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Five years of local control of subscapularis aggressive fibromatosis managed by surgery and imatinib: a case report.

Dao A, Benchakroun N, Jabir H, Taleb A, Bouchbika Z, Tawfiq N, Jouhadi H, Sahraoui S, Benider A - J Med Case Rep (2014)

Bottom Line: We reported no adverse side effects to imatinib regarding Common Terminology Criteria for Adverse Events grading.Systemic treatment with imatinib for unresectable or recurrent tumors with positive c-KIT could be the best therapeutic option.In our case report, the patient was stabilized with imatinib for 30 months and he had a very good quality of life.

View Article: PubMed Central - PubMed

Affiliation: Centre Mohammed VI pour le Traitement des Cancers, CHU Ibn Rochd, Casablanca, Morocco. daocl@yahoo.fr.

ABSTRACT

Introduction: Imatinib, a tyrosine kinase inhibitor, is a major therapeutic option for the management of unresectable aggressive fibromatosis. Unfortunately, for most patients of low or very low average income countries, surgery often is the first treatment option. This is related to unavailability of chemotherapy or targeted therapy, and to a lack of financial resources or surgeons' lack of knowledge of other therapeutic options.

Case presentation: In 2010, a 26-year-old Moroccan man was referred to our oncology and radiotherapy center by his surgeon for the management of a recurrent tumor of his right subscapularis muscle. Before his assessment in our center, two resections were performed by his surgeon after performing an incision biopsy and magnetic resonance imaging. Postoperative magnetic resonance imaging was performed and showed a right axillary nodule size 2.1cm regarding a collection with a residual tumor. We decided to administer imatinib 400mg daily by mouth. Clinical and magnetic resonance imaging evaluation were performed regularly and reported a stable tumor. We reported no adverse side effects to imatinib regarding Common Terminology Criteria for Adverse Events grading.

Conclusions: Recurrences are high during aggressive fibromatosis management. Systemic treatment with imatinib for unresectable or recurrent tumors with positive c-KIT could be the best therapeutic option. In our case report, the patient was stabilized with imatinib for 30 months and he had a very good quality of life.

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Related in: MedlinePlus

Images of the first recurrence. Thoracic computed tomography axial cut (A) and sagittal cut (B). Auxiliary magnetic resonance imaging with axial (C) and sagittal (D) cut showing mass measuring 130mm. This mass was localized on the right chest wall, between scapula and chest wall, hypointensity T1, hypersignal T2; this mass displaces the muscle structures; auxiliary vessels are permeable.
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Fig1: Images of the first recurrence. Thoracic computed tomography axial cut (A) and sagittal cut (B). Auxiliary magnetic resonance imaging with axial (C) and sagittal (D) cut showing mass measuring 130mm. This mass was localized on the right chest wall, between scapula and chest wall, hypointensity T1, hypersignal T2; this mass displaces the muscle structures; auxiliary vessels are permeable.

Mentions: In 2010, a 26-year-old Moroccan man was referred to our oncology and radiotherapy center for management of recurrent tumor of his right subscapularis. No pathological medical or surgical history was reported and no alcohol or tobacco habits. One year before, he had presented to his surgeon with a subscapularis tumor which had appeared gradually and increased in size during 6 months without associated pain or other symptoms. A physical examination reported a mass size 10cm, palpated in his right subscapularis region. A first resection was realized. Histopathological analysis demonstrated spindle-shaped cells with no identifiable nuclear pleomorphism or mitotic activity. There was no necrosis. A benign tumor with spindle-shaped cells is suspected. On immunohistochemical analysis, the cells stained positive for anti-smooth muscle actin, favoring a smooth muscle origin. The diagnostic of AF was retained. Unfortunately, the mass recurred within a period of 10 months and continued to increase in size reaching 13cm on a computed tomography scan and magnetic resonance imaging (MRI; Figure 1) without regional structures involved (bone, muscle or vascular). A second tumor and lymph nodes resection were performed by another surgeon who reported “a very hard resection without cleavage plane”. Histological analysis regarding two fragments size 4×3cm and 14×10×8cm led to a conclusion of AF tumor; hormonal receptor was not found. The resections of margins were narrowed and lymph node resections regarding four nodes were not involved. At assessment in our oncology and radiotherapy center after this second resection, his World Health Organization performance status was zero, weight 84kg, height 177cm. There was no induration or palpable mass. A MRI performed after the repeat surgery was normal. During follow up, a recurrence was suspected 1 year after the second resection. An axillary MRI (Figure 2A and 2B) was performed and a large mass was found in the last tumor site, measuring 12.6cm involving deltoid muscles and extending to axillary area. A third resection was realized and a histopathological examination showed the same AF tumor with a low positivity of c-KIT. Margins were narrowed. Post-surgery MRI (Figure 2C and 2D) was performed and showed a right axillary residual tumor measuring 2.1cm. Diagnosis of recurrent AF tumor with positive c-KIT and without hormonal receptor was retained. The decision to administrate imatinib 400mg daily by mouth was taken. His follow up was performed by clinical examination and was normal during 6 months. When we stopped imatinib administration during 1 month, the tumor grew to a size of 4cm. Retreatment with imatinib was decided. Clinical and MRI evaluation were performed regularly and they reported a stable tumor. The last MRI (Figure 3A) was performed in January 2014; it showed a stabilized tumor and many intratumoral calcifications. We reported no side effects regarding Common Terminology Criteria for Adverse Events. Currently he is feeling well but he continues to have ankylosis (90°) in his right upper limb due to the particular localization of AF and surgery (Figure 3B).Figure 1


Five years of local control of subscapularis aggressive fibromatosis managed by surgery and imatinib: a case report.

Dao A, Benchakroun N, Jabir H, Taleb A, Bouchbika Z, Tawfiq N, Jouhadi H, Sahraoui S, Benider A - J Med Case Rep (2014)

Images of the first recurrence. Thoracic computed tomography axial cut (A) and sagittal cut (B). Auxiliary magnetic resonance imaging with axial (C) and sagittal (D) cut showing mass measuring 130mm. This mass was localized on the right chest wall, between scapula and chest wall, hypointensity T1, hypersignal T2; this mass displaces the muscle structures; auxiliary vessels are permeable.
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4307220&req=5

Fig1: Images of the first recurrence. Thoracic computed tomography axial cut (A) and sagittal cut (B). Auxiliary magnetic resonance imaging with axial (C) and sagittal (D) cut showing mass measuring 130mm. This mass was localized on the right chest wall, between scapula and chest wall, hypointensity T1, hypersignal T2; this mass displaces the muscle structures; auxiliary vessels are permeable.
Mentions: In 2010, a 26-year-old Moroccan man was referred to our oncology and radiotherapy center for management of recurrent tumor of his right subscapularis. No pathological medical or surgical history was reported and no alcohol or tobacco habits. One year before, he had presented to his surgeon with a subscapularis tumor which had appeared gradually and increased in size during 6 months without associated pain or other symptoms. A physical examination reported a mass size 10cm, palpated in his right subscapularis region. A first resection was realized. Histopathological analysis demonstrated spindle-shaped cells with no identifiable nuclear pleomorphism or mitotic activity. There was no necrosis. A benign tumor with spindle-shaped cells is suspected. On immunohistochemical analysis, the cells stained positive for anti-smooth muscle actin, favoring a smooth muscle origin. The diagnostic of AF was retained. Unfortunately, the mass recurred within a period of 10 months and continued to increase in size reaching 13cm on a computed tomography scan and magnetic resonance imaging (MRI; Figure 1) without regional structures involved (bone, muscle or vascular). A second tumor and lymph nodes resection were performed by another surgeon who reported “a very hard resection without cleavage plane”. Histological analysis regarding two fragments size 4×3cm and 14×10×8cm led to a conclusion of AF tumor; hormonal receptor was not found. The resections of margins were narrowed and lymph node resections regarding four nodes were not involved. At assessment in our oncology and radiotherapy center after this second resection, his World Health Organization performance status was zero, weight 84kg, height 177cm. There was no induration or palpable mass. A MRI performed after the repeat surgery was normal. During follow up, a recurrence was suspected 1 year after the second resection. An axillary MRI (Figure 2A and 2B) was performed and a large mass was found in the last tumor site, measuring 12.6cm involving deltoid muscles and extending to axillary area. A third resection was realized and a histopathological examination showed the same AF tumor with a low positivity of c-KIT. Margins were narrowed. Post-surgery MRI (Figure 2C and 2D) was performed and showed a right axillary residual tumor measuring 2.1cm. Diagnosis of recurrent AF tumor with positive c-KIT and without hormonal receptor was retained. The decision to administrate imatinib 400mg daily by mouth was taken. His follow up was performed by clinical examination and was normal during 6 months. When we stopped imatinib administration during 1 month, the tumor grew to a size of 4cm. Retreatment with imatinib was decided. Clinical and MRI evaluation were performed regularly and they reported a stable tumor. The last MRI (Figure 3A) was performed in January 2014; it showed a stabilized tumor and many intratumoral calcifications. We reported no side effects regarding Common Terminology Criteria for Adverse Events. Currently he is feeling well but he continues to have ankylosis (90°) in his right upper limb due to the particular localization of AF and surgery (Figure 3B).Figure 1

Bottom Line: We reported no adverse side effects to imatinib regarding Common Terminology Criteria for Adverse Events grading.Systemic treatment with imatinib for unresectable or recurrent tumors with positive c-KIT could be the best therapeutic option.In our case report, the patient was stabilized with imatinib for 30 months and he had a very good quality of life.

View Article: PubMed Central - PubMed

Affiliation: Centre Mohammed VI pour le Traitement des Cancers, CHU Ibn Rochd, Casablanca, Morocco. daocl@yahoo.fr.

ABSTRACT

Introduction: Imatinib, a tyrosine kinase inhibitor, is a major therapeutic option for the management of unresectable aggressive fibromatosis. Unfortunately, for most patients of low or very low average income countries, surgery often is the first treatment option. This is related to unavailability of chemotherapy or targeted therapy, and to a lack of financial resources or surgeons' lack of knowledge of other therapeutic options.

Case presentation: In 2010, a 26-year-old Moroccan man was referred to our oncology and radiotherapy center by his surgeon for the management of a recurrent tumor of his right subscapularis muscle. Before his assessment in our center, two resections were performed by his surgeon after performing an incision biopsy and magnetic resonance imaging. Postoperative magnetic resonance imaging was performed and showed a right axillary nodule size 2.1cm regarding a collection with a residual tumor. We decided to administer imatinib 400mg daily by mouth. Clinical and magnetic resonance imaging evaluation were performed regularly and reported a stable tumor. We reported no adverse side effects to imatinib regarding Common Terminology Criteria for Adverse Events grading.

Conclusions: Recurrences are high during aggressive fibromatosis management. Systemic treatment with imatinib for unresectable or recurrent tumors with positive c-KIT could be the best therapeutic option. In our case report, the patient was stabilized with imatinib for 30 months and he had a very good quality of life.

Show MeSH
Related in: MedlinePlus