Possible Estrogen Dependency in the Pathogenesis of Branchial Cleft Cysts.

BACKGROUND: Even though branchial cleft cysts are currently accepted as a congenital anomaly, there is often a long delay until clinical presentation; branchial cleft cysts classically appear in the second to fourth decade of life. Our observation of their occurrence in three pregnant women encouraged us to contemplate a possible hormonal influence. METHODS: Immunohistological analysis was performed for the evaluation of the estrogen receptor alpha (ERα) in paraffin-embedded tissue specimens of 16 patients with a diagnosis of branchial cleft cyst, with three of them being pregnant. RESULTS: Expression of ERα was detected within epithelial cells only in branchial cleft cysts in pregnant females; moreover, higher growth fractions (Ki-67/Mib1) were found. CONCLUSION: The fact that the estrogen receptor was expressed only in pregnant women, in contrast to 13 investigated cases, may suggest that the high level of estrogen in pregnancy is a possible explanation for the spontaneous growth of branchial cleft cysts.

Branchiootic syndrome--a clinical case report and review of the literature.

Branchiootoic syndrome is part of the spectrum of brachiootorenal disorders. Brachiootorenal disorder is a rare autosomal dominant condition, characterized by malformations of the outer, middle, and inner ear, which are associated with branchial and renal anomalies. We describe a case of bilateral branchiootoic syndrome and discuss the anatomy of second branchial cleft fistulae and the surgical management of this uncommon condition. We report the case of a 6-year-old girl referred to our department with bilateral intermittently discharging neck swellings. Clinical examination revealed bilateral branchial fistulae and preauricular sinuses, on a background of a positive family history of branchial fistulae. A magnetic resonance imaging scan confirmed the diagnosis of bilateral second branchial cleft fistulae. In view of her symptoms, she underwent bilateral branchial fistula excision and tonsillectomy with an uneventful postoperative recovery.

Branchial cleft or cervical lymphoepithelial cysts: etiology and management.

BACKGROUND: The cervical lymphoepithelial or branchial cleft cyst is a developmental cyst that has a disputed pathogenesis. The objective of this article is to provide a brief review of the literature and to define diagnostic terms related to this anomaly, as well as to describe its etiology, clinical presentation and treatment. CASE DESCRIPTION: The cervical lymphoepithelial or branchial cleft cyst usually presents as a unilateral, soft-tissue fluctuant swelling that typically appears in the lateral aspect of the neck, anterior to the sternocleidomastoid muscle, and becomes clinically evident late in childhood or in early adulthood. Clinicians can diagnose the cyst with appropriate imaging to assess the extent of the lesion before definitive surgical treatment. The authors describe a patient who underwent excision of a well-encapsulated cystic structure that was diagnosed as a branchial cleft cyst. CLINICAL IMPLICATIONS: The cervical lymphoepithelial or branchial cleft cyst can be easily misdiagnosed as a parotid swelling or odontogenic infection. It is imperative that clinicians make an accurate diagnosis so that appropriate treatment (that is, surgical excision) can be performed. If the cysts are treated properly, recurrences are rare.

Lateral cervical cleft: a previously unreported anomaly resulting from incomplete disappearance of the second pharyngeal (branchial) cleft.

The authors present a 2-year-old boy with a skin defect located in the right lateral side of the neck. They suggest the defect is a partial failure of disappearance of the second pharyngeal (branchial) cleft and propose a name of lateral cervical cleft.

Lymphoepithelial cyst of the parotid gland--a case report.

The lymphoepithelial cyst has been commonly termed branchial cleft cyst or branchial cyst. Although many theories, including the branchial apparatus theory, thymic duct theory, and inclusion theory, have been put forward, the etiology is still controversial. Parotid lymphoepithelial cysts are rare and could be divided into two groups, AIDS-related and non-AIDS related, by the relationship with HIV infection. A non-AIDS related lymphoepithelial cyst of left parotid gland in an 81-year-old man presenting as a parotid tumor is described. A left partial parotidectomy was carried out to remove the lesion. There was no recurrence after 2 years. Lymphoepithelial cysts of the parotid gland are often misdiagnosed as other benign tumors in spite of detailed preoperative investigations. Thus a pathological proof is needed for precise diagnosis. In addition, we also need to take the possibility of HIV infection into consideration, although Taiwan is not an AIDS epidemic area.

Lateral neck cysts--the branchial theory revisited. A critical review and clinicopathological study of 97 cases with special emphasis on cytokeratin expression.

Theories regarding the origin of lateral neck cysts (LNC) range from congenital branchial pouch abnormalities to acquired salivary gland inclusions within lymph nodes. We analyzed 97 LNC histologically and evaluated their cytokeratin (CK) profile in a search for their derivation. 77/97 LNC were located in soft tissues, 20/97 within lymph nodes. LNC of young patients and of recent symptomatic presentation in older patients were lined by respiratory epithelium with scant lymphoid tissue, with expression of "simple epithelial" CK in ciliated cells and bimodal expression of "simple" and "stratified-epithelial-type" CK in basal cells. In longer standing symptomatic LNC, respiratory epithelium alternated with transitional-type pseudostratified epithelium with intraepithelial Langerhans cells and lymphoid hyperplasia, or consisted exclusively of squamous epithelium. We propose that respiratory epithelium is the "native" epithelium of LNC and squamous metaplasia results from inflammation induced stem cell hyperplasia in respiratory epithelium, evidenced by co-expression of "simple" and "stratified-epithelial-type" CK in all cells of transitional-type pseudostratified epithelium, the early stage in metaplastic transformation. Respiratory epithelium predominates in early LNC, lines pharyngeal tonsils and the recessus tonsillo-tubalis, and is a minor constituent of palatine tonsil but is not present in salivary glands. None of the LNC contained dysplasia, atypia, or carcinoma, or were associated with a primary carcinoma of tonsils or head and neck. We demonstrate that LNC arise from developmental remnants (congenital) of the 2nd branchial pouch, which may lie dormant for many years. Symptomatic enlargement, squamous metaplasia and lymphoid hyperplasia ensue as a consequence of immunologic stimulation a development reflected in hyperplastic palatine tonsils.

First branchial complex anomalies: report of 3 cases.

Three cases of first branchial cleft anomalies are presented. The embryology and pathology of first branchial complex anomalies, their imaging characteristics and differential diagnosis are discussed.

Lesiones quísticas del cuello: correlación clínica y de imagen / Cystic lesions of the neck image and clinical correlation

Se hace una revisión retro y prospectiva en 14 pacientes (8 mujeres, 6 hombres) con lesiones quísticas congénitas a nivel del cuello. Los autores analizan las lesiones más frecuentes encontradas a nivel del cuello supra e infrahioideo y que incluyen quiste del conducto tirogloso, quiste del segundo arco branquial, linfangioma o higroma quístico, sus manifestaciones clínicas correlacionadas con las características observadas a través de los métodos de imagen

Metastatic papillary thyroid carcinoma presenting as a typical branchial cyst.

Two cases of papillary thryoid carcinoma presenting as a cystic lateral neck mass are reported. This tumour characteristically presents in patients under 40-years-old and in the presence of an occult primary tumour may mimic a branchial cyst. In such cases simple aspiration of the cyst will produce a chocolate-brown serous fluid which excludes the diagnosis of a branchial cyst and is characteristic of papillary thyroid carcinoma. Cytological examination of the fluid has a high degree of sensitivity and specificity in the diagnosis of thyroid malignancy and should avoid delay in diagnosis and unnecessary surgical exploration prior to definitive treatment.

Branchiogenic carcinoma--a case for radiation induced carcinogenesis.

From the time the first case of bronchial cleft carcinoma was reported over a century ago, very few cases have been reported which prove to be a cancer of primary branchial origin, while many metastatic cystic squamous cell carcinomas in the neck have been wrongly categorized as branchial carcinoma. Here, we describe a case of primary branchiogenic carcinoma in a 53 year old Libyan female showing a transformation of a benign epithelium into a malignant type. We further report that this transformation is perhaps brought about by an irregular radiotherapy to the head and neck region.

A possible inflammatory reaction in a lateral neck cyst (branchial cyst) because of odontogenic infection.

We present the case of a woman who suffered from an acutely infected diffuse mass in the right neck. This mass had grown rapidly after difficult extraction of a tooth. Histologic analysis of the excised material revealed a lateral neck cyst with a lymph node that showed signs of an acute inflammation near the cyst. These findings support the theory that a preexisting lateral neck cyst may be "activated" by an intraoral inflammation. The different theories of the origin of lateral neck cysts are presented and discussed in the context of the case description.

The aetiology of lateral cervical (branchial) cysts: past and present theories.

Four theories have been suggested to explain the aetiology of lateral cervical cysts. Ascherson (1832) suggested that the cysts arose due to incomplete obliteration of branchial cleft mucosa, which remained dormant until stimulated to grow later in life. His (1886) suggested these cysts were vestiges of the precervical sinus. Wenglowski (1912) believed lateral cervical cysts developed from the third pharyngeal pouch (thymopharyngeal duct). A number of investigators during the 19th century noted the close relationship between lateral cervical cysts and lymphoid tissue (Lucke, 1861). Luschka (1848) suggested that cystic degeneration of cervical lymph nodes was the mechanism by which lateral cervical cysts were formed. This theory received little support until King (1949) studied the histology of a large number of lateral cervical cysts and concluded that these cysts resulted from cystic transformation of cervical lymph nodes. The evidence for and against these theories of aetiology is discussed. The debate is centred on a study of 20 patients with lateral cervical cysts operated on in the Department of Otolaryngology, Bedford Hospital, between January 1986 and December 1991. In all twenty cases the wall of the cyst was found to be composed of lymphoid tissue, histologically identical to the present in lymph nodes. The mean age of presentation was 31 years, and in no case was a tract or cord found which connected the cyst to the skin or pharynx. The evidence strongly suggests that lateral cervical cysts develop from the cystic transformation of cervical lymph nodes. Mechanisms by which this may occur are discussed.

Lympho-epithelial cysts: a maxillofacial surgeon's perspective.

Cervical swellings are referred to Oral and Maxillofacial Surgeons and may subsequently be diagnosed as lympho-epithelial cysts. The clinical presentation of such swellings appears to differ from that seen in other specialties also treating these cysts in that a larger proportion of lympho-epithelial cysts presenting to our specialty are infected, which complicates the diagnosis and also the subsequent management. We present our recent experience of 5 patients who underwent excision of a lympho-epithelial cyst, all of which were infected on initial presentation and four of which were associated with concurrent ipsilateral dento-alveolar infection. The fifth case presented with rapid development of a cervical swelling in the immediate post-natal period. As these cysts contain abundant lymphoid tissue as an integral part of the lymph drainage system of the head and neck, we propose that the management of an infected lympho-epithelial cyst should include a full clinical and radiographic examination of the oral cavity for a possible focus of infection and indeed any patient awaiting surgery for a non-infected cyst would benefit from an examination of the mouth and treatment of any sites of chronic infection.

Branchial cysts: congenital or acquired?

Branchial cysts are one of the commonest causes of neck lumps in the young adult. Their aetiology is the subject of some controversy, although available evidence suggests that they are derived from lymphoid tissue. We present two cases of branchial cysts, and emphasise the importance of excluding more sinister causes of neck lumps before excision, even when the clinical features suggest benign pathology.

[Castleman's disease. Report of 2 cases with otorhinolaryngologic involvement]. / La maladie de Castleman. A propos de deux observations ORL.

Two cases of head and neck Castleman disease are reported. Diagnosis is always confirmed by histopathologic examination of the specimen. Head and neck and general localisations are described and discussed. Surgery remains the only treatment of this benign tumor with a high success rate.

Aetiology of branchial cysts.

The aetiology of branchial cysts is unknown. It is possible that they arise by more than one mechanism. The major theories are that they originate either from the branchial apparatus or from lymphoid tissues. A retrospective review was conducted of 61 cases occurring over a recent 14-year period. Eighty-five percent were diagnosed after the age of 10 years, 80% occurred in the 'classical' position, 80% had a squamous epithelial lining and 87% had lymphoid tissue in the wall. The clinical and histological features strongly support the lymphoid aetiology theory for the majority of branchial cysts.

Immunohistochemical and histologic study of cervical lymphoepithelial cysts.

Four cases of lateral cervical cysts were reported. All cyst walls were lined with keratinized stratified squamous epithelium, and contained lymph follicles with well-defined germinal centers. Ectopic salivary gland tissue was found in one case. Histochemical examination by the immunoperoxidase method revealed the existence of carcinoembryonic antigen (CEA). However, alpha-feto protein (AFP) and amylase were not detected in any of the cysts. The cyst fluid contained high levels of CEA and high titers of salivary type amylase, but AFP was negative. These results do not support any one of the theories concerning the etiology of cervical lymphoepithelial cysts. It seems likely that cysts derive from both epithelial remnants of the branchial apparatus and from salivary gland inclusions in lymph nodes.