Available online at www.sciencedirect.com
British Journal of Oral and Maxillofacial Surgery 52 (2014) 379–381
Short communication
Spontaneous resolution of a branchial cleft cyst without intervention: a previously unreported outcome Peter A. Brennan ∗ , Angela Critchlow, Sara Rehman, Deepak Thomas Maxillofacial Unit, Queen Alexandra Hospital, Portsmouth PO6 3LY, UK Accepted 5 February 2014 Available online 7 March 2014
Abstract Branchial cleft cysts typically present as a persistent mass in the anterior triangle of the neck. Diagnosis is made by imaging and fine needle aspiration cytology and is confirmed histologically after excision. We report a branchial cleft cyst in a 27-year-old woman that resolved spontaneously over several weeks without surgical intervention. To the best of our knowledge this is the first report of such a case. © 2014 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Branchial cleft cyst; Spontaneous regression; Ultrasound; Management
Introduction Branchial cleft cysts are epithelial lined cysts that arise as a result of incomplete obliteration of the branchial apparatus, and are the most common cystic lesion to occur in the neck.1–4 Anomalies of the second branchial cleft are the most common and can include fistulas, sinuses, or cysts. Lesions of the third and fourth cleft are comparatively rare.1–4 Although they can occur at any age, branchial cleft cysts present most often in adolescents and young adults. They have no sex predilection and are rarely found bilaterally.5 Diagnosis is made with computed tomography (CT), magnetic resonance imaging (MRI), or ultrasound, and usually with ultrasound-guided fine needle aspiration cytology (FNAC).6,7 A necrotic or cystic metastatic node should always be considered in the differential diagnosis, and rarely, carcinoma can also arise in the wall of the cyst. Prompt management is therefore necessary. These cysts do not resolve spontaneously, and unless surgically excised may become infected and result in the formation of a sinus. We present a case of spontaneous
∗
Corresponding author. Tel.: +44 2393 286099; fax: +44 2292 286089. E-mail address: [email protected] (P.A. Brennan).
resolution of a branchial cleft cyst, which occurred after a delay in the planned operation because of acute cholecystitis.
Case report A 27-year-old woman presented with a 5-week history of an asymptomatic soft mass in the right upper neck. Medically she was otherwise well. She was an ex-smoker, but had stopped several years previously, and had given birth to her first child 3 months earlier. Physical examination found a soft 3 cm mass anterior to the sternocleidomastoid in the right level II region. Intraoral examination and flexible nasendoscopy were normal, and no fluid was aspirated on FNAC. Cytological examination showed squamous epithelial cells with no atypia, features that are suggestive of a branchial cleft cyst (Fig. 1). CT 10 days later showed a non-specific cystic mass measuring 2.9 cm (Fig. 2). The wall of varying thickness suggested that it might not be a simple cyst and that a metastatic necrotic node should also be considered. An examination under anaesthesia with panendoscopy was planned to exclude a primary tumour, followed by excision of the mass. The operation had to be deferred as the patient developed acute cholecystitis. However, on review 3 months later, the mass was barely palpable. Ultrasound showed 3 small
0266-4356/$ – see front matter © 2014 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
http://dx.doi.org/10.1016/j.bjoms.2014.02.004
380
P.A. Brennan et al. / British Journal of Oral and Maxillofacial Surgery 52 (2014) 379–381
Fig. 1. Cytology slide showing squamous epithelial cells with no atypia consistent with a branchial cleft cyst (Giemsa, original magnification ×200).
reactive nodes measuring up to 15 mm in long axis in level II, all with normal hilar and Doppler signals. There was no evidence of the cyst (Fig. 3A) or the tract from which it might have arisen. Clinical review and ultrasound 6 months later (9 months after initial presentation) once again confirmed no evidence of a cyst or tract, and only a few small reactive lymph nodes (Fig. 3B).
Discussion This interesting case shows spontaneous resolution of a presumed branchial cleft cyst, which to our knowledge has not been reported before. It is difficult to speculate how it occurred, but as no fluid was obtained on FNAC and the lesion did not reduce in size afterwards (confirmed by CT 10
Fig. 2. Computed tomogram showing suspected branchial cleft cyst (arrow) in the right upper neck with irregular thickening of the wall.
Fig. 3. Ultrasound scans of the right neck showing no evidence of a branchial cleft cyst and some reactive lymph nodes 3 months (A), and 9 months (B) after initial presentation.
days later), it is unlikely that the contents leaked out and that the cyst subsequently collapsed. Those that do collapse on FNAC usually refill again quite quickly. One possibility might be that the cyst enlarged after childbirth, perhaps as a result of the hormonal changes that occur after pregnancy. However, we have not found any similar cases or any to confirm that branchial cysts are affected by hormonal changes. It is possible that the presumed tract from which the cyst had arisen was still present although it was not seen on serial ultrasound scans. Another possibility is the lymph node inclusion theory, which suggests that it arose from the cystic alteration of epithelium trapped in the cervical lymph nodes.8 However, as the lesion was not excised and resolved spontaneously, it is not possible to be certain about its pathogenesis. Since the differential diagnosis includes metastatic carcinoma of different origins as well as carcinoma arising within the wall of a branchial cleft cyst,9 excision is recommended. Our patient had close follow-up with serial ultrasound scans. Recently, sclerotherapy has been reported as a successful treatment.10
P.A. Brennan et al. / British Journal of Oral and Maxillofacial Surgery 52 (2014) 379–381
Regardless of the pathogenesis, the reason for the unusual resolution in our case is unknown and it would be interesting to learn if colleagues have had any similar cases.
Acknowledgement We are grateful to Dr AV Spedding, Consultant Histopathologist for her help with this case.
References 1. Woo EK, Connor SE. Computed tomography and magnetic resonance imaging appearances of cystic lesions in the suprahyoid neck: a pictorial review. Dentomaxillofac Radiol 2007;36:451–8. 2. Mossa-Basha M, Yousem DM. Congenital cystic lesions of the neck. Appl Radiol 2013;42:8–22.
381
3. Saki N, Rahim F, Nickakhlagh S, et al. Anatomical variation of branchial clefts: a retrospective study. J Med Sci 2009;9:108–12 http://scialert.net/abstract/?doi=jms.2009.108.112 4. Flanagan PM, Roland NJ, Jones AS. Cervical node metastases presenting with features of branchial cysts. J Laryngol Otol 1994;108:1068–71. 5. Munoz-Fernández N, Mallea-Canizares I, Fernández-Julián E, et al. Double second branchial cleft anomaly. Acta Otorrinolaringol Esp 2011;62:68–70 (in Spanish). 6. Papadogeorgakis N, Petsinis V, Parara E, et al. Branchial cleft cysts in adults. Diagnostic procedures and treatment in a series of 18 cases. Oral Maxillofac Surg 2009;13:79–85. 7. Coppens F, Peene P, Lemahieu SF. Diagnosis and differential diagnosis of branchial cleft cysts by CT scan. J Belge Radiol 1990;73: 189–96. 8. Upile T, Jerjes W, Al-Khawalde M, et al. Branchial cysts within the parotid salivary gland. Head Neck Oncol 2012;4:24. 9. Saito T, Sato T, Usui H, et al. A case of squamous cell carcinoma arising from branchial cleft cyst. Oral Sci Int 2008;5:135–40. 10. Kim MG, Lee NH, Ban JH, et al. Sclerotherapy of branchial cleft cysts using OK-432. Otolaryngol Head Neck Surg 2009;141:329–34.
British Journal of Oral and Maxillofacial Surgery 52 (2014) 379–381
Short communication
Spontaneous resolution of a branchial cleft cyst without intervention: a previously unreported outcome Peter A. Brennan ∗ , Angela Critchlow, Sara Rehman, Deepak Thomas Maxillofacial Unit, Queen Alexandra Hospital, Portsmouth PO6 3LY, UK Accepted 5 February 2014 Available online 7 March 2014
Abstract Branchial cleft cysts typically present as a persistent mass in the anterior triangle of the neck. Diagnosis is made by imaging and fine needle aspiration cytology and is confirmed histologically after excision. We report a branchial cleft cyst in a 27-year-old woman that resolved spontaneously over several weeks without surgical intervention. To the best of our knowledge this is the first report of such a case. © 2014 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Branchial cleft cyst; Spontaneous regression; Ultrasound; Management
Introduction Branchial cleft cysts are epithelial lined cysts that arise as a result of incomplete obliteration of the branchial apparatus, and are the most common cystic lesion to occur in the neck.1–4 Anomalies of the second branchial cleft are the most common and can include fistulas, sinuses, or cysts. Lesions of the third and fourth cleft are comparatively rare.1–4 Although they can occur at any age, branchial cleft cysts present most often in adolescents and young adults. They have no sex predilection and are rarely found bilaterally.5 Diagnosis is made with computed tomography (CT), magnetic resonance imaging (MRI), or ultrasound, and usually with ultrasound-guided fine needle aspiration cytology (FNAC).6,7 A necrotic or cystic metastatic node should always be considered in the differential diagnosis, and rarely, carcinoma can also arise in the wall of the cyst. Prompt management is therefore necessary. These cysts do not resolve spontaneously, and unless surgically excised may become infected and result in the formation of a sinus. We present a case of spontaneous
∗
Corresponding author. Tel.: +44 2393 286099; fax: +44 2292 286089. E-mail address: [email protected] (P.A. Brennan).
resolution of a branchial cleft cyst, which occurred after a delay in the planned operation because of acute cholecystitis.
Case report A 27-year-old woman presented with a 5-week history of an asymptomatic soft mass in the right upper neck. Medically she was otherwise well. She was an ex-smoker, but had stopped several years previously, and had given birth to her first child 3 months earlier. Physical examination found a soft 3 cm mass anterior to the sternocleidomastoid in the right level II region. Intraoral examination and flexible nasendoscopy were normal, and no fluid was aspirated on FNAC. Cytological examination showed squamous epithelial cells with no atypia, features that are suggestive of a branchial cleft cyst (Fig. 1). CT 10 days later showed a non-specific cystic mass measuring 2.9 cm (Fig. 2). The wall of varying thickness suggested that it might not be a simple cyst and that a metastatic necrotic node should also be considered. An examination under anaesthesia with panendoscopy was planned to exclude a primary tumour, followed by excision of the mass. The operation had to be deferred as the patient developed acute cholecystitis. However, on review 3 months later, the mass was barely palpable. Ultrasound showed 3 small
0266-4356/$ – see front matter © 2014 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
http://dx.doi.org/10.1016/j.bjoms.2014.02.004
380
P.A. Brennan et al. / British Journal of Oral and Maxillofacial Surgery 52 (2014) 379–381
Fig. 1. Cytology slide showing squamous epithelial cells with no atypia consistent with a branchial cleft cyst (Giemsa, original magnification ×200).
reactive nodes measuring up to 15 mm in long axis in level II, all with normal hilar and Doppler signals. There was no evidence of the cyst (Fig. 3A) or the tract from which it might have arisen. Clinical review and ultrasound 6 months later (9 months after initial presentation) once again confirmed no evidence of a cyst or tract, and only a few small reactive lymph nodes (Fig. 3B).
Discussion This interesting case shows spontaneous resolution of a presumed branchial cleft cyst, which to our knowledge has not been reported before. It is difficult to speculate how it occurred, but as no fluid was obtained on FNAC and the lesion did not reduce in size afterwards (confirmed by CT 10
Fig. 2. Computed tomogram showing suspected branchial cleft cyst (arrow) in the right upper neck with irregular thickening of the wall.
Fig. 3. Ultrasound scans of the right neck showing no evidence of a branchial cleft cyst and some reactive lymph nodes 3 months (A), and 9 months (B) after initial presentation.
days later), it is unlikely that the contents leaked out and that the cyst subsequently collapsed. Those that do collapse on FNAC usually refill again quite quickly. One possibility might be that the cyst enlarged after childbirth, perhaps as a result of the hormonal changes that occur after pregnancy. However, we have not found any similar cases or any to confirm that branchial cysts are affected by hormonal changes. It is possible that the presumed tract from which the cyst had arisen was still present although it was not seen on serial ultrasound scans. Another possibility is the lymph node inclusion theory, which suggests that it arose from the cystic alteration of epithelium trapped in the cervical lymph nodes.8 However, as the lesion was not excised and resolved spontaneously, it is not possible to be certain about its pathogenesis. Since the differential diagnosis includes metastatic carcinoma of different origins as well as carcinoma arising within the wall of a branchial cleft cyst,9 excision is recommended. Our patient had close follow-up with serial ultrasound scans. Recently, sclerotherapy has been reported as a successful treatment.10
P.A. Brennan et al. / British Journal of Oral and Maxillofacial Surgery 52 (2014) 379–381
Regardless of the pathogenesis, the reason for the unusual resolution in our case is unknown and it would be interesting to learn if colleagues have had any similar cases.
Acknowledgement We are grateful to Dr AV Spedding, Consultant Histopathologist for her help with this case.
References 1. Woo EK, Connor SE. Computed tomography and magnetic resonance imaging appearances of cystic lesions in the suprahyoid neck: a pictorial review. Dentomaxillofac Radiol 2007;36:451–8. 2. Mossa-Basha M, Yousem DM. Congenital cystic lesions of the neck. Appl Radiol 2013;42:8–22.
381
3. Saki N, Rahim F, Nickakhlagh S, et al. Anatomical variation of branchial clefts: a retrospective study. J Med Sci 2009;9:108–12 http://scialert.net/abstract/?doi=jms.2009.108.112 4. Flanagan PM, Roland NJ, Jones AS. Cervical node metastases presenting with features of branchial cysts. J Laryngol Otol 1994;108:1068–71. 5. Munoz-Fernández N, Mallea-Canizares I, Fernández-Julián E, et al. Double second branchial cleft anomaly. Acta Otorrinolaringol Esp 2011;62:68–70 (in Spanish). 6. Papadogeorgakis N, Petsinis V, Parara E, et al. Branchial cleft cysts in adults. Diagnostic procedures and treatment in a series of 18 cases. Oral Maxillofac Surg 2009;13:79–85. 7. Coppens F, Peene P, Lemahieu SF. Diagnosis and differential diagnosis of branchial cleft cysts by CT scan. J Belge Radiol 1990;73: 189–96. 8. Upile T, Jerjes W, Al-Khawalde M, et al. Branchial cysts within the parotid salivary gland. Head Neck Oncol 2012;4:24. 9. Saito T, Sato T, Usui H, et al. A case of squamous cell carcinoma arising from branchial cleft cyst. Oral Sci Int 2008;5:135–40. 10. Kim MG, Lee NH, Ban JH, et al. Sclerotherapy of branchial cleft cysts using OK-432. Otolaryngol Head Neck Surg 2009;141:329–34.
