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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

ORL J Otorhinolaryngol Relat Spec 2005 1;67(6):331-4. Epub 2005 Dec 1.

Department of Pediatric Surgery, Gazi University Medical Faculty, Emek, Turkey.

Aim: The aim of this study was to evaluate the results of surgical treatment of cervicofacial cystic hygromas in children.

Patients And Methods: Medical records of 17 patients who were operated for cervicofacial cystic hygroma between 1985 and 2004 were evaluated in terms of age, gender, symptoms, diagnostic workups, outcomes and complications.

Results: There was a slight male predominance -- 10 (59%) boys and 7 (41%) girls. Read More

Arch Otolaryngol Head Neck Surg 2005 Nov;131(11):990-5

Department of Otolaryngology-Head and Neck Surgery, Northwestern University Feinberg School of Medicine, Chicago, Ill, USA.

Objective: To determine the role of angiogenesis in the clinical behavior and pathogenesis of lymphangioma tumors.

Design: A retrospective study. Median follow-up period was 44. Read More

J Otolaryngol 2004 Oct;33(5):283-8

Department of Pediatric Otolaryngology, 3333 Burnet Avenue, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229-3039, USA.

Objective: To review our experience with the planned intrapartum management of fetuses with suspected severe airway obstruction.

Methods: Retrospective review of prenatal presentations, intrapartum airway procurement methods, outcomes, and complications.

Results: All cases (N = 11) at our tertiary airway referral institution between 1995 and 2002 were reviewed. Read More

Int J Pediatr Otorhinolaryngol 2005 Nov;69(11):1509-13

University of California Irvine, Aesthetic and Plastic Surgery Institute, Irvine, CA, USA.

Introduction: Tissue expanders have long been used for reconstructing large cutaneous and fascio-cutaneous defects in children. Previous studies have examined tissue expansion for all body regions, touching upon the head and neck regions. We present a focused review of our experience with cervicofacial tissue expansion in the pediatric population. Read More

J Pediatr Orthop B 2004 Jul;13(4):284-6

Department of Orthopedics and Traumatology, Medical Faculty, Osmangazi University, Meşelik, Eskişehir, Turkey.

Lymphangiomas arising outside of the cervicofacial, thoracic, and abdominal areas are extremely rare. We describe a child with a giant cystic lymphangioma cavernousum occurring in the lower leg with overlying lymphangioma circumscriptum, along with its magnetic resonance imaging findings. Read More

Rev Laryngol Otol Rhinol (Bord) 2002 ;123(1):27-32

Hôpital 20 Aout, Service d'ORL et de chirurgie cervico-faciale, Casablanca, Maroc.

Introduction: Cystic cervico-facial lymphangioma (DDFL) are relatively rare. Because of their size and site, they pose a real problem in treatment. The authors present a retrospective study about 10 cases. Read More

Laryngoscope 2001 Nov;111(11 Pt 1):1929-37

Department of Otolaryngology-Head and Neck Surgery, Geisinger Medical Center, Danville, Pennsylvania 17822-1333, USA.

Objective: To outline a rational approach to the management of cystic hygroma based on the authors' experience, the natural history of the disease, and the results of surgical treatment.

Study Design And Methods: A retrospective review of all patients with the diagnosis of lymphangioma or cystic hygroma from 1958 to 2000 was performed.

Results: Seventy-four patients were identified with 46 cases confined to the cervicofacial region. Read More

Arch Otolaryngol Head Neck Surg 2001 Aug;127(8):997-1002

Department of Pediatric Otolaryngology, The Children's Hospital of Buffalo, 219 Bryant St, Buffalo, NY 14222, USA.

Background: Heterotopic neuroglial (brain) tissue is a rare cause of airway obstruction in newborns. Fewer than 30 cases have been reported in the English literature. Brain heterotopias can mimic more common congenital anomalies of the head and neck. Read More

Head Neck 2001 Apr;23(4):326-37

Department of Otolaryngology, Head and Neck Service, Université Catholique de Louvain (UCL), Saint-Luc University Hospital, Hippocrate Avenue 10, B-1200, Brussels, Belgium.

Background: Lymphatic malformations (LM) are rare benign congenital tumors appearing mainly in the head and neck with a considerably variable outcome. A need exists to validate a staging system, taking into account the prognosis of the malformation, including preoperative and postoperative complications, long-term sequelae, and persistence of the disease to improve parental counseling and evaluate the outcome of a surgical treatment of such tumors.

Methods: Twenty-two patients treated for LM were selected from a series of 129 patients operated on for congenital malformations of the head and neck between 1986 and 1997 at St-Luc University Hospital, Brussels, Belgium. Read More

Arch Otolaryngol Head Neck Surg 1999 Jun;125(6):643-8

Department of Otorhinolaryngology, University of Ulm, Medical School, Germany.

Objectives: To compare different surgical interventions for the treatment of extensive cervicofacial lymphangiomas and to define the minimal extent of surgery necessary to control disease.

Design: Retrospective study. Mean +/- SD follow-up was 31+/-4 months after surgery. Read More

J Pediatr Surg 1996 Dec;31(12):1648-50

Department of Surgery, University of Washington School of Medicine, Seattle 98195-6410, USA.

Giant cervicofacial lymphatic malformation, a potentially life-threatening congenital malformation, historically has been removed in staged resections. However, complete surgical extirpation is desirable and can be achieved with the aid of a multidisciplinary team. The authors present the case of a 12-year-old girl who had an extensively enlarged cervicofacial lymphatic malformation. Read More

J Pediatr Surg 1996 Oct;31(10):1463-4

Skeletal Radiol 1996 Feb;25(2):201-4

Department of Orthopedics, Madigan Army Medical Center, Fort Lewis, WA 98431, USA.

Cystic hygromas arising outside of the cervicofacial, thoracic, and abdominal areas are extremely rare. Detailed descriptions of this lesions along with its MRI findings, in the extremities are lacking in the literature. The following case report describes such a lesion occurring in the arm. Read More

J Pediatr Surg 1995 Aug;30(8):1159-60

Dr Charles A. Janeway Child Health Centre, St John's, Newfoundland.

The authors present two cases of lymphangioma of the cervicofacial region, treated with a new investigational drug in North America, OK-432 (picibanil), a sclerosing agent intralesionally injected. Both patients had been treated surgically and had recurrence of the tumor. Intralesional injection of OK-432 without aspiration was employed for the first patient, and after aspiration in the second patient. Read More

Plast Reconstr Surg 1995 May;95(6):951-60

Division of Plastic Surgery, Children's Hospital, Boston, Mass., USA.

This is a retrospective review of the clinical course and long-term soft-tissue/skeletal problems in 17 patients with large cervicofacial lymphatic malformations. Morbidity included infection (71 percent), airway compromise requiring tracheostomy (65 percent), poor dental health with aggressive caries (53 percent), abnormal articulatory patterns (47 percent), and episodic bleeding (35 percent). All patients underwent soft-tissue excision (mean four procedures per patient). Read More

Am J Pediatr Hematol Oncol 1994 Nov;16(4):325-8

Department of Pediatrics, College of Medicine, University of Florida, Gainesville 32610-0296.

Purpose: Surgical resection of cervicofacial cystic hygromas and lymphangiomas rarely effects complete reduction because of severe anatomic restrictions.

Patients And Methods: With prior knowledge of cyclophosphamide activity against lesions of this type, a formal trial of cyclophosphamide was initiated.

Results: Overall dose escalation therapy resulted in 50% reduction in mass without recurrence after cessation of therapy and with minimal and readily reversible toxicity. Read More

Ann Pediatr (Paris) 1992 Oct;39(8):491-4

Service d'Otorhinolaryngologie, Hôpital d'Enfants Armand Trousseau, Paris.

Twenty-four cases of cervicofacial lymphangioma treated between 1984 and 1991 are reported. The therapeutic approach is discussed. Because the therapeutic problem depends on whether or not the airways are involved, an endoscopic evaluation and a CT scan study should be included in the workup. Read More

J Magn Reson Imaging 1991 Sep-Oct;1(5):579-84

Department of Neuroradiology and Therapeutic Angiography, Hôpital Lariboisière, Paris, France.

Between 1980 and 1990, 150 patients with cervicofacial vascular malformations were studied at the authors' institution with computed tomography, plain radiography, and angiography. Since 1989, 34 of these patients have also undergone magnetic resonance (MR) imaging. Capillary-venous hemangiomas seem to be the best indication for the adjunctive use of MR imaging. Read More

Arch Otolaryngol Head Neck Surg 1991 May;117(5):546-53

Department of Otolaryngology-Head Neck Surgery, University of Washington, Seattle 98195.

Cystic hygromas usually present in infancy or early childhood as compressible masses that may rapidly and intermittently enlarge. While they may arise in any anatomic location, hygromas of the head and neck are especially difficult to manage since enlargement may cause serious sequela such as airway obstruction, feeding difficulties, and speech pathology. Complete extirpation of these lesions is often impossible, and recurrence rates are accordingly high. Read More

J Chir (Paris) 1991 Mar;128(3):145-52

Service O.R.L. et Chirurgie Maxillo-Cervico-Faciale, C.H.U. Ibnou Rochd, Casablanca, Maroc.

The authors report 7 cases of cervico-facial cystic hygromas. Cystic hygromas are rare affections that can pose redoubtable therapeutic problems because of their size and localisation. Their diagnosis is clinically easy but must be affirmed by an histologic investigation. Read More

Ann Otolaryngol Chir Cervicofac 1991 ;108(4):227-30

Service ORL et de Chirurgie Cervico-Faciale, Hôpital Trousseau, Paris.

The authors report a series of 22 cervicofacial lymphagiomas treated from 1984 to 1990, and try to define a therapeutic strategy. In 11 cases, facial, laryngeal or pharyngeal extension was observed, and 1 cas showed mediastinal extension. These cases are not easily treated, and it is impossible to perform complete surgery, thus leading to recurrence. Read More

Rev Stomatol Chir Maxillofac 1990 ;91(5):385-9

Service ORL, Hôpital Charles-Nicolle, Tunis, Tunisie.

A review of fifteen new cases of cystic cervicofacial lymphangiomas is presented. While discussing the different localizations of these malformations, the authors emphasize the relationships existing between this type of malformation and both the facial nerve and the vasculonervous axis of the neck. They recommend surgical management. Read More

Acta Stomatol Belg 1987 Oct;84(3):257-64

Cesk Otolaryngol 1986 Apr;35(3):179-83

Ann Otolaryngol Chir Cervicofac 1986 ;103(2):113-7

Of 41 children, 22 girls and 19 boys, with cervicofacial lymphangioma, 31 had congenital lymphangiomas, 30 lymphangiomas with cervical expression but only 9 were restricted to the neck region. Operation in 35 children showed that in 12 cases the lymphangioma was limited in extent and failed to extend to the neck or parotidojugular region, and excision was complete and there were no recurrences. Partial excision was performed in 23 children in whom the lymphangioma extended to various degrees into the tongue, pharynx or larynx, and tracheotomy was necessary in 10 of these cases. Read More

Rev Stomatol Chir Maxillofac 1986 ;87(1):6-13

After an embryological review, the author describes the macroscopic and surgical aspects of cervicofacial lymphangiomas and hemolymphangiomas. Therapeutic difficulties are variable and are discussed with regard to different clinical forms. There are all degrees between benign forms which are easy to treat and major forms which raise difficult therapeutic problems because of their extent. Read More

Ann Otolaryngol Chir Cervicofac 1985 ;102(7):525-30

Among the various congenital cervicofacial swellings observed in children, three types are rarely encountered in general practice: auriculobranchial cysts and fistulae, thymic cysts and cystic lymphangiomas. To resolve diagnostic and therapeutic problems related to these rare lesions it is essential that their etiopathogenic bases be understood. Auriculobranchial cysts and fistulae present clinical symptoms (cervical, parotid, auricular) that are only poorly evidenced and are related mainly to infection. Read More