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Ann Med Surg (Lond) 2022 Jun 19;78:103835. Epub 2022 May 19.

ENT Head and Neck Surgery Department, Ibn Rochd University Hospital, Faculty of Medicine and Pharmacy, Hassan II University, Casablanca, Morocco.

Introduction: Cystic lymphangiomas are rare dysembrioplasias that occur mostly in children. Although benign, these tumors remain potentially life-threatening, due to the possible compression of the upper airway. The management of cystic lymphangiomas is still somewhat controversial, with surgery generally being the first-line treatment. Read More

J Craniofac Surg 2022 May 17;33(3):850-854. Epub 2021 Sep 17.

Department of Burns and Plastic Surgery, Children's Hospital of Nanjing Medical University, Nanjing, China.

Background: The cervicofacial lymphatic malformations (LMs) often have poor outcomes due to their microcystic component and diffuse infiltration. Mostly, traditional treatments are inadequate for these refractory cases. Recent researches have shown that sirolimus is effective in the treatment of complicated LMs, however, there is still no standard strategy. Read More

Front Pediatr 2021 18;9:709223. Epub 2021 Aug 18.

Neonatal Surgery Unit, Medical and Surgical Department of the Fetus, Newborn, and Infant, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.

Lymphatic malformations (LMs) are rare entities, sometimes difficult to treat, that may be life-threatening when intricately connected to airway structures. Invasive treatments are occasionally required, with sclerotherapy considered the treatment of choice and surgery as a second-line approach. The aim of the present study was to evaluate our multidisciplinary team experience in treating newborns affected by LMs requiring operative management, while defining early outcomes. Read More

J Pediatr Surg 2021 May 30;56(5):1062-1067. Epub 2020 Sep 30.

Division of Pediatric Surgery, Department of Surgery, Children's Hospital Los Angeles, 4650 Sunset Blvd., MS#100, Los Angeles, CA, USA 90027; Department of Surgery, Keck School of Medicine, University of Southern California, 1975 Zonal Ave, Los Angeles, CA, USA 90089. Electronic address:

Background/purpose: Cervicofacial lymphatic malformations (CFLM) are rare, potentially life-threatening vascular anomalies, yet reports on multidisciplinary treatment strategies are lacking. We evaluated outcomes for CFLMs following sclerotherapy, surgical resection, and/or medical management.

Methods: We identified children with a CFLM at a vascular anomalies center from 2004 to 2019. Read More

Pediatrics 2020 12;146(6)

Plastic Surgery Unit,

The treatment of microcystic and combined lymphangiomas, especially in the head and neck region, is still a challenge because the lymphangiomas do not respond to conventional therapies and their recurrence rate is high, regardless of the treatment choice. Complete surgical resection is the main treatment of lymphangiomas, but because of localization perioperative complications, such as bleeding, neural damage and airway obstruction are common disadvantages of this method. Bleomycin-based sclerotherapy is another common therapeutic approach, in which the lymphocysts are aspirated, and 25% to 50% of their volumes are replaced with a sclerotisant drug. Read More

Front Pediatr 2020 28;8:418. Epub 2020 Jul 28.

Service de Réanimation Néonatale, HFME, Hospices Civils de Lyon, Bron, France.

Cystic lymphatic malformations result from an abnormal embryological development of the lymphatic structures. Here we report on a case of a preterm female baby, born at 34 weeks of gestation, with a voluminous cervicofacial cystic lymphatic malformation responsible for an airway obstruction. An mTOR inhibitor, sirolimus, was started from the first day of life, and was combined with iterative sclerotherapy procedures. Read More

Rev Med Liege 2020 Jul;75(7-8):541-543

Service de Chirurgie cardiovasculaire, CHU Liège, Belgique.

Cystic lymphangioma is a rare and benign lymphatic malformation found most often in the first two years of life. Clinical manifestations are diverse and depend on size and site of the lesion. The most frequently affected sites are cervicofacial and axillary areas. Read More

J Surg Case Rep 2020 Jul 31;2020(7):rjaa179. Epub 2020 Jul 31.

U.O.S. Maxillofacial Surgery, S. Andrea Hospital, Rome, Italy.

Cystic lymphangioma (CL) in adult is a very rare pathology. Its etiology remains unclear, but it is supposed to be congenital or to be a result of obstruction and lymph fluid retention of developing lymphatic vessels. It generally occurs in the head and neck region, probably because of the rich lymphatics in this area. Read More

J Craniofac Surg 2019 Sep;30(6):e553-e555

Department of Otolaryngology-Head and Neck Surgery, Shanghai Children's Hospital, Shanghai Jiao Tong University.

Objective: Lymphatic malformations (LMs) are rare and benign diseases. This article aimed to present a series of surgically treated giant cervicofacial LMs in children.

Methods: A retrospective analysis of giant cervicofacial LMs (tumor diameter greater than 10 cm) in children over the past 8 years was performed in our department. Read More

Vasc Endovascular Surg 2019 Feb 29;53(2):170-176. Epub 2018 Nov 29.

4 ENT Clinic of Red Cross Hospital, Athens, Greece.

Lymphangiomas are rare benign malformations of the lymphatic system, commonly present in children, over the head and neck area. Occasionally, they can grow significantly in size and especially those located over the cervical region can cause airway obstruction and become life-threatening. Recurrent lymphangiomas usually occur during the early postsurgical period and 80% of them within the first 3 to 5 years. Read More

Pan Afr Med J 2018 6;29:202. Epub 2018 Apr 6.

Service de Dermatologie, CHU Hassan II, Fes, Maroc.

Superficial circumscribed lymphangiomas are predominantly benign lesions occuring in the cervicofacial area. These malformations of the lymphatic vessels are frequently diagnosed in children. We report the clinical case of lymphangioma of the tongue that occurred in an old man. Read More

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

Arch Plast Surg 2016 Jan 15;43(1):10-8. Epub 2016 Jan 15.

Department of Plastic and Reconstructive Surgery, Kyungpook National University School of Medicine, Daegu, Korea.

Background: Lymphatic malformation (LM) is a form of congenital vascular malformation with a low incidence. Although LM has been studied, no consensus has emerged regarding its cause or treatment.

Methods: In this study, we retrospectively evaluated 40 patients who visited our vascular anomalies center for the treatment of cervicofacial LM, which is a common manifestation of LM. Read More

Int J Clin Exp Pathol 2015 1;8(5):5924-8. Epub 2015 May 1.

Department of Pathology, Fujieda Municipal General Hospital Shizuoka, Japan.

Cystic lymphangioma, a lymphatic system malformation, is usually observed in infants and children and is rarely found in adults. It most commonly occurs in the cervicofacial region, followed by the axilla. Mediastinal cystic lymphangioma is rare, accounting for 1. Read More

Curr Pediatr Rev 2014 ;10(3):238-48

Princess Margaret Hospital, Roberts Road, Subiaco, Western Australia 6008, Australia.

Introduction: Lymphangioma is a rare benign cyst caused by congenital malformation of the lymphatic systems that often occurs in the cervicofacial region. There is no consensus on its management: Observation, aspiration, injection, cryotherapy, electrocautery, radiation, laser, ligation and excision.

Methods: We performed a literature search with the keywords "cystic hygroma", "lymphangioma", "management", "OK 432" and "picibanil" from Medline, Embase and PubMed databases. Read More

J Craniofac Surg 2013 Jul;24(4):1307-9

Department of Maxillofacial Surgery and ENT, Avicenne University Medical Center, Rabat, Morocco.

Purpose: Lymphatic malformations (LMs) are benign lesions. Most of them are found in head and neck regions as asymptomatic mass, but giant lymphangiomas may affect breathing or swallowing and constitute a major therapeutic challenge.

Methods: A retrospective analysis of giant head and neck LMs with impairment of respiration or swallow for the past 11 years was performed in the Department of Maxillofacial Surgery and ENT of the Avicenne Medical University Center. Read More

J Maxillofac Oral Surg 2011 Jun 20;10(2):152-4. Epub 2011 Apr 20.

Surgical treatment of lymphangioma of the face is a difficult task to achieve due to close vicinity of the lesion to the facial nerve and possibility of scar tissue formation. Inefficient surgical removals generally will give rise to high recurrence rates because of infiltrative and diffuse extension of the lesion. However, complete cure has been described by non-surgical methods. Read More

Indian J Dermatol 2011 May;56(3):321-3

Department of Dermatology, Venereology and Leprosy, Grant Medical College and Sir J J Group of Hospitals, Mumbai, India .

Primary cutaneous actinomycosis caused by Actinomyces israelii occurs most commonly in the cervicofacial area. It commonly presents as "lumpy jaw" with draining sinuses which discharge the characteristic "sulfur granules". A low index of suspicion and a low sensitivity in culturing the organism, due to its fastidious nature often delays the diagnosis. Read More

J Indian Med Assoc 2010 Sep;108(9):606, 608

Department of Paediatric Medicine, NRS Medical College and Hospital, Kolkata 700014.

Lymphatic malformations are the second most common benign vascular tumour in children, after haemangioma. They may be localised or generalised and commonly occur in the cervicofacial region, axilla and thorax. They appear early in life with almost all cases evident by the age of 3 years. Read More

Int J Pediatr Otorhinolaryngol 2011 May 21;75(5):673-7. Epub 2011 Mar 21.

Department of Paediatric Otolaryngology, Great Ormond Street Hospital, Great Ormond Street, London, WC1N 3JH, United Kingdom.

Objective: There has been much recent focus on sclerotherapy treatment of lymphatic malformations with OK432. Surgical treatment however can have a number of advantages, including complete curative excision. The aim of this study was to evaluate the results of surgical excision as the primary (first) treatment for this condition. Read More

Rev Laryngol Otol Rhinol (Bord) 2010 ;131(2):75-82

Hôtel-Dieu, Service d'O.R.L. et de Chirurgie Cervico-Faciale, Place A. Ricordeau, BP 1005, 44093 Nantes Cedex 01, France.

The authors propose an update on cervicofacial congenital cysts and fistulas' symptomatology. Embryological data, epidemiology and clinical manifestations are described. A reminder of the therapeutic principles is proposed as well as the evolution of these congenital pathologies, which may or may not involve the branchial system. Read More

J Pediatr Surg 2010 Aug;45(8):1627-32

Department of Pediatric Surgery, Hacettepe University Faculty of Medicine, Ankara 06100, Turkey.

Background And Purpose: Sclerotherapy with bleomycin sulfate (BS) is currently used in the management of cervicofacial cystic lymphatic malformations in children. Neurotoxic adverse effects of BS after intraventricular or intracavitary administration have been reported; however, the effects of intralesionally administered BS on the adjacent peripheral neural structures have not been previously investigated. The authors conducted a clinical experimental study to evaluate facial nerve function in children who have undergone BS sclerotherapy for cervicofacial cystic lymphatic malformation. Read More

Zhonghua Yi Xue Za Zhi 2009 Dec;89(48):3413-6

Department of Otolaryngology, Qilu Hospital, Shandong University, Jinan 250012, China.

Objective: To discuss the methods of surgical treatment and their timing choices of cervical lymphangioma.

Methods: A retrospective review of 53 patients with cervicofacial lymphangioma were treated surgically from July 1990 to December 2008. The age at operation was from 6. Read More

Int J Pediatr Otorhinolaryngol 2010 Apr 18;74(4):398-403. Epub 2010 Feb 18.

Our Lady's Hospital for Sick Children, Crumlin, Dublin, D12, Republic of Ireland.

Objective: The aim of this study was to develop a management algorithm for cervicofacial lymphatic malformations, based on the authors' experience in managing these lesions as well as current literature on the subject.

Study Design And Methods: A retrospective medical record review of all the patients treated for lymphatic malformations at our institution during a 10-year period (1998-2008) was performed.

Data Collected: age at diagnosis, location and type of lesion, radiologic investigation performed, presenting symptoms, treatment modality used, complications and results achieved. Read More

Int J Pediatr Otorhinolaryngol 2009 Jun 25;73(6):833-7. Epub 2009 Mar 25.

Department of Oral and Maxillofacil Surgery, China.

Objectives: Various modalities have been used to treat lymphatic malformations, such as steroids, the injection of sclerosing agents, aspiration, and surgery. Giant macrocystic lymphangiomas involving the cervicofacial region in infants and children constitute a major therapeutic challenge.

Methods: This was a retrospective review of 47 pediatric patients with giant macrocystic lymphatic malformations of the cervicofacial region that underwent surgical resections. Read More

J Otolaryngol Head Neck Surg 2008 Apr;37(2):169-73

Division of Pediatric Otolaryngology & Head/Neck Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA.

Objective: To provide clinicians with an overview of the complications and functional and clinical outcomes of children affected by massive lymphatic malformations affecting multiple sites in the head, neck, and/or chest.

Methods: We reviewed the medical records of all patients who received treatment for massive cervicofacial lymphatic malformations at our tertiary care pediatric institution during a 17-year period. Massive lesions were defined as (1) unilateral lesions that infiltrated at least 50% of two anatomic regions, including the head, neck, or chest; (2) unilateral lesions in which there was involvement of the midline structures of the head or neck; or (3) bilateral lesions in which 50% of each side was affected. Read More

Ann Otolaryngol Chir Cervicofac 2007 Dec;124(6):277-84

Service d'ORL et de chirurgie cervicofaciale, hôpital d'instruction des armées Omar-Bongo-Ondimba, BP 20404, Libreville, Gabon.

Objectives: To review the etiopathogenesis, diagnosis, and the role of surgery in the management of childhood cystic cervicofacial lymphangioma in Gabon.

Material And Methods: Our retrospective study concerned 16 cases of childhood cystic cervicofacial lymphangioma treated surgically between 1990 and 2004.

Results: The sample included ten girls and six boys whose average age was 30. Read More

Acta Otorrinolaringol Esp 2007 May;58(5):222-4

Servicio de Cirugía Oral y Maxilofacial, Hospital del Río Hortega, Valladolid, España.

The treatment of cervicofacial lymphangiomas has been changing in recent years. OK-432 appears to be a relatively safe and effective treatment option for patients of all ages. An intracystic injection of OK-432 produces a local inflammatory reaction, leading to resolution of the lesion. Read More

Laryngorhinootologie 2006 Dec;85(12):883-91

Klinik für Hals-Nasen- und Ohrenheilkunde der Philipps-Universität Marburg.

The successful treatment of vascular anomalies depends on profound knowledge of the biologic behavior of vascular lesions and their correct classification. On the base of the clinical course Mulliken and Glowacki developed a biologic classification that was accepted as official classification by the ISSVA (International Society for the Study of Vascular Anomalies). Based on an extended literature research, this manuscript will give an overview of different internationally accepted treatment concepts. Read More

Shanghai Kou Qiang Yi Xue 2005 Dec;14(6):553-6

According to the classification of ISSVA, Waner and Suen, the traditionally called lymphangiomas are now referred to as lymphatic malformations, including both macrocystic and microcystic lesions. They are commonly seen vascular anomalies, and most frequently diagnosed at birth and most often occur in the head and neck area. The etiology of lymphatic malformations is still unknown, resulting in a variety of treatment modalities. Read More