| Presentation of first branchial cleft
anomalies: the Sheffield experience.J Laryngol Otol.
2007 May;121(5):455-9. Epub 2006 Nov 24.
Abnormalities of the
first branchial cleft are rare. They may present with a cutaneous
defect in the neck, parotid region, external auditory meatus or peri-auricular
area, or with inflammatory or infective lesions at these sites. A
retrospective case note review of the patients treated by the senior
author is presented. This group consisted of 18 patients and
represents the largest published UK series to date. Eleven patients
(65 per cent) had undergone incomplete surgery prior to referral. Over
half the patients had a clinically apparent lesion in relation to the
external auditory meatus. There was a variable relationship between
the tract and the facial nerve, which was identified at surgery in 15
cases. These findings are consistent with those of previously
published series. Clinicians should keep this diagnosis in mind when
assessing patients with infected lesions in the neck and parotid area.
Surgeons should be familiar with parotid surgery, in children where
appropriate, and be prepared to expose the facial nerve before
embarking on the surgical management of these lesions.
First branchial cleft anomaly, a case for misdiagnosis.Wien
Klin Wochenschr. 2004;116 Suppl 2:72-4.
First branchial cleft anomaly is a rare condition that is often
misdiagnosed and falsely mistreated before complete and definitive
surgical treatment. Its origin is uncertain and the presence of
ectodermal and sometimes also mesodermal elements has led some authors
to the conclusion that it represents buried nests of cells forming the
first branchial cleft and the underlying mesoderm. First branchial
cleft anomaly can be presented as a cystic lesion, fistula or sinus
extending towards the membranous external ear canal. The sinus tract
runs through the parotid gland in close association with the facial
nerve. There is no imaging method capable of identifying a first
branchial cleft anomaly with certainty. The danger of facial nerve
injury during surgery and the failure to identify the sinus tract
running to the external ear canal are the main reasons for incomplete
excision. The facial nerve must be identified and preserved and the
lesion completely excised. Facial nerve injury is more common in
attempts to remove recurrent branchial cleft lesions.
Unusual association of
congenital middle ear cholesteatoma and first branchial cleft anomaly:
management and embryological concepts.
Int J Pediatr Otorhinolaryngol. 2005 Feb;69(2):279-82.
OBJECTIVE: To report two cases
of an undescribed association of first branchial cleft fistula and
middle ear congenital cholesteatoma and to discuss management and
embryological hypothesis. DESIGN: Retrospective study and review of
the literature RESULTS: Both patients were young girls free of past
medical or surgical history. Surgical removal of the first cleft
anomaly found in the two cases a fistula routing underneath the facial
nerve. Both cholesteatomas were located in the hypotympanum,
mesotympanum. In one case, an anatomical link between the two
malformations was clearly identified with CT scan. DISCUSSION: The
main embryological theories and classification are reviewed. A
connection between Aimi's and Michaels' theories (congenital
cholesteatoma) and Work classification might explain the reported
clinical association.
First branchial cleft fistula
presenting with external opening on earlobe.
Eur Arch Otorhinolaryngol. 2006 Jul;263(7):685-7.
First branchial cleft fistula
is a rare congenital malformation of the head and neck with an
incidence of less than 10% of all branchial cleft defects. We herein
report a 15-year-old girl who had a cystic mass in the postauricular
region with an external opening on the posterior face of the earlobe.
Surgical exploration revealed that a second sinus tract was passing
through the conchal cartilage without going beyond the skin of the
external acoustic meatus. The mass and the tract were excised along
with the opening on the earlobe as well as the skin island surrounding
the opening. The case was treated surgically with success .The
significance of our case was the location of external opening on the
earlobe.
First branchial cleft sinus presenting with cholesteatoma and
external auditory canal atresia.Int
J Pediatr Otorhinolaryngol. 2003 Jul;67(7):811-4.
First branchial cleft abnormalities are rare. They may involve the
external auditory canal and middle ear. We describe a 6-year-old girl
with congenital external auditory canal atresia, microtia, and
cholesteatoma of mastoid and middle ear in addition to the first
branchial cleft abnormalities. Clinical features of the patient are
briefly described and the embryological relationship between first
branchial cleft anomaly and external auditory canal atresia is
discussed. The surgical management of these lesions may be performed,
both the complete excision of the sinus and reconstructive otologic
surgery.
Updating concepts of first branchial cleft defects: a literature
reviewInt J Pediate Otorhinolaryngol.2002 Feb
1;62(2):103-9.
OBJECTIVE: The Sinuses and fistulae of first
branchial cleft origin have been widely reported in the literature and
their variable relationship to the facial nerve has been described.
Most published series however are too small to allow a detailed
analysis of the relative frequency of various relationships of these
lesions to the facial nerve and therefore enabling the determination
of risks to the nerve at surgery. The aim of this study was to perform
a comprehensive review of literature in an attempt to identify those
patients with a deep tract (lying deep to the main trunk of the facial
nerve and/or its branches, and/or between the branches) and to
recognize the incidence of the complications of surgical management.
METHODS: Available English, French and German literature between 1923
and 2000 was reviewed and variables including patient's age, sex, side
and type of anomaly, opening of the lesion and the relationship of the
tract are analyzed in relation to the position of the facial nerve.
The complications due to their surgical excision are also reported.
RESULTS: Of the total number of cases with fistulae and sinuses
identified (n=158) fistulous tracts were more likely to lie deep to
the facial nerve compared with sinus tracts (P=0.01). Lesions with
openings in the external auditory meatus are associated with a tract
superficial to the facial nerve (P=0.05). Patients presenting at a
younger age were more likely to have a deep tract with consequent
increased risk of facial nerve damage. CONCLUSION: Identification of
the facial nerve trunk at an early stage of dissection is critical.
Extra care and caution should be exercised in younger patients (<6
months), those with fistulous tracts and in patients with a tract
opening elsewhere other than the external auditory canal.
Malformations of
the first branchial cleft: a case report. Acta otorhinolaryngol
Ital.2000 Jun;20(3):192-5.
First
branchial cleft anomalies are extremely rare and account for less than
8% of all branchial anomalies. They are generally found in infancy
although they arise during the embryonal period because of an
incomplete closure of the first branchial cleft. In 1972 Work
classified first branchial cleft anomalies into two types: Type I,
ectodermal cysts, is a duplication of the external auditory duct; Type
II, originate in both ectodermal and endodermal components and contain
cartilage. The present case report describes a Type II branchial
malformation. G.M., a 2-year-old male, presented painful tumefaction
in the left parotid area with cutaneous fistulization between the
sternocleidomastoid muscle and the mandibular angle. Cranial CT
permitted diagnosis and made it possible to stage surgery. The
difficulties encountered in recognizing and diagnosing first branchial
cleft anomalies are often responsible for application of the wrong
surgical approach and the resulting frequency in recurrences.
First branchial cleft anomalies: a
study of 39 cases and a review of the literature.
Arch Otolaryngol Head Neck Surg.1998 Mar;124(3):291-5.
OBJECTIVES:
To identify the clinical and anatomical presentations and to discuss
the guidelines for surgical management of anomalies of the first
branchial cleft. DESIGN: Retrospective study. SETTING: Three tertiary
care centers. PATIENTS: Thirty-nine patients with first branchial
cleft anomalies operated on between 1980 and 1996. INTERVENTION: All
patients were treated surgically. Complete removal of the lesion
required superficial parotidectomy with facial nerve dissection in 36
cases. The relationship of the facial nerve and anomalies is
discussed. RESULTS: Anatomically, 3 types of first branchial cleft
anomalies are identified: fistulas (n=11), sinuses (n=20), and cysts
(n=8). Clinically, 3 types of presentation are noted: chronic purulent
drainage from the ear (n=12), periauricular swelling in the parotid
area (n=18), and abscess or persistent fistula in the neck located
above a horizontal plane passing through the hyoid bone (n=21). A
membranous attachment between the floor of the external auditory canal
and the tympanic membrane was observed in 10% of cases. The facial
nerve was located lateral to the anomaly in 39% of cases. CONCLUSIONS:
Before definitive surgery, many patients (n=17) underwent incision and
drainage for infection owing to the difficulties in diagnosing this
anomaly. Wide exposure is necessary in most cases, and a standard
parotidectomy incision allows adequate exposure of the anomaly and
preservation of the facial nerve. Complete removal without
complications depends on a good understanding of regional
embryogenesis, a knowledge of the circumstances surrounding discovery,
an awareness of the different anatomical presentations, and a
readiness to identify and protect the facial nerve during resection.
Duplication of the
auditory canal. A vestige of the 1st branchial
cleft. Laryngo rhinootologie1989 Dec;68(12):694-7.
Anomalies of the first branchial cleft appear as
duplicated auditory canals; they are rare clinical entities. Patients
will present with a history of recurrent fistulas of the neck or
parotid gland close to the angle of the mandible. There are two
distinct malformations associated with the first cleft: A simple sinus
which is lined with squamous epithelium and runs parallel to the
auditory canal (Type I); the Type II anomaly, on the other hand, has a
close, though variable relationship to the facial nerve and contains
cartilage, a mesodermal derivate, within the wall. Embryologically it
appears that the point of time at which the disorder developed
dictates whether the malformation is of Type I or II. The authors
present six cases of first branchial cleft anomalies, two patients
with Type I and four with Type II lesions. The sex distribution was
striking: all the patients were female. In case of a Type II defect,
three variations of the facial nerve with split main trunks were
found. Recurrent operations and infections lead to scar tissue and
subsequent surgery is more difficult, with serious hazard to the
facial nerve. Where positive identification of the facial nerve is a
major problem it is advisable to identify the nerve in the mastoid
cavity and trace it to the stylomastoid foramen and parotid gland.
First branchial cleft cysts:
clinical update. Laryngoscope.1987 Feb;97(2):136-40.
First branchial cleft cysts develop as a result of
incomplete fusion of the cleft between the first and second branchial
arches and give rise to two distinct anomalies, termed type I and type
II anomalies. Type I anomalies are purely ectodermal while type II
anomalies exhibit ectodermal and mesodermal elements. The type II
anomaly incorporates some portion of the first and second arch as well
as the cleft. Type I lesions are extremely rare. They appear
histologically as cysts lined by squamous epithelium. Clinically, they
present as a cystic mass or fistula posterior to the pinna and concha.
The cyst is usually superior to the main trunk of the facial nerve and
ends in a cul-de-sac on or near a bony plate at the level of the
mesotympanum. Type II lesions are more numerous and represent a
duplication of both membranous and cartilaginous portions of the
external auditory canal. They contain skin as well as adnexal
structures and cartilage and may be associated with the parotid gland.
Most frequently they are associated with fistulae in the concha or
external auditory canal as well as fistulous openings in the neck.
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