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Bilateral nasolabial cyst
J Stomatol Oral Maxillofac Surg 118 (2017) 385–388
Available online at
ScienceDirect www.sciencedirect.com
Case Report
Bilateral nasolabial cyst S. Dghoughi Department of oral surgery, faculty of dentistry, university Mohamed V, Rabat, Morocco A B S T R A C T A R T I C L E I N F O
Historique de l’article : Received 1st May 2017 Accepted 6 July 2017 Available online 22 August 2017
Nasolabial cyst is an uncommon non-odontogenic extraosseous cyst located in the nasolabial fold. Bilateral nasolabial cysts are rarer and only few cases have been reported. We describe a case of two asymptomatic masses of the nasal ala areas, which prove to be nasolabial cysts in a 30-year-old Moroccan woman.
Keywords: Bilateral nasolabial cyst Non-odontogenic cyst Maxillary cyst
1. Introduction Nasolabial cyst (NLC) is a very rare developmental nonodontogenic cyst that arises in the maxillofacial soft tissue [1,2]. It is classically related to the nasolabial fold or nasal alar soft tissue [3] and may involve bone secondarily [4].
2. Observation
fold. The lesions measured 12 19 mm and 13 18 mm on the right and left sides (Fig. 3). In view of the above findings the diagnosis of bilateral nasoalveolar cyst was made. The cysts were enucleated using intraoral approach under local anesthesia. A full-thickness flap was reflected along the gingivolabial sulcus in the canine-to-canine region revealing two cystic swellings causing an erosion of the anterior part of the maxilla (Fig. 4). The incisions were closed primarily and postoperative recovery was uneventful.
A 30-year-old female patient with no medical history referred to our division, complaining of an asymptomatic fluctuant mass in both right and left nasal ala areas perceived a year before consultation. The masses caused an elevation of the upper lip (Fig. 1). The patient had previously sought a dentist at a private dental clinic who raised the possibility of an infectious cause. Antibiotics have been prescribed and an incision drainage attempt which did not help the lesion to regress. The lesion was asymptomatic, but the patient was concerned by the possible severity of the lesion and also the aesthetic discomfort. Intraoral examination revealed bilateral smooth fluctuant masses lateral to the central incisors in the maxillary labial vestibule. The mucosal surface was normal (Fig. 2). Central and lateral incisors were vital and had no evidence of any recent or past dental intervention. There was no history of any nasal discharge. Panoramic radiographs revealed no obvious radiolucency computed tomography of the face revealed two well-defined hyperdense lesions on either side of the nasal ala at the nasolabial
E-mail address: [email protected] http://dx.doi.org/10.1016/j.jormas.2017.07.007 C 2017 Elsevier Masson SAS. All rights reserved. 2468-7855/
Fig. 1. Extraoral view. Two round masses in both left and right areas of the ala of the nose causing a disappearance of the nasolabial fold and en elevation of the upper lip.
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S. Dghoughi / J Stomatol Oral Maxillofac Surg 118 (2017) 385–388
Fig. 2. Intraoral view. Bilateral smooth fluctuant masses lateral to the central incisors in the maxillary labial vestibule covered by normal mucosa.
Fig. 5. Microscopic examination of the surgical specimen. The cyst is lined by pseudo-stratified squamous epithelium enclosing mucous secreting goblet cells. Capsule showed fibrous tissue infiltrated with mild polymorphic chronic inflammatory infiltrate and congestive vessels. (HES 400 ).
The histology indicated a cyst lined by pseudo-stratified squamous epithelium enclosing many cilia and mucous secreting goblet cells. Capsule showed fibrous tissue infiltrated with mild polymorphic chronic inflammatory infiltrate and congestive vessels. There were no granulomas or any evidence of malignancy (Fig. 5). Diagnosis confirmed the presence of bilateral nasolabial cysts. The postoperative course was uneventful and improvement of the cosmetic aspect was noticed (Fig. 6). After a year of follow-up the lesion did not recur. 3. Discussion
Fig. 3. Axial section computed tomography (CT) in soft tissue window of the face
NLC was first described by Zuckerkandl in 1882 [1,5–7]. It is a rare soft tissue cyst that occurs as a swelling in the nasolabial fold at the base of the alae of the nose [6]. Its frequency is around 0.7% of cysts of the jaws [1,6,8] and 2.5% of the non-odontogenic cysts [1,5,6]. Many names have been used to describe this cyst such as Klestadt’s cyst, nasoalveolar cyst, nasal vestibular cyst, mucoid cyst of the nose, nasal wing cyst, maxillary cyst, subalar cyst and nasoglobular cyst [6,7]. Currently, the term ‘‘nasolabial cyst’’ is more commonly used. The mean age of patients is in the fourth or
Fig. 4. Intraoperative view. Reflection of a full-thickness flap along the gingivolabial sulcus revealing two cystic lesions with erosion of the anterior part of the maxilla.
S. Dghoughi / J Stomatol Oral Maxillofac Surg 118 (2017) 385–388
Fig. 6. Extraoral view. Three weeks after surgery, an improvement of the esthetic aspect is noted. The teeth are covered by the upper lip at rest position compared to Fig. 1.
fifth decade [1,3,5–8] with a significantly higher ratio of women (4:1) [1–3,5]. It is usually unilateral, with no prevalence of side occurrence but bilateral cases have been also reported, though very rare [6,8]. It has been estimated that approximately 10 to 11% of the cases are bilateral [3,5–8]. Because of its rarity, it is often misdiagnosed and not treated properly. This is also shown in our case where an earlier misdiagnosis led to inappropriate treatment for several months [8]. This lesion occurs more commonly in Afro-Americans [6,8]. The origin of NLC has been controversial. In 1913, Klestadt suggested that the NLC arose from epithelium entrapped at the point where the maxillary, medial nasal, and lateral nasal processes fuse. From his concept came the term fissural cyst [1,3,6,7]. This theory has been abandoned because embryologic evidence shows that epithelium does not exist between these processes [2,8] and because those cysts previously labeled as globulomaxillary cysts were actually found to be cysts and tumors of other origins [2]. The most accepted theory is the one raised by Bruggemann in 1920, which suggests that the NLC arises from the epithelial rests of the lower anterior part of the nasolacrimal duct [6,9] due to histological similarities. The lining of the cyst is a pseudo-stratified columnar epithelium similar to the lining of the nasolacrimal duct. Though these cysts are referred to as developmental cysts [1–4,6,7]. The NLC presents as a painless round mass within the soft tissue adjacent to the ala of the nose around the uppermost portion of the nasolabial crease [1,2,4]. The mass is compressible suggesting a cyst. The overlying skin is normal without area of drainage [2]. The cyst may enlarge towards the nasolabial fold, the vestibule of the nose and the labioalveolar sulcus. This may result in a disappearance of the nasolabial fold, an elevation of the ala of the nose [6,10], a swelling of the floor of the nasal cavity often bulging through the mucosa in the buccal sulcus in the region of the lateral upper incisor or canine [10]. The cyst is asymptomatic unless nasal obstruction, infection or deformity occur [3,6–8,10]. In our case the cyst was asymptomatic, but the cosmetic defect pushed the patient to seek medical care. The cyst may interfere with the upper denture by its growth eroding the maxillary alveolus and displacing the incisor teeth [6,10]. Variations in size can occur and could be explained by the observation that it can spontaneously drain into the nose or mouth, [3,5,6,10]. Cysts may cause bulging on the lateral wall of the nasal fossa floor, which becomes evident on occlusal maxillary radiographs. Osteolytic lesions, when present, may eventually involve the maxillary sinus [7].
387
Since the NLC is a soft tissue cyst, panoramic and intraoral radiographs usually reveal no abnormalities except when it causes significant maxillary bone erosion [4,7,8,10]. In that case the cyst being external to the alveolar bone, produces ‘‘cupping’’ of the underlying cortical plate [4]. Computed tomography and magnetic resonance imaging, may reveal the cystic nature of these lesions in greater detail and reliability, their relation with the nasal alae and the maxillary bone, as well as bone involvement, which facilitate the diagnosis [1,7,8]. The most common differential diagnosis is a canine space abscess from an odontogenic infection, which can easily be ruled out by vitality testing of the maxillary teeth of the affected area [2,5]. Periapical, dentigerous, nasopalatine duct cysts can be excluded on account of the radiographic findings, these are typically osseous lesions [1,3,4,10]. Epidermoid cysts or implanted epidermal inclusion cysts clinically and histologically resemble the nasolabial cyst [2,5], a thorough histopathologic review of the epithelial lining is necessary to distinguish them. In the NLC, the majority of its lining is pseudo-stratified columnar cells rather than stratified squamous cells as are found in the other two [2]. Benign or malignant salivary gland tumours can usually be excluded on the strength of the clinical and radiological features, such as their longstanding nature and in very limited bone involvement [5]. Other uncommon differentials include nasolacrimal mucocele, which occurs secondary to non-canalization of the nasolacrimal duct. It is commonly seen in infants and they present with epiphora and dacryocystitis [3]. Grossly, when resected in toto, the nasolabial cysts are soft to firm soft tissue mass with a smooth surface. Contents are variably cystic with clear fluid, hemorrhage, or purulent material if infected. The commonest type of epithelium encountered is pseudostratified columnar, followed by combined columnar, cuboidal and columnar, and stratified squamous epithelium. Intraepithelial goblet cells are a consistent feature. It is seen in up to 52% cases [3]. The cyst wall stroma is a hypocellular, collagen-rich fibrovascular tissue with or without chronic inflammatory cells in the subepithelial region [3,8]. Several treatment modalities have been described in the management of the nasolabial cyst, for example surgical excision, endoscopic marsupialization, simple aspiration, incision and drainage, injection of sclerotic agents and marsupialization. With the exception of complete surgical excision and endoscopic marsupialization, all other treatments are associated with a high recurrence rate [5]. Therefore, a surgical excision via an intraoral approach is the treatment of choice. The sublabial approach is the most popular and well-established procedure with a wider surgical field and more assurance of a complete excision. The transnasal endoscopic marsupialization seems to be a simple and effective alternative [1]. It is less invasive and easier to perform in large lesions [1,3]. However, It has been shown that after successful marsupialization the nasolabial cyst is converted to an air-containing paranasal sinus [1,5]. Because this cyst is usually closely related to the floor of the nose perforation of the nasal mucosa may be expected during its removal. When very small perforations are caused, they can be left untreated, however, larger ones must be sutured [6,10]. After excision, recurrence does not develop [2]. This paper presents a case of a bilateral NLC, which was misdiagnosed for several months. Its features must be known by all practitioners to avoid useless treatments as it is usually mixed up with a periapical infection. References [1] Chandrasekharan R, Varghese AM, Mathew J, Ashish G. A rare case of bilateral nasolabial cysts in a postpartum lady. Indian J Dent Res 2014;25:225–7. [2] Marx RE, Stern D. Oral and maxillofacial pathology: a rationale for diagnosis and treatment, 2nd ed., Illinois: Quintessence Publishing; 2012.
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S. Dghoughi / J Stomatol Oral Maxillofac Surg 118 (2017) 385–388
[3] Patil AR, Singh AP, Nandikoor S, Meganathan P. Bilateral nasolabial cysts – case report and review of literature. Indian J Radiol Imaging 2016;26:241–4. [4] Parwani R, Parwani S, Wanjari S. Diagnosis and management of bilateral nasolabial cysts. J Oral Maxillofac Pathol 2013;17:443–6. [5] Marcoviceanu MP, Metzger MC, Deppe H, Freudenberg N, Kassem A, Pautke C, et al. Report of rare bilateral nasolabial cysts. J Craniomaxillofac Surg 2009;37:83–6. [6] Venkatesh VK, Satelur K, Yerlagudda K. Nasolabial cysts—report of four cases including two bilateral occurrences and review of literature. Indian J Dent 2011;2:156–9.
[7] Aquilino RN, Bazzo VJ, Faria RJA, Eid NLM, Bo´scolo FN. Nasolabial cyst: presentation of a clinical case with CT and MR images. Braz J Otorhinolaryngol 2008;74:467–71. [8] Sato M, Morita K, Kabasawa Y, Harada H. Bilateral nasolabial cysts: a case report. J Med Case Rep 2016;10:246. [9] Wesley RK, Scannell T, Nathan LE. Nasolabial cyst: presentation of a case with a review of the literature. J Oral Maxillofac Surg 1984;42:188–92. [10] Allard RHB. Nasolabial cyst. Review of the literature and report of 7 cases. Int J Oral Surg 1982;11:351–9.
Available online at
ScienceDirect www.sciencedirect.com
Case Report
Bilateral nasolabial cyst S. Dghoughi Department of oral surgery, faculty of dentistry, university Mohamed V, Rabat, Morocco A B S T R A C T A R T I C L E I N F O
Historique de l’article : Received 1st May 2017 Accepted 6 July 2017 Available online 22 August 2017
Nasolabial cyst is an uncommon non-odontogenic extraosseous cyst located in the nasolabial fold. Bilateral nasolabial cysts are rarer and only few cases have been reported. We describe a case of two asymptomatic masses of the nasal ala areas, which prove to be nasolabial cysts in a 30-year-old Moroccan woman.
Keywords: Bilateral nasolabial cyst Non-odontogenic cyst Maxillary cyst
1. Introduction Nasolabial cyst (NLC) is a very rare developmental nonodontogenic cyst that arises in the maxillofacial soft tissue [1,2]. It is classically related to the nasolabial fold or nasal alar soft tissue [3] and may involve bone secondarily [4].
2. Observation
fold. The lesions measured 12 19 mm and 13 18 mm on the right and left sides (Fig. 3). In view of the above findings the diagnosis of bilateral nasoalveolar cyst was made. The cysts were enucleated using intraoral approach under local anesthesia. A full-thickness flap was reflected along the gingivolabial sulcus in the canine-to-canine region revealing two cystic swellings causing an erosion of the anterior part of the maxilla (Fig. 4). The incisions were closed primarily and postoperative recovery was uneventful.
A 30-year-old female patient with no medical history referred to our division, complaining of an asymptomatic fluctuant mass in both right and left nasal ala areas perceived a year before consultation. The masses caused an elevation of the upper lip (Fig. 1). The patient had previously sought a dentist at a private dental clinic who raised the possibility of an infectious cause. Antibiotics have been prescribed and an incision drainage attempt which did not help the lesion to regress. The lesion was asymptomatic, but the patient was concerned by the possible severity of the lesion and also the aesthetic discomfort. Intraoral examination revealed bilateral smooth fluctuant masses lateral to the central incisors in the maxillary labial vestibule. The mucosal surface was normal (Fig. 2). Central and lateral incisors were vital and had no evidence of any recent or past dental intervention. There was no history of any nasal discharge. Panoramic radiographs revealed no obvious radiolucency computed tomography of the face revealed two well-defined hyperdense lesions on either side of the nasal ala at the nasolabial
E-mail address: [email protected] http://dx.doi.org/10.1016/j.jormas.2017.07.007 C 2017 Elsevier Masson SAS. All rights reserved. 2468-7855/
Fig. 1. Extraoral view. Two round masses in both left and right areas of the ala of the nose causing a disappearance of the nasolabial fold and en elevation of the upper lip.
386
S. Dghoughi / J Stomatol Oral Maxillofac Surg 118 (2017) 385–388
Fig. 2. Intraoral view. Bilateral smooth fluctuant masses lateral to the central incisors in the maxillary labial vestibule covered by normal mucosa.
Fig. 5. Microscopic examination of the surgical specimen. The cyst is lined by pseudo-stratified squamous epithelium enclosing mucous secreting goblet cells. Capsule showed fibrous tissue infiltrated with mild polymorphic chronic inflammatory infiltrate and congestive vessels. (HES 400 ).
The histology indicated a cyst lined by pseudo-stratified squamous epithelium enclosing many cilia and mucous secreting goblet cells. Capsule showed fibrous tissue infiltrated with mild polymorphic chronic inflammatory infiltrate and congestive vessels. There were no granulomas or any evidence of malignancy (Fig. 5). Diagnosis confirmed the presence of bilateral nasolabial cysts. The postoperative course was uneventful and improvement of the cosmetic aspect was noticed (Fig. 6). After a year of follow-up the lesion did not recur. 3. Discussion
Fig. 3. Axial section computed tomography (CT) in soft tissue window of the face
NLC was first described by Zuckerkandl in 1882 [1,5–7]. It is a rare soft tissue cyst that occurs as a swelling in the nasolabial fold at the base of the alae of the nose [6]. Its frequency is around 0.7% of cysts of the jaws [1,6,8] and 2.5% of the non-odontogenic cysts [1,5,6]. Many names have been used to describe this cyst such as Klestadt’s cyst, nasoalveolar cyst, nasal vestibular cyst, mucoid cyst of the nose, nasal wing cyst, maxillary cyst, subalar cyst and nasoglobular cyst [6,7]. Currently, the term ‘‘nasolabial cyst’’ is more commonly used. The mean age of patients is in the fourth or
Fig. 4. Intraoperative view. Reflection of a full-thickness flap along the gingivolabial sulcus revealing two cystic lesions with erosion of the anterior part of the maxilla.
S. Dghoughi / J Stomatol Oral Maxillofac Surg 118 (2017) 385–388
Fig. 6. Extraoral view. Three weeks after surgery, an improvement of the esthetic aspect is noted. The teeth are covered by the upper lip at rest position compared to Fig. 1.
fifth decade [1,3,5–8] with a significantly higher ratio of women (4:1) [1–3,5]. It is usually unilateral, with no prevalence of side occurrence but bilateral cases have been also reported, though very rare [6,8]. It has been estimated that approximately 10 to 11% of the cases are bilateral [3,5–8]. Because of its rarity, it is often misdiagnosed and not treated properly. This is also shown in our case where an earlier misdiagnosis led to inappropriate treatment for several months [8]. This lesion occurs more commonly in Afro-Americans [6,8]. The origin of NLC has been controversial. In 1913, Klestadt suggested that the NLC arose from epithelium entrapped at the point where the maxillary, medial nasal, and lateral nasal processes fuse. From his concept came the term fissural cyst [1,3,6,7]. This theory has been abandoned because embryologic evidence shows that epithelium does not exist between these processes [2,8] and because those cysts previously labeled as globulomaxillary cysts were actually found to be cysts and tumors of other origins [2]. The most accepted theory is the one raised by Bruggemann in 1920, which suggests that the NLC arises from the epithelial rests of the lower anterior part of the nasolacrimal duct [6,9] due to histological similarities. The lining of the cyst is a pseudo-stratified columnar epithelium similar to the lining of the nasolacrimal duct. Though these cysts are referred to as developmental cysts [1–4,6,7]. The NLC presents as a painless round mass within the soft tissue adjacent to the ala of the nose around the uppermost portion of the nasolabial crease [1,2,4]. The mass is compressible suggesting a cyst. The overlying skin is normal without area of drainage [2]. The cyst may enlarge towards the nasolabial fold, the vestibule of the nose and the labioalveolar sulcus. This may result in a disappearance of the nasolabial fold, an elevation of the ala of the nose [6,10], a swelling of the floor of the nasal cavity often bulging through the mucosa in the buccal sulcus in the region of the lateral upper incisor or canine [10]. The cyst is asymptomatic unless nasal obstruction, infection or deformity occur [3,6–8,10]. In our case the cyst was asymptomatic, but the cosmetic defect pushed the patient to seek medical care. The cyst may interfere with the upper denture by its growth eroding the maxillary alveolus and displacing the incisor teeth [6,10]. Variations in size can occur and could be explained by the observation that it can spontaneously drain into the nose or mouth, [3,5,6,10]. Cysts may cause bulging on the lateral wall of the nasal fossa floor, which becomes evident on occlusal maxillary radiographs. Osteolytic lesions, when present, may eventually involve the maxillary sinus [7].
387
Since the NLC is a soft tissue cyst, panoramic and intraoral radiographs usually reveal no abnormalities except when it causes significant maxillary bone erosion [4,7,8,10]. In that case the cyst being external to the alveolar bone, produces ‘‘cupping’’ of the underlying cortical plate [4]. Computed tomography and magnetic resonance imaging, may reveal the cystic nature of these lesions in greater detail and reliability, their relation with the nasal alae and the maxillary bone, as well as bone involvement, which facilitate the diagnosis [1,7,8]. The most common differential diagnosis is a canine space abscess from an odontogenic infection, which can easily be ruled out by vitality testing of the maxillary teeth of the affected area [2,5]. Periapical, dentigerous, nasopalatine duct cysts can be excluded on account of the radiographic findings, these are typically osseous lesions [1,3,4,10]. Epidermoid cysts or implanted epidermal inclusion cysts clinically and histologically resemble the nasolabial cyst [2,5], a thorough histopathologic review of the epithelial lining is necessary to distinguish them. In the NLC, the majority of its lining is pseudo-stratified columnar cells rather than stratified squamous cells as are found in the other two [2]. Benign or malignant salivary gland tumours can usually be excluded on the strength of the clinical and radiological features, such as their longstanding nature and in very limited bone involvement [5]. Other uncommon differentials include nasolacrimal mucocele, which occurs secondary to non-canalization of the nasolacrimal duct. It is commonly seen in infants and they present with epiphora and dacryocystitis [3]. Grossly, when resected in toto, the nasolabial cysts are soft to firm soft tissue mass with a smooth surface. Contents are variably cystic with clear fluid, hemorrhage, or purulent material if infected. The commonest type of epithelium encountered is pseudostratified columnar, followed by combined columnar, cuboidal and columnar, and stratified squamous epithelium. Intraepithelial goblet cells are a consistent feature. It is seen in up to 52% cases [3]. The cyst wall stroma is a hypocellular, collagen-rich fibrovascular tissue with or without chronic inflammatory cells in the subepithelial region [3,8]. Several treatment modalities have been described in the management of the nasolabial cyst, for example surgical excision, endoscopic marsupialization, simple aspiration, incision and drainage, injection of sclerotic agents and marsupialization. With the exception of complete surgical excision and endoscopic marsupialization, all other treatments are associated with a high recurrence rate [5]. Therefore, a surgical excision via an intraoral approach is the treatment of choice. The sublabial approach is the most popular and well-established procedure with a wider surgical field and more assurance of a complete excision. The transnasal endoscopic marsupialization seems to be a simple and effective alternative [1]. It is less invasive and easier to perform in large lesions [1,3]. However, It has been shown that after successful marsupialization the nasolabial cyst is converted to an air-containing paranasal sinus [1,5]. Because this cyst is usually closely related to the floor of the nose perforation of the nasal mucosa may be expected during its removal. When very small perforations are caused, they can be left untreated, however, larger ones must be sutured [6,10]. After excision, recurrence does not develop [2]. This paper presents a case of a bilateral NLC, which was misdiagnosed for several months. Its features must be known by all practitioners to avoid useless treatments as it is usually mixed up with a periapical infection. References [1] Chandrasekharan R, Varghese AM, Mathew J, Ashish G. A rare case of bilateral nasolabial cysts in a postpartum lady. Indian J Dent Res 2014;25:225–7. [2] Marx RE, Stern D. Oral and maxillofacial pathology: a rationale for diagnosis and treatment, 2nd ed., Illinois: Quintessence Publishing; 2012.
388
S. Dghoughi / J Stomatol Oral Maxillofac Surg 118 (2017) 385–388
[3] Patil AR, Singh AP, Nandikoor S, Meganathan P. Bilateral nasolabial cysts – case report and review of literature. Indian J Radiol Imaging 2016;26:241–4. [4] Parwani R, Parwani S, Wanjari S. Diagnosis and management of bilateral nasolabial cysts. J Oral Maxillofac Pathol 2013;17:443–6. [5] Marcoviceanu MP, Metzger MC, Deppe H, Freudenberg N, Kassem A, Pautke C, et al. Report of rare bilateral nasolabial cysts. J Craniomaxillofac Surg 2009;37:83–6. [6] Venkatesh VK, Satelur K, Yerlagudda K. Nasolabial cysts—report of four cases including two bilateral occurrences and review of literature. Indian J Dent 2011;2:156–9.
[7] Aquilino RN, Bazzo VJ, Faria RJA, Eid NLM, Bo´scolo FN. Nasolabial cyst: presentation of a clinical case with CT and MR images. Braz J Otorhinolaryngol 2008;74:467–71. [8] Sato M, Morita K, Kabasawa Y, Harada H. Bilateral nasolabial cysts: a case report. J Med Case Rep 2016;10:246. [9] Wesley RK, Scannell T, Nathan LE. Nasolabial cyst: presentation of a case with a review of the literature. J Oral Maxillofac Surg 1984;42:188–92. [10] Allard RHB. Nasolabial cyst. Review of the literature and report of 7 cases. Int J Oral Surg 1982;11:351–9.