Inflammatory myofibroblastic tumors of the lung are rare, here, an unusual case is described. A 14-year-old boy presented with a history of weight loss and clubbing and was found to have a solitary circumscribed mass in right lower lobe treated with lobectomy. This case indicates the need for early and complete removal of the inflammatory myofibroblastic tumor of the lung

Fatimi, Saulat H.

Sheikh, S

Yaqoob, N

English

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eCommons@AKUSection of Cardiothoracic Surgery Department of SurgeryJanuary 2006Inflammatory myofibroblastic tumor of the lung inan adolescent boyS SheikhAga Khan UniversitySaulat H. FatimiAga Khan University, saulat.fatimi@aku.eduN YaqoobAga Khan UniversityFollow this and additional works at: https://ecommons.aku.edu/pakistan_fhs_mc_surg_cardiothoracicPart of the Surgery CommonsRecommended CitationSheikh, S., Fatimi, S., Yaqoob, N. (2006). Inflammatory myofibroblastic tumor of the lung in an adolescent boy. Journal of College ofPhysicians and Surgeons Pakistan, 16(11), 736-737.Available at: https://ecommons.aku.edu/pakistan_fhs_mc_surg_cardiothoracic/78CASE REPORT 1NFLAlYI1Y1ATORY MYOFlBROBLAST1C TUiviOR OF THE LUNG 1l\J AN ADOLESCENT BOY Sadaf Sheikh, Saulat Hasnain Fatimi and Nausheen Yaqoob• ABSTRACT Inflammatory myofibroblastic tumors of the lung are rare, here, an unusal case is described. A 14-year-old boy prese 1 with a history of weight loss and clubbing and was found to have a solitary circumscribed mass in right lower lobe Ire nted with lobectomy. This case indicates the need for early and complete removal of the inflammatory myofibroblastic tum~re: ~lu~. f KEY WORDS: Pseudotumor. Spindle cells. Lobectomy. INTRODUCTION Inflammatory pseudotumors (IPTs), inflammatory myofibroblastic tumors or plasma cell granulomas are benign, essentially non-neoplastic, tumor-like masses which are inflammatory in origin.' Although they have been widely reported under different names (e.g., postinflammatory tumors, histiocytoma, xanthoma, fibroxanthoma, xantho-granuloma, plasma cell tumor), their first detailed description was made in 1973.2 We present a case that illustrates the diversity of clinical presentations and unpredictability of the natural course of IPTs. CASE REPORT A 14-year-old physically fit boy, who was a non-smoker, presented with history of weight loss for 4 months. He had been well until 4 months back when he noticed some weight loss over the course of two years. He did not have any other complaints or insignificant past medical and drug histories. His physical examination was unremarkable except of clubbing of the fingers. Chest radiograph (Figure 1) showed a well circumscribed 5 em right lower lobe mass. Computed tomographic scan (Figure 2) showed a circumscribed sharply defined mass in the right lower lobe located in the peripheral portion of the lung away from the main bronchus. It was a solid lesion with the differential possibility of slowly growing neoplasm or hamartoma. In view of the uncertain diagnosis and prognosis, triple-regimen antibiotics was started. Repeated radiological examination showed no change in the mass size. A right thoracotomy and right lower lobectomy were performed. Histologic examination revealed proli-ferating spindle cells consistent with the features of Division ot Cardiothoracic Surgery, Department of Surgery. The Aga Khan University Hospital, Karachi. *Department ~.::~~log~~~:-~~~-~_l:l_?n University Hospital, Karadli. Correspondence: Dr. Sadaf Sheikh, Bioethics, SON, Room No. 118, The Aga Khan University Hospital, Stadium Road, Karachi-74800, Pakistan. E-mail: sheikh.sadaf@gmail.oom ·-----·-·------·--·----···-··· Received June 15, 2005; accepted September 2, 2006. 736 Figure 1: Chest X-ray showed :i rounded mass in the right !ower lobe mass Figure 2: Computed tomographic scan showed a rounded right lower lobe mass treated by right lower lobectomy. inflammatory pseudotumor of the lung (Figure 3). It wa~ further studied by immunohistochemistry which shoW~ strong reactivity of spindle cells of the lesion for sll\00 muscle actin. He remained well with no tumor recurrence U1 his follow-up visits. JCPSP 2006, Vol. 1 6 (1 1 ): 736·731 --Photomicrograph shows an inflammatory myofibroblastic tumor. 1 is composed of spindle cells arranged in interlacing fascicles, with inflammatory cellR The spindle cells have cellular atypia activity and the inflammatory cells are mature. {Hematoxylin-magnification 400X). pseudotumors of the lung are rare, with no lisp<JSition to sex or race. More than half of the patients are 40 years of age and 15% are between the ages of 1 and Their etiology and pathogenesis remain uncertain, prior lung infection was recorded in one-third of the 2 Suggestions include immune mediated response or~:anizi.ng intraalveolar pneumonia with progression to hist.op<•thc>log;ic types.' IPTs are regarded as inflammatory or reactive rather than neoplasms, they may have features such as local recurrence, distant metastases and ff~ogenetic clonal changes.J-5 IPTs are well circumscribed, non-firm, white yellow masses. Most are enchyrru1l but some are endobronchial and may cause . <ucuuu. Less than 5% invade the mediastinum and chest Microscopically, the lesions consist of variable of fibroblasts and granulation tissue, fibrous tissue inflammatory cells including lymphocytes, histiocytes, cells, macrophages, neutrophils, eosinophils and large number of plasma cells.1.2 Immuno-itocli1ernistry has demonstrated the polyclonal nature of the cells with immunoglobulin G predominance.s patients are asymptomatic and the IPTs are discovered ~~ti~;;;~,,~~~~~ findings on examination of radiographs; if the ··1 are symptomatic, cough, hemoptysis, shortness of clubbing, chest pain and arthralgia may be noted.l Radiologically IPTs typically present as solitary circumscribed masses. Multiple lung masses, pneumonic consolidation, atelectasis, hilar masses and cavitation are quite unusual.l2,6 Percutaneous fine needle aspiration biopsy is considered insufficient for diagnosis and frozen section is also subject to errors.' Early and complete 'surgical resection of the IPTs remains the best treatment option to exclude malignancy and to achieve cure. Delay in diagnosis and treatment may increase considerably the magnitude of the surgical intervention required. Non-surgical treatment modalities including radiotherapy, chemotherapy and steroids may have a place in the setting of incomplete surgical resection, multifocal disease, postoperative tumor recurrence or contraindication to lung resection.?. B The natural history of IPTs is unpredictable. Although spontaneous regression may occur, local expansion may cause significant morbidity and occasionally death.' Although the most common picture of the IPTs is one of an asymptomatic, well-circumscribed lung mass that may mimic cancer, clinicians need to bear in mind their diverse clinical presentations. Surgical excision is usually indicated to reach a firm diagnosis and to attempt cure. i:.•. __ ; ·. 1. Melloni G, Carretta A, Ciriaco P, Arrigoni G, Fieschi S, Rizzo N, et al. Inflammatory pseudotumor of the lung in adults. Ann Tborac Surg 2005; 79: 426 · 32. 2. Kelly SJ, Lambie NK, Singh HP. Inflammatory myofibroblastic tumor of the left ventricle in an older adult. Ann Tborac Surg 2003; 75: 1971-3. 3. Copin MC, Gosselin BH, Ribet ME. Plasma cell granuloma of the lung: difficulties in diagnosis and prognosis. Ann Tborac Surg 1 996; 61: 1477·82. 4. Sakurai H, Hasegawa T, Watanabe S, Suzuki K, Asamura H, Tsuchiya R. Inflammatory myofibroblastic tumor of the lung. Eur J Cardiothorac Surg 2004; 25: 155-9 . 5. Yildirim Z, Soysal 0, G6kirmak M. Clhan HB, Mizrak B, Hasanou HC. Endobronchial inflammatory pseudotumor: a cause of chronic cough. Asian Cardiovasc Tborac Ann 1 999; 7: 326-8. 6. Bahadori M, Liebow AA. Plasma eel! granulomas of the lung. Cancer 1973; 31: 191·208. 7. Matsubara 0, Xan-Liu NS, Kenney RM, Mark EJ. Inflammatory pseudotumors of the lung: progression from organizing pneumonia to fibrous histiocytoma or to plasma cell granuloma in 32 cases.HumParha/1988; 19:807-14. 8. Warter A, Satge 0, Roeslin N. Angioinvasive plasma cell granulomas of the lung. Cancer 1987; 59: 435-43 . ..... ;lc ... P 2006, Vol. 16 (11): 736-737 737 

Inflammatory myofibroblastic tumor of the lung in an adolescent boy

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Inflammatory myofibroblastic tumor of the lung in an adolescent boy

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