Angiomatoid fibrous histiocytoma: A case of local recurrence and metastases to loco‐regional lymph nodes that responded to chemotherapy

Abstract

Angiomatoid fibrous histiocytoma (AFH) is a rare soft tissue tumour with intermediate malignant potential. We report the case of a patient with local recurrence of AFH in two locations and lymph node metastases 18 months after primary surgical excision. The patient was treated with six cycles of ifosfamide and doxorubicin chemotherapy and a further three courses of ifosfamide monotherapy. Reassessment imaging showed a good response to chemotherapy with reduction in size of the two tumours of local recurrence and the lymph node metastases. This case demonstrates that AFH can respond to chemotherapy, even though it is rarely used.

PD-L1 expression in angiomatoid fibrous histiocytoma

“…In a case with unresectable and metastatic AFH, chemotherapy with vincristine, doxorubicin, dactinomycin, and cyclophosphamide achieved a complete response 16 . Six cycles of ifosfamide and doxorubicin followed by three courses of ifosfamide monotherapy were tried with a good response in a AFH with local recurrence and metastases 17 . More recently, Potter et al 23 described a clinical response to tocilizumab, an IL-6 receptor inhibitor antibody, in a child with metastatic AFH, whose disease progressed during treatment with traditional chemotherapeutic agents (vincristine, dactinomycin, and cyclophosphamide).…”

“…Albeit less frequently, a small subset of AFHs (less than 5%) metastasize, and distant metastasis is associated with mortality 1 , 12 – 15 . Treatment options for unresectable and or metastatic tumors are very limited 16 , 17 .…”

Therapeutic response of metastatic angiomatoid fibrous histiocytoma carrying EWSR1‐CREB1 fusion to the interleukin‐6 receptor antibody tocilizumab

“…Additionally, there is some rationale for considering its use either in combination with or in place of traditional chemotherapeutic agents for patients with metastatic AFH with EWSR1‐CREB1 fusion, paraneoplastic syndrome, and elevated serum IL‐6. While there are case reports of metastatic AFH responding to chemotherapy, given the high mortality rate of this disease and the risk of significant adverse effects from traditional chemotherapeutic agents, tocilizumab, which is generally well tolerated, may be a suitable alternative. The rarity and specificity of this entity precludes adequately powered randomized clinical trials of tocilizumab for this indication; however, as the EWSR1‐CREB1 fusion gene is prevalent not only in AFH, but also in other malignancies (such as pulmonary myxoid sarcoma and clear cell sarcoma), this is an area that warrants further investigation.…”

Angiomatoid fibrous histiocytoma in children: case report and the review of literature

“…Роль химиотерапии и лучевой терапии в лечении АФГ не ясна. В литературе встречаются единичные сообщения об эффективности полихимиотерапии, как правило, у пациентов с рецидивами заболевания [19,20]. Так, S. Ogden et al описали эффективность комбинации ифосфамида и доксорубицина у пациента с локорегиональным рецидивом АФГ [19].…”

“…В литературе встречаются единичные сообщения об эффективности полихимиотерапии, как правило, у пациентов с рецидивами заболевания [19,20]. Так, S. Ogden et al описали эффективность комбинации ифосфамида и доксорубицина у пациента с локорегиональным рецидивом АФГ [19]. Также описано Г Е М АТ ОЛ О Г И И и О Н КОЛ О Г И И Д Е Т С КО Й оссийский Р урнал Ж 94 2018 ТОМ 5 1 С л у ч а й и з п р а к т и к и использование лучевой терапии у пациента с третьим рецидивом опухоли, в результате которой была достигнута длительная бессобытийная выживаемость [21].…”