Two months after commencing tildrakizumab, the patient demonstrated a significant reduction in his abscess and nodule count from 45 to 6, although his VAS and DLQI scores were still high at 9 and 13, respectively. Further clinical improvement was observed at month 5 (Figs 1 and 2, with an abscess and nodule count of 5 and VAS score of 7, but a DLQI score of 19. He was able to slowly wean his prednisolone at month 2 over 3 months with no relapse of his pyoderma gangrenosum. During this time, he reported no adverse events and did not have any haematologic, hepatic or renal laboratory abnormalities. There were no other concomitant procedures or treatment prescribed, and the patient did not report any other lifestyle or behavioural changes. Although the improvement in the patient's hidradenitis suppurativa could partially be contributed by the surgical procedures he underwent prior to starting tildrakizumab, this case represents a promising therapeutic strategy for controlling pyoderma gangrenosum and treating hidradenitis suppurativa in PASH syndrome. It would be important, however, to gather more clinical data in the future, evaluating its response in a larger cohort of patients, to further determine its safety and efficacy.
BackgroundRetroperitoneal liposarcoma (RPLS) is a rare malignancy that is notorious for recurrence. Surgical resection with clean margin is the current treatment of choice. However, owing to the large retroperitoneal space, RPLSs often grow to significant sizes before being diagnosed. Neoadjuvant and adjuvant therapies have potentials to improve long term treatment outcome.Case presentationA 55-year-old Han Chinese male presented to the general surgery department with a one-year history of abdominal fullness and a one-week history of palpable right inguinal mass. At first, he was diagnosed with incarcerated inguinal hernia. However, abdominal computer tomography (CT) and biopsy confirmed his final diagnosis to be retroperitoneal well-differentiated liposarcoma, cT2bN0M0, stage IIb. The tumor, which measured 44.5cm in maximum diameter, was too large for primary surgical resection. Neoadjuvant radiotherapy with 70 Gy in 35 fractions was delivered to the tumor, which shrunk the target volume from 6300 cc to 4800 cc, as observed in the middle of the radiotherapy course. The right testicular mass also received 70Gy/35Fx. Conversion surgery was performed after radiotherapy. Unfortunately, due to residual tumor, adjuvant chemotherapy consisting of AIM (ifosfamide, Mesna, and doxorubicin) and MAID (Mesna, doxorubincin, ifosfamide, and dacarbazine) regimens were administered sequentially. Afterward, debulking surgery was conducted, plus another 18 cycles of ifosfamide monotherapy when residual tumor was still seen on CT. Since the completion of ifosfamide chemotherapy, the patient has been cancer free with no evidence of tumor recurrence for more than 26 months.ConclusionDespite conflicting evidence in the literature, our case supports the use of high dose neoadjuvant radiotherapy and adjuvant chemotherapy in treating large, unresectable RPLSs. It also highlights the importance of using individualized, multidisciplinary approach in achieving cure for large, unresectable rare tumors.
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