Inflammatory myofibroblastic tumor‐CNS is an important differential diagnosis for dural-based lesions in children and young adults due to its propensity for recurrence and malignant degeneration.

scholarly journals A rare cause of chronic dysphagia: pulmonary inflammatory myofibroblastic tumor with distal esophagus invasion

2021 ◽  
Vol 16 (1) ◽  
Author(s):  
Yi-Min Gu ◽  
Long-Qi Chen
Keyword(s):  
Inflammatory Myofibroblastic Tumor ◽  
Distal Esophagus ◽  
Case Presentation ◽  
Contrast Enhanced ◽  
Rare Lesion ◽  
Children And Young Adults ◽  
Enhanced Computed Tomography ◽  
Contrast Enhanced Computed Tomography ◽  
Pulmonary Inflammatory Myofibroblastic Tumor

Abstract Background Inflammatory myofibroblastic tumor (IMT) is rare intermediate tumor, which happens mostly in children and young adults. Case presentation Reported is the successful treatment of a 29-year-old man presented with progressively dysphagia and weight loss. No other abnormal symptoms were observed. The contrast enhanced computed tomography (CT) revealed a dumbbell-shaped lesion between lung and esophagus. Finally, it was pathologically diagnosed as pulmonary IMT invading to the distal esophagus after operation. The patient underwent partial esophagectomy and left lower lobectomy, and was discharged on 10th postoperative day. Conclusions IMT is a rare lesion that usually occurs in the lung, but pulmonary IMT with distal esophagus invasion has not been described previously. Discriminating untypical symptom, completed resection, pathological expertise and closed follow-up will reach the successful diagnosis and treatment.

scholarly journals Inflammatory Myofibroblastic Tumor of the Nasal Septum

2013 ◽  
Vol 2013 ◽  
pp. 1-4 ◽  
Author(s):  
Yuri Okumura ◽  
Kazuhiro Nomura ◽  
Takeshi Oshima ◽  
Atsuko Kasajima ◽  
Takahiro Suzuki ◽  
...  
Keyword(s):  
Nasal Septum ◽  
Inflammatory Myofibroblastic Tumor ◽  
Clinical Presentation ◽  
Smooth Muscle Actin ◽  
Imaging Studies ◽  
Posterior Edge ◽  
Juvenile Angiofibroma ◽  
Anaplastic Lymphoma ◽  
Spindle Cells ◽  
One Year

We report an extremely rare case of inflammatory myofibroblastic tumor of the posterior edge of the nasal septum. An 11-year-old boy presented with frequent epistaxis and nasal obstruction persisting for one year. Based on the clinical presentation and imaging studies, juvenile angiofibroma was suspected, but angiography suggested the possibility of another type of tumor. Transnasal endoscopic surgery found that the tumor protruded into the nasopharynx from the posterior end of the nasal septum. Histological examination identified spindle cells with immunoreaction for vimentin, smooth muscle actin, and anaplastic lymphoma kinase (ALK), but not for desmin and cytokeratin. This is a report of inflammatory myofibroblastic tumor mimicking juvenile angiofibroma. This case suggests that angiography is helpful in the differential diagnosis of epipharyngeal tumor in adolescence.

scholarly journals Intraosseous Inflammatory Myofibroblastic Tumor in the Mandible: A Rare Pathologic Case Report

2014 ◽  
Vol 2014 ◽  
pp. 1-4
Author(s):  
Dale E. Stringer ◽  
Chad N. Allen ◽  
Katina Nguyen ◽  
Rahul Tandon
Keyword(s):  
Case Report ◽  
Rare Case ◽  
Inflammatory Myofibroblastic Tumor ◽  
Strong Association ◽  
Current Mode ◽  
Surgical Removal ◽  
Maxillofacial Region ◽  
Anaplastic Lymphoma ◽  
Rare Case Report ◽  
Rare Lesion

Inflammatory myofibroblastic tumor (IMT) is an extremely rare lesion found in the maxillofacial region. Its frequency diminishes further when found in the bone. Although classification has varied throughout its history, the histologic features are often diagnostic, particularly with its strong association with anaplastic lymphoma kinase-1 (ALK-1) staining. The current mode of treatment for such a lesion is surgical removal with careful followup. In this rare case report, we describe the diagnosis and treatment in a 16-year-old male. Although this rare pathology can present as—and at times mimic—more serious pathologies, it is important for the attending surgeon to initially manage the pathology conservatively.

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