ABSTRACT
BACKGROUND: Desmoid tumors are intermediary malignant, fibrous lesions occurring in various soft tissues. Surgical treatment is relentlessly challenging because of the propensity for local aggressive behavior and high risk of recurrence. Consequently, a wide range of oncological drugs and radiation therapy are being used; however, outcomes are unpredictable. We investigated whether local treatment with an oncolytic peptide could be beneficial in a patient with an unresectable desmoid tumor. CASE PRESENTATION: In a young 29-year-old Caucasian woman who was diagnosed with a retromammary desmoid tumor infiltrating deeply into the anterior thoracic wall, surgery was considered excessively mutilating, and observation was recommended. The lesion progressed, however, and caused debilitating pain, despite nonsteroidal anti-inflammatory medication. Subcutaneous injections of human interferon-α (Multiferon®) resulted in reduced growth kinetics but had to be terminated because of development of symptomatic pneumonitis. Frequently used oncological treatment was withheld because of the toxicity profile, and the patient was instead included in a phase I study investigating transdermal intratumoral injection of LTX-315, an oncolytic peptide that induces anticancer immune responses ( ClinicalTrials.gov , NCT01986426 ). A marked increase of CD8+ tumor-infiltrating T cells in the lesion was complemented by upregulation of immune gene signature (including effector T-cell, T-helper type 1 cell, chemokine, and cytokine genes). These changes were followed by gradual symptom relief and long-term disease stabilization, indicating clinical benefit. LTX-315 was well tolerated until termination in week 16 after a serious allergic reaction. CONCLUSIONS: Our patient was treated with repeated intratumoral injections of LTX-315, resulting in tumor regression accompanied by upregulation of immune genes and T-cell infiltration. Local application of immunotherapy, minimizing systemic side effects, represents a novel treatment modality in desmoid tumors that should be tested in further clinical trials.
Subject(s)
Fibromatosis, Aggressive , Oligopeptides/administration & dosage , Thoracic Wall , Adult , Antineoplastic Agents/administration & dosage , CD8-Positive T-Lymphocytes/immunology , Diagnosis, Differential , Female , Fibromatosis, Aggressive/immunology , Fibromatosis, Aggressive/pathology , Fibromatosis, Aggressive/physiopathology , Fibromatosis, Aggressive/therapy , Humans , Injections, Intralesional , Positron Emission Tomography Computed Tomography/methods , Remission Induction , Thoracic Wall/diagnostic imaging , Thoracic Wall/pathology , Tumor BurdenABSTRACT
Context: Autonomous cortisol secretion (ACS) can be unilateral or bilateral irrespective of the presence of an adrenal tumor. A reliable method to distinguish between unilateral and bilateral ACS is lacking. Objective: Evaluate the use of adrenal venous sampling (AVS) to distinguish between unilateral and bilateral ACS. Design and Methods: This was a prospective study of AVS in patients with adrenal tumors who received a diagnosis of ACS or adrenal Cushing syndrome (CS). Unilateral secretion was defined as >2.3-fold difference in cortisol levels between the two adrenal veins. Metanephrine levels were used to ascertain correct catheter position. Results were correlated with findings on CT and iodine-131-cholesterol scintigraphy. Results: Thirty-nine patients underwent AVS; there were no complications. The procedure was inconclusive in six patients and repeated with success in one, giving a success rate of 85%, and leaving 34 procedures for evaluation (adrenal CS, n = 2; ACS, n = 32). Of 14 patients with bilateral tumors, 10 had bilateral and 4 had unilateral overproduction. Of 20 patients with unilateral tumors, 11 had lateralization to the side of the tumor and the remaining had bilateral secretion. Cholesterol scintigraphy findings were concordant with those of AVS in 13 of 18 cases (72%) and discordant in 5 (28%). Conclusion: Laterality of ACS does not always correspond to findings on CT images. AVS is a safe and valuable tool for differentiation between unilateral and bilateral cortisol secretion and should be considered when operative treatment of ACS is a possibility.
Subject(s)
Adrenal Gland Neoplasms/diagnosis , Adrenal Glands/metabolism , Cushing Syndrome/diagnosis , Hydrocortisone/blood , Hyperaldosteronism/diagnosis , Adrenal Gland Neoplasms/blood , Adrenal Gland Neoplasms/complications , Adrenal Glands/blood supply , Adrenal Glands/diagnostic imaging , Adult , Aged , Catheterization/methods , Cholesterol/administration & dosage , Cholesterol/chemistry , Contrast Media/administration & dosage , Cushing Syndrome/blood , Cushing Syndrome/complications , Diagnosis, Differential , Feasibility Studies , Female , Femoral Vein/surgery , Humans , Hydrocortisone/metabolism , Hyperaldosteronism/blood , Hyperaldosteronism/etiology , Iodine Radioisotopes/administration & dosage , Iodine Radioisotopes/chemistry , Male , Middle Aged , Prospective Studies , Radionuclide Imaging/methods , Tomography, X-Ray ComputedABSTRACT
Intravenous drug abusers commonly develop endocarditis due to injection of particulate matter that can cause endothelial damage to the valves. The frequent need to access the venous system can result in vascular traumas with potential complications including arteriovenous (AV) fistulas. Here, we present the case of an intravenous drug abuser with endocarditis and an unusually large AV fistula in the groin. The patient was successfully operated for endocarditis. However, the AV fistula was at the time not acknowledged. The combination of ileofemoral vein thrombosis and a large AV fistula led to pulmonary septic embolism and life-threating, right-sided heart failure. Computed tomography scan did not reveal the AV fistula, but suspicion was raised. Ultrasound diagnosed and revealed the magnitude of the AV fistula, and the patient was treated with a minimally invasive percutaneous technique.
