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Clinicopathological Characteristics of Cervical Chondrocutaneous Branchial Remnant: a Single-Institutional Experience

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Clinicopathological Characteristics of Cervical Chondrocutaneous Branchial Remnant: a Single-Institutional Experience Int J Clin Exp Pathol 2017;10(9):9866-9877 www.ijcep.com /ISSN:1936-2625/IJCEP0059151 Original Article Clinicopathological characteristics of cervical chondrocutaneous branchial remnant: a single-institutional experience Ha Young Woo, Hyun-Soo Kim Department of Pathology, Severance Hospital, Yonsei University College of Medicine, Seoul, Republic of Korea Received June 11, 2017; Accepted June 22, 2017; Epub September 1, 2017; Published September 15, 2017 Abstract: Cervical chondrocutaneous branchial remnant (CCBR) is an uncommon developmental anomaly typically seen on the lateral neck. We recently experienced four cases of CCBR and initiated a comprehensive review of previously published cases. During a 10-year period, four (0.4%) of the 1,096 patients who underwent excision of branchial cleft anomalies were diagnosed as having CCBR at our institution. Patient age ranged from 2-6 years and patients presented with asymptomatic cutaneous masses present since birth measuring approximately 1 cm on the lateral neck. Three patients had congenital thyroid hemiagenesis, subependymal cyst, and tongue tie, respectively. We identified 76 previously published cases of CCBR. The median age of these patients was 18 months. CCBR de- veloped more often in males (48/80; 60.0%). Most of the masses were located on the left (34/80; 42.5%) or right (18/80; 22.5%) lateral neck, whereas 23 (28.75%) involved bilateral lesions. Lesion size ranged from 0.3-3.5 cm. Grossly, the overlying skin of the masses was similar to the surrounding skin of the neck. Histologically, the lesions were covered by keratinizing squamous epithelium and had skin appendages and cartilage. Thirty-nine (48.75%) and 12 (15.0%) patients were found to have elastic and hyaline cartilage, respectively. Twenty-eight patients had single (13/28; 46.4%) or multiple (15/28; 53.6%) congenital anomalies. Forty-four different types of anomalies were reported. The most frequent anomalies were problems with cardiovascular and auditory systems. Our observa- tions suggest that CCBR is a visible marker for more serious associated congenital anomalies. We recommend that clinicians and pediatricians further evaluate patients with CCBR through complete physical examination, abdominal and cardiac ultrasound, karyotyping, and biochemical marker analysis. Keywords: Cervical chondrocutaneous branchial remnant, congenital cartilaginous rest of the neck, cervical ac- cessory tragus, branchial cleft anomaly, congenital malformation Introduction pears. Persistence of the branchial cleft or pouch results in cervical anomalies along the The branchial arches represent the embryologi- anterior border of the sternocleidomastoid cal precursors of the face, neck, and pharynx. muscle from the tragus to the clavicle. They Anomalies of branchial arches are the second may present as cysts, sinuses, fistulae, or carti- most common congenital lesions of the head laginous remnants. and neck in children, accounting for approxi- mately 20% of pediatric congenital head and Accessory tragi are fairly common, congenital neck lesions and occasionally forming parts of anomalies of the external ear. They usually complex syndromes, especially those of the appear as small, skin-colored, preauricular tags first and second branchial arches [1]. During or nodules and consist of skin, subcutaneous the third to fifth week of embryonic develo- fat, and/or cartilage [2]. During the fifth to sixth pment, the second branchial arch grows cau- week of embryonic development, the formation dally and covers the third, fourth, and sixth of the auricle of the ear is initiated from the first branchial arches. When it fuses to the skin ca- and second branchial arches. These arches udal to these arches, the cervical sinus is then form the six hillocks (mesenchymal tuber- formed. Eventually, the edges of cervical sinus cles), and subsequently fuse to form the auricle fuse and the ectoderm within the tube disap- structures [3]. The formation of accessory tragi Clinicopathological characteristics of CCBR is due to errors during this period. The diverse cleft fistula”, “branchial cleft sinus”, “cervical clinical manifestations of accessory tragi may chondrocutaneous branchial remnants”, “con- be unilateral or bilateral, single or multiple, and genital cartilaginous rests of the neck”, “cervi- soft or firm. These anomalies may be isolated cal accessory tragus”, “cervical auricles”, “neck or associated with other congenital anomalies auricles”, and “wattle” among archival surgi- of the first and second branchial arches. cal pathology cases. Clinical and pathological information were obtained from the electrical Similar to preauricular accessory tragi in ap- medical record system and pathology reports. pearance but located in the lower neck, cer- The clinical details that were reviewed included vical chondrocutaneous branchial remnants age at the time of diagnosis, sex, presenting (CCBRs) are rather less common congenital symptom, side of lesion, number of lesion, size lesions. They are among the rarest of branchial of lesion, associated congenital anomaly, his- cleft anomalies. Owing to lack of consensus, tological type of cartilage, and coexisting there are numerous synonyms for CCBRs unusual histopathological finding. This study including branchial cartilages, cervical auricles, was reviewed and approved by the Institutional cervical accessory tragi, cervical skin tags, Review Board at Severance Hospital, Yonsei cervical vestiges, choristomas, papillomas, University Health System, Seoul, Republic of fibromas, wattles, and congenital cartilaginous Korea (2016-1518-001). rests of the neck [4-32]. In 1997, however, Atlan and colleagues [8] suggested the term Histopathology and histochemistry CCBRs as a clear, widely acceptable name for these anomalies. The resected specimens were fixed in 10% neutral-buffered formalin and embedded in At present, 76 cases of CCBR have been docu- paraffin blocks. Each formalin-fixed, paraffin- mented in the English literature [4-32]. Existing embedded block was sectioned at 4 μm on a confusion regarding the origin and the correct standard rotary microtome, and slices were nomenclature is caused by the difficulty of clini- brought from a water bath on cleaned slides cal differential diagnosis of CCBRs, which main- and stained with hematoxylin and eosin. Mas- ly include sentinel tags next to the branchial son trichrome staining and elastic van Gieson sinus tracts or fistulae, acrochordons, and staining were also performed. The final histo- benign papillomas. Recently, some patients pathological diagnoses of the lesions were presented to our institution with CCBRs. Upon made by a board-certified pathologist. review of the previous literature and standard textbooks, we found very little detailed, clini- Literature review cally useful information concerning these lesions. Therefore, we decided to examine our The Medline database was thoroughly search- institutional experience and review previously ed using the PubMed retrieval service. Searches published cases thoroughly with the goal of were performed at August 2016, using the key clarifying their clinical and pathological charac- words “branchial cartilages”, “cervical acces- teristics, anatomical and surgical characteris- sory tragus”, “cervical auricle”, “cervical chon- tics, and any associated local or distant con- drocutaneous branchial remnant”, “cervical genital anomalies. Comprehensive analyses of skin tag”, “congenital cartilaginous rest of the cases may expand our knowledge regarding neck”, “neck auricle”, and “wattle”. CCBRs. Results Materials and methods Patient demographics Case selection During the period from August 2007 to July The cases were selected from the computer- 2016, 1,096 patients underwent excision for ized files of Department of Pathology, Seve- branchial cleft anomalies. The ages of the rance Hospital, Yonsei University College of 1,096 patients ranged between 1 month and Medicine, Seoul, Republic of Korea. A thorough 83 years (median, 12 years). The majority search was performed using the key words (972/1,096; 88.7%) of these patients were “branchial”, “branchial cleft cyst”, “branchial diagnosed with first or second branchial cleft 9867 Int J Clin Exp Pathol 2017;10(9):9866-9877 Clinicopathological characteristics of CCBR Figure 1. Image findings of cervical chondrocutaneous branchial remnants (case 2). A: Axial view of neck computed tomography reveals a polypoid mass (red circle) in the left neck. B: The mass (red circle) appears to extend to the subcutaneous layer, but there is no connection to other deep structures of the neck. C: A coronal view reveals right thyroid hemiagenesis. tongue tie (case 4). Neck computed tomogra- phy (CT) was performed in one (25.0%) case (case 2) and revealed a tiny pedunculated mass on the left neck. The mass extended to the sub- cutaneous layer, but did not appear to be con- nected with deep underlying structures (Figure 1A, 1B). In this case, the right thyroid was not identified Figure( 1C). All patients underwent surgical excision. The masses and underlying cartilage were removed with some subcutane- ous tissue. There were no connections between the lesions and the deep soft tissue of the neck. Grossly, they were pedunculated masses Figure 2. Gross findings of cervical chondrocutane- covered with normal-appearing skin (Figure 2). ous branchial remnants (case 1). An ovoid, peduncu- The cut surfaces showed centrally located car- lated mass is located at the right anterior neck. It is tilaginous tissues.
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