Journal List > Ann Dermatol > v.31(1) > 1126771

Cheon, Kim, Park, Lee, Jin, Shim, Kim, Kim, Ko, Kim, Kim, and You: A Case of Cervical Chondrocutaneous Branchial Remnant Comprised of Hyaline Cartilage
Dear Editor:
Cervical chondrocutaneous branchial remnants (CCBRs) are rare, congenital, benign neck masses, and are derived from dislocated branchial apparatus components comprised of cartilage tissues1.
Herein, we describe a 44-year-old female who presented with a solitary asymptomatic skin-colored nodule on the lower part of right side of the neck anterior to SCM (Fig. 1A). The patient denied any history of trauma, surgery, or injection. No remarkable findings except for the skin lesion were observed. Ultrasonography showed a hyperechoic nodule (0.8×0.3 cm) in the subcutaneous layer; No internal vascularity, fistula, or sinus connection with the deep underlying structures of the neck was found (Fig. 1B). Histopathological examination after surgical excision showed a hyaline cartilage core in the dermis with isogenous chondrocytes, a glassy extracellular matrix and absence of elastic fiber, which characterize hyaline cartilage (Fig. 1C, D). A diagnosis of CCBR was confirmed, and the patient showed no recurrence during 9 months of follow-up.
CCBRs have been reported under numerous names, such as wattle, cervical auricle, accessory tragus, cervical skin tag, and congenital cartilaginous rests of the neck1. Several pervious authors identified CCBRs comprised of elastic cartilage, suggesting that CCBRs arise from ectopic auricular tissue2. However, Begovic et al.1 reported numerous cases of CCBRs comprised of hyaline cartilage. Because the second branchial arch can differentiate into both elastic and hyaline cartilage, the authors insisted that the origin of CCBRs is the second branchial arch. In addition, CCBRs are located in the middle or lower portion of the SCM and are deeply connected with the superficial fascia of the neck. CCBRs are considered a second branchial remnant disorder rather than an ectopic auricular migratory disorder3. Therefore, the use of particular terms such as cervical auricle and accessory tragus should be avoided.
Recent studies have revealed more detailed histological features of CCBRs. Large nerves and cluster of Pacinian corpuscles have been observed in the periphery of CCBRs4. Pacinian corpuscles are primary mechanoreceptors that are usually located in the deep dermis and detect gross pressure changes and vibration. Researchers in that study hypothesized that CCBRs attract sensory axons and neural crest cells that organize as Pacinian corpuscles.
CCBRs are often associated with numerous congenital anomalies; auditory, gastrointestinal, genitourinary, cardiovascular, musculoskeletal, and visual anomalies, as well as complex syndromes, occur in up to 76% of cases2. Thus, detailed additional examinations, such as abdominal and cardiac ultrasonography, are recommended for patients with CCBRs. However, the prevalence of associated anomalies varies greatly. Begovic et al.1 reported that 29% of CCBR patients exhibit anomalies. Compared to those in the studies of Atlan et al.2 and Begovic et al.1, all patients included in the study of Atlan et al.2 exhibited CCBRs composed of elastic cartilage. Meanwhile, Begovic et al.1 found that more than half of the patients in their study exhibited CCBRs composed of hyaline cartilage. Retrospective analysis revealed that among 11 cases of CCBRs composed of hyaline cartilage, only one case involved an associated anomaly (vesicoureteral reflux, which is common in normal neonates) (Table 1)135. Although the cause remains uncertain, the presence of hyaline cartilage in CCBRs can be considered a favorable marker, indicating a low possibility of associated anomalies.
This rare case involving a CCBR comprised of hyaline cartilage further supports the current knowledge regarding the embryogenesis and associated anomalies of CCBRs.

Figures and Tables

Fig. 1

(A) Tiny (about 0.3×0.8 cm in size) pedunculated skin-colored to yellowish nodule on the lower part of right side of the neck anterior to sternocleidomastoid muscle (SCM) is shown (red dotted line: right SCM area of the neck). (B) Ultrasonography reveals a well-defined hypoechoic nodule (approximately 0.8×0.3 cm) in the subcutaneous fat layer (white arrows). (C) Histopathologic examination of the skincolored nodule shows a central cartilaginous core in the subcutaneous fat layer (H&E, ×20). (D) There was no elastic fiber in extracellular matrix of cartilaginous core (Verhoeff's-van Gieson stain, ×200). We received the patient's consent form about publishing all photographic materials.

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Table 1

Reported cases of CCBRs composed of hyaline cartilage and associated anomalies

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Case Reference Sex/age Location Associated anomalies
1 Tamir et al.5 (2008) F/5 yr Bilateral NA
2 Choi et al.3 (2012) F/4 yr Left NA
3 Begovic et al.1 (2014) M/2 mo Left NA
4 F/5 mo Left NA
5 F/6 mo Right NA
6 M/13 mo Left NA
7 M/15 mo Right Vesicoureteral reflux
8 F/4 mo Right NA
9 M/7 yr Left NA
10 M/15 yr Bilateral NA
11 Present case F/44 yr Left NA

CCBRs: cervical chondrocutaneous branchial remnants, F: female, M: male, NA: not available.

Notes

CONFLICTS OF INTEREST The authors have nothing to disclose.

References

1. Begovic N, Simic R, Vlahovic A, Kravljanac D, Djuricic S, Mijovic T. Cervical chondrocutaneous branchial remnants--report of 17 cases. Int J Pediatr Otorhinolaryngol. 2014; 78:1961–1964.
2. Atlan G, Egerszegi EP, Brochu P, Caouette-Laberge L, Bortoluzzi P. Cervical chrondrocutaneous branchial remnants. Plast Reconstr Surg. 1997; 100:32–39.
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3. Choi HJ, Lee JC, Kim JH. Cervical branchial cartilaginous remnant. J Craniofac Surg. 2012; 23:611–613.
crossref
4. Feito J, Ramos-García JL, Gago Á, Cobo JL, García-Suárez O, Junquera LM, et al. Pacinian corpuscles in a cervical chondrocutaneous remnant: a case report and update of pacinian corpuscles. Am J Dermatopathol. 2016; 38:231–235.
5. Tamir S, Nidal M, Constantin R, Perez R, Sichel JY. Bilateral cervical chondrocutaneous branchial remnants. Int J Pediatr Otorhinolaryngol Extra. 2008; 3:117–119.
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ORCID iDs

Sang-Jin Cheon
https://orcid.org/0000-0002-6099-4460

Tae-Wook Kim
https://orcid.org/0000-0002-8138-7993

Seong-Min Park
https://orcid.org/0000-0003-4847-235X

Hyun-Ju Lee
https://orcid.org/0000-0002-1696-0976

HyunJu Jin
https://orcid.org/0000-0002-0343-1629

Woo-Haing Shim
https://orcid.org/0000-0002-5182-5294

Gun-Wook Kim
https://orcid.org/0000-0003-1599-7045

Hoon-Soo Kim
https://orcid.org/0000-0002-7649-1446

Hyun-Chang Ko
https://orcid.org/0000-0002-3459-5474

Byung-Soo Kim
https://orcid.org/0000-0003-0054-8570

Moon-Bum Kim
https://orcid.org/0000-0001-8801-1369

Hyang-Suk You
https://orcid.org/0000-0002-1697-397X

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