ALK inhibition in two emblematic cases of pediatric inflammatory myofibroblastic tumor: Efficacy and side effects

Brivio, Erica; Zwaan, C. Michel

doi:10.1002/pbc.27645

Cited by 31 publications

(12 citation statements)

References 14 publications

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“…Recently, ALK directed therapy, as an alternative neo-adjuvant therapy, has been developed when the standard surgical treatment is not feasible. Furthermore, partial therapeutic effect on IMT has been described in previous studies (20)(21)(22). Nevertheless, the resistance to ALK inhibitor crizotinib appeared about 6 months after the initiation of the treatment when the patients had IMT with ALK rearrangement (20).…”

Section: Discussionmentioning

confidence: 81%

Inflammatory Myofibroblastic Tumor of the Sigmoid Colon in an Infant: A Case Report

wu¹,

Meng²,

Sun³

et al. 2021

Preprint

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Background: Inflammatory myofibroblastic tumor (IMT), an infrequent mesenchymal lesion composed of myofibroblastic and fibroblastic spindle cells with inflammatory infiltration, mostly occurred in pediatric patients. IMT has been reported in a number of locations throughout the body. However, the cases occurred in the gastrointestinal tract, especially those involving the sigmoid colon in pediatric patients, are very rare and even have not been reported. Here we present a case of a large IMT originating from the sigmoid colon and invading the omentum and the surrounding tissues in an 11-month-old boy. Case presentation: The patient is an 11-month-old boy initially presenting with symptoms of fever and vomiting. Abdominal ultrasound revealed an inhomogeneous echo with a size of 9.9*4.6 cm in the left lower abdomen. Abdomen computed tomography (CT) scanning confirmed that a mass with a size of 10*8*6 cm located in the left middle and lower abdomen. Post-operative pathological examination confirmed the histopathologic diagnosis of IMT. Complete resection of the tumor along with the involved colon segments were performed and followed by end-to-end colon anastomosis. The boy recovered well without postoperative complications or recurrence during one-year follow-up. Conclusions: Colon IMT is extremely rare; its etiology and pathogeny are unclear. Imaging examination may provide reliable evidence for determining IMT. However, postoperative histopathologic examination is also important for the final diagnosis. Complete tumor excision, accompanied by rare recurrence and satisfying survival, is the best therapeutic approach at present. This case report may complete the clinical presentation of IMT by reminding physicians not to ignore the possibility of IMT in the colon.

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Section: Discussionmentioning

confidence: 81%

Inflammatory Myofibroblastic Tumor of the Sigmoid Colon in an Infant: A Case Report

wu¹,

Meng²,

Sun³

et al. 2021

Preprint

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“…Based on the importance of the ALK pathway in the progression of IMT, ALK-targeted inhibitors, such as crizotinib, which are mostly used in the treatment of patients with metastatic or unresectable ALK-positive IMT, are effective and even offer surgical opportunities for these patients[32]. However, tumor cells may acquire resistance during the treatment process, which may limit their efficacy[33]. Anti-inflammatory therapy, such as nonsteroidal anti-inflammatory drugs (NSAIDs), is reported to be effective in ALK-negative patients for whom ALK inhibition is not an option[34].…”

Section: Discussionmentioning

confidence: 99%

Kidney inflammatory myofibroblastic tumor masquerading as metastatic malignancy: A case report and literature review

Zhang¹,

Guo²,

Liang³

et al. 2019

WJCC

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BACKGROUNDInflammatory myofibroblastic tumor (IMT) is a rare mesenchymal tumor that is characterized by spindle cells differentiated from muscle fibroblasts and infiltration of various types of inflammatory cells. IMT can occur at any age and at any anatomic site. The most common location of IMT is the bladder in the genitourinary tract. Only scarce cases of kidney IMT have been reported in the literature.CASE SUMMARYA 77-year-old woman, with a history of bilateral renal calculus for 15 years, was admitted to the Department of Urology of our hospital complaining of recurrent painless gross hematuria for one month. The treatment with cephalosporin was ineffective. Computed tomography imaging showed a mixed density and slightly heterogeneously enhanced lesion in the middle pole of the left kidney and ipsilateral adrenal enlargement. The patient underwent surgical treatment by retroperitoneoscopic left radical nephrectomy plus adrenalectomy. A large number of typical spindle cells surrounded by plasma cells and lymphocytes were observed microscopically. Immunohistochemical analyses indicated that these spindle cells were positive for vimentin, cytokeratin (CK), Ki-67, CK7, CD34, and CD31 and were focally positive for CD10 and anaplastic lymphoma kinase (ALK-1). Thus, a diagnosis of IMT was made definitively. The patient recovered well after operation, and no recurrence or metastasis was noted during the 22-mo follow-up.CONCLUSIONSince kidney IMT is very rare and lacks characteristic clinical manifestation, it is easily misdiagnosed as a malignant tumor before operation. Surgery remains the best choice for diagnosis and treatment, and such cases must be followed carefully because of the uncertain biological behavior of this tumor. This report suggests that renal calculus may be one of the causes of IMT, but further investigation is necessary to prove it.

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“…Согласно данным визуализации, объем опухоли сократился на 70 %, что позволило выполнить радикальную органосохраняющую операцию. Представленные данные показывают высокую эффективность ALK-ингибиторов и подчеркивают важную роль и возможность их применения в качестве неоадъювантной терапии, как было продемонстрировано во 2-м случае [49].…”

Section: терапияunclassified

Inflammatory myofibroblastic tumors in children: literature review

Сулейманова

Качанов

Имянитов

et al. 2020

Ross. ž. det. gematol. onkol.

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Inflammatory myofibroblastic tumor (IMT) is a rare type of neoplasm with undetermined biological potential. IMT can occur in both childhood and adulthood. The standard of therapy is radical surgical treatment, but for patients with inoperable/recurrent or metastatic forms of IMT, therapeutic options are very limited. This literature review describes specific clinical, morphological and biological characteristics of this neoplasm, provides modern approaches to the diagnosis and treatment of IMT.

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ALK inhibition in two emblematic cases of pediatric inflammatory myofibroblastic tumor: Efficacy and side effects

Cited by 31 publications

References 14 publications

Inflammatory Myofibroblastic Tumor of the Sigmoid Colon in an Infant: A Case Report

Inflammatory Myofibroblastic Tumor of the Sigmoid Colon in an Infant: A Case Report

Kidney inflammatory myofibroblastic tumor masquerading as metastatic malignancy: A case report and literature review

Inflammatory myofibroblastic tumors in children: literature review

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