Successful Superficial Blood Sampling to Localize a Fibroblast Growth Factor-23-Producing Tumor.

BACKGROUND Tumor-induced osteomalacia (TIO) is a paraneoplastic syndrome caused by aberrant fibroblast growth factor-23 (FGF-23)-producing tumors. Early surgical resection is the optimal strategy for preventing TIO progression. Thus, tumor localization is a priority for successful treatment. A simple and safe examination method to identify functional endocrine tumors is essential to achieve better outcomes in patients with TIO. CASE REPORT A 64-year-old Japanese man with recurrent fractures, hypophosphatemia, and elevated alkaline phosphatase and FGF-23 levels (109 pg/mL) was admitted to our university hospital and was diagnosed with FGF23-related hypophosphatemic osteomalacia. Notably, the superficial dorsal vein in the patient's left foot exhibited a high FGF-23 level (7510 pg/mL). Octreotide and ¹8F-fluorodeoxyglucose (FDG) scintigraphy and systemic venous sampling revealed that the tumor in the third basal phalanx of the left foot was responsible for FGF-23 overproduction. Tumor resection resulted in a rapid decrease in serum FGF-23 levels and an increase in serum phosphorus levels. CONCLUSIONS Octreotide scintigraphy, FDG-positron emission tomography, and systemic venous sampling are the standard methods for localizing functional endocrine tumors. However, the limited availability and invasive nature of these examinations hinder effective treatment. Here, we highlight the importance of peripheral superficial blood sampling as an alternative to conventional systemic methods for confirming the presence of FGF-23-producing tumors. Clinicians should consider TIO as a potential cause of acquired hypophosphatemic osteomalacia. Furthermore, peripheral superficial vein blood sampling may be useful for confirming the localization of FGF-23-producing tumors.

Head and Neck Arteriovenous Malformations: Clinical Manifestations and Endovascular Treatments.

Arteriovenous malformations (AVMs) are vascular malformations that present high-flow direct communication between the arteries and veins, not involving the capillary beds. They can be progressive and lead to various manifestations, including abnormal skin or mucosal findings, ischemia, hemorrhage, and high-output heart failure in severe cases. AVMs often involve the head and neck region. Head and neck AVMs can present region-specific clinical manifestations, angioarchitecture, and complications, especially in cosmetic appearance and ingestion, respiratory, and neuronal functions. Therefore, when planning endovascular treatment of head and neck AVMs, physicians should consider not only the treatment strategy but also the preservation of the cosmetic appearance and critical functions. Knowledge of the functional vascular anatomy as well as treatment techniques should facilitate a successful management. This review summarizes AVMs' clinical manifestations, imaging findings, treatment strategy, and complications.

Value of fat-suppressed T2-weighted imaging for predicting short-term pain relief after sclerotherapy for venous malformations in the extremities.

PURPOSE: This study aimed to evaluate the value of fat-suppressed T2-weighted imaging (FS-T2WI) for predicting short-term pain relief after polidocanol sclerotherapy for painful venous malformations (VMs) in the extremities. MATERIALS AND METHODS: This retrospective study included patients with painful VMs in the extremities between October 2014 and September 2021, had their first sclerotherapy without history of surgical therapy, and underwent magnetic resonance imaging before sclerotherapy. Pain relief was assessed 2 months after 3% polidocanol sclerotherapy and was categorized as follows: progression, no change, partial relief, or free of pain. The associations between pain relief and imaging features on FS-T2WI were analyzed. RESULTS: The study included 51 patients. The no change, partial relief, and free of pain groups included 6 (11.8%), 25 (49.0%), and 20 (39.2%) patients, respectively. No patient experienced progressive pain. The lesion diameter was ≤ 50 mm in 13 (65.0%) patients in the free of pain group, whereas it was > 50 mm in all patients in the no change group (p = 0.019). The lesions showed well-defined margin in 15 (75.0%) patients in the free of pain group, whereas they showed ill-defined margin in 5 (83.3%) patients in the no change group (p = 0.034). The most common morphological type was cavitary in the free of pain group (14 [70.0%] patients), whereas there was no patient with cavitary type lesion in the no change group (p = 0.003). Drainage vein was demonstrated in 6 (100%), 22 (88.0%), and 11 (55.0%) patients in the no change, partial relief, and free of pain group, respectively (p = 0.011). CONCLUSION: A lesion size of 50 mm or less, a well-defined margin, a cavitary type, and no drainage vein on FS-T2WI were significant features for predicting short-term pain relief after polidocanol sclerotherapy for painful VMs in the extremities.

Utility of non-contrast-enhanced magnetic resonance imaging in predicting preoperative clinical stage and prognosis in patients with thymic epithelial tumor.

PURPOSE: The purpose of this study was to find useful imaging features on non-contrast-enhanced magnetic resonance imaging (MRI) that can divide patients with thymic epithelial tumor (TET) into clinical stage I-II and III-IV groups under assumption that contrast media are contraindicated. MATERIALS AND METHODS: This retrospective study included 106 patients (median age, 60 years; range, 27-82 years; 62 women) with surgically resected TET who underwent MRI between August 1986 and July 2015. All cases were classified according to the 2015 WHO classification and staged using the eighth edition of the TNM system. Two radiologists independently evaluated 14 categories of MRI findings; the findings in patients with stage I-II were compared with those of patients with stage III-IV using a logistic regression model. Disease-specific survival associated with significant findings was calculated using the Kaplan-Meier method. RESULTS: Univariate analysis showed that stage III-IV patients were more likely to have tumors with an irregular contour, heterogeneity on T1WI, low-signal intensity on T2WI, irregular border with lung, findings of great vessel invasion (GVI) (hereafter, GVI sign), pericardial thickening/nodule, and lymphadenopathy (all, P < 0.01). On multivariable analysis, only two findings, irregular border between tumor and lung (odds ratio [OR], 272.8; 95% CI 26.6-2794.1; P < 0.001) and positive GVI sign (OR, 49.3; 95% CI 4.5-539.8; P = 0.001) remained statistically significant. Patients with one or both features had significantly worse survival (log-rank test, P < 0.001). CONCLUSION: For patients with TET who are unable to receive contrast for preoperative staging, the two image findings of an irregular border between tumor and lung and the positive GVI sign on non-contrast-enhanced MRI could be helpful in determining stage III-IV disease which is associated with a worse survival.

Super-selective and staged glue embolization for labral AVMs using ultra-thin microcatheters: report of two cases.

BACKGROUND: Treating arteriovenous malformation (AVM) is challenging because of the high recurrence rate and because incomplete resection or embolization can induce aggressive growth. However, a standard strategy is not fully established. Although transcatheter arterial embolization (TAE) is currently almost always part of the treatment, in many cases, single treatment is not curative and only palliative. Additionally, the success and complication rates associated with TAE alone are unclear, and there has been limited study of staged TAE for facial AVMs. Furthermore, few reports have described the details of the procedure. CASE-PRESENTATION: We report two cases of AVM of the upper lip in patients who were successfully treated by staged super-selective TAE at several-month intervals using ultra-thin microcatheters and n-butyl-2-cyanoacrylate. CONCLUSION: Staged and super-selective TAE may prevent complications and provide high curability and might be a useful treatment in cases of AVM.

Transarterial Embolization of Neovascularity for Refractory Nighttime Shoulder Pain: A Multicenter, Open-Label, Feasibility Trial.

PURPOSE: To assess the feasibility of transarterial embolization (TAE) for recalcitrant nighttime shoulder pain in a multicentric study. MATERIALS AND METHODS: This prospective, open-label, feasibility trial included 100 patients treated at 5 institutions. TAE was performed in 76 patients with adhesive capsulitis (AC) and 24 patients with symptomatic rotator cuff tears (sRCTs). The ipsilateral radial artery was punctured, and imipenem/cilastatin sodium was infused as an embolic agent. Adverse events, 10 point pain numerical rating scale (NRS), range of motion (ROM) of the shoulder joint, and quality of life (via the EuroQol-5D [EQ-5D]) were evaluated. RESULTS: All patients exhibited neovascularity on baseline angiography, and all TAE procedures were performed successfully. No patient experienced a major adverse event. The mean nighttime pain NRS scores at baseline and 1, 3, and 6 months after TAE were 6.4 ± 2.2, 3.4 ± 2.6, 2.3 ± 2.5, and 1.6 ± 2.2, respectively (for all, P < .001). The mean ROM of anterior elevation at baseline and 1, 3, and 6 months after TAE were 97° ± 29°, 119° ± 28°, 135° ± 27°, and 151° ± 17°, respectively (for all, P < .001). The mean EQ-5D scores at baseline and 1, 3, and 6 months after TAE were 0.63 ± 0.17, 0.73 ± 0.16, 0.80 ± 0.17, and 0.84 ± 0.17, respectively (for all, P < .001). There was no significant difference in the clinical success rate between the AC and sRCT groups. CONCLUSIONS: TAE for nighttime shoulder pain caused by AC and sRCTs was feasible with sufficient safety and efficacy.

Stent graft placement guided by intravascular ultrasound for subclavian artery dissecting aneurysm.

Subclavian artery dissecting aneurysm is relatively rare and can be caused by traumatic, nontraumatic, and iatrogenic etiologies. Surgical management of subclavian artery dissecting aneurysm has been sparsely reported. Recently, due advances in endovascular techniques making them less invasive, these approaches have become more standard as treatments. Subclavian artery dissecting aneurysm management usually depends on whether there is ischemia of the tissues supplied by the subclavian artery. Furthermore, treatment strategies depend on which section of the artery is involved. In particular, treatment is difficult if the dissecting aneurysm has branching vessels. In this case report, we show that endovascular repair using a covered stent graft is a promising approach to repair a subclavian artery dissecting aneurysm. In this case, the stent graft was highly effective, and follow-up examinations showed good patency of the subclavian artery. Additional use of IVUS (Volcano Inc.; Rancho Cordova, CA, USA) is helpful to obtain the precise location of the true lumen of a dissecting aneurysm.

T1 invasive lung adenocarcinoma: Thin-section CT solid score and histological periostin expression predict tumor recurrence.

Adenocarcinoma is the most common histological type of non-small cell lung cancer (NSCLC), and various biomarkers for predicting its prognosis after surgical resection have been suggested, particularly in early-stage lung adenocarcinoma. Periostin (also referred to as POSTN, PN or osteoblast-specific factor) is an extracellular matrix protein, the expression of which is associated with tumor invasiveness in patients with NSCLC. In the present study, the novel approach, in which the thin-section CT findings prior to surgical resection and periostin expression of resected specimens were analyzed in combination, was undertaken to assess whether the findings could be a biomarker for predicting the outcomes following resection of T1 invasive lung adenocarcinoma. A total of 73 patients who underwent surgical resection between January 2000 and December 2009 were enrolled. A total of seven parameters were assessed in the thin-section CT scans: i) Contour; ii) part-solid ground-glass nodule or solid nodule; iii) percentage of solid component (the CT solid score); iv) presence of air-bronchogram and/or bubble-like lucencies; v) number of involved vessels; vi) shape linear strands between the nodule and the visceral pleura; and vii) number of linear strands between the nodule and the visceral pleura. Two chest radiologists independently assessed the parameters. Periostin expression was evaluated on the basis of the strength and extent of staining. Univariate and multivariate analyses were subsequently performed using the Cox proportional hazards model. There was a substantial to almost perfect agreement between the two observers with regard to classification of the seven thin-section CT parameters (κ=0.64-0.85). In the univariate analysis, a CT solid score >80%, pathological lymphatic invasion, tumor and lymph node status and high periostin expression were significantly associated with recurrence (all P<0.05). Multivariate analysis demonstrated that a CT solid score >80% and high periostin expression were risk factors for recurrence (P=0.002 and P=0.011, respectively). The cumulative recurrence rates among the three groups (both negative, CT solid score >80% or high periostin expression, or both positive) were significantly different (log-rank test, P<0.001). Although the solid component is already known to be a major predictor of outcome in lung adenocarcinomas according to previous studies, the combined analysis of CT solid score and periostin expression might predict the likelihood of tumor recurrence more precisely.

Vessel Occlusion using Hydrogel-Coated versus Nonhydrogel Embolization Coils in Peripheral Arterial Applications: A Prospective, Multicenter, Randomized Trial.

PURPOSE: To evaluate the safety and effectiveness of hydrogel-coated coils for vessel occlusion in the body trunk. MATERIALS AND METHODS: A total of 77 patients with various peripheral vascular lesions, treatable by embolization with coils, were randomized (hydrogel group, n = 38; nonhydrogel group, n = 39). In the hydrogel group, embolization of the target vessel was conducted using 0.018-inch hydrogel-coated coils (AZUR 18; Terumo Medical Corporation, Tokyo, Japan) with or without bare platinum coils. The nonhydrogel group received both bare platinum coils and fibered coils without the use of hydrogel-coated coils. RESULTS: Complete target vessel occlusion was accomplished in 36 patients in the hydrogel group and 37 patients in the nonhydrogel group. No major adverse events were observed in either group. The median number of coils/vessel diameter and the median total coil length/vessel diameter were significantly larger in the nonhydrogel group than in the hydrogel group (P = .005 and P = .004, respectively). The median embolization length was significantly longer in the nonhydrogel group (31.95 mm) than in the hydrogel group (23.43 mm) (P = .002). If no expansion was assumed, the median packing density in the hydrogel group was 44.9%, which was similar to that in the nonhydrogel group (46.5%) (P = .79). With full expansion assumed, the median packing density in the hydrogel group was 125.7%. CONCLUSIONS: Hydrogel-coated coils can be safely used for peripheral vascular coil embolization, and hydrogel-coated and conventional coils in combination allow for a shorter embolization segment and shorter coil length.

Natural History of Unruptured Visceral Artery Aneurysms Due to Segmental Arterial Mediolysis and Efficacy of Transcatheter Arterial Embolization: A Retrospective Multiinstitutional Study in Japan.

OBJECTIVE. The purpose of this study was to clarify the natural history of unruptured visceral artery aneurysms due to segmental arterial mediolysis and the efficacy of transcatheter arterial embolization. MATERIALS AND METHODS. Patients with a pathologic or clinical diagnosis of visceral artery aneurysms due to segmental arterial mediolysis between 2005 and 2015 were enrolled. For patients with clinical diagnoses, images were collected and assessed by central radiologic review. To clarify the natural history of unruptured aneurysms, the morphologic changes were assessed. The efficacy and safety of transcatheter arterial embolization for aneurysms due to segmental arterial mediolysis were evaluated. RESULTS. Forty-five patients with 123 aneurysms due to segmental arterial mediolysis were enrolled. Among the 123 aneurysms, 70 unruptured aneurysms were evaluated for natural history. Forty-five of the 70 (64%) aneurysms had no change in morphology. Among the other 25 aneurysms, nine (13% of the 70) were reduced in size, 13 (19%) disappeared, and three (4%) were newly found at follow-up. Aneurysms of the middle colic artery were ruptured in 10 of 11 (91%) cases. Transcatheter arterial embolization was performed on 45 aneurysms and was successful in all cases but caused slight arterial injury in three cases (6.7%). CONCLUSION. At initial diagnosis, unruptured aneurysms due to segmental arterial mediolysis are likely to be stable or to resolve, but the risk of rupture of aneurysms of the middle colic artery appears high. Transcatheter arterial embolization is a useful treatment, but careful manipulation is necessary.

Location of embolization affects patency after coil embolization for pulmonary arteriovenous malformations: importance of time-resolved magnetic resonance angiography for diagnosis of patency.

OBJECTIVES: This study aimed to assess the diagnostic accuracy of computed tomography (CT) and time-resolved magnetic resonance angiography (TR-MRA) for patency after coil embolization of pulmonary arteriovenous malformations (PAVMs) and identify factors affecting patency. METHODS: Data from the records of 205 patients with 378 untreated PAVMs were retrospectively analyzed. Differences in proportional reduction of the sac or draining vein on CT between occluded and patent PAVMs were examined, and receiver operating characteristic analysis was performed to assess the accuracy of CT using digital subtraction angiography (DSA) as the definitive diagnostic modality. The accuracy of TR-MRA was also assessed in comparison to DSA. Potential factors affecting patency, including sex, age, number of PAVMs, location of PAVMs, type of PAVM, and location of embolization, were evaluated. RESULTS: The sensitivity, specificity, positive predictive value (PPV), negative predictive value (NPV), and accuracy of CT were 82%, 81%, 77%, 85%, and 82%, respectively, when the reduction rate threshold was set to 55%, which led to the highest diagnostic accuracy. The sensitivity, specificity, PPV, NPV, and accuracy of TR-MRA were 89%, 95%, 89%, 95%, and 93%, respectively. On both univariable and multivariable analyses, embolization of the distal position to the last normal branch of the pulmonary artery was a factor that significantly affected the prevention of patency. CONCLUSIONS: TR-MRA appears to be an appropriate method for follow-up examinations due to its high accuracy for the diagnosis of patency after coil embolization of PAVMs. The location of embolization is a factor affecting patency. KEY POINTS: • Diagnosis of patency after coil embolization for pulmonary arteriovenous malformations (PAVMs) is important because a patent PAVM can lead to neurologic complications. • The diagnostic accuracies of CT with a cutoff value of 55% and TR-MRA were 82% and 93%, respectively. • The positioning of the coils relative to the sac and the last normal branch of the artery was significant for preventing PAVM patency.

Follow-up non-contrast MRA after treatment of intracranial aneurysms using microcoils with prominent metallic artifact: a comparative study of TOF-MRA and Silent MRA.

PURPOSE: Some of the detachable microcoils are associated with the prominent metallic artifact. We have applied Silent MRA to reduce the artifact. In this study, we present a retrospective study in which Silent MRA is used for cases showing prominent metallic artifact on conventional TOF-MRA due to a detachable bare platinum microcoil (Barricade coil). MATERIALS AND METHODS: Fifteen patients, who had undergone endosaccular embolization using Barricade coil and other detachable microcoils up to 3 days previously, were scanned with TOF-MRA and silent MRA at the same time. The treatment DSA and follow-up MRA images were graded by two experienced neuroradiologists, focusing on the visibility of residual aneurysm and parent arterial lumen. RESULTS: DSA images showed residual aneurysm (RA) in four, residual neck (RN) in six, and complete occlusion (CO) in five patients. TOF-MRA images showed RN in five, CO in four, mild defect (MD) in one, severe defect (SD) in three, and complete defect in two. In contrast, on Silent MRA, the grades were RA in two, RN in five, CO in five, and MD in three. CONCLUSION: Barricade coils are associated with prominent metallic artifact on TOF-MRA. Silent MRA is useful for follow-up MRA after embolization using Barricade coils. The metallic artifacts were compared between TOF-MRA and Silent MRA in patients treated by using Barricade coils. Barricade coils are associated with more metallic artifact on TOF-MRA than Silent MRA. Silent MRA is useful for follow-up MRA after embolization using Barricade coils.

Internal coil packing method for the Amplatzer vascular plug 4.

The Amplatzer vascular plugs (AVPs), including AVP 4, have excellent occlusion; however, insufficient embolization or recanalization may occasionally occur. This study aimed to evaluate the feasibility and effectiveness of internal coil packing method to insert microcoils into the AVP 4 using an experimental vascular model. The insertion of a 1.7 F microcatheter through the nitinol mesh of the AVP 4 was not possible. Only 0.010-inch electrolytic detachable microcoils could be inserted through catheter tips in contact with nitinol mesh. The internal coil packing of the AVP 4 may be accomplished by inserting 0.010-inch microcoils into the AVP 4 in cases of continued perfusion or recanalization with required short-segment embolization.

A Rare Case of Cerebral Air Embolism Caused by Pulmonary Arteriovenous Malformation After Removal of a Central Venous Catheter.

Cerebral air embolism following central venous catheter (CVC) removal is extremely rare. We report a case of cerebral air embolism with loss of consciousness after removal of CVC caused by pulmonary arteriovenous malformation (PAVM). Computed tomography revealed air bubbles in the internal carotid arteries along the sulci in the cerebral hemispheres, as well as a PAVM. The cerebral air embolism was treated with hyperbaric oxygen and intravenous thrombolytic therapy, and transcatheter embolization of the PAVM was performed. When inserting/removing CVC in a patient with a small PAVM, treatment of the PAVM, irrespective of its size, could prevent the type of complication that occurred in our present case.

Successful treatment of a patient with chyluria due to lymphangioleiomyomatosis using sirolimus.

Lymphangioleiomyomatosis (LAM) is a rare and progressive neoplastic disease of young woman, characterized by the proliferation of abnormal smooth muscle-like cells (LAM cells) in the lungs and axial lymphatics. A 44-year-old woman was referred to our hospital because pleural effusion was detected during a health checkup. She had chylothorax, chylous ascites, and chyluria, and her computed tomography scan showed a solid tumor in the pelvis. Surgical biopsy was performed; she was diagnosed as having LAM. We could not control the fluid collection and chyluria using standard medical treatments. Therefore, we chose to administer sirolimus, and her symptoms dramatically improved. The mechanism of chyluria presumably involved LAM cell infiltrates in the ureter via the lymphatic vessel flow, which causes LAM to develop because of ureter wall exposure.

Treatment of a Large Pharyngeal Venous Malformation using Transoral Sclerotherapy.

Management of venous malformation (VM) in the pharynx is challenging because of difficulties in needle access and the presence of nerves as well as other critical anatomical structures around the lesion. We describe the successful treatment of a large pharyngeal VM using transoral sclerotherapy. The patient, a 21-yearold male, visited our hospital for sclerotherapy treatment consultation after complaining of difficulty in swallowing because of a pharyngeal VM. Transoral sclerotherapy was successful, and the symptoms showed complete alleviation. Using a flat detector-equipped angiographic C-arm CT (CACT) enabled the precise assessment of sclerosant placement, facilitating safer sclerotherapy in the pharynx.

A Retrograde Transvenous Embolization Technique with Balloon-Assisted Arterial Aspiration for a Peripheral Arteriovenous Malformation with a Venous Pouch.

Management of arteriovenous malformations (AVMs) is challenging, and there is no consensus regarding either the ideal approach or the treatment timing. Percutaneous embolization is the most frequent approach currently used and is considered the first-line technique for AVMs. There is an ongoing discussion about the best technical approach to embolize AVMs. AVMs associated with a dominant outflow vein (DOV) are rare. Embolization of both the DOV and the nidus is considered more effective. Herein, we report a novel technique of transvenous embolization of a DOV under negative pressure from an arterial balloon catheter in a case of a peripheral AVM. This technique allows the embolization of the DOV and the nidus retrogradely.