Received: 18 April 2013 Accepted: 25 September 2013 Disponible en ligne 9 January 2014
Available online at
ScienceDirect www.sciencedirect.com
Case report
Ganglion cyst of the temporomandibular joint Ganglion kystique de l’articulation temporomandibulaire W. Heng-Kuna,*, G. Yan-Lingb, Z. Wen-Fengc, S. Zhec, W. Ren-Xina, Z. Xiao-Taoa a
Department of oral and maxillofacial surgery, Weihai municipal hospital, Weihai 264200, Shandong province, PR China b Department of stomatology, Weihai women and children hospital, Weihai 264200, Shandong province, PR China c Department of oro-maxillofacial head and neck surgery, hospital of stomatology, Wuhan university, Wuhan 430079, Hubei Province, China
Summary Introduction. Ganglion cyst of the temporomandibular joint is a rare disease, which may arise from myxoid degeneration of the collagenous tissue of the temporomandibular joint capsule, without epithelial or endothelial lining. We report a case of cystic lesion in a 40-year-old female patient. Observation. The patient had a left pre-auricular oval-shaped swelling without any articular symptoms. The pathological analysis after surgical removal allowed diagnosing the lesion as a ganglion cyst of the left temporomandibular joint. Discussion. We made a literature review and noted that this condition was predominant in female patients. We recommend using MRI for diagnostic purposes and surgery as the best therapeutic alternative. ß 2013 Elsevier Masson SAS. All rights reserved.
Re´sume´ Introduction. Le ganglion kystique de l’articulation temporomandibulaire est une maladie rare, qui peut re´sulter de la de´ge´ne´rescence myxoı¨de du tissu collage`ne de la capsule de l’articulation sans reveˆtement e´pithe´lial ou endothe´lial. Nous en rapportons un cas chez une femme de 40 ans. Observation. La patiente avait une tume´faction ovalaire pre´auriculaire gauche, sans symptomatologie articulaire. Le diagnostic anatomopathologique apre`s exe´re`se chirurgicale a e´te´ celui d’un ganglion kystique de l’articulation temporomandibulaire gauche. Discussion. Cette pathologie rare pre´domine chez la femme. Il n’y a pas de consensus sur les explorations a` pre´voir et sur le traitement. Nous recommandons l’IRM et proˆnons la chirurgie comme meilleure alternative the´rapeutique. ß 2013 Elsevier Masson SAS. Tous droits re´serve´s.
Keywords: Temporomandibular joint, Ganglion cyst
Mots cle´s : Articulation temporomandibulaire, Ganglion kystique
Introduction
occurring in the temporomandibular joint (TMJ) are rare. Twenty-seven cases of ganglion cysts in TMJ have been reported in the English literature [1–8] (listed in table I). We report a new case of and describe its clinic-pathological features.
A ganglion cyst is a benign cystic lesion that is closely associated with joints and tendon sheaths. This lesion is commonly encountered subcutaneously along the extensor surface of the wrist and ankle joints. However, ganglion cysts
Case report * Corresponding author. E-mail addresses: [email protected], [email protected] (W. Heng-Kun).
A 40-year-old female patient consulted with a chief complaint of swelling in the left pre-auricular region, first noticed
2213-6533/$ - see front matter ß 2013 Elsevier Masson SAS. All rights reserved. http://dx.doi.org/10.1016/j.revsto.2013.09.004 Rev Stomatol Chir Maxillofac Chir Orale 2013;115:62-64
62
Ganglion cyst of the temporomandibular joint
Table I Reported cases of temporomandibular joint (TMJ) ganglion cysts. Case
Authors
Age
Gender
Location
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28
Heydt (in 1977) Ethell (in 1979) Patel (in 1979) Kinkead (in 1981) Kenney (in 1987) Shiba (in 1987) Copeland (in 1988) Singer (in 1988) El-Massy (in 1989) Tom (in 1990) Hopper (in 1991) Lopes (in 1994) Chang (in 1997) Goudot (in 1999) Albright (in 2000) Nahlieli (in 2000) Takaku (in 2001) Kim (in 2003) Silva (in 2005) Junko (in 2006) Ali (in 2006) Shinawi (in 2007) Deng (in 2010) Khachi S (in 2011) Wu (in 2011) Yang (in 2011) Mumert (in 2012) Present case
47 28 45 36 44 28 60 ND 33 22 58 33 32 35 51 57 50 37 51 66 28 11 45 56 59 58 63 40
F F F F F M F ND F M F F F M F F F F F M F M F M F F F F 6M21F
L R R R L R L ND R R R L L R R L L L L L R L+R L L R R L L 15R13L
ND: no description; M: male; F: female; L: left; R: right.
3 months before. There was neither joint pain nor TMJ dysfunction. Her medical history was unremarkable, with no history of trauma in TMJ region. The mass was 2.0 cm 2.0 cm 1.0 cm in the left pre-auricular region. It had a smooth contour, was not mobile, was firm, and painless. The movement of the mandible was normal without any click or noise. There was no regional lymphadenopathy. The complete blood count and the differential leukocyte count were normal. A color Doppler revealed an 11 mm 6 mm cystic lesion, over the left parotid gland (fig. 1). The mass was excised under general anesthesia with a preauricular incision. The cystic lesion was adjacent to the left TMJ capsule, without communication with the joint or parotid extension. After suturing, a pressure bandage was applied for 7 days. The postoperative course was uneventful. After 1 month, the mandibular movements were normal, and there was no facial palsy. There was no recurrence at 30 months. The hematoxylin and eosin stained section revealed a cystic lesion surrounded by dense fibrous connective tissue. Myxoid degeneration was observed in the fibrous connective tissue
without epithelial lining (fig. 2). All these findings were consistent with the diagnosis of a ganglion cyst.
Discussion Ganglion cysts of the TMJ are rare. The first case was reported in 1977 [1]. The etiology is unknown, and the commonly accepted theory is ganglion formation associated with chronic, repetitive trauma. In the past, the terms ‘‘ganglion cyst’’ and ‘‘synovial cyst’’ were often synonymous [2,3]. Their histological features and origins are different. Ganglion cysts may arise from the myxoid degeneration or softening of the collagenous tissue of joint capsule or tendon sheath. Ganglion cysts are pseudocysts encapsulated by fibrous tissue, without any connection with the joint cavity. Synovial cysts are true cysts surrounded by cuboidal or flat synovial cells, which may be connected with the joint cavity. Synovial cysts may be due to an increase of intra-articular pressure, due to trauma or inflammation, causing synovial herniation. Another hypothesis is the retention of embryogenesis synovial nests [4,5]. In our case, as in most reported cases, there was no history of TMJ trauma, but our patient chewed only on one side, which could be a possible etiology. The pre-auricular region localization suggests various differential diagnoses, such as parotid gland tumor, hemangioma, neurofibroma, branchial cyst, and sebaceous cyst. Ganglion cysts have been misdiagnosed as a parotid tumor and treated by superficial parotidectomy [5]. Proper preoperative diagnosis, intraoperative observation, and frozen section are important to avoid inadequate treatment. There is no consensus regarding the best imaging technique for the ganglion cyst, although there are many imaging methods, including panoramic X-ray, ultrasound, CT scan, and MRI. The panoramic X-ray is not contributive. Our patient did not undergo CT and MRI examination due to economic reasons. In our case, the diagnosis was made by ultrasound; it indicated an anechoic cystic lesion, located next to the joint capsule, without any communication with the joint. It was different from the case reported by Nahlieli et al. [5], for which ultrasound revealed a hypoechoic cystic process. Albright et al. [1] concluded that CT was a useful diagnostic tool for ganglion cysts. For Sugiura et al., MRI revealed a mass located next to the joint capsule, while CT scan examination did not reveal any abnormality in this region [4]. We also believe MRI is the best imaging examination, because it can distinctly reveal the location, size, and density of the lesion, as well as its connection with surrounding structures. It is difficult to diagnose a ganglion cyst before biopsy, with only physical and imaging examination. Ganglion cysts usually present as a unilocular mass as in our case, and sometimes as a multilocular mass. The microscopic examination revealed a cystic structure, with dense fibrous connective tissue wall, without endothelial lining. The cysts were filled
63
W. Heng-Kun et al.
Rev Stomatol Chir Maxillofac Chir Orale 2014;115:62-64
bacteria and viruses, which could be related to myxoid degeneration of the muscle. Crushing, repeated puncture, ethyl alcohol absolute injection, carbolic acid injection, steroid injection, and radiotherapy have been used in the treatment of ganglion cysts, however the cysts recurred. Surgical excision is currently considered as the best procedure for a ganglion cyst [7,8]. The facial nerve must be located and preserved, prior to cyst resection. It is crucial to resect the cyst roots and peripheral part of the normal tendon and ligament to prevent recurrence. The resected tendon and ligament of the TMJ must be repaired.
Disclosure of interest Figure 1. Color Doppler revealing an anechoic cyst 11 mm 6 mm, over the left parotid gland.
The authors declare that they have no conflicts of interest concerning this article.
References [1]
[2]
[3]
[4]
[5]
Figure 2. Photomicrograph showing the dense fibrous connecting tissue of the cyst wall. There is no epithelial lining (HE 400).
with viscous or gelatinous fluid, consistent with the diagnosis of ganglion cyst. Kim et al. [6] analyzed the cystic content with high performance liquid chromatography and mass spectrophotometry, and identified some proteins originating from
64
[6] [7]
[8]
Albright JT, Diecidue RJ, Johar A, Keane WM. Intraosseous ganglion of the temporomandibular joint presenting with otorrhea. Arch Otolaryngol Head Neck Surg 2000;126:665–8. Goudot P, Jaquinet AR, Richter M. Cysts of the temporomandibular joint. Report of two cases. Int J Oral Maxillofac Surg 1999;28:338–40. Shinawi M, Hicks J, Guillerman RP, Jones J, Brandt M, Perez M, et al. Multiple ganglion cysts (‘cystic ganglionosis’): an unusual presentation in a child. Scand J Rheumatol 2007;36:145–8. Sugiura J, Itol H, Yamasaki A, Koitabashi T. Ganglion of the temporomandibular joint: a case report and review of the literature. Oral Med Pathol 2006;11:55–8. Nahlieli O, Lewkowicz A, Hasson O, Vered M. Ganglion cyst of the temporomandibular joint: report of case and review of literature. J Oral Maxillofac Surg 2000;58:216–9. Kim SG, Cho BO, Lee YC, Hong SP, Chae CH. Ganglion cyst of the temporomandibular joint. J Oral Pathol Med 2003;32:310–3. Takaku S, Sano T, Komine Y, Fukazawa N. Ganglion of the temporomandibular joint: case report. J Oral Maxillofac Surg 2001;59:224–8. Silva EC, Guimaraes AL, Gomes CC, Gomez RS. Ganglion cyst of the temporomandibular joint. Br J Oral Maxillofac Surg 2005;43:77–80.
Available online at
ScienceDirect www.sciencedirect.com
Case report
Ganglion cyst of the temporomandibular joint Ganglion kystique de l’articulation temporomandibulaire W. Heng-Kuna,*, G. Yan-Lingb, Z. Wen-Fengc, S. Zhec, W. Ren-Xina, Z. Xiao-Taoa a
Department of oral and maxillofacial surgery, Weihai municipal hospital, Weihai 264200, Shandong province, PR China b Department of stomatology, Weihai women and children hospital, Weihai 264200, Shandong province, PR China c Department of oro-maxillofacial head and neck surgery, hospital of stomatology, Wuhan university, Wuhan 430079, Hubei Province, China
Summary Introduction. Ganglion cyst of the temporomandibular joint is a rare disease, which may arise from myxoid degeneration of the collagenous tissue of the temporomandibular joint capsule, without epithelial or endothelial lining. We report a case of cystic lesion in a 40-year-old female patient. Observation. The patient had a left pre-auricular oval-shaped swelling without any articular symptoms. The pathological analysis after surgical removal allowed diagnosing the lesion as a ganglion cyst of the left temporomandibular joint. Discussion. We made a literature review and noted that this condition was predominant in female patients. We recommend using MRI for diagnostic purposes and surgery as the best therapeutic alternative. ß 2013 Elsevier Masson SAS. All rights reserved.
Re´sume´ Introduction. Le ganglion kystique de l’articulation temporomandibulaire est une maladie rare, qui peut re´sulter de la de´ge´ne´rescence myxoı¨de du tissu collage`ne de la capsule de l’articulation sans reveˆtement e´pithe´lial ou endothe´lial. Nous en rapportons un cas chez une femme de 40 ans. Observation. La patiente avait une tume´faction ovalaire pre´auriculaire gauche, sans symptomatologie articulaire. Le diagnostic anatomopathologique apre`s exe´re`se chirurgicale a e´te´ celui d’un ganglion kystique de l’articulation temporomandibulaire gauche. Discussion. Cette pathologie rare pre´domine chez la femme. Il n’y a pas de consensus sur les explorations a` pre´voir et sur le traitement. Nous recommandons l’IRM et proˆnons la chirurgie comme meilleure alternative the´rapeutique. ß 2013 Elsevier Masson SAS. Tous droits re´serve´s.
Keywords: Temporomandibular joint, Ganglion cyst
Mots cle´s : Articulation temporomandibulaire, Ganglion kystique
Introduction
occurring in the temporomandibular joint (TMJ) are rare. Twenty-seven cases of ganglion cysts in TMJ have been reported in the English literature [1–8] (listed in table I). We report a new case of and describe its clinic-pathological features.
A ganglion cyst is a benign cystic lesion that is closely associated with joints and tendon sheaths. This lesion is commonly encountered subcutaneously along the extensor surface of the wrist and ankle joints. However, ganglion cysts
Case report * Corresponding author. E-mail addresses: [email protected], [email protected] (W. Heng-Kun).
A 40-year-old female patient consulted with a chief complaint of swelling in the left pre-auricular region, first noticed
2213-6533/$ - see front matter ß 2013 Elsevier Masson SAS. All rights reserved. http://dx.doi.org/10.1016/j.revsto.2013.09.004 Rev Stomatol Chir Maxillofac Chir Orale 2013;115:62-64
62
Ganglion cyst of the temporomandibular joint
Table I Reported cases of temporomandibular joint (TMJ) ganglion cysts. Case
Authors
Age
Gender
Location
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28
Heydt (in 1977) Ethell (in 1979) Patel (in 1979) Kinkead (in 1981) Kenney (in 1987) Shiba (in 1987) Copeland (in 1988) Singer (in 1988) El-Massy (in 1989) Tom (in 1990) Hopper (in 1991) Lopes (in 1994) Chang (in 1997) Goudot (in 1999) Albright (in 2000) Nahlieli (in 2000) Takaku (in 2001) Kim (in 2003) Silva (in 2005) Junko (in 2006) Ali (in 2006) Shinawi (in 2007) Deng (in 2010) Khachi S (in 2011) Wu (in 2011) Yang (in 2011) Mumert (in 2012) Present case
47 28 45 36 44 28 60 ND 33 22 58 33 32 35 51 57 50 37 51 66 28 11 45 56 59 58 63 40
F F F F F M F ND F M F F F M F F F F F M F M F M F F F F 6M21F
L R R R L R L ND R R R L L R R L L L L L R L+R L L R R L L 15R13L
ND: no description; M: male; F: female; L: left; R: right.
3 months before. There was neither joint pain nor TMJ dysfunction. Her medical history was unremarkable, with no history of trauma in TMJ region. The mass was 2.0 cm 2.0 cm 1.0 cm in the left pre-auricular region. It had a smooth contour, was not mobile, was firm, and painless. The movement of the mandible was normal without any click or noise. There was no regional lymphadenopathy. The complete blood count and the differential leukocyte count were normal. A color Doppler revealed an 11 mm 6 mm cystic lesion, over the left parotid gland (fig. 1). The mass was excised under general anesthesia with a preauricular incision. The cystic lesion was adjacent to the left TMJ capsule, without communication with the joint or parotid extension. After suturing, a pressure bandage was applied for 7 days. The postoperative course was uneventful. After 1 month, the mandibular movements were normal, and there was no facial palsy. There was no recurrence at 30 months. The hematoxylin and eosin stained section revealed a cystic lesion surrounded by dense fibrous connective tissue. Myxoid degeneration was observed in the fibrous connective tissue
without epithelial lining (fig. 2). All these findings were consistent with the diagnosis of a ganglion cyst.
Discussion Ganglion cysts of the TMJ are rare. The first case was reported in 1977 [1]. The etiology is unknown, and the commonly accepted theory is ganglion formation associated with chronic, repetitive trauma. In the past, the terms ‘‘ganglion cyst’’ and ‘‘synovial cyst’’ were often synonymous [2,3]. Their histological features and origins are different. Ganglion cysts may arise from the myxoid degeneration or softening of the collagenous tissue of joint capsule or tendon sheath. Ganglion cysts are pseudocysts encapsulated by fibrous tissue, without any connection with the joint cavity. Synovial cysts are true cysts surrounded by cuboidal or flat synovial cells, which may be connected with the joint cavity. Synovial cysts may be due to an increase of intra-articular pressure, due to trauma or inflammation, causing synovial herniation. Another hypothesis is the retention of embryogenesis synovial nests [4,5]. In our case, as in most reported cases, there was no history of TMJ trauma, but our patient chewed only on one side, which could be a possible etiology. The pre-auricular region localization suggests various differential diagnoses, such as parotid gland tumor, hemangioma, neurofibroma, branchial cyst, and sebaceous cyst. Ganglion cysts have been misdiagnosed as a parotid tumor and treated by superficial parotidectomy [5]. Proper preoperative diagnosis, intraoperative observation, and frozen section are important to avoid inadequate treatment. There is no consensus regarding the best imaging technique for the ganglion cyst, although there are many imaging methods, including panoramic X-ray, ultrasound, CT scan, and MRI. The panoramic X-ray is not contributive. Our patient did not undergo CT and MRI examination due to economic reasons. In our case, the diagnosis was made by ultrasound; it indicated an anechoic cystic lesion, located next to the joint capsule, without any communication with the joint. It was different from the case reported by Nahlieli et al. [5], for which ultrasound revealed a hypoechoic cystic process. Albright et al. [1] concluded that CT was a useful diagnostic tool for ganglion cysts. For Sugiura et al., MRI revealed a mass located next to the joint capsule, while CT scan examination did not reveal any abnormality in this region [4]. We also believe MRI is the best imaging examination, because it can distinctly reveal the location, size, and density of the lesion, as well as its connection with surrounding structures. It is difficult to diagnose a ganglion cyst before biopsy, with only physical and imaging examination. Ganglion cysts usually present as a unilocular mass as in our case, and sometimes as a multilocular mass. The microscopic examination revealed a cystic structure, with dense fibrous connective tissue wall, without endothelial lining. The cysts were filled
63
W. Heng-Kun et al.
Rev Stomatol Chir Maxillofac Chir Orale 2014;115:62-64
bacteria and viruses, which could be related to myxoid degeneration of the muscle. Crushing, repeated puncture, ethyl alcohol absolute injection, carbolic acid injection, steroid injection, and radiotherapy have been used in the treatment of ganglion cysts, however the cysts recurred. Surgical excision is currently considered as the best procedure for a ganglion cyst [7,8]. The facial nerve must be located and preserved, prior to cyst resection. It is crucial to resect the cyst roots and peripheral part of the normal tendon and ligament to prevent recurrence. The resected tendon and ligament of the TMJ must be repaired.
Disclosure of interest Figure 1. Color Doppler revealing an anechoic cyst 11 mm 6 mm, over the left parotid gland.
The authors declare that they have no conflicts of interest concerning this article.
References [1]
[2]
[3]
[4]
[5]
Figure 2. Photomicrograph showing the dense fibrous connecting tissue of the cyst wall. There is no epithelial lining (HE 400).
with viscous or gelatinous fluid, consistent with the diagnosis of ganglion cyst. Kim et al. [6] analyzed the cystic content with high performance liquid chromatography and mass spectrophotometry, and identified some proteins originating from
64
[6] [7]
[8]
Albright JT, Diecidue RJ, Johar A, Keane WM. Intraosseous ganglion of the temporomandibular joint presenting with otorrhea. Arch Otolaryngol Head Neck Surg 2000;126:665–8. Goudot P, Jaquinet AR, Richter M. Cysts of the temporomandibular joint. Report of two cases. Int J Oral Maxillofac Surg 1999;28:338–40. Shinawi M, Hicks J, Guillerman RP, Jones J, Brandt M, Perez M, et al. Multiple ganglion cysts (‘cystic ganglionosis’): an unusual presentation in a child. Scand J Rheumatol 2007;36:145–8. Sugiura J, Itol H, Yamasaki A, Koitabashi T. Ganglion of the temporomandibular joint: a case report and review of the literature. Oral Med Pathol 2006;11:55–8. Nahlieli O, Lewkowicz A, Hasson O, Vered M. Ganglion cyst of the temporomandibular joint: report of case and review of literature. J Oral Maxillofac Surg 2000;58:216–9. Kim SG, Cho BO, Lee YC, Hong SP, Chae CH. Ganglion cyst of the temporomandibular joint. J Oral Pathol Med 2003;32:310–3. Takaku S, Sano T, Komine Y, Fukazawa N. Ganglion of the temporomandibular joint: case report. J Oral Maxillofac Surg 2001;59:224–8. Silva EC, Guimaraes AL, Gomes CC, Gomez RS. Ganglion cyst of the temporomandibular joint. Br J Oral Maxillofac Surg 2005;43:77–80.
