Giant cell tumors (GCTs) of the skull base are rare entities. Although considered histologically benign, GCTs are locally aggressive with a high rate of local recurrence. The present case describes a 14-year-old girl with a clival GCT who underwent long-term therapy with denosumab after local relapse. To our knowledge, it is the second case described with a follow-up term > 2 years from the start of denosumab and who did not receive any other adjuvant treatment besides denosumab. The patient achieved a local control of the disease. According to the few available data, radical excision with adjuvant therapy helps in long-term control in uncommon sites, such as the skull. However, the definitive treatment is still controversial because of their rarity and few follow-up data. The present case highlights the benefit of denosumab and its safety as long-term therapy and contributes to the existing literature with analysis and evaluation of the management strategies and prognosis.
11520 Background: Currently, there is no accepted standard chemotherapy regimen for recurrent osteosarcoma. We performed a retrospective study to evaluate the effectiveness, toxicity and clinical benefit of second line therapy with Gemcitabine (G) in combination with Docetaxel (D), in patients with relapsed high-grade osteosarcoma who have been previously treated with High-dose Ifosfamide (HD-IFO) during first-line treatment (ClinicalTrials ID: NCT04651179). Methods: Patients were eligible to the analysis according to the following criteria: episode of first relapse of osteosarcoma, previous first line treatment according to poor responders’ arm of ISG-AIEOP OS 2 Protocol based on methotrexate, cisplatin, doxorubicin and HD-IFO (3gr/m2/day, day 1-5) ± Mifamurtide, presence of measurable disease according to RECIST 1.1, treatment with at least two cycles of Gemcitabine 900 mg/m2/day, days 1 and 8 and Docetaxel 75 mg/m2 day 8 every 21 days. Primary objective: overall survival (OS) at 12 months; secondary objectives: overall response rate (ORR) (complete response + partial response), progression-free survival (PFS) at 6-12 months, toxicity and quality of life. Results: 14 patients were included in the analysis. All patients were previously treated according to poor responders’ arm of ISG-AIEOP OS 2 Protocol, 7/14 (50%) of them received Mifamurtide. Most of patients (12/14, 86%) received 4 GD cycles and the total number of cycles administered was 60. Median OS was 20 months (range 11-69); OS at 12 and 24 months was 84% (95% CI 65-100) and 51% (95% CI 22-79), respectively; ORR were 35,7% after six cycles. Median PFS was 12 months (range 2-69); 6-months and 12-months PFS were 86% (95% CI 67-100) and 56% (30-83) respectively. For 8/14 (57%) patients a surgical approach was feasible after 2 and 6 GD cycles. Previous use of Mifamurtide correlate with a better 6-months PFS rate (100% vs 57%, P = 0.0011). Also, amelioration of ECOG/Lansky score showed a positive correlation with both PFS (P = 0.01) and OS (P = 0.04). No extra-hematological toxicities grade ≥3 was observed according to CTCAE v4.03 criteria. Conclusions: This trial confirms that GD combination has an anti-tumor activity for relapsed high-grade osteosarcoma with a well-tolerated toxicity profile. Furthermore, GD guarantee a surgical tumor remotion for patients with a chemo-resistant disease such as patients previously treated with a poor responders’ arm improving their OS and PFS rate. Promising results for patients previously treated with Mifamurtide need to be confirmed in future trials. Clinical trial information: NCT04651179.
The paper describes the case report of a sepsis due to Yersinia enterocolitica in an ex preterm infant of 32 weeks’ gestational age with birth weight of 1,220 g. The infant was admitted at 39 weeks postconceptual age to the emergency room for fever, feeding’s difficulty and abdominal pain with bloody stools. The physicians suspected the diagnosis of enterocolitis and Yersinia enterocolitica finding in the blood stream confirmed the diagnosis. The infant’s conditions improved under antibiotic treatment. Family history was negative, except for the particular anamnestic data of the domestic coexistence with dog puppies. Y. enterocolitica is an uncommon pathogen affecting infants, causing illness ranging from self-limited enteritis to life-threatening systemic infection, mostly in neonates. It is responsible for zoonosis, acquired from the ingestion of contaminated food or the contact with sick pets. This case-report provides an example of clinical presentation of Y. enterocolitica in early childhood.
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