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    1 OF 132

    Treatment of Lymphatic Malformations with the mTOR Inhibitor Sirolimus: A Systematic Review.
    Lymphat Res Biol 2018 Jun 20. Epub 2018 Jun 20.
    Department of Otolaryngology, Head and Neck Surgery, University of Leipzig , Leipzig, Germany .
    Background: Extensive lymphatic malformations are low-flow vascular malformations that can cause devastating complications. Treatment of these malformations is challenging. This systematic review presents current use of sirolimus in patients with extensive lymphatic malformations. Read More

    CT Lymphangiography (CTL) in Primary Intestinal Lymphangiectasia (PIL): A Comparative Study with Intraoperative Enteroscopy (IOE).
    Acad Radiol 2018 Jun 6. Epub 2018 Jun 6.
    Department of Radiology, Beijing Shijitan Hospital, Capital Medical University, No. 10 Tieyi St, Haidian District, Beijing 100038, China. Electronic address:
    Rationale And Objectives: To investigate the clinical feasibility of CT lymphangiography (CTL) in primary intestinal lymphangiectasia (PIL) by comparison with intraoperative enteroscopy (IOE) during exploratory laparotomy.

    Materials And Methods: Eleven PIL patients (F/M, two/nine, age range 10-37 years) were recruited in this study, and they were performed IOE during exploratory laparotomy for suspected serious lymphatic-intestinal leakages. All the patients were performed CTL before surgery, and the imaging data were reviewed by two radiologists separately. Read More

    Ectopic cervical thymic cyst in a seven year old: a diagnostic challenge.
    J Pak Med Assoc 2018 May;68(5):797-800
    Aga Khan University Hospital, Karachi.
    Cervical thymus cyst is a rare entity and resembles lymphangioma or branchial cleft cyst therefore, presents as a diagnostic challenge. It has a slight male predominance and presents on the left side of the neck in up to 70% of cases. It is a painless mass that progressively increases. Read More

    A young woman with steroid-responsive, IgG4-positive plasma cell-enriched cystic lymphangioma and chylous ascites.
    Clin Case Rep 2018 Jun 17;6(6):1098-1100. Epub 2018 Apr 17.
    Division of Hematology Department of Medicine University of British Columbia Vancouver British Columbia Canada.
    Lymphangiomas are benign tumors of the lymphatic vessels, which can be inflammatory and occasionally steroid-responsive. IgG4-related disease (IgG4-RD) is a recently defined fibro-inflammatory condition. We describe a novel association between reactive IgG4+ plasma cells and cystic lymphangioma in a young woman who had a dramatic clinical response to steroids. Read More

    Symptomatic lymphangioma of the adrenal gland: a case report.
    J Surg Case Rep 2018 May 18;2018(5):rjy106. Epub 2018 May 18.
    Department of General and Visceral Surgery, Cantonal Hospital of Lucerne, Lucerne, Switzerland.
    Lymphangiomas (LAs) are rare benign tumors of the lymphatic vessels. In total, 95% of all reported LAs are located in the head, neck and the mediastinum. LAs of the adrenal gland are very rare and currently, only ~54 cases have been reported in literature. Read More

    A Supernumerary Nipple-Like Clinical Presentation of Lymphangioma Circumscriptum.
    Case Rep Dermatol Med 2018 11;2018:6925105. Epub 2018 Feb 11.
    Department of Dermatology, University of Texas McGovern Medical School, Houston, TX, USA.
    Lymphangioma circumscriptum is a superficially localized variant of lymphangioma. The characteristic clinical presentation is a "frogspawn" grouping of vesicles or papulovesicles on the proximal limb or limb girdle areas. Though most lymphangiomas develop congenitally, the lymphangioma circumscriptum subtype is known to present in adults. Read More

    Efficacy and absorption of topical sirolimus for the treatment of vascular anomalies in children: A case series.
    Pediatr Dermatol 2018 May 23. Epub 2018 May 23.
    Department of General Pediatrics, Centre Hospitalier Universitaire Sainte-Justine, Université de Montréal, Montréal, QC, Canada.
    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. Read More

    Blaschko-linear "Congenital Mixed Hemato-lymphangio-keratoma Serpiginosum" Naeviforme: A New Hybrid Entity or Various Morphological Reflections of the Two Different Vascular Abnormalities?
    J Clin Aesthet Dermatol 2018 May 1;11(5):43-47. Epub 2018 May 1.
    Dr. Tas is with the Health Sciences University, Istanbul Bagcilar Research and Training Hospital, Department of Dermatology in Istanbul, Turkey.
    We present the case of a 15-year-old girl who presented with an unusual grouping of lesions on her upper left leg. The lesions had been present since birth. The patient had five different types of lesions: 1) transparent grouped or scattered yellowish vesicles; 2) keratotic-surfaced, grouped dark-yellowish papules; 3) bright-red grouped papules; 4) keratotic-surfaced grouped dark-red papules; and 5) patchy, punctate, and erythematous red macules. Read More

    Fatal Airway Obstruction in a Man With a Cystic Hygroma.
    Am J Forensic Med Pathol 2018 May 3. Epub 2018 May 3.
    From the Department of Pathology and Laboratory Medicine, Spectrum Health Blodgett Campus, Grand Rapids, MI.
    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. Read More

    Lymphangioma Circumscriptum Post Radiotherapy for Penile Cancer Treated with CO₂ Laser.
    Acta Dermatovenerol Croat 2018 Apr;26(1):53-57
    Ancuta Proca Nicula, MD, PhD student, "Victor Babes" University of Medicine and Pharmacy, Dermatology Department, Eftimie Murgu No 2, 300041 Timisoara, Romania;
    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. Read More

    intrapartum treatment procedure in two fetuses with airway obstruction.
    Obstet Gynecol Sci 2018 May 9;61(3):417-420. Epub 2018 Apr 9.
    Department of Otolaryngology, University of Ulsan College of Medicine, Asan Medical Center, Seoul, Korea.
    The intrapartum treatment (EXIT) procedure was introduced to reduce fetal hypoxic damage while establishing an airway in fetuses with upper and lower airway obstruction. Delivery of the fetal head and shoulders while maintaining the uteroplacental circulation offers time to secure the fetal airway. Here, we report two cases of EXIT procedure for fetal airway obstruction, which were successfully managed with extensive preoperative planning by a professional multidisciplinary team. Read More

    Lymphangioma on 68Ga-NOTA-Evans Blue PET/MRI.
    Clin Nucl Med 2018 Jul;43(7):553-555
    From the Departments of Nuclear Medicine and Beijing Key Laboratory of Molecular Targeted Diagnosis and Therapy in Nuclear Medicine.
    A 17-year-old adolescent boy with a history of hypoproteinemia underwent Ga-NEB PET/MRI to evaluate possible lymphatic disorders suggested by FDG PET/CT imaging. The images revealed multiple lesions with increased Ga-NEB uptake and high signal on T2-weighted images, consistent with likely lymphatic disorder. Histopathological examination from biopsied specimen confirmed lymphangioma. Read More

    Efficacy and Safety of a Novel Method of Insulated Intralesional Radiofrequency Ablation for Deep Dermal and Subcutaneous Lesions: A 3-Year Institutional Experience.
    Dermatol Surg 2018 May;44(5):714-720
    All authors are affiliated with the Department of Dermatology and Venereology, All India Institute of Medical Sciences, New Delhi, India.
    Background: Although insulated intralesional radiofrequency ablation (IL-RFA) is being increasingly used in other specialties, not much information on its safety and efficacy in dermatology is available.

    Objective: To describe our experience with insulated IL-RFA for various dermatological conditions.

    Methods: This is a retrospective review of the patients who underwent IL-RFA in the past 3 years. Read More

    Cystic Lymphangioma: Are Triglycerides Always Measurable?
    Case Rep Gastrointest Med 2018 1;2018:9591420. Epub 2018 Mar 1.
    Universitair Ziekenhuis Brussel (UZ Brussel), Vrije Universiteit Brussel (VUB), Department of Gastro-Enterology, Laarbeeklaan 101, 1090 Brussels, Belgium.
    The presence of chylous fluid with high triglycerides levels on endoscopic ultrasound- (EUS-) guided fine needle aspiration (FNA) is very pathognomonic for the diagnosis of cystic lymphangiomas of the pancreas. In our case report the puncture of the pancreatic cyst showed a typical milky fluid though measurable triglyceride concentrations were absent in the laboratory. Two possible explanations were found. Read More

    Synovial sarcoma complicating Maffucci syndrome.
    Indian J Dermatol Venereol Leprol 2018 Apr 17. Epub 2018 Apr 17.
    Department of Dermatology, Farhat Hached University Hospital, Sousse, Tunisia.
    Maffucci syndrome is a rare nonhereditary disorder comprising of lymphovascular malformations and multiple enchondromas, which may be associated with several internal malignancies. This report describes a new association of Maffucci syndrome with pedal synovial sarcoma. Our case is also remarkable as lymphangioma circumscriptum is the sole lymphovascular component, which has been rarely reported. Read More

    Experience with OK-432 in lymphatic vascular malformations in a hospital from northern Mexico.
    Bol Med Hosp Infant Mex 2018 ;75(2):89-93
    Escuela de Medicina, Tecnológico de Monterrey, Monterrey. Nuevo León, México.
    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. Read More

    Percutaneous full endoscopic treatment of cystic lymphangioma of cauda equina: a case report.
    Br J Neurosurg 2018 Apr 16:1-3. Epub 2018 Apr 16.
    c Department of Neurosurgery , Nanoori Incheon Hospital , Incheon , South Korea.
    Intraspinal epidural lymphangioma of cauda equina are extremely rare, only three cases have been reported in the past. We report a 63-year-old female with lymphangioma at S1-S2 level which was resected under epidural anesthesia using a percutaneous full endoscope with no evidence of recurrence at 19 months follow-up. Read More

    Isolated splenic lymphangioma presenting as a huge mass causing anemia and abdominal distension in an adult patient: a case report.
    J Med Case Rep 2018 Apr 16;12(1):97. Epub 2018 Apr 16.
    Department of Pathology, Hassan II University Hospital, Fez, Morocco.
    Background: Lymphangiomas are uncommon benign lesions of lymphatic vessels very rarely affecting the spleen. Isolated involvement of the spleen in adult patients is rarely reported.

    Case Presentation: We report a case of a 40-year-old Arabic woman who presented with a 25-cm abdominal mass, fatigue, and anemia evolving for 6 months. Read More

    Robotic resection of benign nonadrenal retroperitoneal tumors: A consecutive case series.
    Int J Surg 2018 Apr 12;55:188-192. Epub 2018 Apr 12.
    Faculty of Medicine, The Chinese University of Hong Kong, Shatin, New Territories, Hong Kong Special Administrative Region. Electronic address:
    Background: The deep location, narrow operative space and proximity to major vessels make minimally invasive resection of nonadrenal retroperitoneal tumors (NTRs) challenging and rarely reported. This study aimed to evaluate the safety and feasibility of robotic resection of benign nonadrenal retroperitoneal tumors.

    Methods: The demographics and perioperative outcomes of a consecutive series of patients who underwent robotic NTR resection between January 1, 2015, and August 30, 2017, were analyzed. Read More

    A case of extensive acquired progressive lymphangioma.
    Pediatr Dermatol 2018 Apr 6. Epub 2018 Apr 6.
    Department of Dermatology, Mayo Clinic, Rochester, MN, USA.
    Acquired progressive lymphangioma (benign lymphangioendothelioma) is a rare lymphatic anomaly of unclear pathogenesis. Excision is generally advised for local disease, although other therapies have been tried. This report describes a unique case of extensive acquired progressive lymphangioma involving the abdomen, genitalia, and lower extremity of a 1-year-old boy. Read More

    Unilateral Straight Hair-A Symptom of Acquired Horner's Syndrome in a Neonate.
    European J Pediatr Surg Rep 2018 Jan 6;6(1):e32-e36. Epub 2018 Apr 6.
    Department of Pediatric Surgery, University Children's Hospital Basel, Basel, Switzerland.
    A multicystic tumor of the right neck was detected in a girl at 29 weeks of gestation by fetal ultrasound and magnetic resonance imaging (MRI). The baby was delivered by cesarean section at week 37 of gestation. The newborn adapted well, with minimal compromise of breathing and drinking. Read More

    Interpreting body MRI cases: classic findings in abdominal MRI.
    Abdom Radiol (NY) 2018 Mar 28. Epub 2018 Mar 28.
    Radiology, Thomas Jefferson University Hospital, 132 South 10th Street, Suite 1087, Main Building, Philadelphia, PA, 19107, USA.
    Few things in radiology are "pathognomonic" in their appearance or presentation. However, having an awareness of those findings which are specific to a certain entity is important when interpreting imaging studies. These classic findings can be identified with many imaging modalities, but no modality provides as many recognizable observations as an MRI. Read More

    Orbital Lymphaticovenous Malformation with Intradural Extension: Rare Case.
    World Neurosurg 2018 Jun 16;114:151-154. Epub 2018 Mar 16.
    Department of Clinical Neurosciences, University of Calgary, Foothills Medical Centre, Calgary, Alberta, Canada. Electronic address:
    Background: Orbital lymphaticovenous malformations (LVMs) are congenital vascular lesions that are typically infiltrative in nature. There have been reports of orbital LVMs extending intracranially through orbital fissures, but there have been no reports of intradural extension.

    Case Description: We report the first case of orbital LVM extending intradurally through a bony defect in the medial orbital roof. Read More

    Small bowel lymphangioma causing ileo-ileal intussusception in adults.
    Int J Surg Case Rep 2017 21;41:469-472. Epub 2017 Nov 21.
    Department of Surgery, Montefiore New Rochelle Hospital, New York, USA.
    Introduction: Lymphangioma is a rare benign tumor found in gastrointestinal tract. Most lymphangiomas can occur at any age and but mostly are found in children and infants. They are mainly due to malformation of the lymphatic system. Read More

    [Unusual macrocystic lymphatic malformation in an adult patient].
    Pan Afr Med J 2017 10;28:128. Epub 2017 Oct 10.
    Service de Dermatologie-Vénéréologie, CHU Ibn Rochd, Université Hassan II, Casablanca, Maroc.
    Macrocystic lymphatic malformations (MLMs) constitute a circumscribed variation in deep lymphangiomas. They are characterized by a rare occurrence in adults, by their rapid expansion and by their frequent occurrence at the level of the cervicofacial region (75%) and axillary region (20%). They rarely occur in lower limbs. Read More

    Oral lymphangiomas - clinical and histopathological relations: An immunohistochemically analyzed case series of varied clinical presentations.
    J Oral Maxillofac Pathol 2018 Jan;22(Suppl 1):S108-S111
    Department of Oral Pathology and Microbiology, Sarjug Dental College, Darbhanga, Bihar, India.
    Lymphangiomas are benign malformations that represent hamartoma of malformed lymphatics and are composed of cystically dilated lymphatic channels which do not communicate or drain into other lymphatic channels or veins leading to accumulation of lymph. They are congenital malformations with rare occurrence in oral cavity within which tongue dorsum is the most affected site. Venous malformations are often difficult to differentiate from lymphatic malformations leading to the development of several lymphatic markers like D2-40 which seems to be useful for distinguishing vascular from lymphatic vessels. Read More

    Congenital lymphangioma circumscriptum of vulva presenting as multiple giant mass lesions: a case report and literature review.
    J Obstet Gynaecol Res 2018 May 27;44(5):978-982. Epub 2018 Feb 27.
    Department of Pathology, Cancer Institute, Imam Khomeini Hospital Complex, Tehran, Iran.
    A G2L2 33-year-old woman presented to our clinic with large verrucous warty masses in labia major, perinea and the end portion of her spine measuring about 15 × 7, 9 × 7 and 8 × 8 cm, which had been enlarged following puberty and pregnancy. Her right upper and left lower limbs had gross congenital lymphedema. The masses were removed by superficial partial vulvectomy with a qualified margin and repaired without skin graft. Read More

    Triparametric ultrasound in differentiating multicystic renal masses: a rare presentation of unilateral focal renal lymphangioma.
    Radiol Case Rep 2017 Dec 18;12(4):731-737. Epub 2017 Aug 18.
    Radiology department, Southend University Hospital, Essex, UK.
    We describe a rare case of renal lymphangioma presenting as a focal unilateral multicystic renal mass and document the first reported use of triparametric ultrasound (B-mode, Doppler, and contrast-enhanced ultrasound) in its diagnosis and discrimination from other focal multicystic lesions. Renal lymphangiomas are rare, benign, typically developmental lesions composed of cystic dilatation of the lymphatic ducts, usually occurring bilaterally as perinephric collections or parapelvic cysts mimicking hydronephrosis. Radiologists have an important role in suggesting the diagnosis, as clinical presentation can be nonspecific. Read More

    Lymphangioma cavernous of the small bowel mesentery, an infrequent cause of acute abdomen in adult.
    J Surg Case Rep 2018 Feb 14;2018(2):rjy018. Epub 2018 Feb 14.
    Resident General Surgery, P.U.C.E., Quito-Ecuador.
    Lymphangioma is a benign mass lesion characterized by numerous thin-walled lymphatic spaces that usually manifests in the first few years of life. They generally appear in the head, neck and axillary regions. Abdominal lymphangiomas have been reported however they are rare 5%. Read More

    Bleomycin Sclerotherapy in Lymphangiomas of Head and Neck: Prospective Study of 8 Cases.
    Indian J Otolaryngol Head Neck Surg 2018 Mar 8;70(1):145-148. Epub 2018 Jan 8.
    Department of ENT, Tata Main Hospital, Bistupur, Jamshedpur, 831001 India.
    The aim of this study is to evaluate the role of bleomycin as a primary mode of nonsurgical treatment in lymphangiomas of head and neck and study their complications. This is a prospective study of 8 patients diagnosed with lymphangioma of head and neck presenting to ENT department of Tata main Hospital from December 2014 to January 2017. They were clinically and radiologically evaluated and treated with intralesional injection of bleomycin diluted in normal saline. Read More

    Thirty-day perioperative outcomes in resection of cervical lymphatic malformations.
    Int J Pediatr Otorhinolaryngol 2018 Mar 3;106:31-34. Epub 2018 Jan 3.
    Ann & Robert H Lurie Children's Hospital of Chicago, Department of Otolarynology Head and Neck Surgery, Northwestern Feinberg School of Medicine, 225 E Chicago Ave, Chicago, IL 60610-11, United States.
    Introduction: Limited information exists regarding short-term morbidity in the resection of lymphatic malformations. In order to make informed collaborative medical decisions, clinicians and families would benefit from information on 30-day outcomes and the expected course associated with surgical excision of lymphatic malformations.

    Methods: A retrospective chart review was conducted to develop a case series of patients who underwent resection of lymphatic malformation at a pediatric tertiary care center between June 1, 2007 and September 30, 2016. Read More

    Sirolimus-induced regression of a large orbital lymphangioma.
    Orbit 2018 Feb 6:1-2. Epub 2018 Feb 6.
    b Center for Orbital Diseases, Medical Center and Medical Faculty , University of Freiburg , Freiburg , Germany.
    Microcystic lymphatic malformations are difficult to treat surgically, especially when located in the orbital apex. Recently, pharmacologic inhibition of the mTOR pathway by sirolimus was reported as a safe and efficacious treatment option for lymphatic malformations (also known as lymphangiomas). We report the case of a young male patient in which a unilateral, retrobulbar lymphatic malformation regressed to a large extent under treatment with 1 mg sirolimus given orally twice a day over a period of six months. Read More

    The use of sirolimus in the treatment of giant cystic lymphangioma: Four case reports and update of medical therapy.
    Medicine (Baltimore) 2017 Dec;96(51):e8871
    Neonatal Intensive Care Unit.
    Rationale: Lymphatic malformations (LMs) are rare and benign anomalies resulting from the defective embryological development of the primordial lymphatic structures. Due to their permeative growth throughout all tissue layers, treatment is often challenging. Small asymptomatic lesions can be conservatively managed, while symptomatic lesions require active management. Read More

    Ileal Cystic Lymphangioma presenting with Acute Appendicitis.
    J Indian Assoc Pediatr Surg 2018 Jan-Mar;23(1):36-38
    Department of Pediatric Surgery, National University of Hospital, Singapore.
    Mesenteric lymphatic malformations are rare benign tumors that are most commonly found in children. The presentation of these tumors can be variable. It has been suggested that mesenteric lymphatic malformations are congenital; however, there is evidence that their size may be increased by infection. Read More

    Two cases of hemolymphangioma in the thoracic spinal canal and spinal epidural space on MRI: The first report in the literature.
    Medicine (Baltimore) 2017 Dec;96(52):e9524
    Department of Radiology.
    Rationale: Hemolymphangioma is a rare, noninvasive benign tumor of mesenchymal origin resulting from malformation of vascular and lymphatic vessels. The incidence of hemolymphangioma in the spinal canal is low.

    Patient Concerns: This report describes 2 patients with a lesion located in the thoracic spinal canal or spinal epidural space, who were misdiagnosed with suspected meningioma or suspected schwannoma, respectively, based on magnetic resonance imaging (MRI). Read More

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