Journal of Otolaryngology and Ophthalmology of Shandong University ›› 2024, Vol. 38 ›› Issue (2): 98-102.doi: 10.6040/j.issn.1673-3770.0.2022.505

• Original Article • Previous Articles     Next Articles

Congenital midline cervical cleft: a case report and literature review

PENG Yaqi, CHEN Liangsi, XU Mimi, HUNAG Yunye, SHENG Xiaoli   

  1. Department of Otorhinolaryngology & Head and Neck Surgery, Guangdong Provincial People's Hospital (Guangdong Academy of Medical Sciences), Southern Medical University, Guangzhou 510080, Guangdong, China
  • Online:2024-03-20 Published:2024-03-29

Abstract: Objective To explore the characteristics, therapy, and embryological etiology of congenital midline cervical cleft(CMCC). Methods After pathological diagnosis revealed a skin fistula, the patient underwent surgical treatment: resection of a contractile, longitudinal neck sinus, skin tag, skin lesion and followed by double Z-plasty. And the clinical data of a case of CMCC was retrospectively reviewed along with a review of related literature. Results The postoperative neck incision of the patient healed in one stage. No evidence of recurrence was found during the 1-year follow-up. Conclusion CMCC is a rare congenital abnormality of the neck. CMCC is thought to occur through a failure of the first or second branchial arch fusion. Double Z-plasty is a common technique for the prevention of contracture and restoration of the anterior contour of the neck.

Key words: Congenital abnormalities, Branchial cleft abnormalities, Midline cervical cleft, Double Z-plasty

CLC Number: 

  • R762
[1] Eastlack JP, Howard RM, Frieden IJ. Congenital midline cervical cleft: case report and review of the English language literature[J]. Pediatr Dermatol, 2000, 17(2): 118-122. doi:10.1046/j.1525-1470.2000.01727.x
[2] Shaye DA, Nteyumwete H, Kamaliza A, et al. Congenital midline cervical cleft: a rare window to the neck[J]. Facial Plast Surg Aesthetic Med, 2023, 25(2): 184-185. doi:10.1089/fpsam.2022.0101
[3] Puscas L. Midline cervical cleft: review of an uncommon entity[J]. Int J Pediatr, 2015: 209418. doi:10.1155/2015/209418
[4] 赵利敏, 李晓艳. 先天性颈正中裂1例[J]. 中国耳鼻咽喉头颈外科, 2012, 19(4): 224. doi:10.16066/j.1672-7002.2012.04.020 ZHAO Limin, LI Xiaoyan. Congenital median cervical fissure: a case report[J]. Chinese Archives of Otolaryngology-Head and Neck Surgery, 2012, 19(4): 224. doi:10.16066/j.1672-7002.2012.04.020
[5] 沈为民, 崔杰, 陈建斌, 等, 先天性颈正中裂二例[J]. 中华整形外科杂志, 2009, 25(5): 393. doi: 10.3760/cma.j.issn.1009-4598.2009.05.025
[6] 董玉英, 黄振东, 况光仪. 先天性鳃源性颈正中裂和囊肿1例[J]. 中国眼耳鼻喉科杂志, 2002, 2(1): 46. doi:10.3969/j.issn.1671-2420.2002.01.028 DONG Yuying, HUANG Zhendong, KUANG Guangyi. Congenital branchial median cervical fissure and cyst: a case report[J]. Chinese Journal of Ophthalmology and Otolaryngology, 2002, 2(1): 46. doi:10.3969/j.issn.1671-2420.2002.01.028
[7] Achard S, Leroy X, Fayoux P. Congenital midline cervical cleft: a retrospective case series of 8 children[J]. Int J Pediatr Otorhinolaryngol, 2016, 81: 60-64. doi:10.1016/j.ijporl.2015.12.008
[8] Cudzilo D, Matthews-Brzozowska T, Obloj B. Craniofacial morphology in midline cervical cleft: case report and review of literature[J]. Cleft Palate Craniofacial J, 2017, 54(2): 235-241. doi:10.1597/15-126
[9] Salawu AI, Aremu SK, Olakunle BF, et al. A delayed diagnosis of congenital midline cervical cleft[J]. Clin Case Rep, 2022, 10(3): 05540. doi:10.1002/ccr3.5540
[10] Morton CB. Median cleft of lower lip and mandible, cleft sternum and absence of basihyoid[J]. Arch Surg, 1935, 30(4): 647. doi:10.1001/archsurg.1935.01180100093010
[11] Gross RE, Connerley ML. Thyroglossal cysts and sinuses[J]. N Engl J Med, 1940, 223(16): 616-624. doi:10.1056/nejm194010172231602
[12] Ayache D, Ducroz V, Roger G, et al. Midline cervical cleft[J]. Int J Pediatr Otorhinolaryngol, 1997, 40(2/3): 189-193. doi:10.1016/S0165-5876(97)00028-1
[13] Kara CO, Kara IG. Congenital midline cervical cleft[J]. Otolaryngol Head Neck Surg, 2006, 135(6): 953-954. doi:10.1016/j.otohns.2006.05.024
[14] Jakobsen LP, Pfeiffer P, Andersen M, et al. Genetic studies in congenital anterior midline cervical cleft[J]. Am J Med Genet, 2012, 158A(8): 2021-2026. doi:10.1002/ajmg.a.35466
[15] Masood M.M., Piotr Mieczkowski, Ewa P Malc, et al. Congenital midline cervical cleft: first report and genetic analysis of two related patients[J]. Ann Otol Rhinol Laryngol, 2020, 129(7): 653-656. doi: 10.1177/0003489420906180
[16] Riba M, Bejarano M, Hernández C, et al. Midline cervical cleft: an anatomical finding and a proposal for a new approach[J]. Cleft Palate Craniofacial J, 2020, 57(12): 1422-1427. doi:10.1177/1055665620947957
[17] Goldfisher R, Bawa P, Ibrahim Z, et al. Clinical and imaging features of a congenital midline cervical cleft in a neonate: a rare anomaly[J]. Case Rep Pediatr, 2015: 439596. doi:10.1155/2015/439596
[18] French WE, Bale GF. Midline cervical cleft of the neck with associated branchial cyst[J]. Am J Surg, 1973, 125(3): 376-381. doi:10.1016/0002-9610(73)90067-6
[19] 黄舒玲, 陈良嗣, 张贝, 等. 双侧先天性第二鳃裂畸形四例[J]. 中华耳鼻咽喉头颈外科杂志, 2016, 51(6): 464-465. doi: 10.3760/cma.j.issn.1673-0860.2016.06.017 HUANG Shuling, CHEN Liangsi, ZHANG Bei, et al. Bilateral congenital second branchial cleft deformity: report of four cases and literature review[J]. Chinese Journal of Otorhinolaryngology Head and Neck Surgery, 2016, 51(6): 464-465. doi: 10.3760/cma.j.issn.1673-0860.2016.06.017
[20] 王莹莹, 张立庆, 周涵, 等. 74例鳃裂囊肿及瘘管临床分析[J]. 山东大学耳鼻喉眼学报, 2020, 34(4): 111-116. doi: 10.6040/j.issn.1673-3770.0.2019.523 WANG Yingying, ZHANG Liqing, ZHOU Han, et al. Clinical analysis of 74 cases of branchial cleft cysts and fistulas[J]. Journal of Otolaryngology and Ophthalmology of Shandong University, 2020, 34(4): 111-116. doi: 10.6040/j.issn.1673-3770.0.2019.523
[21] 姚丹勉, 陈良嗣, 梁璐, 等. 颈前中线分区清扫治疗儿童复发性甲状舌管囊瘘[J]. 临床耳鼻咽喉头颈外科杂志, 2016, 30(19): 1545-1547. doi:10.13201/j.issn.1001-1781.2016.19.010 YAO Danmian, CHEN Liangsi, LIANG Lu, et al. The selective anterior neck dissection for treating children's recurrent thyroglossal duct cysts[J]. Journal of Clinical Otorhinolaryngology Head and Neck Surgery, 2016, 30(19): 1545-1547. doi:10.13201/j.issn.1001-1781.2016.19.010
[22] 李育广, 刘凤英. 手术治疗甲状舌管囊肿与瘘管65例[J]. 山东大学耳鼻喉眼学报, 2008, 22(6): 575-576 LI Yuguang, LIU Fengying. Surgical treatment of 65 cases of thyroglossal duct cyst and fistula[J]. Journal of Otolaryngology and Ophthalmology of Shandong University, 2008, 22(6): 575-576
[23] Annapoorni CV, Varkey S. Congenital midline cervical cleft-A case report on a rare anomaly of the neck[J]. J Indian Assoc Pediatr Surg, 2022, 27: 627-628. doi:10.4103/jiaps.jiaps_206_21
[24] Moreno SD, Christopher P, Kloostra P. Congenital midline cervical cleft(CMCC): z-plasty versus linear cutaneous repair[J]. J Craniofacial Surg, 2022, 34(1): e38-e41. doi:10.1097/scs.0000000000008940
[25] D'Souza JN, Valika T, Maddalozzo J. Surgical management of midline cervical cleft[J]. Int J Pediatr Otorhinolaryngol, 2019, 127: 109657. doi:10.1016/j.ijporl.2019.109657
[26] Bahakim A, Francois M, Van Den Abbeele T. Congenital midline cervical cleft and W-plasty: our experience[J]. Int J Otolaryngol, 2018: 1-5. doi:10.1155/2018/5081540
[1] Chengcheng LIANG,Xin WANG,Dapeng LEI. Electronic nasopharyngoscopy for the diagnosis and treatment of otorhinolaryngologic diseases: A retrospective study of 27400 cases [J]. Journal of Otolaryngology and Ophthalmology of Shandong University, 2019, 33(5): 107-110.
[2] Junxiao GAO,Qianhui QIU. Efficacy of endoscopic surgery using two types of flaps to reconstruct the skull base after radiotherapy for nasopharyngeal carcinoma [J]. Journal of Otolaryngology and Ophthalmology of Shandong University, 2019, 33(2): 46-50.
[3] SONG Xicheng. Application value of enhanced recovery after surgery in head and neck cancer surgery [J]. Journal of Otolaryngology and Ophthalmology of Shandong University, 2018, 32(5): 1-4.
[4] SUN Shasha, QIU Jie, ZANG Chuanshan, SUN Yan. Behcets disease misdiagnosed as acute epiglottitis: a report of two cases. [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2017, 31(5): 107-109.
[5] GUO Yingyuan, ZHANG Dejun, GUAN Guofang, WEN Lianji, ZHANG Hui, YU Duojiao, MA Shuaishuai. Exploration of the application of three-dimensional printing technology in the standardized training of doctors in the otolaryngological department. [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2017, 31(3): 119-122.
[6] LIU Wenjing. Influence of TransPRK and LASEK on postoperative vision, diopter and corneal biomechanical properties in myopic eyes with thin cornea. [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2016, 30(6): 70-74.
[7] WANG Yalin, CHEN Jianqiu. Discussion on the relationship between vocal cord leukoplakia and premalignant lesions and research progress of vocal cord leukoplakia. [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2016, 30(6): 90-93.
[8] XIE Chang, YAN Bin, LI Jianxing, LU Yongtian. Anatomical and clinical evaluation of the endoscopic transoral transpharyngeal approach to the atlantoaxial. [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2016, 30(3): 47-51.
[9] MENG Qingguo, LU Yongtian, WU Xiaoliu, LI Jianxing, SUN Huanji, ZHANG Baoying, XIE Chang. The groove behind the palatovaginal canal: a landmark for identification of the palatovaginal canal [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2015, 29(5): 55-61.
[10] LI Danfeng, CHEN Fuquan, SHI Li, CHEN Jun. Application of stepped multi-model merged methodology in ENT clinical teaching for under-graduates. [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2015, 29(4): 77-79.
[11] YAN Yu, ZHU Jiang. Advances in the diagnosis of early laryngeal carcinoma and precancerous laryngeal lesions. [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2015, 29(4): 80-85.
[12] DONG Yi-yuan, LING Yue-fu, ZHANG Yao-ting. Effect of melatonin on the cTnT and Ca2+-ATPase of chronic intermittent hypoxia cardiac muscular tissues [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2014, 28(5): 1-3.
[13] GOU Dong-ming1, WANG Hai1, CHEN Jian-chao2. Applicaiton of near hairline coronal incision in otolaryngology and head and neck surgery [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2014, 28(1): 62-64.
[14] WU Xi-fu, HUANG Jian-cong, ZHUANG Shi-min, HUANG Zi-zhen, YE Jin, ZHANG Ge-hua. Cervical necrotizing fasciitis: case report and literature review [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2014, 28(1): 89-90.
[15] QIN He, SUN Jian-jun. Application of acellular dermal matrix in otorhinolaryngological reconstructive surgery [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2012, 26(4): 75-77.
Viewed
Full text


Abstract

Cited

  Shared   
  Discussed   
[1] . [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2006, 20(2): 116 -118 .
[2] ZHOU Zi-ning,JIN Guo-wei . [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2006, 20(5): 462 -465 .
[3] ZHOU Bin,LI Bin . Endoscopic sinus surgery for 75 patients with chronic sinusitis and nasal polyps[J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2006, 20(1): 24 -26 .
[4] XU Sainan,YANG Lei . Apoptosis of epithelial cells in nasal polyps promoted by erythromycin[J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2006, 20(1): 27 -29 .
[5] . [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2006, 20(1): 94 -95 .
[6] LIU Lian-he . Treatment of deep neck abscess in 37 cases[J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2008, 22(2): 180 -181 .
[7] . [J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2008, 22(3): 200 -203 .
[8] QIAO Yi,NI Guan-sen,CHEN Wen-wen . Effect of H-UPPP and nasal operations on obstructive sleep apnea syndrome in 38 cases
[J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2008, 22(3): 206 -208 .
[9] WANG Xiao-feng,LIN Chang,CHENG Jin-mei . Expression of ABAD in inner ears and its clinical significance in different age mice[J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2008, 22(3): 207 -211 .
[10] FAN Qi-jun,HUANG Zhi-wu,MEI Ling,XIAO Bo-kui . Expression of the heat shock protein 27 in rat cochlea induced by sodium salicylate injection by the FQ-PCR technique[J]. JOURNAL OF SHANDONG UNIVERSITY (OTOLARYNGOLOGY AND OPHTHALMOLOGY), 2008, 22(3): 212 -214 .