Case Report
Pelvic inflammatory myofibroblastic tumour mimicking scrotal mass: an extremely rare case
Scottish Medical Journal 2015, Vol. 60(1) e8–e10 ! The Author(s) 2014 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0036933014563240 scm.sagepub.com
Serdar Aykan1, Yigit Akin2, Isil Basara3, Mutlu Ates4, Murat Tuken1, Meral Uyar5, Baris Nuhoglu6 and Atilla Semercioz1
Abstract Inflammatory myofibroblastic tumour (IMT) is a rare benign mesenchymal tumour. However, IMT may arise from a wide variety of tissues and is very rare in the elderly. IMT may mimic the mass in which it originates. Although IMT has been defined as uncertain behaviour, it is treated surgically. We present a-65-year old man whose mass was diagnosed as IMT extending from scrotum to pelvis. The mass was independent of any surrounding anatomic structures. According to our best knowledge this is the first case in the literature that pelvic IMT was diagnosed in an elderly man and successfully treated surgically with a long term follow-up period. Aetiology of IMT is still unknown, and more studies are needed for exact continuum of IMT.
Keywords Adults, inflammatory myofibroblastic tumour, surgery
Case report A 65-year-old man was admitted to a urology outpatient clinic with a painless scrotal mass existing about three months. He had no comorbidity except history of appendectomy more than 40 years previously. In the physical examination, there was a mass found in the right side of scrotum which was independent from the testis and epididymis. Additionally it was painless and swelling extending from scrotum to pelvis. The laboratory tests including blood analyses, prostate specific antigen, a-fetoprotein and human chorionic gonadotropin (hCG) levels were normal. Moreover, there was an obstructive pattern in uroflowmetry. Ultrasonography revealed the mass measured 12 15 cm extending from scrotum to pelvis. MRI was performed. There was an enhancing mass extending from scrotum to pelvis (Figure 1). After the clinical and radiological evaluations, and due to the lack of history of comorbidity, we investigated for a source and/or a metastasis in thorax and abdomen. In computed tomography (CT), there was no metastasis and/or source.
After written informed consent was obtained from the patient, the mass was excised surgically in lithotomy position (Figure 1). The mass seemed like it was independent from the surrounding tissues and anatomic structures in both the pelvis and scrotum. There were spindle cells with low-grade mitosis in pathology examinations with haematoxylin eosin stain
1 M.D., Department of Urology, Bagcilar Research and Training Hospital, Turkey 2 Assistant Professor of Urology, Department of Urology, School of Medicine, Harran University, Turkey 3 M.D., Department of Radiology, Harput State Hospital, Turkey 4 Associated Professor of Urology, Department of Urology, School of Medicine, Afyon Kocatepe University, Turkey 5 M.D., Department of Pathology, Bagcilar Research and Training Hospital, Turkey 6 Professor of Urology, Department of Urology, School of Medicine, Erzincan University, Turkey
Corresponding author: Yigit Akin, Assistant Professor of Urology, Department of Urology, School of Medicine, Harran University, 63100 Sanliurfa, Turkey. Email: [email protected]
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(Figure 1). Additionally, surgical margin was negative. Moreover CD34, CD99, Bcl2 were positive and Desmin, S100 were negative in immunohistochemistry (Figure 1). In the light of the clinical, radiological and pathological evaluations, inflammatory myofibroblastic tumour (IMT) was diagnosed. After the operation, the patient has been in follow-up in the urology outpatient clinic without recurrence.
Discussion The IMT is a rare tumour of unknown cause. IMT grows very slowly. IMT was a benign tumour of childhood and may occur very rarely in the elderly. It may also develop from every part of the body, and mimics the mass in which it originated. In pathological examinations, spindle cells and inflammatory myofibroblasts are classical features of diagnosis.1 IMT is a pathological description of a benign mesenchymal tumour. There has been still ambiguity in description and pathogenesis. IMT is particularly prevalent in males, with a predominance of 3:1 male.2 However, metastasis of IMT is rare and it is usually diagnosed as benign. There is potential for malignant transformation and
its rate is unknown. Therefore, it should be treated when it is diagnosed. Herein, we presented an extremely rare case with scrotal IMT extending from scrotum to pelvis, mimicking a scrotal mass. Brunn described the first cases of IMT in the literature. These cases were with IMT in lungs. However, IMT is a rare tumour and usually occurs in childhood as Cheng et al., described.1 Our case was an extremely rare case in an elderly patient. Roth described the first genitourinary IMT in a 32-year-old woman in 1980.3 After the genitourinary IMT was reported by Roth, there were some reports of IMT in the bladder.1 However, IMT in our case was in the pelvis. According to the report by Cheng et al., IMT mostly occurs in the bladder or genitourinary area.1 Additionally, Houben et al. reported pelvic and/or scrotal IMT might occur after urethral instrumentation.4 Our case had no history of urethral instrumentation as well as no comorbidities. Chakrabarti and Shetty reported a scrotal IMT.5 In their case, the IMT originated from the spermatic cord.5 When a mass is diagnosed in the scrotum in the elderly, differential diagnosis comes into question. We performed additional laboratory tests, and also performed CT for metastasis and/or sources of tumour.
Figure 1. Radiologic, operational and pathological features of the case (a) Sagittal T2 weighted image. There is a iso-hiperintense, well-defined lesion at the infrapubic localization, extending from right scrotum to pelvis (arrow) *B: Bladder, (b) axial nonenhanced T1weighted image, intrascrotal part of the lesion is seen. The lesion is hypointense (arrow), (c) axial postcontrast, fat saturated T1 weighted image, there is diffuse enhancement in the lesion (arrow), (d) The mass was excised surgically, it was independent from testis, epididymis and the other anatomical structures around pelvis (arrow). (e) In the light microscopy, low mitosis spindle cells are seen and interlacing fascicles of myofibroblastic spindle cells are accompanied by a lymphoplasmacytic inflammatory infiltrate with hematoxylin eosin stain (100). (f) Immunochemistry, CD34 was positive.
Downloaded from scm.sagepub.com at Glasgow University Library on June 25, 2015
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Scottish Medical Journal 60(1)
The tumour was single and independent from anatomical structures such as the testis, epididymis and radix penis in the pelvis and scrotum. Surgical treatment options are widely used for IMT. Other treatment options include radiotherapy and chemotherapy with methotrexate or steroids but these are generally used for patients with recurrence.6 Follow-up is essential as well as surgical treatment. We excised the tumour from the pelvis five years ago, in follow-up period there was no recurrence in physical examination and also MRI evaluations.
Conclusion We presented an IMT case which is extremely rare in the elderly. The mass was extending from the scrotum to the pelvis. It was treated surgically and the case is now in the follow-up period. To our best knowledge, this is a unique IMT case extending from scrotum to pelvis with long-term follow-up in the literature. Clinicians should be aware of scrotal masses in the elderly. Detailed differential diagnosis should be performed. However, whilst diagnosis and treatment options of IMT are defined, aetiology is still unknown. Therefore, there is a need for more studies investigating aetiology and exact continuum of IMT.
Declaration of conflicting interests None declared.
Funding This research received no specific grant from any funding agency in the public, commercial or not-for-profit sectors.
References 1. Cheng L, Foster SR, MacLennan GT, et al. Inflammatory myofibroblastic tumours of the genitourinary tract – single entity or continuum? J Urol 2008; 180: 1235–1240. 2. Yalcinkaya I, Demirtas I, Ayakta H, et al. Inflammatory pseudotumor of the lung associated with anemia: a case report and review of the literature. Turk J Haematol 2000; 17: 37–40. 3. Roth JA. Reactive pseudosarcomatous response in urinary bladder. Urology 1980; 16: 635–637. 4. Houben CH, Chan A, Lee KH, et al. Inflammatory myofibroblastic tumour of the bladder in children: what can be expected? Pediatr Surg Int 2007; 23: 815–819. 5. Chakrabarti N and Shetty R. Inflammatory myofibroblastic sarcoma of the spermatic cord. Ind J Surg 2010; 72: 152–154. 6. Meis JM and Enzinger FM. Inflammatory fibrosarcoma of the mesentery and retroperitoneum: a tumour closely simulating inflammatory pseudotumour. Am J Surg Pathol 1991; 15: 1146–1156.
Downloaded from scm.sagepub.com at Glasgow University Library on June 25, 2015
Pelvic inflammatory myofibroblastic tumour mimicking scrotal mass: an extremely rare case
Scottish Medical Journal 2015, Vol. 60(1) e8–e10 ! The Author(s) 2014 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0036933014563240 scm.sagepub.com
Serdar Aykan1, Yigit Akin2, Isil Basara3, Mutlu Ates4, Murat Tuken1, Meral Uyar5, Baris Nuhoglu6 and Atilla Semercioz1
Abstract Inflammatory myofibroblastic tumour (IMT) is a rare benign mesenchymal tumour. However, IMT may arise from a wide variety of tissues and is very rare in the elderly. IMT may mimic the mass in which it originates. Although IMT has been defined as uncertain behaviour, it is treated surgically. We present a-65-year old man whose mass was diagnosed as IMT extending from scrotum to pelvis. The mass was independent of any surrounding anatomic structures. According to our best knowledge this is the first case in the literature that pelvic IMT was diagnosed in an elderly man and successfully treated surgically with a long term follow-up period. Aetiology of IMT is still unknown, and more studies are needed for exact continuum of IMT.
Keywords Adults, inflammatory myofibroblastic tumour, surgery
Case report A 65-year-old man was admitted to a urology outpatient clinic with a painless scrotal mass existing about three months. He had no comorbidity except history of appendectomy more than 40 years previously. In the physical examination, there was a mass found in the right side of scrotum which was independent from the testis and epididymis. Additionally it was painless and swelling extending from scrotum to pelvis. The laboratory tests including blood analyses, prostate specific antigen, a-fetoprotein and human chorionic gonadotropin (hCG) levels were normal. Moreover, there was an obstructive pattern in uroflowmetry. Ultrasonography revealed the mass measured 12 15 cm extending from scrotum to pelvis. MRI was performed. There was an enhancing mass extending from scrotum to pelvis (Figure 1). After the clinical and radiological evaluations, and due to the lack of history of comorbidity, we investigated for a source and/or a metastasis in thorax and abdomen. In computed tomography (CT), there was no metastasis and/or source.
After written informed consent was obtained from the patient, the mass was excised surgically in lithotomy position (Figure 1). The mass seemed like it was independent from the surrounding tissues and anatomic structures in both the pelvis and scrotum. There were spindle cells with low-grade mitosis in pathology examinations with haematoxylin eosin stain
1 M.D., Department of Urology, Bagcilar Research and Training Hospital, Turkey 2 Assistant Professor of Urology, Department of Urology, School of Medicine, Harran University, Turkey 3 M.D., Department of Radiology, Harput State Hospital, Turkey 4 Associated Professor of Urology, Department of Urology, School of Medicine, Afyon Kocatepe University, Turkey 5 M.D., Department of Pathology, Bagcilar Research and Training Hospital, Turkey 6 Professor of Urology, Department of Urology, School of Medicine, Erzincan University, Turkey
Corresponding author: Yigit Akin, Assistant Professor of Urology, Department of Urology, School of Medicine, Harran University, 63100 Sanliurfa, Turkey. Email: [email protected]
Downloaded from scm.sagepub.com at Glasgow University Library on June 25, 2015
Aykan et al.
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(Figure 1). Additionally, surgical margin was negative. Moreover CD34, CD99, Bcl2 were positive and Desmin, S100 were negative in immunohistochemistry (Figure 1). In the light of the clinical, radiological and pathological evaluations, inflammatory myofibroblastic tumour (IMT) was diagnosed. After the operation, the patient has been in follow-up in the urology outpatient clinic without recurrence.
Discussion The IMT is a rare tumour of unknown cause. IMT grows very slowly. IMT was a benign tumour of childhood and may occur very rarely in the elderly. It may also develop from every part of the body, and mimics the mass in which it originated. In pathological examinations, spindle cells and inflammatory myofibroblasts are classical features of diagnosis.1 IMT is a pathological description of a benign mesenchymal tumour. There has been still ambiguity in description and pathogenesis. IMT is particularly prevalent in males, with a predominance of 3:1 male.2 However, metastasis of IMT is rare and it is usually diagnosed as benign. There is potential for malignant transformation and
its rate is unknown. Therefore, it should be treated when it is diagnosed. Herein, we presented an extremely rare case with scrotal IMT extending from scrotum to pelvis, mimicking a scrotal mass. Brunn described the first cases of IMT in the literature. These cases were with IMT in lungs. However, IMT is a rare tumour and usually occurs in childhood as Cheng et al., described.1 Our case was an extremely rare case in an elderly patient. Roth described the first genitourinary IMT in a 32-year-old woman in 1980.3 After the genitourinary IMT was reported by Roth, there were some reports of IMT in the bladder.1 However, IMT in our case was in the pelvis. According to the report by Cheng et al., IMT mostly occurs in the bladder or genitourinary area.1 Additionally, Houben et al. reported pelvic and/or scrotal IMT might occur after urethral instrumentation.4 Our case had no history of urethral instrumentation as well as no comorbidities. Chakrabarti and Shetty reported a scrotal IMT.5 In their case, the IMT originated from the spermatic cord.5 When a mass is diagnosed in the scrotum in the elderly, differential diagnosis comes into question. We performed additional laboratory tests, and also performed CT for metastasis and/or sources of tumour.
Figure 1. Radiologic, operational and pathological features of the case (a) Sagittal T2 weighted image. There is a iso-hiperintense, well-defined lesion at the infrapubic localization, extending from right scrotum to pelvis (arrow) *B: Bladder, (b) axial nonenhanced T1weighted image, intrascrotal part of the lesion is seen. The lesion is hypointense (arrow), (c) axial postcontrast, fat saturated T1 weighted image, there is diffuse enhancement in the lesion (arrow), (d) The mass was excised surgically, it was independent from testis, epididymis and the other anatomical structures around pelvis (arrow). (e) In the light microscopy, low mitosis spindle cells are seen and interlacing fascicles of myofibroblastic spindle cells are accompanied by a lymphoplasmacytic inflammatory infiltrate with hematoxylin eosin stain (100). (f) Immunochemistry, CD34 was positive.
Downloaded from scm.sagepub.com at Glasgow University Library on June 25, 2015
e10
Scottish Medical Journal 60(1)
The tumour was single and independent from anatomical structures such as the testis, epididymis and radix penis in the pelvis and scrotum. Surgical treatment options are widely used for IMT. Other treatment options include radiotherapy and chemotherapy with methotrexate or steroids but these are generally used for patients with recurrence.6 Follow-up is essential as well as surgical treatment. We excised the tumour from the pelvis five years ago, in follow-up period there was no recurrence in physical examination and also MRI evaluations.
Conclusion We presented an IMT case which is extremely rare in the elderly. The mass was extending from the scrotum to the pelvis. It was treated surgically and the case is now in the follow-up period. To our best knowledge, this is a unique IMT case extending from scrotum to pelvis with long-term follow-up in the literature. Clinicians should be aware of scrotal masses in the elderly. Detailed differential diagnosis should be performed. However, whilst diagnosis and treatment options of IMT are defined, aetiology is still unknown. Therefore, there is a need for more studies investigating aetiology and exact continuum of IMT.
Declaration of conflicting interests None declared.
Funding This research received no specific grant from any funding agency in the public, commercial or not-for-profit sectors.
References 1. Cheng L, Foster SR, MacLennan GT, et al. Inflammatory myofibroblastic tumours of the genitourinary tract – single entity or continuum? J Urol 2008; 180: 1235–1240. 2. Yalcinkaya I, Demirtas I, Ayakta H, et al. Inflammatory pseudotumor of the lung associated with anemia: a case report and review of the literature. Turk J Haematol 2000; 17: 37–40. 3. Roth JA. Reactive pseudosarcomatous response in urinary bladder. Urology 1980; 16: 635–637. 4. Houben CH, Chan A, Lee KH, et al. Inflammatory myofibroblastic tumour of the bladder in children: what can be expected? Pediatr Surg Int 2007; 23: 815–819. 5. Chakrabarti N and Shetty R. Inflammatory myofibroblastic sarcoma of the spermatic cord. Ind J Surg 2010; 72: 152–154. 6. Meis JM and Enzinger FM. Inflammatory fibrosarcoma of the mesentery and retroperitoneum: a tumour closely simulating inflammatory pseudotumour. Am J Surg Pathol 1991; 15: 1146–1156.
Downloaded from scm.sagepub.com at Glasgow University Library on June 25, 2015
