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Branchial cleft anomalies: hybrid “Branchial Inclusion” theory

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Abstract

Purpose

Branchial cleft anomalies (BCAs) are developmental malformations of the head and neck region. Their histogenesis has been the subject of controversy and is not fully understood. This study aimed to test all present developmental theories (“branchial apparatus,” “precervical sinus,” “thymopharyngeal,” and “inclusion” theories) on a sample of 48 BCAs from a single institution.

Methods

We performed a retrospective analysis of clinical–epidemiological and anatomical–pathological characteristics of BCAs treated over a 12-year period in our hospital.

Results

Overall, 46 patients (24 [52.17%] women and 22 men [47.83%]) underwent surgical excision of 48 BCAs. The mean patient age at presentation was 31.65 ± 19.40 years. Branchial cleft cysts were found in 42 (87.50%) cases, and branchial cleft sinuses were found in six (12.50%) cases. Eight (16.67%) BCAs were distributed in the preauricular region, 34 (70.83%) at the anterior border of the sternocleidomastoid muscle (SCM), three (6.25%) at the posterior border of the SCM, two (4.17%) in the suprasternal notch, and one (2.08%) in the retrosternal space. Histopathologically, 39 (81.25%) BCAs had a lymphoepithelial structure and nine (18.75%) BCAs had solitary epithelial cells. Inflammation and infection were observed in 24 (50%) and 12 (25%) cases, respectively.

Conclusion

None of the hypothesized developmental theories fully explain the embryonic origin of BCA in our study sample. A possible explanation of BCA histogenesis is through the hybrid “branchial inclusion” theory.

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References

  1. Agaton-Bonilla FC, Gay-Escoda C (1996) Diagnosis and treatment of branchial cleft cysts and fistulae. A retrospective study of 183 patients. Int J Oral Maxillofac Surg 25(6):449–452. https://doi.org/10.1016/s0901-5027(96)80081-6

    Article  CAS  PubMed  Google Scholar 

  2. Adams A, Mankad K, Offiah C, Childs L (2016) Branchial cleft anomalies: a pictorial review of embryological development and spectrum of imaging findings. Insights Imaging 7(1):69–76. https://doi.org/10.1007/s13244-015-0454-5

    Article  PubMed  Google Scholar 

  3. Ascherson GM (1832) De fistulis colli congenitis adjecta fissurarum branchialium in mammalibus avibusque historia succincta. In: Jonas CH (ed) Habilitationsschrift. Berolini, Berlin, pp 1–21

    Google Scholar 

  4. His W (1886) Ueber der Sinus praecervicalis und uber die Thymusanlage. Archiv fur Anatomie und Entwickelungsgescichte 9:421–433

    Google Scholar 

  5. Wenglowski R (1912) Ueber die Halsfisteln und Cysten. Arch Klin Chir 98:151–208

    Google Scholar 

  6. Luschka H (1848) Ueber fistula colli congenita. Archiv fur Physiologische Heilkunde 7:25–27

    Google Scholar 

  7. Lucke AI (1861) Ueber atheromysten der Lymphdrusen. Arch Klin Chir 1:356–365

    Google Scholar 

  8. King ES (1949) The lateral lympho-epithelial cyst of the neck; branchial cyst. Aust N Z J Surg 19(2):109–121. https://doi.org/10.1111/j.1445-2197.1949.tb03690.x

    Article  CAS  PubMed  Google Scholar 

  9. Bhaskar SN, Bernier JL (1959) Histogenesis of branchial cysts; a report of 468 cases. Am J Pathol 35(2):407–443

    CAS  PubMed  PubMed Central  Google Scholar 

  10. Pupic-Bakrac J, Pupic-Bakrac A, Novakovic J, Skitarelic N (2020) Congenital Neck Masses. J Craniofac Surg. https://doi.org/10.1097/SCS.0000000000007122

    Article  PubMed  Google Scholar 

  11. Kenealy JF, Torsiglieri AJ Jr, Tom LW (1990) Branchial cleft anomalies: a five-year retrospective review. Trans Pa Acad Ophthalmol Otolaryngol 42:1022–1025

    CAS  PubMed  Google Scholar 

  12. Smith OD, Ellis PD, Bearcroft PW, Berman LH, Grant JW, Jani P (2000) Management of neck lumps–a triage model. Ann R Coll Surg Engl 82(4):223–226

    CAS  PubMed  PubMed Central  Google Scholar 

  13. McLean T, Iseli C, Amott D, Taylor M (2015) Case report of a p16INK4A-positive branchial cleft cyst. J Laryngol Otol 129(6):611–613. https://doi.org/10.1017/S0022215114003223

    Article  CAS  PubMed  Google Scholar 

  14. Hirshoren N, Fried N, Weinberger JM, Eliashar R, Korem M (2019) The microbiology characteristics of infected branchial cleft anomalies. OTO Open 3(3):2473. https://doi.org/10.1177/2473974x19861065

    Article  Google Scholar 

  15. Rickles NH, Little JW (1967) The histogenesis of the branchial cyst. II. A study of the lining epithelium. Am J Pathol 50(5):765–777

    CAS  PubMed  PubMed Central  Google Scholar 

  16. Work WP (2015) Newer concepts of first branchial cleft defects. 1972. Laryngoscope 125(3):520–532. https://doi.org/10.1002/lary.25202

    Article  PubMed  Google Scholar 

  17. Magdy EA, Ashram YA (2013) First branchial cleft anomalies: presentation, variability and safe surgical management. Eur Arch Otorhinolaryngol 270(6):1917–1925. https://doi.org/10.1007/s00405-012-2287-x

    Article  PubMed  Google Scholar 

  18. Liu H, Cheng A, Ward BB, Wang C, Han Z, Feng Z (2020) Clinical manifestations, diagnosis, and management of first branchial cleft fistula/sinus: a case series and literature review. J Oral Maxillofac Surg. https://doi.org/10.1016/j.joms.2019.12.017

    Article  PubMed  Google Scholar 

  19. Thomaidis V, Seretis K, Tamiolakis D, Papadopoulos N, Tsamis I (2006) Branchial cysts. A report of 4 cases. Acta Dermatovenerol Alp Pannonica Adriat 15(2):85–89

    CAS  PubMed  Google Scholar 

  20. Maturo SC, Michaelson PG, Faulkner JA (2007) Primary branchiogenic carcinoma: the confusion continues. Am J Otolaryngol 28(1):25–27. https://doi.org/10.1016/j.amjoto.2006.06.005

    Article  PubMed  Google Scholar 

  21. Roche JP, Younes MN, Funkhouser WK, Weissler MC (2010) Branchiogenic carcinoma of a first branchial cleft cyst. Otolaryngol Head Neck Surg 143(1):167–168. https://doi.org/10.1016/j.otohns.2010.01.031

    Article  PubMed  Google Scholar 

  22. Ustun M, Risberg B, Davidson B, Berner A (2002) Cystic change in metastatic lymph nodes: a common diagnostic pitfall in fine-needle aspiration cytology. Diagn Cytopathol 27(6):387–392. https://doi.org/10.1002/dc.10201

    Article  PubMed  Google Scholar 

  23. Flanagan PM, Roland NJ, Jones AS (1994) Cervical node metastases presenting with features of branchial cysts. J Laryngol Otol 108(12):1068–1071. https://doi.org/10.1017/s0022215100128907

    Article  CAS  PubMed  Google Scholar 

  24. Martin H, Morfit HM, Ehrlich H (1950) The case for branchiogenic cancer (malignant branchioma). Ann Surg 132(5):867–887. https://doi.org/10.1097/00000658-195011000-00002

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  25. Khafif RA, Prichep R, Minkowitz S (1989) Primary branchiogenic carcinoma. Head Neck 11(2):153–163. https://doi.org/10.1002/hed.2880110209

    Article  CAS  PubMed  Google Scholar 

  26. Goldenberg D, Begum S, Westra WH, Khan Z, Sciubba J, Pai SI, Califano JA, Tufano RP, Koch WM (2008) Cystic lymph node metastasis in patients with head and neck cancer: an HPV-associated phenomenon. Head Neck 30(7):898–903. https://doi.org/10.1002/hed.20796

    Article  PubMed  Google Scholar 

  27. Briggs RD, Pou AM, Schnadig VJ (2002) Cystic metastasis versus branchial cleft carcinoma: a diagnostic challenge. Laryngoscope 112(6):1010–1014. https://doi.org/10.1097/00005537-200206000-00014

    Article  PubMed  Google Scholar 

  28. Mokhtari S (2012) Mechanisms of cyst formation in metastatic lymph nodes of head and neck squamous cell carcinoma. Diagn Pathol 7:6. https://doi.org/10.1186/1746-1596-7-6

    Article  PubMed  PubMed Central  Google Scholar 

  29. Huang YH, Yeh CH, Cheng NM, Lin CY, Wang HM, Ko SF, Toh CH, Yen TC, Liao CT, Ng SH (2017) Cystic nodal metastasis in patients with oropharyngeal squamous cell carcinoma receiving chemoradiotherapy: relationship with human papillomavirus status and failure patterns. PLoS ONE 12(7):e0180779. https://doi.org/10.1371/journal.pone.0180779

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  30. Yehuda M, Schechter ME, Abu-Ghanem N, Golan G, Horowitz G, Fliss DM, Abu-Ghanem S (2018) The incidence of malignancy in clinically benign cystic lesions of the lateral neck: our experience and proposed diagnostic algorithm. Eur Arch Otorhinolaryngol 275(3):767–773. https://doi.org/10.1007/s00405-017-4855-6

    Article  PubMed  Google Scholar 

  31. Gourin CG, Johnson JT (2000) Incidence of unsuspected metastases in lateral cervical cysts. Laryngoscope 110(10 Pt 1):1637–1641. https://doi.org/10.1097/00005537-200010000-00012

    Article  CAS  PubMed  Google Scholar 

  32. Lee DH, Yoon TM, Lee JK, Lim SC (2018) Clinical study of second branchial cleft anomalies. J Craniofac Surg 29(6):e557–e560. https://doi.org/10.1097/SCS.0000000000004540

    Article  PubMed  Google Scholar 

  33. Schroeder JW Jr, Mohyuddin N, Maddalozzo J (2007) Branchial anomalies in the pediatric population. Otolaryngol Head Neck Surg 137(2):289–295. https://doi.org/10.1016/j.otohns.2007.03.009

    Article  PubMed  Google Scholar 

  34. Papadogeorgakis N, Petsinis V, Parara E, Papaspyrou K, Goutzanis L, Alexandridis C (2009) Branchial cleft cysts in adults. Diagnostic procedures and treatment in a series of 18 cases. Oral Maxillofac Surg 13(2):79–85. https://doi.org/10.1007/s10006-009-0156-6

    Article  PubMed  Google Scholar 

  35. Magdy EA, Hamza A, Youssef A, Yoneis A (2020) Second branchial cleft fistula/sinus tract endoscopy: a novel intraoperative technique assisting complete surgical resection. Eur Arch Otorhinolaryngol. https://doi.org/10.1007/s00405-020-06158-6

    Article  PubMed  Google Scholar 

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Funding

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Author information

Authors and Affiliations

  1. Department of Otorhinolaryngology and Maxillofacial Surgery, General Hospital Zadar, Bože Peričića 5, 23 000, Zadar, Croatia

    Jure Pupić-Bakrač, Neven Skitarelić, Ivan Baraka & Mladen Srzentić

  2. Department of Ophthalmology, General Hospital Zadar, Bože Peričića 5, 23 000, Zadar, Croatia

    Ana Pupić-Bakrač

  3. Department of Otorhinolaryngology and Maxillofacial Surgery, University Clinical Hospital Mostar, Bijeli Brijeg bb, 88 000, Mostar, Bosnia and Herzegovina

    Josip Novaković

  4. Department of Infectious Diseases, General Hospital Zadar, Bože Peričića 5, 23 000, Zadar, Croatia

    Vedrana Terkeš

Authors
  1. Jure Pupić-Bakrač
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  2. Neven Skitarelić
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  3. Ana Pupić-Bakrač
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  4. Ivan Baraka
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  6. Josip Novaković
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Contributions

JPB: Conceptualization, Methodology, Investigation, Writing. APB: Investigation and writing. IB, MS, JN, VT-Resources. NS: Validation, supervision.

Corresponding author

Correspondence to Jure Pupić-Bakrač.

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None to declare.

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A retrospective analysis was conducted. Only archived patient data and samples processed for diagnostic purposes were used.

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Prior to the processing, informed consent was obtained from all individual participants for whom identifying information was included in this article.

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The authors assert that all procedures contributing to this work were approved and complied with the ethical standards of the relevant national and institutional committees on human experimentation and with the Helsinki Declaration of 1964, as revised in 2013.

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Electronic supplementary material

Supplement video file 1. Multi-slice computed tomography of the neck in the axial projection showing large branchial cleft cyst extending from hyoid to upper thoracic aperture (MP4 1198 KB)

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Pupić-Bakrač, J., Skitarelić, N., Pupić-Bakrač, A. et al. Branchial cleft anomalies: hybrid “Branchial Inclusion” theory. Eur Arch Otorhinolaryngol 278, 2593–2601 (2021). https://doi.org/10.1007/s00405-020-06551-1

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