Third branchial cleft anomaly presenting as a retropharyngeal abscess

doi:10.1016/S0165-5876(00)00355-4

International Journal of Pediatric Otorhinolaryngology

Volume 54, Issues 2–3, 31 August 2000, Pages 167-172

https://doi.org/10.1016/S0165-5876(00)00355-4 Get rights and content

Abstract

Branchial cleft anomalies are congenital developmental defects that typically present as a soft fluctuant mass or fistulous tract along the anterior border of the sternocleidomastoid muscle. However, branchial anomalies can manifest atypically, presenting diagnostic and therapeutic challenges. Error or delay in diagnosis can lead to complications, recurrences, and even life-threatening emergencies. We describe a case of an infected branchial cleft cyst that progressed to a retropharyngeal abscess in a 5-week-old female patient. The clinical, radiographic, and histologic findings of this rare presentation of branchial cleft cyst are discussed.

Introduction

Branchial cleft anomalies are congenital developmental defects which arise from the primitive branchial apparatus. There are three general types, sinus, fistula, and cyst. These lesions most often are present as a soft fluctuant mass or draining tract located along the anterior border of the sternocleidomastoid muscle [1], [2], [3]. Since most patients present with signs and symptoms of a cystic neck mass or draining fistula, the diagnosis is usually straightforward. Complete surgical excision of the cyst and cyst tract is the treatment of choice for branchial remnants.

Branchial cleft anomalies may also have atypical presentations. This can result in a misdiagnosis of thyroglossal duct cyst, dermal inclusion cyst, lymphatic malformation, or malignant neoplasm [2]. In addition, a branchial cleft anomaly can become infected and develop into an abscess. Patients present with tender erythematous infected neck masses and are diagnosed with simple neck abscesses. Such errors in diagnosis can lead to complications and recurrences.

Abscesses due to third branchial cleft anomalies are uncommon. The progression of the infection into the retropharyngeal space is even more unusual. We describe a case of a third branchial remnant presenting as a retropharyngeal abscess in a 5-week-old female patient. Few such cases have been described in the literature [4], [5]. The clinical, radiographic, and histologic findings of this rare presentation of branchial cleft cyst will be discussed. This case illustrates the potential difficulties in the diagnosis of branchial cleft anomalies. Awareness of atypical presentations enables surgeons to provide the optimal treatment and timely surgical excision.

Section snippets

Case report

A 5-week-old female presented to a community hospital with an enlarging left neck mass, fever, and dysphagia of 1-week duration. She had been given oral cefixime and intramuscular ceftriaxone for several days without clinical improvement. On admission, she had a temperature of 37.9°C. A left sided 4×4 cm erythematous neck mass was palpable at the mid-anterior cervical region. Laboratory examination revealed her white blood cell (WBC) count to be 21,400 cells/mm⁻³. A lateral neck radiograph

Discussion

Branchial remnants are common congenital cervical anomalies. They account for 17% of all the pediatric cervical masses [2] and remain the most common congenital cervical anomalies in adults [3]. There are three general types of branchial cleft anomalies, cysts, sinuses, and fistulae. Branchial cleft cysts are epithelium lined cavities with no external or visceral opening. [6] They usually present in adulthood [2], [3], [5], [7]. Branchial cleft sinuses are tracts with or without a cyst

References (18)

S.A. Deane et al.
Surgery for thyroglossal duct and branchial cleft anomalies
Am. J. Surg.
(1978)
O. Doi et al.
Branchial remnants: a review of 58 cases
J. Pediatr. Surg.
(1988)
T.N. Morrish et al.
Branchial anomaly in a newborn presenting as stridor
Int. J. Pediatr. Otorhinolaryngol.
(1991)
R.E. Sonnino et al.
Unusual patterns of congenital neck masses in children
J. Pediatr. Surg.
(1989)
J. Golledge et al.
The etiology of lateral cervical cysts: past and present theories
J. Laryngol. Otol.
(1994)
K. Hewel et al.
Infected third branchial cleft cyst: retropharyngeal extension to the superior mediastinum
Can. Assoc. Radiol. J.
(1996)
J.R. Chandler et al.
Branchial cleft cysts, sinuses, and fistulas
Otol Clinics North Am.
(1981)
D.J. Halvorson et al.
Branchial cleft cyst posterior to the carotid vessels
ENT J.
(1995)
C.J. Ingoldby
Unusual presentations of branchial cysts: a trap for the unwary
Ann. R. Coll. Surg. Engl.
(1985)

There are more references available in the full text version of this article.

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A Branchial Cyst in a Diamond Python (Morelia spilota)
2019, Journal of Comparative Pathology
A 9-year-old female diamond python (Morelia spilota) was presented with a submandibular swelling. The cytological, macroscopic and histological features of this lesion indicated a diagnosis of branchial (pharyngeal) cyst. Branchial cysts are benign lesions caused by anomalous development of the branchial apparatus and are described rarely in veterinary medicine. We suggest that possible persistence of branchial remnants should be included in the consideration of differential diagnoses for neck masses in adult snakes.
Imaging in Otolaryngology
2018, Imaging in Otolaryngology
Diagnostic Imaging: Head and Neck
2017, Diagnostic Imaging: Head and Neck
Fourth branchial cleft anomaly: Management strategy in acute presentation
2014, International Journal of Pediatric Otorhinolaryngology
Citation Excerpt :
A cyst or a sinus can be observed, while tracts connecting the skin to the pharyngeal mucosa are less frequent and several authors assume that the fourth branchial anomalies should not have a skin orifice, due to the exclusive endobranchial origin [4] and when present, the skin orifice is usually secondary to recurrent infection and/or inadequate surgery [3]. On the contrary some authors believe that these remnants may show complete or incomplete communication between skin and mucosa [8]. Differentiation from fourth to third branchial cleft origin can be difficult since these malformations share the most part of their development area, have a similar clinical presentation and the main differential criterium is the lower location of the internal opening into the pharynx [6,9] of fourth branchial cleft remnants.
Branchial malformations are common congenital head and neck lesions usually diagnosed in childhood during the first decade of life. Acute presentation is usually managed with conservative protocols before a definitive surgical procedure although the risk of life-treating septic complications may influence the physician’s decision. Surgery is the treatment of choice with the removal of the lesion alone, nevertheless more aggressive approaches must be considered in complicated cases. Selective neck dissection including the removal of part of the thyroid lobe with the congenital lesion should be considered as the “ultima ratio” treatment to avoid recurrence.
We reviewed literature and report our experience concerning two patients with fourth branchial cleft sinus.
A three-year-old child with a clinical history of recurrent neck abscess was referred to our department after several drainages performed in another centre. A three-year-old child referred to our department for a left side lower primary neck abscess. In both cases the diagnosis of a complicated fourth cleft remnant was confirmed by rigid endoscopic visualization of the mucosal orifice of the sinus in the pyriform fossa. Surgical management during acute presentation was challenging; in one patient the early fasciitis required an emergency procedure to remove the infected sinus that were strictly adherent to the deep vascular-nervous axis.
Surgery was the definitive treatment in both cases and at 12 and 25 months follow-up respectively no recurrences were observed.
Bilateral pharyngeal apparatus (branchial) sinuses in a set of identical twins - A case report
2012, Journal of Pediatric Surgery
Citation Excerpt :
Few cases of bilateral pharyngeal (branchial) cleft anomalies have been reported in literature [7-9]. Huang et al [10] reported 3 bilateral cases in their series of 37 patients. However, only 6 cases of bilateral branchial cleft fistulas have been reported in the English-language literature [11-14].
Defective closure of the pharyngeal apparatus during the second week of gestation results in pharyngeal arch anomalies. Although pharyngeal apparatus (branchial) anomalies are frequently seen, bilateral cases are rare (only 2% to 3%). Bilaterality has a familial predisposition. Our patients, a set of identical twins, presented with bilateral discharging cervical sinuses noticed at 3 months of age. Physical examination was suggestive of bilateral pharyngeal apparatus (branchial) sinuses. Both patients were managed with surgical excision in the same operative sitting. To the best of our knowledge, ours is the first case of bilateral pharyngeal apparatus (branchial) sinuses in a set of identical twins.
Management of congenital third branchial arch anomalies: A systematic review
2010, Otolaryngology - Head and Neck Surgery
Citation Excerpt :
Nevertheless, these analyses produced results generally congruent with the crude odds ratios, as detailed in the footnote to Table 2. The 82 selected publications,2,3,6-85 presenting 202 cases of third branchial arch anomalies, are summarized in Table 1. Most third branchial arch anomalies were diagnosed in childhood (Table 3).
To systematically review the existing literature on third branchial arch anomalies and suggest guidelines for their management.
We searched PubMed, Medline, and Embase using Scopus, and collected additional publications cited in bibliographies. We included all English-language articles and all foreign-language articles with an English abstract.
Two investigators reviewed all cases explicitly identified as third arch anomalies or meeting anatomical criteria for third arch anomalies; they assessed presentation, diagnostic methods, intervention, and outcome.
We found 202 cases of third arch anomalies; they presented primarily on the left side (89%), usually as neck abscess (39%) or acute suppurative thyroiditis (33%). Barium swallow, direct laryngoscopy, and magnetic resonance imaging were the most useful diagnostic tools. The recurrence rate varied among the treatment options: incision and drainage, 94 percent; endoscopic cauterization of the sinus tract opening, 18 percent; open-neck surgery and tract excision, 15 percent; and partial thyroidectomy during open-neck surgery, 14 percent. Complications after surgery appeared somewhat more frequently in children eight years of age or younger.
Third arch anomalies are more common than previously reported. They appear to be best treated by complete excision of the cyst, sinus, or fistula during a quiescent period. Repeated incision and drainage yields high rates of recurrence and should be avoided. Complications might be minimized by first initiating antibiotic treatment, delaying surgical treatment until the inflammatory process is maximally resolved, and by using endoscopic cauterization.

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^☆: Presented at the 81st Annual Meeting of the Pacific Coast Oto-Ophthalmological Society, June 22, 1997, Coronado, CA.

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Case reportThird branchial cleft anomaly presenting as a retropharyngeal abscess☆

Abstract

Introduction

Section snippets

Case report

Discussion

Am. J. Surg.

J. Pediatr. Surg.

Int. J. Pediatr. Otorhinolaryngol.

J. Pediatr. Surg.

The etiology of lateral cervical cysts: past and present theories

J. Laryngol. Otol.

Infected third branchial cleft cyst: retropharyngeal extension to the superior mediastinum

Can. Assoc. Radiol. J.

Branchial cleft cysts, sinuses, and fistulas

Otol Clinics North Am.

Branchial cleft cyst posterior to the carotid vessels

ENT J.

Unusual presentations of branchial cysts: a trap for the unwary

Ann. R. Coll. Surg. Engl.

Case report
Third branchial cleft anomaly presenting as a retropharyngeal abscess☆