Treatment of Inflammatory Myofibroblastic Tumor of the Chest: The Extent of Resection

doi:10.1016/j.athoracsur.2007.03.037

The Annals of Thoracic Surgery

Volume 84, Issue 1, July 2007, Pages 221-224

https://doi.org/10.1016/j.athoracsur.2007.03.037 Get rights and content

Background

Owing to the rarity of inflammatory myofibroblastic tumor, its pathology and clinical course are poorly understood.

Methods

We performed a retrospective chart review of patients diagnosed with inflammatory myofibroblastic tumor in the chest who underwent surgical resection.

Results

From 1995 to 2006, 15 patients (10 males and 5 females) underwent surgical procedures owing to inflammatory myofibroblastic tumors. The mean age of these patients was 31.3 years (range, 7 months to 61 years). Among them, 13 patients (86.7%) presented with respiratory symptoms such as cough, dyspnea, and hemoptysis. Seven patients presented with the tumor located in the lung parenchyma, 4 in the trachea, 2 in the main bronchus, 1 in the segmental bronchus, and 1 in the chest wall. The diagnosis was confirmed before surgery for only 1 patient (6.3%). The types of operations performed included lobectomies for 4 patients, wedge resections using video-assisted thoracic surgery for 4 patients, tracheal/bronchial resections with end-to-end anastomoses for 6 patients, and chest wall resection for 1 patient. Only 2 patients received adjuvant radiation therapy. We followed up with all patients postoperatively for a mean of 33.3 months (range, 1.2 months to 8.4 years). Fourteen patients were free of local recurrence or distant metastasis during the follow-up period.

Conclusions

Inflammatory myofibroblastic tumor usually requires surgical resection for both proper diagnosis and adequate treatment. Complete resection and achieving negative margins leads to excellent outcome.

Section snippets

Patients and Methods

The Samsung Medical Center Institutional Review Board approved this retrospective study with the use of a database from the Department of Thoracic Surgery (IRB No. 2006-12-023), and waived the need for patient consent. Between 1994 and 2006, 12,355 general thoracic procedures were performed at Samsung Medical Center, and 15 patients (0.12%) underwent surgery owing to IMBT. Medical records were reviewed for sex, age, presenting symptoms, location and size of the lesions, methods of treatment,

Results

Fifteen patients, 10 men and 5 women, underwent surgery for IMBT. The mean age was 31.3 ± 18.1 years (range, 7 months to 61 years). Thirteen patients (86.7%) were symptomatic and their presenting symptoms included cough (20.0%), dyspnea (26.7%), hemoptysis (20.0%), chest pain or discomfort (13.3%), and a palpable chest wall mass (6.7%). The other 2 patients (13.3%) were asymptomatic and their lesions were noticed on routine examinations.

On preoperative chest CT scans, the lesions manifested as

Comment

Inflammatory myofibroblastic tumor in the chest is a rare finding, and making the histopathologic diagnosis is difficult when obtaining only a small biopsy tissue sample for frozen section. Inflammatory myofibroblastic tumor in the chest can be roughly classified into two groups according to location: tumors that develop in the lung parenchyma (parenchymal IMBT) and tumors that develop within the airway (airway IMBT). Previous studies have reported that most IMBTs manifest as a solitary mass in

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Diverse cases of inflammatory myofibroblastic tumors (IMTs) in the lung (pleural, endobronchial, and parenchymal) are presented while discussing the (preoperative) diagnostic challenges and treatment modalities. Other objectives include emphasizing the significance of gene rearrangements and highlighting the multidisciplinary approach in addressing IMTs.
Four cases of IMT in the lung are presented, including a young adolescent girl with an ETV6-neurotrophic tyrosine receptor kinase 3 (NTRK3) gene rearrangement, a 5-year-old boy with challenging preoperative diagnosis, and 2 middle-aged women with respectively pleural and endobronchial tumors with one peribronchial relapse.
The cases demonstrate the diverse clinical presentations and diagnostic complexities associated with IMT in the lung. Surgical resection remains the primary treatment modality, with complete resection leading to a cure in most patients. Unfortunately, aggressive relapse can occur, as in our last case of an endobronchial tumor. Frozen section may confirm the presence of malignant cells perioperatively and impact further treatment. The presence of gene rearrangements, such as ETV6-NTRK3, suggests potential therapeutic implications.
Early detection and complete surgical removal of IMT are crucial for effective treatment. Identifying gene rearrangements such as ETV6-NTRK3 holds promise for targeted therapies. Diagnostic challenges, including the controversy of biopsies and preoperative evaluations, underscore the importance of a multidisciplinary approach. Anatomopathological recognition of IMT stays demanding. Close surveillance is necessary due to potential relapse, whereas frozen section perioperatively can help further treatment. This case series emphasizes the diagnostic challenges and therapeutic considerations for IMT in the lung.
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It is a slow-growing quasi-neoplastic lesion. Although its clinical course is benign it can mimic malignancy both clinically and radiologically [5,6]. Recently, rare cases with aggressive clinical picture, such as local recurrence, malignant transformation, or distant metastasis, were reported.
Inflammatory myofibroblastic tumors (IMTs) are rare childhood neoplasms, with benign clinical course. Although etiology of IMTs are not clear, recent studies have reported that IMT is a true neoplasm rather than a reactive or inflammatory lesion. IMTs are rarely seen in adults and tracheal involvement is also rare both in adults and also in children. We describe a 16-year old female patient who was misdiagnosed and treated as asthma in another center for a few months and presented with acute respiratory distress due to upper airway obstruction. Computerized tomography (CT) of the chest and rigid bronchoscopy revealed a mass lesion that was nearly totally obliterating tracheal lumen. Bronchoscopic resection was performed under general anesthesia and the final pathological diagnosis was tracheal IMT.
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However, a 60% recurrence rate in those receiving incomplete resection has been reported in the same study. Lee and colleagues [7] mentioned a case of IMT originating from the pleura and stated that a tumor measuring 27 × 22 mm was successfully removed from the adjacent rib tissue without performance of rib resection, and the patient's 6.5-year follow-up visit was uneventful. Smaller tumors without chest wall invasion may allow total excision of the tumor without rib resection.
Inflammatory myofibroblastic tumor (IMT) is a rare benign neoplasm. It is a challenging disease because the symptoms and radiologic findings are diverse and nonspecific. Although pulmonary IMT is the most common form, pleural origin is an extremely rare clinical entity. Nuchal fibroma (NF) is another rare benign neoplasm. We report herein a case of pleural IMT with concomitant NF in a 15-year-old girl. To the best of our knowledge, this is the first report suggesting an association between IMT and NF, and our case had the largest reported intrathoracic IMT. Moreover, we found a possible association between IMT and increased CA-125 levels.
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Original articleGeneral thoracicTreatment of Inflammatory Myofibroblastic Tumor of the Chest: The Extent of Resection

Background

Methods

Results

Conclusions

Section snippets

Patients and Methods

Results

Comment

J Thorac Surg

Hum Pathol

Ann Thorac Surg

Ann Thorac Surg

Inflammatory pseudotumor

Radiographics

Original article
General thoracic
Treatment of Inflammatory Myofibroblastic Tumor of the Chest: The Extent of Resection