Publications by authors named "Wibke Uller"

41 Publications

Evaluation of treatment options for postoperative and spontaneous chylothorax in adults.

Interact Cardiovasc Thorac Surg 2021 May 17. Epub 2021 May 17.

Department of Thoracic Surgery, University Medical Center Regensburg, Regensburg, Germany.

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Objectives: Both postoperative and spontaneous chylothorax remain therapeutic challenges without recommendations for a standardized treatment approach. Regardless of its aetiology, patients with chylothorax experience prolonged hospitalization and suffer from the associated complications or the invasive therapy administered.

Methods: We conducted a retrospective, observational review of adult patients with chylothorax treated between January 2010 and September 2019. The primary end point was successful management with sustained cessation and/or controlled chylous output. Therapy duration, inpatient stay and the incidence of complications were evaluated as secondary end points.

Results: Of the 36 patients included (22 men; median age 63 years), 24 patients (67%) suffered from a postoperative accumulation of chylous fluid in the pleural space; in the remaining 12 (33%) patients, chylothoraces occurred spontaneously. Initial conservative treatment was successful in 42% (n = 15); in the other 20 cases (56%) additional invasive therapeutic strategies were followed. A complicated course requiring more than 1 treatment was seen in 54% (n = 13) of the postoperative and in 58% (n = 7) of the spontaneous cases. The median length of hospitalization was significantly longer in the postoperative group (37.5 vs 15.5 days; P = 0.016). Serious complications were observed only in the postoperative group (P = 0.28). There were no in-hospital deaths.

Conclusions: Basic treatment of both postoperative and spontaneous chylothorax should include dietary measures in all patients. Additional sclerosing radiotherapy and interventional or surgical therapy are often necessary. The choice of therapeutic approach should be indicated, depending on the aetiology and development of the chylothorax. Early, multimodal treatment is recommended.
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http://dx.doi.org/10.1093/icvts/ivab127DOI Listing
May 2021

Erroneous placement of central venous catheters in subclavian artery: Retrieval and successful hemostasis with a femoral closure device.

J Vasc Access 2021 Apr 7:11297298211007704. Epub 2021 Apr 7.

Department of Radiology, University Hospital Regensburg, Regensburg, Germany.

Background: Retrospective evaluation of the safety and efficacy of the retrieval of misplaced central venous catheters (CVCs) in subclavian arteries using the femoral closure device Angio-Seal™.

Methods: The clinical data of five patients (female,  = 2; mean age, 55.0 years ± 11.9) in whom a misplaced CVC within a subclavian artery was removed followed by closure of the vessel entry site with 8-French (F) Angio-Seal™ was analyzed.

Results: In 4/5 patients (80%; CVC diameter, 7-8F) the procedure was technically successful without complications. In 1/5 patients (20%; CVC diameter, 11.5F) the procedure failed and an additional covered stent was placed for successful closure of the vessel entry site. There were no complications associated with the Angio-Seal™ or stent implantation during follow-up.

Conclusion: Retrieval of a misplaced CVC within a subclavian artery using the percutaneous closure device Angio-Seal™ is quite safe and effective; however, caution is required if there is a mismatch in the diameter of the Angio-Seal™ and CVC. In the case of procedure failure, successful closure of the vessel entry site can be achieved by covered stent placement.
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http://dx.doi.org/10.1177/11297298211007704DOI Listing
April 2021

Sequential CT arterioportography-arteriosplenography depicts individual haemodynamic changes in children with portal hypertension without cirrhosis.

Eur Radiol Exp 2020 12 2;4(1):65. Epub 2020 Dec 2.

Department of Radiology, University Hospital Regensburg, Franz-Josef-Strauss-Allee 11, 93053, Regensburg, Germany.

We evaluated sequential computed tomography (CT) arterioportography-arteriosplenography for the assessment of venous pathways in children with portal hypertension without cirrhosis. Institutional Review Board approval was obtained for this retrospective, single-centre study. CT was performed after contrast application via catheters placed in the superior mesenteric artery (CT arterioportography) and the splenic artery (CT arteriosplenography) consecutively. Venous pathways in 22 children were evaluated. In all patients, the detailed haemodynamic consequences of portal hypertension could be characterised. The supply of varices at different locations could be assigned to the superior mesenteric vein or splenic vein system. Retrograde blood flow through the splenic vein and inferior mesenteric vein, portosystemic shunting, and patency of splanchnic veins were determined. CT arterioportography-arteriosplenography allowed a complete evaluation of individual haemodynamic pathways in children with portal hypertension.
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http://dx.doi.org/10.1186/s41747-020-00193-yDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7708570PMC
December 2020

Additive value of transarterial embolization to systemic sirolimus treatment in kaposiform hemangioendothelioma.

Int J Cancer 2021 05 3;148(9):2345-2351. Epub 2020 Dec 3.

Klinik und Poliklinik für Radiologie, Klinikum der Universität München, Munich, Germany.

Kaposiform hemangioendothelioma (KHE) is a rare vascular tumor in children, which can be accompanied by life-threatening thrombocytopenia, referred to as Kasabach-Merritt phenomenon (KMP). The mTOR inhibitor sirolimus is emerging as targeted therapy in KHE. As the sirolimus effect on KHE occurs only after several weeks, we aimed to evaluate whether additional transarterial embolization is of benefit for children with KHE and KMP. Seventeen patients with KHE and KMP acquired from 11 hospitals in Germany were retrospectively divided into two cohorts. Children being treated with adjunct transarterial embolization and systemic sirolimus, and those being treated with sirolimus without additional embolization. Bleeding grade as defined by WHO was determined for all patients. Response of the primary tumor at 6 and 12 months assessed by magnetic resonance imaging (MRI), time to response of KMP defined as thrombocyte increase >150 × 10 /μL, as well as rebound rates of both after cessation of sirolimus were compared. N = 8 patients had undergone additive embolization to systemic sirolimus therapy, sirolimus in this group was started after a mean of 6.5 ± 3 days following embolization. N = 9 patients were identified who had received sirolimus without additional embolization. Adjunct embolization induced a more rapid resolution of KMP within a median of 7 days vs 3 months; however, tumor response as well as rebound rates were similar between both groups. Additive embolization may be of value for a more rapid rescue of consumptive coagulopathy in children with KHE and KMP compared to systemic sirolimus only.
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http://dx.doi.org/10.1002/ijc.33406DOI Listing
May 2021

Fast TWIST with iterative reconstruction improves diagnostic accuracy of AVM of the hand.

Sci Rep 2020 10 1;10(1):16355. Epub 2020 Oct 1.

Department of Radiology, University Medical Center Regensburg, 93042, Regensburg, Germany.

Very high temporal and spatial resolution is mandatory for the diagnosis of arteriovenous malformations (AVM) of the hand. Until now, magnetic resonance imaging (MRI) has not fulfilled both requirements simultaneously. This study presents how the combination of a very fast TWIST MRI (time-resolved angiography with interleaved stochastic trajectories) sequence and iterative reconstructions optimizes temporal as well as spatial resolution. 11 patients were examined at a 3-T MRI scanner with two different TWIST protocols: the standard and the study protocol, acquiring a data set every 5.57 s and 1.44 s respectively. The study data was retrospectively iteratively reconstructed with different regularization factors (0.001, 0.002, 0.004, 0.008). Results were compared using the sign-test. P-values < 0.05 were regarded statistically significant. With a low amount of contrast medium, the temporal resolution of the study protocol enabled the differentiation of arteries from veins in all patients whereas the signal-to-noise ratio (SNR) deteriorated. Depending on the regularization factors, SNR, delineation of arterial feeders and non-involved hand and interdigital arteries, as well as artefact levels varied. Overall, iterative reconstruction with regularization factor 0.004 achieved the best results, consequently showing the ability of MRI as a reliable diagnostic method in AVMs of the hand.
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http://dx.doi.org/10.1038/s41598-020-73331-6DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7529883PMC
October 2020

3T MRI of Peripheral Vascular Malformations: Characteristics and Comparison of Two Fat-Saturated sequences: Short Tau Inversion Recovery Versus Three-Dimensional High-Resolution Volume Interpolated Gradient Recalled Echo.

Rofo 2021 Apr 1;193(4):446-458. Epub 2020 Oct 1.

University Clinic and Policlinic of Radiology, Martin Luther University Hospital Halle-Wittenberg, Halle (Saale), Germany.

Purpose:  To assess morphological and hemodynamic characteristics of peripheral vascular malformations on 3 T magnetic resonance imaging (MRI) including qualitative comparison of two fat-saturated sequences: short tau inversion recovery (STIR) and three-dimensional high-resolution volume interpolated gradient recalled echo (GRE).

Materials And Methods:  During 9 months, 100 patients with suspected or known vascular malformations were prospectively assessed on a 3 T scanner using T2-weighted STIR and turbo spin echo (TSE), T1-weighted TSE, time-resolved contrast-enhanced magnetic resonance angiography (MRA) with interleaved stochastic trajectories (TWIST) and T1-weighted volume interpolated breath-hold examination (VIBE) after contrast enhancement. The analysis included signal behavior and morphologic and hemodynamic characteristics. Additionally, the image quality of the fat-saturated sequences was evaluated by 2 radiologists.

Results:  86 patients (14 dropouts; 57 female, 29 male; mean age 26.8 years, age range 1-56) were analyzed. 22 had high-flow and 64 low-flow malformations, including 14 with a lymphatic component. In 21 of 22 patients with high-flow malformations, typical characteristics (flow voids, hyperdynamic arteriovenous fistula, dilated main/feeder-arteries and draining veins) were documented. Patients with low-flow malformations had phleboliths in 35 cases, fluid-fluid levels in 47 and dilated draining veins in 23. Lymphatic malformations showed peripheral contrast enhancement of cyst walls in the volume interpolated GRE. The comparison of fat-saturated sequences showed significantly better results of the volume interpolated GRE in all categories except the presence of artifacts which were significantly reduced in the STIR (p < 0.05).

Conclusion:  3 T MRI with MRA provides detailed morphological and hemodynamic information of different types of peripheral vascular malformations. Contrast-enhanced high-resolution volume interpolated GRE proved superior to STIR in differentiating morphologic features and to be diagnostic in the differentiation of lymphatic parts and joint involvement.

Key Points:   · 3 T MRI with MRA offers detailed information about vascular malformations.. · Fat-saturated MRI provides especially information about morphological characteristics, extent and tissue involvement.. · Volume interpolated GRE proved superior in almost all categories compared to STIR.. · Volume interpolated GRE showed more artifacts.. · Volume interpolated GRE additionally allows differentiation of lymphatic parts and evaluation of joint involvement..

Citation Format: · Höhn F, Hammer S, Fellner C et al. 3T MRI of Peripheral Vascular Malformations: Characteristics and Comparison of Two Fat-Saturated sequences: Short Tau Inversion Recovery Versus Three-Dimensional High-Resolution Volume Interpolated Gradient Recalled Echo. Fortschr Röntgenstr 2021; 193: 446 - 458.
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http://dx.doi.org/10.1055/a-1253-8422DOI Listing
April 2021

[Diagnostic Criteria and Treatment of Hereditary Hemorrhagic Telangiectasia].

Laryngorhinootologie 2020 10 28;99(10):682-693. Epub 2020 Sep 28.

HNO-Klinik, Universitätsklinik Regensburg, Germany.

Hereditary hemorrhagic telangiectasia (HHT; Osler-Weber-Rendu syndrome; Morbus Osler) represents a syndrome affecting capillary vessels, leading to arteriovenous shunting. With an average worldwide prevalence of 1:5.000-8.000 HHT is considered an orphan disease. Arteriovenous shunts involve predominantly the nasal mucosa, the intestine, lung, liver and central nervous system. Epistaxis is the primary and most bothersome complaint of patients with HHT. A multistage therapeutic concept includes nasal ointment, laser therapy under local anesthesia and surgery under general anesthesia, as well as drug therapies. In addition, screening to determine affection of internal organs is carried out. Lesions that require therapy should be treated in an interdisciplinary setting. Treatment of lesions of the skin, oral and gastrointestinal mucosa and liver is carried out in regard to patients' symptoms, whereas vascular malformations of the lung and brain might need treatment without being symptomatic, due to possible life-threatening complications.
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http://dx.doi.org/10.1055/a-1220-7045DOI Listing
October 2020

MR imaging of venous malformations: sciatic nerve infiltration patterns and involved muscle groups.

Sci Rep 2020 09 3;10(1):14618. Epub 2020 Sep 3.

Department of Radiology and Policlinic of Radiology, University Hospital Halle (Saale), Ernst-Grube-Straße 40, 06120, Halle (Saale), Germany.

The aim of this retrospective cross-sectional study was to provide an MRI-based examination framework of venous malformations (VMs) infiltrating the sciatic nerve and determine the frequency of nerve infiltration patterns and muscle involvement in correlation to the patients' quality of life. Pelvic and lower limb MR images of 378 patients with vascular malformations were examined retrospectively. Pain levels and restriction of motion were evaluated with a questionnaire. Cross-sectional areas of affected nerves were compared at standardized anatomical landmarks. Intraneural infiltration patterns and involvement of muscles surrounding the sciatic nerve were documented. Sciatic nerve infiltration occurred in 23/299 patients (7.7%) with VM. In all cases (23/23; 100%), gluteal or hamstring muscles surrounding the nerve were affected by the VM. Infiltrated nerves were enlarged and showed signal alterations (T2-hyperintensity) compared to the unaffected side. Enlarged nerve cross-sectional areas were associated with elevated pain levels. Three nerve infiltration patterns were observed: subepineurial (12/23; 52.2%), subparaneurial (6/23; 26.1%) and combined (5/23; 21.7%) infiltration. This study provides a clinically relevant assessment for sciatic nerve infiltration patterns and muscle involvement of VMs, while suggesting that VMs in gluteal and hamstring muscles require closer investigation of the sciatic nerve by the radiologist.
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http://dx.doi.org/10.1038/s41598-020-71595-6DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7471964PMC
September 2020

Congenital Disseminated Pyogenic Granuloma: Characterization of an Aggressive Multisystemic Disorder.

J Pediatr 2020 Jul 2. Epub 2020 Jul 2.

Division of Vascular and Interventional Radiology, Boston Children's Hospital, Boston, MA; Harvard Medical School, Boston, MA.

Objective: To describe the clinical, radiologic, and histopathologic features of "congenital disseminated pyogenic granuloma" involving various organs with high morbidity related to cerebral hemorrhagic involvement.

Study Design: We searched the database of the Vascular Anomalies Center at Boston Children's Hospital from 1999 to 2019 for patients diagnosed as having multiple vascular lesions, visceral vascular tumors, congenital hemangiomatosis, multiple pyogenic granulomas, or multiple vascular lesions without a definite diagnosis. A retrospective review of the medical records, photographs, histopathologic, and imaging studies was performed. Only patients with imaging studies and histopathologic diagnosis of pyogenic granuloma were included.

Results: Eight children (5 male, 3 female) had congenital multifocal cutaneous vascular tumors. Lesions also were found in the brain (n = 7), liver (n = 4), spleen (n = 3), muscles (n = 4), bone (n = 3), retroperitoneum (n = 3), and intestine/mesentery (n = 2). Less commonly affected were the spinal cord, lungs, kidneys, pancreas, and adrenal gland (n = 1 each). The mean follow-up period was 21.8 months. The cerebral and visceral lesions were hemorrhagic with severe neurologic sequelae. The histopathologic diagnosis was pyogenic granuloma with prominent areas of hemorrhage and necrosis. The endothelial cells had enlarged nuclei, pale cytoplasm and were immunopositive for CD31 and negative for D2-40 and glucose transporter 1.

Conclusions: Congenital disseminated pyogenic granuloma is a distinct multisystemic aggressive disorder that primarily affects the skin, brain, visceral organs, and musculoskeletal system. Differentiation of this entity from other multiple cutaneous vascular lesions is critical because of possible cerebral hemorrhagic involvement.
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http://dx.doi.org/10.1016/j.jpeds.2020.06.079DOI Listing
July 2020

Fluoroscopy-guided endoscopic sclerotherapy: a novel hybrid approach for symptomatic rectosigmoidal venous malformation (with video).

Gastrointest Endosc 2021 02 15;93(2):496-502. Epub 2020 Jun 15.

Department of Internal Medicine I, Martin Luther University, Halle (Saale), Germany.

Background And Aims: Recommendations for the treatment of lower GI bleeding do not include bleeding from venous malformations (VMs). The aim of this study was to delineate the usefulness of a novel hybrid intervention (fluoroscopy-guided endoscopic sclerotherapy) for the treatment of symptomatic VMs in the rectosigmoidal colon with bleeding.

Methods: The magnetic resonance images of 421 patients with VM, referred to multicenter vascular anomaly centers from 2009 to 2017, were analyzed retrospectively. Treatment was performed for all patients who experienced bleeding from rectosigmoidal VMs using fluoroscopy-guided endoscopic sclerotherapy with polidocanol foam as a novel approach.

Results: A total of 27 patients displayed VM in the rectosigmoidal area. Eleven of these presented with acute or previous bleeding and received treatment. Active bleeding was observed in 8 patients (72.7%), whereas 3 patients (27.3%) had signs of previous bleeding. Six of the 11 patients had anemia (54.5%). There were no adverse events within 24 hours of the intervention. In a 2-year follow-up period, only 1 patient (9.1%) presented with recurrent bleeding after 13 months and was successfully treated again with fluoroscopy-guided endoscopic sclerotherapy.

Conclusions: Fluoroscopy-guided endoscopic sclerotherapy was shown to be a safe and effective treatment of symptomatic VMs of the rectosigmoidal area. Thus, fluoroscopy-guided endoscopic sclerotherapy should be considered for patients with bleeding from VMs of the rectosigmoid after a comprehensive workup and interdisciplinary case discussion.
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http://dx.doi.org/10.1016/j.gie.2020.06.027DOI Listing
February 2021

Safety and effectiveness of percutaneous sclerotherapy for venous disorders of the labia majora in patients with vascular malformations.

J Vasc Surg Venous Lymphat Disord 2020 11 19;8(6):1083-1089. Epub 2020 Mar 19.

Department of Radiology and Policlinic of Radiology, University Hospital Halle (Saale), Halle (Saale), Germany.

Objective: The aim of this study was to evaluate the safety and clinical outcomes of percutaneous sclerotherapy of venous disorders of the labia majora in patients with vascular malformations of the lower limbs.

Methods: Thirty percutaneous sclerotherapy treatments were performed over a 6-year period among 17 female patients with symptomatic venous malformation (VM) or secondary varicosis of the labia majora. Four patients were treated with sclerotherapy alone, 13 patients had additional procedures to control the VM before sclerotherapy. Polidocanol was used as sclerosant. Indications for sclerotherapy included pain, bleeding, thrombophlebitis, and swelling. Genitourinary symptoms were recorded. The number of treatments and procedure-related complications were registered. Complications were classified according to the Society of Interventional Radiology (SIR) classification system (grade A-E). The 3-month postintervention follow-up included magnetic resonance imaging, clinical examination, and a symptom-related questionnaire. If no reintervention was necessary, consultation was scheduled biannually.

Results: All patients had local swelling and pain; only a fraction of the patients had further symptoms with bleeding or thrombophlebitis (47% each). Eight patients required reintervention. No major complications were observed; minor complications such as postprocedural swelling occurred in 29% (SIR grade A), pain occurred in 17% (SIR grade B), and skin blistering developed in 5% (SIR grade B). Upon follow-up examination after a median of 40 months, 76% showed complete relief of symptoms, and 23% reported partial relief. All patients reported a substantial reduction in pain (75% >5 points in visual analogue scale) and swelling (88% complete cessation).

Conclusions: Percutaneous sclerotherapy is a safe and effective treatment option of VM and secondary varicosis of the labia majora.
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http://dx.doi.org/10.1016/j.jvsv.2020.01.008DOI Listing
November 2020

Author Correction: Rectosigmoidal manifestations of venous malformations: MR imaging findings and interdisciplinary therapeutic modalities.

Sci Rep 2020 Feb 7;10(1):2458. Epub 2020 Feb 7.

Universitätsklinikum Halle (Saale), Martin-Luther-Universität Halle-Wittenberg, Department of Radiology and Policlinic of Radiology, Ernst-Grube-Straße 40, D-06120, Halle (Saale), Germany.

An amendment to this paper has been published and can be accessed via a link at the top of the paper.
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http://dx.doi.org/10.1038/s41598-020-59025-zDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7005778PMC
February 2020

Complicated chylous pericardial and thoracic effusion as the first clinical manifestation of thoracic lymphatic malformation.

Interact Cardiovasc Thorac Surg 2020 04;30(4):654-655

Department of Thoracic Surgery, University Medical Center Regensburg, Regensburg, Germany.

Lymphatic malformations are benign focal proliferations of lymphatic vessels with a congenital origin. We present a case of an 18-year-old patient with post-traumatic chylopericardium and recurrent left-sided chylothorax, who was unresponsive to a variety of therapeutic measures until he was diagnosed with a complicated thoracic lymphatic malformation.
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http://dx.doi.org/10.1093/icvts/ivz301DOI Listing
April 2020

Rectosigmoidal manifestations of venous malformations: MR imaging findings and interdisciplinary therapeutic modalities.

Sci Rep 2019 12 27;9(1):19916. Epub 2019 Dec 27.

Universitätsklinikum Halle (Saale), Martin-Luther-Universität Halle-Wittenberg, Department of Radiology and Policlinic of Radiology, Ernst-Grube-Straße 40, D-06120, Halle (Saale), Germany.

The aim of this study was to identify the frequency of rectosigmoidal involvement in patients with venous malformations (VM) of the lower extremities and to demonstrate multidisciplinary therapeutic options. The medical records and magnetic resonance images (MRI) of patients with VM of the lower extremities, over a six-year period, were reviewed retrospectively in order to determine the occurrence of rectosigmoidal involvement. Vascular interventions, surgical treatments, percutaneous and hybrid (endoscopy-guided angiography) sclerotherapy and procedural complications (according to Clavien-Dindo classification) were also noted. Of the 378 patients with vascular malformation of the lower limbs, 19 patients (5%) had documented venous rectosigmoidal malformation. All of these 19 patients reported episodes of rectal bleeding, while seven patients (36.8%) also had anemia. All patients underwent endoscopy. By endoscopy, seven patients (36.8%) showed discreet changes, and 12 patients (63.2%) showed pronounced signs of submucosal VM with active (47.3%) or previous (15.7%) bleeding. Treatment was performed in all patients with pronounced findings. Six patients underwent endoscopy-guided hybrid sclerotherapy, one patient underwent endoscopic tissue removal, one patient received percutaneous sclerotherapy and one patient received a combination of transvenous embolization and hybrid sclerotherapy. Three patients required open surgery. No complications occurred after conservative treatments; however, one complication was reported after open surgery. None of the treated patients reported further bleeding and anemia at the end of the follow-up period. In this cohort, rectosigmoidal VM occurred in 5% of patients presenting with a high incidence of rectal bleeding. Percutaneous or endoscopy-guided hybrid sclerotherapy appeared to be a safe and effective alternative to surgery.
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http://dx.doi.org/10.1038/s41598-019-56217-0DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6934467PMC
December 2019

Do patients clinically diagnosed with vascular malformations of 1 lower extremity benefit from imaging of both legs from pelvis to toe? A prospective MRI study.

J Am Acad Dermatol 2020 04 3;82(4):981-984. Epub 2019 Jul 3.

Interdisciplinary Center for Vascular Anomalies, University Clinic and Polyclinic of Radiology, University Hospital Halle, Halle (Saale), Germany.

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http://dx.doi.org/10.1016/j.jaad.2019.06.1305DOI Listing
April 2020

[Management of Visceral Artery Aneurysms with Preservation of Organ Perfusion: More Than Twenty Years Experience].

Zentralbl Chir 2018 Oct 24;143(5):516-525. Epub 2018 Oct 24.

Abteilung für Gefäßchirurgie, Universitätsklinikum Regensburg, Deutschland.

Background: Visceral artery aneurysms (VAA) are rare and often incidental findings. Indications for treatment are symptomatic patients, pseudoaneurysms and a true aneurysm of diameter of ≥ 2.5 cm for mesenteric arteries and ≥ 3 cm for renal artery aneurysms. Pregnancy and liver transplantation play an important role in aneurysm rupture. Technical success after open or endovascular procedure and maintenance of organ perfusion are crucial. The aim of this review is to evaluate our data and experience of more than 20 years and to develop a strategy to deal with visceral artery aneurysm in elective and emergency cases.

Patients: Between 1995 and 2018, 179 patients (84 males, 95 females, median age 62 [18 - 87] years) were diagnosed with VAA at the Regensburg University Hospital. The site of aneurysm was the splenic artery in 113 (63%) patients (pts), hepatic and renal arteries in 22 and 21 pts (12% each), and gastropancreaticoduodenal artery in 14 (8%) and superior/inferior mesenteric artery in 9 (5%) cases. Surveillance without intervention occurred in 110 (62%) pts, and 34 (19%) pts underwent open and 35 (19%) endovascular repair. In all patients, preoperative imaging was performed, preferably by computed tomography angiography (CTA).

Results: A total of 69 patients underwent open or endovascular repair. 51 (74%) pts were treated electively, 18 (26%) pts presented urgently with acute bleeding. 16 emergency pts received endovascular treatment, and in 2 pts open surgery was performed. After emergency treatment, two pts exhibited segmental liver malperfusion without consequences. In one case, segmental bowel resection was necessary. 32/51 (63%) patients were treated electively by open surgery, 19/51 (37%) by endovascular procedures. There were no liver or bowel infarctions. Four splenectomies and one unilateral nephrectomy were necessary in patients with splenic or renal artery aneurysms. Moreover, three partial renal infarctions were noticed postoperatively (overall 8/21 [38%]). After endovascular repair of splenic or renal artery aneurysms, two cases of splenic and three cases of renal segmental infarction were observed. Splenectomy had to be performed twice (overall 7/14 [50%]). Organ perfusion was monitored by CTA, and preferentially by contrast enhanced ultrasound.

Conclusion: The endovascular approach is the preferred option in an emergency to control bleeding in pseudoaneurysms. Patients for elective splenic or renal artery aneurysm repair have to be evaluated very carefully to achieve technical success with occlusion of the aneurysm and excellent organ perfusion. Imaging techniques such as ultrasound, especially CEUS, are strongly recommended in postoperative follow-up. Partial or complete splenic infarction leads to vaccination.
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http://dx.doi.org/10.1055/a-0750-6016DOI Listing
October 2018

Venous Malformations: Phleboliths Correlate With the Presence of Arteriovenous Microshunts.

AJR Am J Roentgenol 2018 12 9;211(6):1390-1396. Epub 2018 Oct 9.

1 Department of Radiology, University Hospital Regensburg, Franz-Josef-Strauβ-Allee 11, 93053 Regensburg, Germany.

Objective: The objective of this study was to identify differences between venous malformations (VMs) with arteriovenous (AV) microshunts and VMs without AV microshunts.

Materials And Methods: In this retrospective study, 83 patients with untreated VMs of the extremities underwent a prospectively defined MRI protocol performed at 3 T. MR images were reviewed to determine localization and extent of each VM and tissue involvement. The VM volume and the lesion volume relative to body surface were measured. The number of phleboliths within the lesions was determined. Using time-resolved MR angiography, the hemodynamic subtype (VM with AV microshunts versus VM without AV microshunts) was classified, and the interval between the onset of arterial enhancement and lesion enhancement (t) was measured. Clinical data were reviewed to determine patient demographic characteristics, the onset of symptoms, the frequency of thrombophlebitis, and the pain score.

Results: Of the 83 patients, 56 (67%) presented with AV microshunts. The t (p ≤ 0.001), absolute and relative lesion volume (p = 0.001), and number of phleboliths (p = 0.038) differed significantly between these two subgroups. With use of a multivariable regression model, the strongest indicator for VM with AV microshunts was the presence of more than two phleboliths (odds ratio, 2.80; p = 0.007). No differences were noted regarding patient demographic characteristics and history or lesion morphologic features or anatomy.

Conclusion: Occurrence of AV microshunts in untreated VM is a frequent phenomenon. This hemodynamic subtype is significantly associated with phleboliths and large lesions. The subtypes did not differ in terms of history and clinical course.
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http://dx.doi.org/10.2214/AJR.18.19703DOI Listing
December 2018

Preoperative Embolization of Venous Malformations Using n-Butyl Cyanoacrylate.

Vasc Endovascular Surg 2018 May 15;52(4):269-274. Epub 2018 Mar 15.

1 Vascular Anomalies Center, Boston Children's Hospital and Harvard Medical School, Boston, MA, USA.

Purpose: The purpose of this study was to evaluate the safety and efficacy of preoperative percutaneous n-butyl cyanoacrylate (nBCA) embolization of venous malformations in children.

Material And Methods: Clinical data were retrospectively reviewed in children who underwent embolization using nBCA followed by resection of venous malformations.

Results: A total of 17 embolizations were performed in 14 patients (9 females, mean age: 5.5 years; median age: 3 years; range 0.1-16 years). The venous malformations involved the lower extremity and the knee joint (n = 7), the trunk (n = 4), head and neck (n = 2), and hand (n = 1). n-Butyl cyanoacrylate was diluted with iodized oil at a ratio of 1:3 to 1:5. The mean and median volume of nBCA per procedure were 2.1 and 2 mL, respectively (range: 0.5-8 mL). There were no complications associated with the procedures. The mean and median time between final embolization and resection were 3.6 and 2 days, respectively. All children underwent successful resection of the symptomatic lesions. The estimated mean and median blood loss were 75 and 50 mL, respectively (range: 5-350 mL). The postprocedure course was uneventful, the days to discharge ranged between 1 and 6 days (mean 3 days).

Conclusion: Initial results suggest that preoperative percutaneous n-butyl cyanoacrylate embolization of venous malformations is safe and effective in children, with the potential for minimizing blood loss and inpatient stay.
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http://dx.doi.org/10.1177/1538574418762192DOI Listing
May 2018

Impact of low dose settings on radiation exposure during pediatric fluoroscopic guided interventions.

Eur J Radiol 2018 Mar 9;100:1-6. Epub 2018 Jan 9.

Institut für Röntgendiagnostik, Universitätsklinikum Regensburg, Franz-Josef-Strauss-Allee 11, Regensburg, D-93053, Germany; Universitätsklinik und Poliklinik für Radiologie, Universitätsklinikum Halle, Ernst-Grube-Str 40, Halle, D-06120, Germany.

Purpose: To evaluate the effects of lowering the detector entrance exposure in children undergoing interventional radiology procedures.

Materials And Methods: The study retrospectively investigated radiation dose levels in pediatric patients aged 0-18 years before (n = 39) and after (n = 26) lowering detector entrance dose, undergoing embolization of peripheral Arteriovenous malformations, Portal Vein Interventions or Percutaneous Transhepatic Cholangio Drainage (PTCD) between 2014 and 2017. Patient characteristics, fluoroscopy time, protocols used as well as resulting Skin Dose and Dose Area Product (DAP) were compared in each cohort. Image quality was assessed by two independent readers.

Results: The two patient cohorts did not differ in terms of patient demographics. Similarly, fluoroscopy time did not differ before and after implementation of the low dose settings. An overall reduction of skin dose of 75.1% for AVM embolizations, 80.5% for Portal Vein Interventions and 85.3% for PTCD placement was observed. The DAP decrease was 82.5% for AVM embolizations, 72.2% for Portal Vein Interventions and 79.8% for PTCD placement. Image quality was generally considered to be good with an insignificant difference between pre and post implementation of the low dose approach and good agreement between the two readers. Manual inroom-switching to higher dose levels was possible, however this was not performed more frequently after implementation of the low dose settings.

Conclusion: Lowering the detector entrance dose in pediatric interventional radiology procedures results in a significant decrease of the radiation dose burden.
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http://dx.doi.org/10.1016/j.ejrad.2018.01.011DOI Listing
March 2018

Ethanol-Gel Sclerotherapy of Venous Malformations: Effectiveness and Safety.

AJR Am J Roentgenol 2017 Dec 20;209(6):1390-1395. Epub 2017 Sep 20.

1 Department of Radiology, University Medical Center Regensburg, 93042 Regensburg, Germany.

Objective: In the treatment of venous malformations, ethanol may be administered in a gelified form to increase local effects and reduce systemic ones. The purpose of this prospective study was to evaluate the efficacy and safety of a commercially available viscous ethanol gel in the treatment of venous malformations.

Subjects And Methods: Thirty-one patients (mean age, 23.4 years; age range, 6.6-46.5 years) with venous malformations were prospectively scheduled for two ethanol-gel sclerotherapy sessions. Venous malformations were located at the lower extremity (n = 18), the upper extremity (n = 9), and the face (n = 4). Questionnaires to assess pain, clinical examinations, professional photographs, and contrast-enhanced MRI of the venous malformations were performed before and after therapy to measure therapy-induced changes. Two experienced radiologists blinded to the examination date and clinical status compared photographs and MR images before and after treatment.

Results: A mean of 4.2 mL of ethanol gel were administered per session. The technical success rate was 100%. Clinical success, defined as improvement or resolution of symptoms, was noted in 81% of patients. Mean pain score decreased, and the difference was statistically significant (3.9 vs 3.1, p = 0.005). In 54 treatment sessions where follow-up was available, four minor complications occurred. Comparison of photographs and MR images before and after treatment showed improvement in 35% and 93% of patients, respectively.

Conclusion: Ethanol gel is an effective and safe sclerosing agent in the treatment of venous malformations.
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http://dx.doi.org/10.2214/AJR.16.17603DOI Listing
December 2017

Ethanolgel sclerotherapy of venous malformations improves health-related quality-of-life in adults and children - results of a prospective study.

Eur Radiol 2017 Jun 4;27(6):2482-2488. Epub 2016 Oct 4.

Institut für Röntgendiagnostik, Universitätsklinikum Regensburg, Franz-Josef-Strauß-Allee 11, 93053, Regensburg, Germany.

Objectives: To assess the treatment-induced changes of health-related quality-of-life (HRQoL) in patients with venous malformations (VM) who underwent ethanol gel sclerotherapy.

Methods: The prospective study in children and adults was approved by the local ethics committee. 31 patients (mean age 23.42 years, range 6.6 - 46.5; 26 female, 5 male) with VM were included. Patients' self-assessed HRQoL was measured before and after treatment using psychometrically validated questionnaires for adults and children. Differences were analysed with a paired t test.

Results: 58 sclerotherapy sessions were performed. The Physical Component Summary (PCS) at baseline was 43.69 and increased significantly (p = 0.01122) to 48.95 after treatment. The bodily pain (BP) scale increased significantly from 37.94 to 48.56 (p = 0.00002), the general health (GH) scale increased significantly from 46.69 to 52.17 (p = 0.00609). Baseline Physical Summary Score (PHS) in children increased significantly after treatment (p < 0.00001) from 25.25 to 45.89. The baseline Psychosocial Summary Score (PSS) in children was 51.08 and increased significantly (p = 0.00031) after treatment to 58.84.

Conclusion: Paediatric and adult patients with VM suffer from bodily pain with overall reduced physical functioning. After sclerotherapy, these restrictions are successfully returning to normal levels with a positive effect on mental and psychosocial domains.

Key Points: • Sclerotherapy in venous malformation patients has an unknown effect on health-related quality-of-life • Prospective study showed improvements in bodily pain and general health in adults • Children and adults improved from treatment with ethanol gel • Sclerotherapy is an appropriate therapy.
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http://dx.doi.org/10.1007/s00330-016-4603-0DOI Listing
June 2017

Direct Effective Dose Calculations in Pediatric Fluoroscopy-Guided Abdominal Interventions with Rando-Alderson Phantoms - Optimization of Preset Parameter Settings.

PLoS One 2016 24;11(8):e0161806. Epub 2016 Aug 24.

Institut für Röntgendiagnostik, Universitätsklinikum Regensburg, Regensburg, Germany.

Objective: The aim of the study was to calculate the effective dose during fluoroscopy-guided pediatric interventional procedures of the liver in a phantom model before and after adjustment of preset parameters.

Methods: Organ doses were measured in three anthropomorphic Rando-Alderson phantoms representing children at various age and body weight (newborn 3.5kg, toddler 10kg, child 19kg). Collimation was performed focusing on the upper abdomen representing mock interventional radiology procedures such as percutaneous transhepatic cholangiography and drainage placement (PTCD). Fluoroscopy and digital subtraction angiography (DSA) acquisitions were performed in a posterior-anterior geometry using a state of the art flat-panel detector. Effective dose was directly measured from multiple incorporated thermoluminescent dosimeters (TLDs) using two different parameter settings.

Results: Effective dose values for each pediatric phantom were below 0.1mSv per minute fluoroscopy, and below 1mSv for a 1 minute DSA acquisition with a frame rate of 2 f/s. Lowering the values for the detector entrance dose enabled a reduction of the applied effective dose from 12 to 27% for fluoroscopy and 22 to 63% for DSA acquisitions. Similarly, organ doses of radiosensitive organs could be reduced by over 50%, especially when close to the primary x-ray beam.

Conclusion: Modification of preset parameter settings enabled to decrease the effective dose for pediatric interventional procedures, as determined by effective dose calculations using dedicated pediatric Rando-Alderson phantoms.
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http://journals.plos.org/plosone/article?id=10.1371/journal.pone.0161806PLOS
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4996450PMC
July 2017

Time-resolved magnetic resonance angiography (MRA) at 3.0 Tesla for evaluation of hemodynamic characteristics of vascular malformations: description of distinct subgroups.

Eur Radiol 2017 Jan 23;27(1):296-305. Epub 2016 Feb 23.

Department of Radiology, University Hospital Regensburg, 93042, Regensburg, Germany.

Objectives: Quantitative evaluation of hemodynamic characteristics of arteriovenous and venous malformations using time-resolved magnetic resonance angiography (MRA) at 3.0 Tesla.

Methods: Time-resolved MRA with interleaved stochastic trajectories (TWIST) at 3.0 Tesla was studied in 83 consecutive patients with venous malformations (VM) and arteriovenous malformations (AVM). Enhancement characteristics were calculated as percentage increase of signal intensity above baseline over time. Maximum percentage signal intensity increase (signal), time intervals between onset of arterial enhancement and lesion enhancement (t), and time intervals between beginning of lesion enhancement and maximum percentage of lesion enhancement (t) were analyzed.

Results: All AVMs showed a high-flow hemodynamic pattern. Two significantly different (p < 0.001) types of venous malformations emerged: VMs with arteriovenous fistulas (AVF) (median signal 737 %, IQR [interquartile range] = 511 - 1182 %; median t 5 s, IQR = 5 - 10 s; median t 35 s, IQR = 26 - 40 s) and without AVFs (median signal 284 %, IQR = 177-432 %; median t 23 s, IQR = 15 - 30 s; median t 60 s, IQR = 55 - 75 s).

Conclusions: Quantitative evaluation of time-resolved MRA at 3.0 Tesla provides hemodynamic characterization of vascular malformations. VMs can be subclassified into two hemodynamic subgroups due to presence or absence of AVFs.

Key Points: • Time-resolved MRA at 3.0 Tesla provides quantitative hemodynamic characterization of vascular malformations. • Malformations significantly differ in time courses of enhancement and signal intensity increase. • AVMs show a distinctive high-flow hemodynamic pattern. • Two significantly different types of VMs emerged: VMs with and without AVFs.
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http://dx.doi.org/10.1007/s00330-016-4270-1DOI Listing
January 2017

Abernethy Malformation.

Radiographics 2015 Sep-Oct;35(5):1623

Division of Vascular and Interventional Radiology, Boston Children's Hospital and Harvard Medical School, 300 Longwood Ave, Boston, MA 02115.

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http://dx.doi.org/10.1148/rg.2015150089DOI Listing
January 2016

The retrograde transvenous push-through method: a novel treatment of peripheral arteriovenous malformations with dominant venous outflow.

Cardiovasc Intervent Radiol 2015 Jun 12;38(3):623-31. Epub 2015 Mar 12.

Department of Radiology, University Medical Center Regensburg, Franz Josef Strauss Allee 11, 93053, Regensburg, Germany,

Purpose: To evaluate the efficacy and safety of a novel retrograde transvenous embolization technique of peripheral arteriovenous malformations (AVMs) using Onyx.

Materials And Methods: We conducted a retrospective analysis of all patients who underwent transvenous retrograde Onyx embolization of peripheral AVMs with dominant venous outflow over a 29-month period. The embolization is aimed at retrograde filling of the nidus after building a solid plug in the dominant venous outflow (push-through). Classification, clinical signs, technical aspects, clinical and technical success rates, and complications were recorded. Short-term outcome was assessed.

Results: 11 Symptomatic patients (8 female; mean age 31.4 years) were treated at our Vascular Anomalies Center with this method between January 2012 and May 2014. The AVMs were located on the upper extremity (n = 3), pelvis (n = 2), buttock (n = 2), and lower extremity (n = 4). Retrograde embolization was successfully carried out after preparatory transarterial-flow reduction in eight cases (73 %) and venous-flow reduction with Amplatzer Vascular Plugs in four cases (36 %). Complete devascularization (n = 10; 91 %) or 95 % devascularization (n = 1; 9 %) led to complete resolution (n = 8; 73 %) or improvement of clinical symptoms (n = 3; 27 %). One minor complication occurred (pain and swelling). During a mean follow-up time of 8 months, one clinically asymptomatic recurrence of AVM was detected.

Conclusion: Initial results suggest that retrograde transvenous Onyx embolization of peripheral AVMs with dominant venous outflow is a safe and effective novel technique with a low complication rate.
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http://dx.doi.org/10.1007/s00270-015-1063-xDOI Listing
June 2015

Lymphatic and other vascular malformative/overgrowth disorders are caused by somatic mutations in PIK3CA.

J Pediatr 2015 Apr 11;166(4):1048-54.e1-5. Epub 2015 Feb 11.

Vascular Anomalies Center, Boston Children's Hospital, Boston, MA. Electronic address:

Objectives: To test the hypothesis that somatic phosphatidylinositol-4,5-bisphospate 3-kinase, catalytic subunit alpha (PIK3CA) mutations would be found in patients with more common disorders including isolated lymphatic malformation (LM) and Klippel-Trenaunay syndrome (KTS).

Study Design: We used next generation sequencing, droplet digital polymerase chain reaction, and single molecule molecular inversion probes to search for somatic PIK3CA mutations in affected tissue from patients seen at Boston Children's Hospital who had an isolated LM (n = 17), KTS (n = 21), fibro-adipose vascular anomaly (n = 8), or congenital lipomatous overgrowth with vascular, epidermal, and skeletal anomalies syndrome (n = 33), the disorder for which we first identified somatic PIK3CA mutations. We also screened 5 of the more common PIK3CA mutations in a second cohort of patients with LM (n = 31) from Seattle Children's Hospital.

Results: Most individuals from Boston Children's Hospital who had isolated LM (16/17) or LM as part of a syndrome, such as KTS (19/21), fibro-adipose vascular anomaly (5/8), and congenital lipomatous overgrowth with vascular, epidermal, and skeletal anomalies syndrome (31/33) were somatic mosaic for PIK3CA mutations, with 5 specific PIK3CA mutations accounting for ∼ 80% of cases. Seventy-four percent of patients with LM from Seattle Children's Hospital also were somatic mosaic for 1 of 5 specific PIK3CA mutations. Many affected tissue specimens from both cohorts contained fewer than 10% mutant cells.

Conclusions: Somatic PIK3CA mutations are the most common cause of isolated LMs and disorders in which LM is a component feature. Five PIK3CA mutations account for most cases. The search for causal mutations requires sampling of affected tissues and techniques that are capable of detecting low-level somatic mosaicism because the abundance of mutant cells in a malformed tissue can be low.
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http://dx.doi.org/10.1016/j.jpeds.2014.12.069DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4498659PMC
April 2015

CT features of early type II endoleaks after endovascular repair of abdominal aortic aneurysms help predict aneurysm sac enlargement.

Radiology 2015 Mar 7;274(3):906-16. Epub 2014 Nov 7.

From the Department of Radiology (R.M.W., S.S., W.U., O.G., C.S., W.A.W.), Center for Clinical Studies (F.Z.), and Department of Surgery (K.P., P.K.), University Medical Center Regensburg, Franz-Josef-Strauss-Allee 11, 93053 Regensburg, Germany.

Purpose: To determine computed tomographic (CT) features of early type II endoleaks associated with aneurysm sac enlargement after endovascular aortic aneurysm repair (EVAR) of abdominal aortic aneurysm.

Materials And Methods: Institutional review board approval was not required for this retrospective study. The authors reviewed imaging and clinical data from 56 patients (seven women, 49 men; mean age ± standard deviation, 71 years ± 7.9; age range, 52-85 years) with early type II endoleak who had undergone EVAR between December 2002 and December 2011 and who had been followed up with imaging and clinical evaluation for at least 6 months. The number and diameter of all feeding and/or draining arteries were measured, and endoleaks were classified according to their sources into simple inferior mesenteric artery (IMA), simple lumbar artery (LA), complex LA, and complex IMA-LA type II endoleaks. Volume and attenuation of the nidus were measured. Aneurysm enlargement was defined as an increase in the aneurysm volume of more than 5% during follow-up. Simple and multivariate logistic regression analyses were performed to identify independent clinical and imaging variables associated with aneurysm enlargement.

Results: Twenty-three of the 56 patients (41%) showed aneurysm sac enlargement during follow-up (mean follow-up, 3.0 years ± 2.0). With the multivariate model, the variables that showed the strongest indicators for aneurysm sac enlargement were complex IMA-LA type II endoleak (odds ratio [OR] = 10.29, P = .004) and the diameter of the largest feeding and/or draining artery (OR = 4.55, P = .013). Patients without complex IMA-LA type II endoleak in whom the largest feeding and/or draining artery was larger than 3.8 mm and patients with a complex IMA-LA type II endoleak in whom the largest feeding and/or draining artery was larger than 2.2 mm were at high risk for aneurysm sac enlargement.

Conclusion: The strongest indicators for aneurysm sac enlargement are complex IMA-LA type II endoleak and the diameter of the largest feeding and/or draining artery.
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http://dx.doi.org/10.1148/radiol.14140284DOI Listing
March 2015

Portomesenteric thrombosis and hypertension in Klippel-Trenaunay syndrome.

Avicenna J Med 2014 Oct;4(4):106

Division of Vascular and Interventional Radiology, Boston Children's Hospital and Harvard Medical School, Boston, MA, USA.

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http://dx.doi.org/10.4103/2231-0770.140663DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4183901PMC
October 2014

Overgrowth syndromes with complex vascular anomalies.

Semin Pediatr Surg 2014 Aug 19;23(4):208-15. Epub 2014 Jun 19.

Vascular Anomalies Center, Boston Children׳s Hospital and Harvard Medical School, Boston, Massachusetts; Division of Vascular and Interventional Radiology, Boston Children׳s Hospital and Harvard Medical School, 300 Longwood Ave, Boston, Massachusetts 02115. Electronic address:

Management of overgrowth syndromes with complex vascular anomalies is challenging. Careful analysis of the various clinical features by an interdisciplinary team of physicians experienced in this field is paramount to proper diagnostic and therapeutic approaches. In this article, we focus on the spectrum of the clinical presentation and the management strategies of the most common overgrowth syndromes with complex vascular anomalies.
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http://dx.doi.org/10.1053/j.sempedsurg.2014.06.013DOI Listing
August 2014
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