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Lymphatic malformation

Debi M Thomas, Minna M Wieck, Christa N Grant, Avafia Dossa, Donna Nowicki, Phillip Stanley, Chadi Zeinati, Lori K Howell, Dean M Anselmo
PURPOSE: To evaluate efficacy of sclerotherapy with doxycycline versus sodium tetradecyl sulfate (STS) for treatment of macrocystic and mixed lymphatic malformations (LMs). MATERIALS AND METHODS: This single-center retrospective review identified 41 children (17 boys; 24 girls; age range, 1 month to 15.4 y) who underwent sclerotherapy with doxycycline (n = 32) or STS (n = 9) for macrocystic (n = 31) or mixed (n = 10) LMs. There were 114 treatments performed, averaging 2...
October 21, 2016: Journal of Vascular and Interventional Radiology: JVIR
C Lorca-García, B Berenguer, E de Tomás Palacios, C Marín Molina
INTRODUCTION: Periorbital lymphatic malformations are a rare entity in which diagnosis and treatment could be a challenge for surgeons. Management may be conservative, or they can be treated by sclerotherapy, surgery or combination of both. CLINICAL OBSERVATION: We present two cases of periorbital lymphatic malformations treated by sclerotherapy (OK-432 or bleomycin) and surgery in which we achieve good results with minimum morbidity. COMMENTS: Surgery combined with sclerotherapy is a good option of treatment for periorbital lymphatic malformations because surgery can be more conservative and therefore, sequelae are lower...
April 20, 2016: Cirugía Pediátrica: Organo Oficial de la Sociedad Española de Cirugía Pediátrica
F Heran Dreyfus, O Galatoire, P Koskas, F Lafitte, E Nau, O Bergès
Orbito-palpebral vascular pathology represents 10% of all the diseases of this area. The lesion may be discovered during a brain CT scan or MRI, or because it causes clinical symptoms such as orbital mass, visual or oculomotor alteration, pain, proptosis, or acute bleeding due to a complication of the lesion (hemorrhage, thrombosis). We present these lesions using an anatomical, clinical, imaging and therapeutic approach. We distinguish four different entities. Vascular tumors have common imaging characteristics (hypersignal on T2 sequence, contrast enhancement, abnormal vascularization well depicted with ultrasound and Doppler, and possible bleeding)...
October 18, 2016: Journal Français D'ophtalmologie
Candace L White, Brandon Olivieri, Ricardo Restrepo
No abstract text is available yet for this article.
October 11, 2016: AJR. American Journal of Roentgenology
Paloma Triana, Mariela Dore, Vanesa Nuñez Cerezo, Manuel Cervantes, Alejandra Vilanova Sánchez, Miriam Miguel Ferrero, Mercedes Díaz González, Juan Carlos Lopez-Gutierrez
Aim of the Study mTOR inhibitors are showing promising results in the management of vascular anomalies. Although current controlled trials remain to be completed, many individual experiences are being published. We present our series of children with complex vascular anomalies treated with sirolimus. Patients and Methods A retrospective review of 41 patients treated with sirolimus between January 2011 and December 2015 was performed: 15% (n = 6) had vascular tumors (4 kaposiform hemangioendotheliomas, 1 PTEN) and 85% (n = 35) had malformations (13 generalized lymphatic anomalies/Gorham-Stout diseases [GSD], 1 kaposiform lymphangiomatosis [KLA], 11 large lymphatic malformations (LMs) in critical areas, 2 lymphedemas, 4 venous malformations, and 4 aggressive arteriovenous malformations [AVM])...
October 10, 2016: European Journal of Pediatric Surgery
María Librada Porriño-Bustamante, José Aneiros-Fernández, Juan Antonio Retámero, María Antonia Fernández-Pugnaire
Hobnail hemangioma, also known as targetoid hemosiderotic hemangioma, is an uncommon vascular proliferation that clinically presents as a small solitary red to purple papule or macule, located on the limbs or trunk. Multiple lesions and atypical locations have been described. Histopathologically, it exhibits a biphasic pattern, with dilated vessels in the superficial dermis and angulated vessels in the deeper dermis, with endothelial cells that show a hobnail appearance. There is controversy about the histogenetic origin of hobnail hemangioma, although recent studies support that it is a lymphatic malformation...
October 5, 2016: Journal of Cutaneous Medicine and Surgery
Joanna H Tu, Huy M Do, Viraat Patel, Kristen W Yeom, Joyce M C Teng
BACKGROUND: Sclerotherapy is one of the most commonly used minimally invasive interventions in the treatment of macrocystic lymphatic malformations (LMs). Several different sclerosing agents and injection protocols have been reported in the literature, each with varying degrees of success. The safety and efficacy of the treatments have not been evaluated comparatively in the pediatric population. METHODS: Chart review of pediatric patients with macrocystic/mixed head and neck LMs who underwent sclerotherapy using OK-432, doxycycline, or ethanolamine oleate at Lucile Packard Children's Hospital at Stanford during 2000-2014...
October 5, 2016: Journal of Neurointerventional Surgery
Hai Wei Wu, Xuan Wang, Jia Wei Zheng, Hai Guang Zhao, Jing Ge, Ling Zhang, Yan An Wang, Li Xin Su, Xin Dong Fan
Treatment of microcystic lymphatic malformations (LMs) is still a great challenge to physicians in the field of managing vascular anomalies. Several kinds of treatment have been proposed for microcystic LMs, but the responses to these treatment modalities vary considerably among individuals. The aim of the study was to investigate the safety and efficacy of intralesional injection of pingyangmycin for microcystic LMs located in the deep facial region.Twenty-one consecutive patients with deep-seated facial microcystic LMs were treated with intralesional injection of pingyangmycin between March 2010 and April 2015...
September 2016: Medicine (Baltimore)
Cameron C Trenor
We have entered an exciting era in the care of patients with vascular anomalies. These disorders require multidisciplinary care and coordination and dedicated centers have emerged to address this need. Vascular tumors have been treated with medical therapies for many years, while malformations have been historically treated with endovascular and operative procedures. The recent serendipitous discoveries of propranolol and sirolimus for vascular anomalies have revolutionized this field. In particular, sirolimus responses are challenging the dogma that vascular malformations are not biologically active...
September 2016: Seminars in Cutaneous Medicine and Surgery
A Yasmine Kirkorian, Anna L Grossberg, Katherine B Püttgen
The fundamental genetics of many isolated vascular anomalies and syndromes associated with vascular anomalies have been elucidated. The rate of discovery continues to increase, expanding our understanding of the underlying interconnected molecular pathways. This review summarizes genetic and clinical information on the following diagnoses: capillary malformation, venous malformation, lymphatic malformation, arteriovenous malformation, PIK3CA-related overgrowth spectrum (PROS), Proteus syndrome, SOLAMEN syndrome, Sturge-Weber syndrome, phakomatosis pigmentovascularis, congenital hemangioma, verrucous venous malformation, cutaneomucosal venous malformation, blue rubber bleb nevus syndrome, capillary malformation-arteriovenous malformation syndrome, Parkes-Weber syndrome, and Maffucci syndrome...
September 2016: Seminars in Cutaneous Medicine and Surgery
Yasmeen Khatib, Madhura Dande, Richa D Patel, Vinod Gite
Vascular malformations (VMs) are structural malformations of vascular development causing soft tissue abnormality with functional and esthetic impairment. They are named by their predominant vessel type as arterial, venous, lymphatic or mixed types. VMs of the parotid gland are extremely rare and constitute a distinct entity of parotid pathology that requires specific diagnostic tools and management. Till 2013, only fifty cases of VMs of the parotid have been described in the literature. We present a case of a venolymphatic malformation of the parotid gland extending into the parapharyngeal space in a 21-year-old male who presented with a swelling on the left side of the face extending into the neck...
May 2016: Journal of Oral and Maxillofacial Pathology: JOMFP
S Vignes
Lymphedema results from impaired lymphatic transport with increased limb volume. Lymphedema are divided in primary and secondary forms. Upper-limb lymphedema secondary to breast cancer treatment is the most frequent in France. Primary lymphedema is sporadic, rarely familial or associated with complex malformative or genetic disorders. Diagnosis of lymphedema is mainly clinical and lymphoscintigraphy is useful in primary form to assess precisely the lymphatic function of the two limbs. Erysipelas (cellulitis) is the main complication, but psychological or functional discomfort may occur throughout the course of lymphedema...
August 31, 2016: La Revue de Médecine Interne
Sebastian Gómez-Galán, Manuel Santiago Mosquera-Paz, Jorge Ceballos, Paola Andrea Cifuentes-Grillo, Laura Gutiérrez-Soriano
BACKGROUND: Lymphangiomas are a heterogeneous group of congenital vascular malformations characterized by cystic dilation of lymphatic vessels. They can occur at any age, but they are more common during childhood and in cutaneous localizations. Hemangiomas and vascular malformations of the gastrointestinal tract are very uncommon. Most are asymptomatic and diagnosis is often made as an incidental finding during endoscopy. On rare occasions the initial manifestation can be chronic anemia due to low grade gastrointestinal bleeding...
2016: BMC Research Notes
Hui Lu, Qiang Chen, Hui Shen, Ganmin Ye
BACKGROUND: Rapidly involuting congenital hemangioma is a congenital soft tissue tumor, which is difficult to excise completely and rather prone to recur. This atypical tumor combined with capillary-lymphatic-venous malformation was not reported in the literature. CASE PRESENTATION: We report an atypical case of a 16-year-old teenager who was born with a mass in his right upper limb. Since there is a recurrence after excision for several times and had a serious impact on daily life, we chose amputation...
2016: World Journal of Surgical Oncology
June K Wu, Ellen D Hooper, Sherelle L Laifer-Narin, Lynn L Simpson, Jessica Kandel, Carrie J Shawber
Lymphatic malformations (LMs) are congenital lymphatic lesions that impose significant and costly morbidities on affected patients. Treatment options are limited due to incomplete understanding of LM pathobiology. Expression of an activated β2-adrenergic receptor has been described in LM tissue, suggesting that this pathway may contribute to the clinical manifestations of LM. We hypothesized that propranolol, a β-adrenergic receptor antagonist, might improve symptoms of patients with LMs and lymphatic anomalies...
September 2016: Pediatrics
Nina Bögershausen, Umut Altunoglu, Filippo Beleggia, Gökhan Yigit, Hülya Kayserili, Peter Nürnberg, Yun Li, Janine Altmüller, Bernd Wollnik
Kabuki syndrome (KS) is a rare developmental disorder characterized by multiple congenital malformations, postnatal growth retardation, intellectual disability, and recognizable facial features. It is mainly caused by mutations in either KMT2D or KDM6A. We describe a 14-year-old boy with KS presenting with an unusual combination of bilateral microphthalmia with orbital cystic venous lymphatic malformation and neonatal cholestasis with bile duct paucity, in addition to the typical clinical features of KS. We identified the novel KMT2D mutation c...
August 17, 2016: American Journal of Medical Genetics. Part A
Marla N Jahnke
Vascular lesions in childhood are comprised of vascular tumors and vascular malformations. Vascular tumors encompass neoplasms of the vascular system, of which infantile hemangiomas (IHs) are the most common. Vascular malformations, on the other hand, consist of lesions due to anomalous development of the vascular system, including the capillary, venous, arterial, and lymphatic systems. Capillary malformations represent the most frequent type of vascular malformation. IHs and vascular malformations tend to follow relatively predictable growth patterns in that IHs grow then involute during early childhood, whereas vascular malformations tend to exhibit little change...
August 1, 2016: Pediatric Annals
Bruce K Rubin
Plastic bronchitis is an uncommon and probably underrecognized disorder, diagnosed by the expectoration or bronchoscopic removal of firm, cohesive, branching casts. It should not be confused with purulent mucous plugging of the airway as seen in patients with cystic fibrosis or bronchiectasis. Few medications have been shown to be effective and some are now recognized as potentially harmful. Current research directions in plastic bronchitis research include understanding the genetics of lymphatic development and maldevelopment, determining how abnormal lymphatic malformations contribute to cast formation, and developing new treatments...
September 2016: Clinics in Chest Medicine
Igor Banzic, Milos Brankovic, Živan Maksimović, Lazar Davidović, Miroslav Marković, Zoran Rančić
OBJECTIVES: Parkes Weber syndrome is a congenital vascular malformation which consists of capillary malformation, venous malformation, lymphatic malformation, and arteriovenous malformation. Although Parkes Weber syndrome is a clinically distinctive entity with serious complications, it is still frequently misdiagnosed as Klippel-Trenaunay syndrome that consists of the triad capillary malformation, venous malformation, and lymphatic malformation. METHODS: We performed a systematic review investigating clinical, diagnostic, and treatment modalities of Parkes Weber syndrome (PubMed/MEDLINE, Embase, and Cochrane databases)...
August 9, 2016: Phlebology
Gabriele Tonni, Roberta Granese, Eduardo Félix Martins Santana, José Pedro Parise Filho, Isabela Bottura, Alberto Borges Peixoto, Annamaria Giacobbe, Andrea Azzerboni, Edward Araujo Júnior
AIM: The aim of this study was to review prenatally diagnosed tumors of the head and neck in the fetus and to report antenatal and postnatal outcomes. METHODS: PubMed/Medline, EMBASE/SCOPUS, Cochrane database and Google Scholar were reviewed over the last 20 years. No language or article type restriction was used. RESULTS: A total of 1940 record were retrieved. Of the 713 records screened, 566 full-text articles were assessed for eligibility...
August 10, 2016: Journal of Perinatal Medicine
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