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Lymphatic malformation

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https://www.readbyqxmd.com/read/29790593/efficacy-and-absorption-of-topical-sirolimus-for-the-treatment-of-vascular-anomalies-in-children-a-case-series
#1
Sophie Le Sage, Michèle David, Josée Dubois, Julie Powell, Catherine C McCuaig, Yves Théorêt, Niina Kleiber
BACKGROUND/OBJECTIVES: Efficacy of topical sirolimus has recently been described in lymphatic anomalies but not in other types of vascular anomalies. To our knowledge, systemic absorption of topical sirolimus in these lesions has not yet been reported. The objective was to evaluate the efficacy, tolerance, and absorption of topical sirolimus 0.1% with different types of vascular anomalies in children. METHODS: Sirolimus 0.1% was applied on cutaneous vascular anomalies in six children aged 2-17...
May 23, 2018: Pediatric Dermatology
https://www.readbyqxmd.com/read/29782340/fatal-airway-obstruction-in-a-man-with-a-cystic-hygroma
#2
Cassandra Maria Wygant, Stephen D Cohle
We describe a 24-year-old man with a cystic hygroma of the left side of the lower neck that led to sudden death. Cystic hygroma (cystic lymphangioma) is a congenital malformation of the lymphatic system. The patient, who had a tracheostomy because of airway obstruction from the cystic hygroma, was found dead with his tracheostomy tube on the floor next to him. Complications of cystic hygroma include infiltration of the neck causing airway obstruction, dysphagia, pain, and obstructive sleep apnea.
May 3, 2018: American Journal of Forensic Medicine and Pathology
https://www.readbyqxmd.com/read/29782301/lymphangioma-circumscriptum-post-radiotherapy-for-penile-cancer-treated-with-co%C3%A2-laser
#3
Dietmar Schulz, Andreas Lein, Ancuta Proca Nicula, Katrin Schierle, Caius Solovan
Lymphangioma circumscriptum (LC) is a rare, benign condition, predominantly characterized by the malformation of lymphatic skin vessels. Its onset may be congenital or due to secondary causes such as radiotherapy, infections, or surgical procedures. We present the case of a 55-year-old patient with a pathologic history of squamous cell carcinoma of the penis followed by radical penectomy. Due to metastasis to the locoregional lymph nodes, the entire affected area was subsequently treated with radiation therapy, receiving a total dose of 55...
April 2018: Acta Dermatovenerologica Croatica: ADC
https://www.readbyqxmd.com/read/29774401/high-levels-of-serum-sclerostin-and-dkk1-in-a-case-of-klippel-tr%C3%A3-naunay-syndrome
#4
P Muto, A Lo Gullo, G Mandraffino, S Loddo, M Atteritano
Klippel-Trénaunay syndrome (KTS) is described as a complex syndrome characterized by various combinations of capillary, venous, and lymphatic malformations associated with bone and soft tissue hypertrophy. We report a case of a 67-year-old postmenopausal Caucasian women with KTS that shows elevated levels of sclerostin and Dickkopf-related protein 1 (DKK1). Dual-energy X-ray absorptiometry (DXA) BMD T-scores at lumbar spine and femur were normal. Serum calcium and phosphorus levels were consistently normal, 25-hydroxyvitamin D (25OHD) < 30 ng/mL, and normal parathyroid hormone (PTH)...
May 17, 2018: Osteoporosis International
https://www.readbyqxmd.com/read/29774173/role-of-interventional-radiology-in-the-management-of-peripheral-vascular-malformations-a-tertiary-care-center-experience
#5
Misbah Tahir, Muhammad Anees Mumtaz, Anum Sultan, Jawaid Iqbal, Raza Sayani
Peripheral vascular malformations (PVMs) represent a wide spectrum of vascular abnormalities occurring due to anomalous connections between arteries, veins, capillaries, and lymphatic channels at the microscopic level, in different combinations. They are rare and challenging to treat. Different operators may have different approaches based on their experience and expertise. Sclerotherapy either alone or in combination with embolization has been used as an independent method for the treatment of PVMs. Purpose The aim of this study is to assess the safety and efficacy of sclerotherapy and embolization, with or without surgery, for the treatment of peripheral vascular malformations, based on our approach...
March 16, 2018: Curēus
https://www.readbyqxmd.com/read/29773423/management-of-giant-embryonic-vein-in-klippel-tr%C3%A3-naunay-syndrome
#6
Animesh Rathore, Peter Gloviczki, Haraldur Bjarnason
Klippel-Trénaunay syndrome is a rare mixed malformation characterized by congenital varicose veins, low-flow venous and lymphatic malformations, hypertrophy of soft tissue and bone, and capillary malformations. A 35-year-old man with a diagnosis of Klippel-Trénaunay syndrome presented to the clinic with significant pain and swelling in the left leg. Initial conservative management with compression therapy failed. He was then managed surgically with preoperative placement of an inferior vena cava filter (because of a history of deep venous thrombosis and pulmonary embolism), followed by resection of the lateral embryonic vein, ligation of large perforators, and excision of smaller varicosities...
May 14, 2018: Journal of Vascular Surgery. Venous and Lymphatic Disorders
https://www.readbyqxmd.com/read/29766551/lower-lip-capillary-malformation-associated-with-lymphatic-malformation-without-overgrowth-part-of-the-spectrum-of-clapo-syndrome
#7
Camila Downey, Juan Carlos López-Gutiérrez, Esther Roé-Crespo, Luis Puig, Eulalia Baselga
Characteristic lower lip capillary malformation of CLAPO syndrome (Capillary malformation of the lower lip, Lymphatic malformations of the face and neck, Asymmetry, and Partial or generalized Overgrowth) may also occur as an isolated lesion or with only minor anomalies, supporting the concept that there is a spectrum of abnormalities in CLAPO syndrome. Preliminary studies have demonstrated mosaic activating mutations in PIK3CA.
May 15, 2018: Pediatric Dermatology
https://www.readbyqxmd.com/read/29697621/vascular-anomalies-from-a-clinicohistologic-to-a-genetic-framework
#8
Arin K Greene, Jeremy A Goss
BACKGROUND: Vascular anomalies currently are classified according to their clinical and histological characteristics. Recent advances in molecular genetics have enabled the identification of somatic mutations in most types of vascular anomalies. The purpose of this study was to collate information regarding the genetic basis of vascular anomalies. METHODS: The PubMed literature was reviewed for all citations that identified a mutation in a vascular anomaly between 1994 and 2017...
May 2018: Plastic and Reconstructive Surgery
https://www.readbyqxmd.com/read/29668117/development-of-molecular-therapies-for-venous-malformations
#9
Jaakko Kangas, Marjut Nätynki, Lauri Eklund
Vascular anomalies are localized defects of morphogenesis that can affect lymphatic and blood vessels. They are generally called birthmarks, typically observed soon after birth and occurring in up to 10% of children. Based on their clinical and histological characteristics, they are classified into vascular tumours and vascular malformations. The most common malformations are venous malformations (VMs) resulting in chronic vascular diseases that can be associated with significant morbidity necessitating often demanding and repeating clinical management...
April 18, 2018: Basic & Clinical Pharmacology & Toxicology
https://www.readbyqxmd.com/read/29665587/interstitial-transpedal-mr-lymphangiography-of-central-lymphatics-using-a-standard-mr-contrast-agent-feasibility-and-initial-results-in-patients-with-chylous-effusions
#10
Claus Christian Pieper, Hans Heinz Schild
PURPOSE:  To describe a novel technique for transpedal magnetic resonance lymphangiography (tMRL) with interstitial injection of a standard gadolinium-based contrast agent, and to assess its ability to visualize the central lymphatic system (CLS) in patients with chylous effusions. MATERIALS AND METHODS:  Experiences with tMRL were retrospectively reviewed in seven patients with chylous effusions. High-resolution three-dimensional multi-echo (mDIXON) images of the entire torso were obtained before and after interstitial injection of an extracellular gadolinium-based contrast agent...
April 17, 2018: RöFo: Fortschritte Auf Dem Gebiete der Röntgenstrahlen und der Nuklearmedizin
https://www.readbyqxmd.com/read/29658945/experience-with-ok-432-in-lymphatic-vascular-malformations-in-a-hospital-from-northern-mexico
#11
Jorge Alberto Cantú Reyes, Erik Antonio Mier Escurra, Joel Cázares Rangel, Julieta Rodríguez De Ita, Jan Lammel Lindemann, Luis Francisco Rendón García, Boris José Chacón López
Background: Lymphatic vascular malformations (LVM) or formerly called lymphangiomas are congenital malformations present in about 1 out of 6000 to -16000 births. The most relevant classification system for lymphangioma management is based on the size of the cysts. Spontaneous resolution is uncommon; thus, expectant management is not recommended. The classic treatment is excisional surgery, but it can affect adjacent structures or have relapses, so, sclerosing substances like OK-432 are being studied...
2018: Boletín Médico del Hospital Infantil de México
https://www.readbyqxmd.com/read/29628618/female-pelvic-vascular-malformations
#12
REVIEW
Aparna Annam
Vascular malformations are classified primarily according to their flow characteristics, slow flow (lymphatic and venous) or fast flow (arteriovenous). They can occur anywhere in the body but have a unique presentation when affecting the female pelvis. With a detailed clinical history and the proper imaging studies, the correct diagnosis can be made and the best treatment can be initiated. Lymphatic and venous malformations are often treated with sclerotherapy while arteriovenous malformations usually require embolization...
March 2018: Seminars in Interventional Radiology
https://www.readbyqxmd.com/read/29627129/-new-nosological-and-therapeutic-perspectives-in-syndromic-vascular-malformations-with-a-vein-lymphatic-component
#13
P Henneton, S Mestre, M Nou, I Quere
Vascular malformations are poorly recognized constitutional anomalies which arises during early childhood. Several classifications tried to draw a distinction across the different entities. Recent advances in molecular biology have contributed to the update of their nosology. Syndromic vascular malformations are an example: while Klippel-Trenaunay syndrome, Proteus or CLOVES syndrome share many common features, understanding of pathological mechanism and specially the role of the PIK3/AKT/mTOR pathway enables us to rethink their classification...
April 4, 2018: La Revue de Médecine Interne
https://www.readbyqxmd.com/read/29610184/a-novel-presentation-of-littoral-cell-angioma-and-lymphatic-malformations-in-a-neonate
#14
Lindsey Gakenheimer-Smith, Jeffrey Mohlman, Kate VandenHeuvel, William Daniel Jackson, William Thomsen, Adam Stevenson, Frank Cipriano
This case report describes a novel presentation of littoral cell angioma (LCA) and lymphatic malformations involving the omentum and mesentery. To our knowledge, these 2 entities have not been reported in the same patient. A 1-month term infant male presented with chylous ascites. During his workup, imaging detected splenic nodules. Biopsies revealed that the nodules were LCA and the chylous ascites was secondary to microscopic mesenteric and omental lymphatic malformations. Evaluation for a secondary malignancy, an underlying immunologic defect, and genetic causes were unrevealing...
April 2018: Pediatrics
https://www.readbyqxmd.com/read/29602374/-venolymphatic-malformation-with-bone-erosion-an-unusual-affectation-in-these-lesions
#15
Ana Teijo, Felipe Sacristán, Jesús Del Pozo, Tania Pérez, Laura López, Ana Reguera, Pilar Casteleiro, Ángeles Vázquez-Barro, Ángel Concha
A 30 year old male presented with a cutaneous lesion on the distal area of the 5th toe with involvement of the outer edges. Macroscopically, it was a 1cm violaceous and keratotic lesion. Radiography showed an increase in soft tissue, possibly due to a vascular lesion. MRI showed a hyper-intense signal with erosion of the distal phalanx compatible with a low-flow vascular malformation. The distal phalanx was amputated. Histopathology revealed a lesion formed by venous and D2-40 positive lymphatic vessels. This case highlights the fact that even minimal skin involvement in vascular malformations may conceal an important deeper lesion, such as erosion of the cortical bone...
April 2018: Revista Española de Patología
https://www.readbyqxmd.com/read/29596549/preoperative-facial-nerve-mapping-to-plan-and-guide-pediatric-facial-vascular-anomaly-resection
#16
Randall A Bly, Robert N Holdefer, Jefferson Slimp, Gregory A Kinney, Vicente Martinez, Scott C Manning, Jonathan A Perkins
Importance: Facial vascular anomalies are surgical challenges due to their vascularity and facial nerve distortion. To assist facial vascular anomaly surgical treatment, presurgical percutaneous facial nerve stimulation and recording of compound motor action potentials can be used to map the facial nerve branches. During surgery, the nerve map and continuous intraoperative motor end plate potential monitoring can be used to reduce nerve injury. Objective: To investigate if preoperative facial nerve mapping (FNM) is associated with intraoperative facial nerve injury risk and safe surgical approach options compared with standard nerve integrity monitoring (NIM)...
March 29, 2018: JAMA Otolaryngology—Head & Neck Surgery
https://www.readbyqxmd.com/read/29571951/-benign-aggressive-vascular-anomalies-in-children
#17
REVIEW
Olivia Boccara, Annabel Maruani, Christine Léauté-Labrèze
Superficial vascular anomalies constitute a large group of malformative and tumoral conditions developed from all types of vessels. Vascular tumors are the result of cellular hyperplasia, whereas vascular malformations (VMs) are constituted of dysplastic vessels. The classification from International Society for the Study of Vascular Anomalies (ISSVA) is based on this pathogenic difference. The most common vascular tumor is infantile hemangioma, which treatment, when necessary, is propranolol. Congenital hemangiomas and tumors that might be complicated with Kasabach-Merritt phenomenon, i...
March 20, 2018: Bulletin du Cancer
https://www.readbyqxmd.com/read/29560340/hennekam-syndrome-a-case-report
#18
Yeong Guk Lee, Seung Chan Kim, Si-Bog Park, Mi Jung Kim
Hennekam syndrome is a rare autosomal recessive disorder resulting from malformation of the lymphatic system. The characteristic signs of Hennekam syndrome are lymphangiectasia, lymph edema, facial anomalies, and mental retardation. This is a case in which a patient presented with left-arm lymphedema, facial-feature anomalies, and multiple organ lymphangiectasia consistent with symptoms of Hennekam syndrome. There is no curative therapy at this time, but rehabilitative treatments including complete decongestive therapy for edema control appeared to be beneficial...
February 2018: Annals of Rehabilitation Medicine
https://www.readbyqxmd.com/read/29533276/giant-pilomatricoma-arising-as-a-rapidly-growing-vascularized-tumor-in-a-child
#19
Alexandra Yannoutsos, Julie Malloizel-Delaunay, Emilie Tournier, Abdelaziz Abid, Alessandra Bura-Rivière
In this observation, we report an unusual presentation of a pilomatricoma in an 8-year-old girl who was initially referred to the department of vascular medicine for diagnosis and care of a suspected mixed lymphatic venous malformation. The lesion on her left shoulder presented as a giant bluish-purple red solitary mass, painful and rapidly growing, measuring 7 cm in anteroposterior diameter. This mass did not present the typical characteristics of a lymphatic venous malformation but exhibited warning signs of malignancy on clinical examination and imaging...
February 27, 2018: American Journal of Dermatopathology
https://www.readbyqxmd.com/read/29515932/successful-treatment-of-a-case-with-cervical-lymphatic-malformation-repeated-bleomycin-sclerotherapy
#20
Mustafa Mert Başaran, Selmin Özgürsoy Karataylı, Arzu Pampal, Şefik Halit Akmansu
Lymphatic malformations (LM) are benign congenital malformations of the lymphatic system. They are frequently seen in the neck and sometimes can be life-threatening due to compression to the airway. Treatment modalities are widespread including surgical excision, radiotherapy, laser therapy, and application of intralesional sclerosing agents. We report the successful treatment of cervical LM in a 3-year-old boy who presented with a sudden onset of a large cystic mass in the posterior cervical triangle and was treated with repeated injections of intralesional bleomycin...
December 2017: Turkish Archives of Otorhinolaryngology
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