IP Archives of Cytology and Histopathology Research

Print ISSN: 2581-5725

Online ISSN: 2456-9267

CODEN : IACHCL

IP Archives of Cytology and Histopathology Research (ACHR) open access, peer-reviewed quarterly journal publishing since 2016 and is published under the Khyati Education and Research Foundation (KERF), is registered as a non-profit society (under the society registration act, 1860), Government of India with the vision of various accredited vocational courses in healthcare, education, paramedical, yoga, publication, teaching and research activity, with the aim of faster and better dissemination of knowledge, we will be publishing the article more...

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Get Permission Kalariya: Inflammatory myofibroblastic tumor of urinary bladder in young male: A case report and literature review


Introduction

Inflammatory myofibroblastic tumor (IMT) of the urinary bladder is an exceptionally rare neoplasm characterized by the proliferation of myofibroblastic spindle cells within a background of chronic inflammation. 1, 2, 3, 4 Despite its infrequency, IMT poses diagnostic and therapeutic challenges due to its diverse clinical presentations and resemblance to other bladder malignancies. This report aims to present a case of IMT of the urinary bladderin young male, detailing its clinical course, diagnostic approach, treatment strategies, and outcome. Furthermore, a comprehensive literature review will be conducted to provide insights into the characteristics, management, and prognosis of this uncommon tumor entity.

Figure 1
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Case Presentation

A 20-year-old male presented with complaints of dysuria and dribbling of urine without any episodes of hematuria. The patient was investigated through CT abdomen, which showed irregular heterogeneously enhancing mass on left dome of urinary bladder. All blood investigations including renal function test were normal. Cystoscopic examination demonstrated a protruding mass originating from the left dome of bladder wall measuring 5x4 cm sized. Partial cystectomy with bilateral iliac lymphnodes dissection was performed. Specimen was sent for histopathological examination. Histopathological examination and Immunohistochemistry study of the excised tissue done. On gross examination weight of specimen was 67.2 gms. 6.4 X 5.0 X 4.8 cm sized part of Urinary bladder with perivesicle fat with mass with surrounding margins received. There was a presence of 5.0 X 4.0 X 4.0 cm sized mass. Mass was single, solid, greyish white with yellowish brown colored areas and hard in consistancy. On microscopic examination Sections from mass show Spindled cell proliferation with infiltration by inflammatory cells predominantly lymphocytes and plasma cells. Spindle cells are stellate myofibroblasts with abundant eosinophilic cytoplasm and elongated nuclei. Areas with hyalinisation present. Lamina propria and muscularis propria were showing involvement by tumor cells. Perivesical fat and surgical margins were free from tumor cells. Total 6 iliac lymphnodes were identified microscopically from the both sides and all were free from tumor cells. On immunohistochemistry study(7,8,9) shows positivity for ALK1, SMA and DESMIN in spindle cells. CD 3, CD 20, CD138, Kappa and lambda were positive in background inflammatiory cells. Negative for myogenin and EMA. So confirmed the diagnosis of inflammatory myofibroblastic tumor. Subsequent evaluation ruled out distant metastasis, and the patient after surgical excision followed by adjuvant therapy. Regular follow-up examinations showed no evidence of tumor recurrence.

Discussion

Inflammatory myofibroblastic tumor 1, 2 of the urinary bladder in young male is a rare entity with a wide spectrum of clinical manifestations, ranging from asymptomatic to hematuria, dysuria, and abdominal pain. Diagnosis 3, 4 relies on histopathological examination demonstrating spindle cell proliferation with inflammatory infiltrates. Differential diagnosis includes other bladder tumors such as sarcomatoid urothelial carcinoma, leiomyosarcoma and rhabdomyosarcoma which can be ruled out by immunohistochemistry study. Treatment strategies encompass surgical resection as the cornerstone, with adjuvant therapy reserved for unresectable or recurrent cases. The literature review highlights the rarity of IMT of the urinary bladder, with limited reported cases at young age and variable treatment outcomes. Various diagnostic modalities and treatment options, including chemotherapy and targeted therapy, have been explored with varying success rates.

Conclusion

An Inflammatory myofibroblastic tumor of urinary bladder is an uncommon benign tumor of urinary bladder of unknown neoplastic potential. Inflammatory myofibroblastic tumor of the urinary bladder 5, 6 at younger age remains a diagnostic and therapeutic challenge due to its rarity and nonspecific clinical features. However, timely recognition and appropriate management, including surgical excision and adjuvant therapy, can lead to favourable outcomes with low recurrence rates. 7, 8, 9, 10, 11 This case underscores the importance of considering IMT in the differential diagnosis of bladder masses and the need for further research to elucidate optimal treatment strategies for this rare tumor entity.

Source of Funding

None.

Conflict of Interest

None.

References

1 

K Machioka Y Kitagawa K Izumi S Kitamura H Ikeda M Namiki Inflammatory myofibroblastic tumor of the urinary bladder with benign pelvic lymph node enlargement: a case reportCase Rep Oncol2014725715

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JY Teoh NH Chan HY Cheung SSM Hou CF Ng Inflammatory myofibroblastic tumors of the urinary bladder: a systematic reviewUrology20148435038

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R Alam M H Johnson T Caldwell Diagnosing and treating inflammatory myofibroblastic tumor of the bladderCase Rep Urol2016572402010.1155/2016/5724020

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K Siemion J Reszec-Gielazyn J Kisluk L Roszkowiak J Zak A Korzynska What do we know about inflammatory myofibroblastic tumors?-A systematic reviewAdv Med Sci202267112938

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B Marais P Eyal K Kesner J John Inflammatory myofibroblastic tumour of the bladder: a case report and review of the literatureTher Adv Urol20221410.1177/17562872221096385

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B Balagobi S Gobishangar A Ginige D Gamlaksha J Sanjeyan L Suvethini Inflammatory myofibroblastic tumour: Case report of a rare form of bladder tumourInt J Surg Case20229210678610.1016/j.ijscr.2022.106786

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CM Coffin JL Hornick CD Fletcher Inflammatory myofibroblastic tumor: Comparison of clinicopathologic, histologic, and immunohistochemical features including ALK expression in atypical and aggressive casesAm J Surg Pathol200731450920

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CM Coffin A Patel S Perkins KS Elenitoba-Johnson E Perlman CA Griffin ALK1 and p80 expression and chromosomal rearrangements involving 2p23 in inflammatory myofibroblastic tumorMod Pathol200114656976

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JR Cook LP Dehner MH Collins Z Ma SW Morris CM Coffin Anaplastic lymphoma kinase (ALK) expression in the inflammatory myofibroblastic tumor: a comparative immunohistochemical studyAm J Surg Pathol20012511136471

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EK Libby LT Ellis S Weinstein RD Hammer KS Murray Metastatic inflammatory myofibroblastic tumor of the bladderUrol Case Rep201810210.1016/j.eucr.2018.11.007

11 

L Gros AP Dei Tos RL Jones A Digklia Inflammatory Myofibroblastic Tumour: State of the ArtCancers20221415366210.3390/cancers14153662



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Article type

Case Report


Article page

59-61


Authors Details

Jigna Jerambhai Kalariya*


Article History

Received : 22-02-2024

Accepted : 29-04-2024


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