Volume 3, Issue 3 , Pages 117-119, September 2008
Bilateral cervical chondrocutaneous branchial remnants
Article Outline
Summary
Cartilaginous branchial development has been studied extensively for many years. Since the first description of these studies done in 1828 [W.B. Matthews, Congenital cartilaginous rests in the neck, Arch. Surg. 28 (1934) 59; G. Atlan, E.P. Egerszegi, P. Brochu, L. Caouette-Laberge, P. Bortoluzzi, Cervical chrondrocutaneous branchial remnants, Plastic Reconstr. Surg. 100 (July (1)) (1997) 32–39; H. Braun, T. Hofmann, H. Wolfgruber, W. Anderhuber, A. Beham, H. Stammberger, Case report of bilateral cervical chondrocutaneous branchial remnants, Int. J. Pediatr. Otorhinolaryngol. 67 (January (1)) (2003) 89–92] and through modern time medical literature bilateral cervical branchial chondrocutaneous remnants have been a subject of very few reports. We present a case of a 5-year-old patient with bilateral cervical chondrocutaneous remnants treated by complete surgical removal.
Keywords: Branchial apparatus, Chondrocutaneous remnant, Hyaline cartilage
1. Introduction
When reviewing the medical literature concerning cervical congenital anomalies one is usually referred to midline cysts or sinuses of branchial origin [1], [2]. There are few descriptions in the literature concerning pure chondrocutaneous rests in the lower neck region and even fewer descriptions concerning bilateral chondrocutaneous rests [3], [4]. During the last century these congenital anomalies received may different names ranging from cervical tags [5], hamartomata [5], skin appendages [5], polyps [5], papillomata [6], fibromata [6], vestiges [6], accessory auricules [7] or tragai [7] and even wattles [7].
This confusion concerning these anomalies was due most probably to the fact that the origin of these anomalies was poorly understood.
With further investigation into this group of anomalies it was decided upon the term choristoma. A choristoma is a generic pathological term for a non-neoplastic tumor-like mass of developmental origin in which are found tissues foreign to the site at which it is located. In the head and neck region there are several descriptions of these congenital anomalies including the oral cavity, nasopharynx, middle ear and eye region [8], [9], [10], [11]. When trying to further classify these anomalies Atlan et al. in a series of 17 patients exhibiting a congenital anomalie in the cervical area proposed the term ‘cervical chondrocutaneous branchial remnant’ [3].
2. Case report
A 5-year-old girl presented to the clinic with bilateral, painless, pediculated masses in the lower neck anterior to the sternocleidomastoid (Fig. 1). No signs of inflammation or infection were noted upon palpation. The mother stated that these masses were noted directly after birth and have not grown since. The mother denied any family history of neck masses. General physical examination was otherwise normal.
Fig. 1.
Bilateral cervical branchial chondrocutaneous remnants (arrows).
The patient was operated on under general anesthesia. Both tumor-like lesions were excised. These lesions appeared to be solid cartilaginous masses covered by normal appearing skin. During the operation no sign of fistulisation or communication to underlying structures was seen.
The histological examination confirmed bilateral ectopic hyaline cartilage remnants (Fig. 2) measuring 1.3
cm and 1cm in length and 0.7cm in width.
Fig. 2.
Histopathological slide of hyaline cartilage of cervical branchial chondrocutaneous remnant.
During patient follow-up no signs of clinical evidence of recurrence on both sides was noted.
3. Discussion
In reviewing the sparse literature concerning cervical chondrocutaneous branchial remnants we note the series that was presented by Atlan et al. [3]. In his series there was shown to be a male predominance, most anomalies were unilateral (only one patient presented with bilateral lesion), all remnants had a cartilaginous core and most patients exhibited associated anomalies. When discussing the possible embryologic origins of these remnants Atlan brings into consideration two components: the location of the remnants (upper versus middle and lower neck) and the type of cartilage found (elastic versus hyaline). When taking into consideration both of these factors Atlan, as most other authors in the literature, states that the possible origin of cervical chondrocutaneous branchial remnants may be divided into two groups. One advocates that these lesions are derivates of auricular tissue, as the other group states that these lesions originate from branchial tissue (lower arches). More precise examination of this problem leads to the inspection of the type of cartilage found in the remnants. While elastic cartilage would suggest an auricular (first or second branchial arch) origin or perhaps a possible derivate of the lower neck (fourth through six branchial arches), hyaline cartilage on the other hand would suggest a cervical source (second or third branchial arches).
When taking this fact into consideration (hyaline cartilage found in histology) and when regarding the location of the remnants in our case (lower neck) one can conclude that the most plausible origin is the second branchial arch.
The surgical treatment of these remnants is a simple excision extending to the superficial neck musculature, which was done in our case and proved sufficient. In the series presented by Atlan et al. [3] he stated that the average age of surgery was 12.8 months. We propose an intervention be done just before entering school allowing both minimization of surgical complications and the full cooperation of the patient yet sparing the patient the psychological complications of having a bilateral visual neck mass.
Although it is stated in the literature that branchial system malformations are commonly associated with other congenital defects [3], [4] in our case no other malformations were found.
4. Conclusion
Bilateral cervical chondrocutaneous branchial remnants are rare malformations sparsely cited in the medical literature. The fact that these lesions are benign and treated with complete surgical excision is an important factor for the patient presenting this malformation.
Our case is the first citing in the literature of bilateral cervical chondrocutaneous branchial remnants of hyaline cartilage source. Thus, this case may be in favor of the theory stating that these anomalies are origin of the second branchial arch (providing both explanation of location and cartilage type).
References
- . Congenital cartilaginous rests in the neck. Arch. Surg. 1934;28:59
- . Nomenclature of developmental tumors. Ann. Otol. Rhinol. Laryngol. 1984;93(January (1)):98–99
- . Cervical chrondrocutaneous branchial remnants. Plastic Reconstr. Surg. 1997;100(July (1)):32–39
- . Case report of bilateral cervical chondrocutaneous branchial remnants. Int. J. Pediatr. Otorhinolaryngol. 2003;67(January (1)):89–92
- . Congenital anomalies of the head and neck. Int. J. Dermatol. 1980;19(November (9)):479–486
- . Accessory tragi. Arch. Dermatol. 1971;104(December (6)):625–631
- . In: fifth ed.. Champion RH, Burton JL, Ebling FJG editor. Textbook of Dermatology. vol. 1:Boston: Blackwell Scientific; 1992;p. 445
- . Choristoma of the soft palate. Eur. Arch. Otorhinolaryngol. 1990;247(1):264–266
- . Choristoma of the nasopharynx in a newborn infant. Arch. Otolaryngol. 1996;347–348
- . Choristoma of the middle ear. Eur. Arch. Otorhinolaryngol. 1996;253(3):182–184
- . Phakomatous choristoma: a case report and review of the literature. Am. J. Dermatopathol. 2000;22(February (1)):55–59
PII: S1871-4048(08)00003-8
doi:10.1016/j.pedex.2008.01.001
© 2008 Elsevier Ireland Ltd. All rights reserved.
Volume 3, Issue 3 , Pages 117-119, September 2008
