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A Patient With Metastatic Sarcoma was Successfully Treated With Radiolabeled Somatostatin Analogs

Crespo-Jara, Aurora MD; González Manzano, Ramón MD, PhD; Lopera Sierra, Maribel MD; Redal Peña, María Carmen MD; Brugarolas Masllorens, Antonio MD, PhD

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doi: 10.1097/RLU.0000000000001288
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A 36-year-old woman with a diagnosis of a left popliteal synovial sarcoma treated with surgery and chemotherapy (adryamicin/ifosfamide). A local recurrence required additional surgery followed by chemoradiotherapy (70 Gy plus ifosfamide). She had a disease-free interval of 4 years until lung metastasis was detected. Between October 2007 and May 2012, she was treated with several drug combinations (in total, she had been resistant to >6 lines of therapy including all the available active drugs in soft tissue sarcomas) and operated by thoracotomy in 3 stages. Pazopanib treatment was initiated in November 2012 and then interrupted because of hemoptysis. She was referred to our institution in February 2013 presenting extreme weight loss and a performance status of 2. The most recent scan showed lung metastasis in more than 50% of the right side of the thorax, with large pleural disease and mediastinum involvement. Traqueal compression at carina level was present. A computed tomography–guided needle biopsy from a lung metastatic lesion was performed for an expression microarray. A highly significant expression of the somatostatin receptor 2 (SSTR2) gene (in excess of 10-fold) and to a lesser extent SSTR5 (>5-fold) as compared with the normal control tissue was apparent in the normalized microarray data. There are references in literature identifying SSTR in more than 80% of the soft tissue tumors analyzed by reverse transcriptase–polymerase chain reaction,1,2 as well as positive uptake in molecular imaging.3,4 Figure 1 shows the high uptake in the right hemithorax in the Octreoscan confirming the potential indication for somatostatin analog–based treatments. She began lanreotide 30 mg intramuscularly every 2 to 3 weeks until May 31, 2013, reaching disease stabilization.
She underwent 2 cycles with Lutathera, a radiolabeled somatostatin analog that has been shown to be an effective and safe treatment for somatostatin receptor–positive gastroenteropancreatic neuroendocrine tumors5–7: 5.55 GBq in July 2013 and 7.4 GBq in September 2013. Figure 2 shows posttherapy 177Lu-octreotate whole-body scans (A, first course, July 2013; B, second course, September 2013). A superior uptake than the baseline Octreoscan can be shown in the first 177Lu-octreotate scan and additionally 2 implants upon the chest wall and a new local recurrence of the primary lesion in the left knee. Tolerance was excellent as it has been previously reported.8,9 After the first cycle, 1 episode of hemoptysis was observed, without any hematologic disturbance, and resolved with symptomatic medication. The patient had a very important clinical improvement from the beginning of the treatment, better quality of life, performance status, and daily life activities. In addition, a significant reduction of lung masses can be shown by comparison of both 177Lu-octreotate scan after the first single course.
Objective partial response based on RECIST criteria (reduction of >50%) was observed after 2 courses of Lutathera. Figure 3 shows baseline thoracic computed tomography scan (A–C) and after 11 months (D–F). The overall duration of her response (stabilization with cold lanreotide and partial response with Lutathera) exceeded 1 year. The objective reduction made possible a surgical approach in an attempt to reduce the tumor load, and therefore a thoracotomy and debulking was carried out in January 2014. Unfortunately, an accidental rupture of the right atrium with profuse bleeding occurred during surgery. She died in April 2014 after a long stay in the intensive care unit. Lutathera is an investigational medical product in late-stage development.


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    microarray expression; Octreoscan; peptide receptor radionuclide therapy; radiolabeled somatostatin analogs; sarcoma; somatostatin receptors; synovial sarcoma

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