{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,12,8]],"date-time":"2025-12-08T16:35:33Z","timestamp":1765211733637,"version":"3.46.0"},"reference-count":15,"publisher":"MDPI AG","issue":"23","license":[{"start":{"date-parts":[[2025,12,1]],"date-time":"2025-12-01T00:00:00Z","timestamp":1764547200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JCM"],"abstract":"<jats:p>Background: Congenital cheek sinuses are exceedingly rare craniofacial anomalies, typically present at birth and often misdiagnosed due to their resemblance to dermal pits or dimples. Only a few cases have been reported in the literature. This case is notable for its histopathological confirmation as a type II first branchial cleft anomaly containing an accessory tragus, highlighting the embryologic and clinical overlap between congenital cheek sinuses and branchial cleft anomalies. Case presentation: We report a three-year-old boy presenting with a congenital dimple along the right nasolabial fold. The lesion was present since birth and occasionally discharged clear fluid but had no history of infection or inflammation. Imaging demonstrated a subcutaneous tract without communication to the oral cavity or parotid gland. Under general anesthesia, surgical excision was performed, and the sinus tract was dissected in continuity to a cartilaginous remnant. Histopathologic analysis confirmed the presence of an accessory tragus consistent with a type II first branchial cleft anomaly. The patient\u2019s postoperative recovery was uneventful, and at three months\u2019 follow-up, there was no recurrence and an excellent cosmetic outcome. Conclusions: This case emphasizes the importance of considering branchial cleft anomalies in the differential diagnosis of congenital cheek sinuses. Although these lesions may appear benign and superficial, histopathological confirmation is critical for accurate classification. Complete surgical excision remains the definitive treatment, ensuring both diagnostic clarity and favorable aesthetic results. Increased awareness of such rare anomalies can improve diagnostic accuracy and surgical planning, particularly with respect to their potential proximity to critical facial structures.<\/jats:p>","DOI":"10.3390\/jcm14238519","type":"journal-article","created":{"date-parts":[[2025,12,8]],"date-time":"2025-12-08T16:20:29Z","timestamp":1765210829000},"page":"8519","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["A Rare Cheek Sinus Presentation of a Type II First Branchial Cleft Anomaly: A Case Report and Literature Review"],"prefix":"10.3390","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0553-2228","authenticated-orcid":false,"given":"Mor","family":"Rittblat","sequence":"first","affiliation":[{"name":"Department of Plastic and Reconstructive Surgery, Hadassah Hebrew University Medical Centre, Jerusalem 91120, Israel"},{"name":"Department of Military Medicine and \u201cTzameret,\u201d Faculty of Medicine, The Hebrew University of Jerusalem, Jerusalem 91120, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bar","family":"Davidov","sequence":"additional","affiliation":[{"name":"Department of Plastic and Reconstructive Surgery, Hadassah Hebrew University Medical Centre, Jerusalem 91120, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1023-341X","authenticated-orcid":false,"given":"Menahem","family":"Gross","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology Head and Neck Surgery, Hadassah-Hebrew University Medical Center, Jerusalem 91120, Israel"},{"name":"School of Medicine, Hebrew University, Jerusalem 91120, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Noam","family":"Armon","sequence":"additional","affiliation":[{"name":"Department of Plastic and Reconstructive Surgery, Hadassah Hebrew University Medical Centre, Jerusalem 91120, Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2025,12,1]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"e7108","DOI":"10.1097\/GOX.0000000000007108","article-title":"Congenital Lateral Upper Lip Sinus: A Review of the Literature","volume":"13","author":"Elias","year":"2025","journal-title":"Plast. Reconstr. Surg. Glob. Open"},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"1351","DOI":"10.1002\/lary.26343","article-title":"The ectopic accessory parotid system with congenital cheek fistula: An overview and current update","volume":"127","author":"Dutta","year":"2017","journal-title":"Laryngoscope"},{"key":"ref_3","first-page":"e15657","article-title":"Congenital Cheek Fistula: A Rare Case of Developmental Abnormality in the Buccal Area","volume":"13","author":"Kitano","year":"2021","journal-title":"Cureus"},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"311","DOI":"10.1054\/bjps.1998.3057","article-title":"Congenital cheek fistula: A report of three cases","volume":"52","author":"Nakano","year":"1999","journal-title":"Br. J. Plast. Surg."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"635","DOI":"10.1136\/jmedgenet-2014-102476","article-title":"Oculo-auriculo-vertebral spectrum: A review of the literature and genetic update","volume":"51","author":"Saraiva","year":"2014","journal-title":"J. Med. Genet."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"667","DOI":"10.1007\/s10792-019-01227-0","article-title":"Ophthalmic features and management outcomes of 30 children having Goldenhar syndrome","volume":"40","author":"Singh","year":"2019","journal-title":"Int. Ophthalmol."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"411","DOI":"10.1016\/S0021-9924(98)00013-6","article-title":"Branchio-oto-renal syndrome","volume":"31","author":"Smith","year":"1998","journal-title":"J. Commun. Disord."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"485","DOI":"10.1097\/00006534-196511000-00001","article-title":"The first and second branchial arch syndrome","volume":"36","author":"Grabb","year":"1965","journal-title":"Plast. Reconstr. Surg."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"2187","DOI":"10.1016\/j.jpedsurg.2011.08.019","article-title":"Congenital ectopic fistula of a minor salivary gland","volume":"46","author":"Yeh","year":"2011","journal-title":"J. Pediatr. Surg."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"360","DOI":"10.1002\/lary.24859","article-title":"Cheek fistula from the ectopic salivary gland: A variant of the oculo-auriculo-vertebral spectrum","volume":"125","author":"Sun","year":"2015","journal-title":"Laryngoscope"},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"1581","DOI":"10.1288\/00005537-197209000-00001","article-title":"Newer concepts of first branchial cleft defects","volume":"82","author":"Work","year":"1972","journal-title":"Laryngoscope"},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"69","DOI":"10.1007\/s13244-015-0454-5","article-title":"Branchial cleft anomalies: A pictorial review of embryological development and spectrum of imaging findings","volume":"7","author":"Adams","year":"2016","journal-title":"Insights Imaging"},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"291","DOI":"10.1001\/archotol.124.3.291","article-title":"First branchial cleft anomalies: A study of 39 cases and a review of the literature","volume":"124","author":"Triglia","year":"1998","journal-title":"Arch. Otolaryngol. Head Neck Surg."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"1453","DOI":"10.5858\/2004-128-1453-PQCAAN","article-title":"Pathologic quiz case: An anterior neck mass in a 5-month-old female infant. Wattle (congenital cervical tragus)","volume":"128","author":"Rund","year":"2004","journal-title":"Arch. Pathol. Lab. Med."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"3107","DOI":"10.1002\/jum.15016","article-title":"Ultrasound Evaluation of Dermal Sinuses\/Fistulas in Pediatric Patients","volume":"38","author":"Hosokawa","year":"2019","journal-title":"J. Ultrasound Med."}],"container-title":["Journal of Clinical Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2077-0383\/14\/23\/8519\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,12,8]],"date-time":"2025-12-08T16:30:57Z","timestamp":1765211457000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2077-0383\/14\/23\/8519"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,12,1]]},"references-count":15,"journal-issue":{"issue":"23","published-online":{"date-parts":[[2025,12]]}},"alternative-id":["jcm14238519"],"URL":"https:\/\/doi.org\/10.3390\/jcm14238519","relation":{},"ISSN":["2077-0383"],"issn-type":[{"value":"2077-0383","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,12,1]]}}}