Case Report: A novel chemotherapy-free regimen combined with photodynamic therapy, target therapy, and immunotherapy in a geriatric male with huge recurrent scalp and facial angiosarcoma: a report of an extremely rare case and literature review

Abstract

Angiosarcomas are sporadic vascular neoplasms among the most aggressive subtypes of soft tissue sarcomas. In addition, vast and multiple recurrent superficial scalp and facial angiosarcomas are very complex and extremely difficult to manage. Their occurrence brings about significant social and emotional distress to affected individuals. To date, no specific therapeutic strategy has been the most effective and reliable. Herein, we report a highly unique case of a geriatric male patient with recurrent scalp and facial angiosarcoma successfully treated by a chemotherapy-free regimen consisting of photodynamic therapy (PDT), immunotherapy, and target therapy. Notably, PDT provided promising remarkable auspicious outcomes and proved to be a better therapeutic option for refractory malignant angiosarcomas.

Keywords: chemotherapy-free; geriatric patient; immunotherapy; photodynamic therapy; recurrent; scalp and facial angiosarcomas; target therapy.

Figure 1

(A) Scalp hemangiosarcoma specimen (4×, by K-Viewer), post-operative pathological findings Hematoxylin and eosin (HE) staining and Immunohistochemical staining of CD31(+), ERG (+), D2-40(+), Ki-67 (20%). (B) Detection of peripheral blood immunological evaluation by a flow cytometric technique.

Figure 2

(A-D) Negative CTLA4+CD8 cells and positive LAG3+CD8, TIM3+CD8, PD-1+CD8, CD-CD19-CD14+CD16-HLA-DR cells were confirmed by a flow cytometric technique.

Figure 3

(A–L) CT scan images and 3D reconstruction printing of the patient before and after PDT treatment: (A–C) CT scan presented bilateral periorbital, bilateral frontal, left parietal, right temporal, and left mandibular paracortical subcutaneous soft tissue lesions of the patient after the first time PDT treatment; (D–F) CT scan showed some obvious and significant decrease in size of the recurrent malignant facial lesions before the second time PDT treatment. (G–I) 3D imaging of the patient’s tumor site before treatment, and (J–L) post-treatment. (G, J) the positive view. (H, K) the right view. (I, L) the left view.

Figure 4

(A-G) The entire multiple recurrent angiosarcoma lesion morphological and appearance changes before and after PDT irradiation laser treatment. (A) the appearance of the tumor the first time on admission before PDT irradiation, (B-E) second visit just after having undergone PDT treatment, day 22, almost diminishing, (F, G) tumor appearance on day 44, last PDT irradiation, only scarring observed, tumor completely diminished and healed.

Figure 5

A schematic diagram of the synergistic effect of PDT, targeted therapy, and immunotherapy. Red arrows: under laser irradiation of PDT, O² in the tumor microenvironment is converted to ¹O₂, generating several numbers of ROS to directly kill tumor cells. Then, neutrophils, macrophages, and dendritic cells are recruited into some numbers to surround the tumor cells, which undergo necrosis, apoptosis, and cell proptosis. Green arrows: The dead cell fragments and associated antigens, DAMPS, stimulate the DCs again, causing activation and dramatic proliferation of T-cells. A large infiltration of CD8+ T-cells and CD4+ T-cells then appears in the tumor tissue. Meanwhile, PDT causes T-cell infiltration. Based on the above, PD-1 inhibitors can better kill tumor cells. Blue arrows: ROS induced by PDT can destroy tumor endothelial tissue, and in combination with targeted therapeutic drugs, will cause massive death of tumor tissue endothelial cells, leading to hypoxic necrosis of tumor microvessels. In summary, the combination of PDT target therapy, and immunotherapy appears to be a very good synergy to achieve a better killing effect on tumor cells.