- Email: [email protected]
Pigmented villonodular synovitis occurring in the temporomandibular joint
G Model
ANL-2534; No. of Pages 9 Auris Nasus Larynx xxx (2018) xxx–xxx Contents lists available at ScienceDirect
Auris Nasus Larynx journal homepage: www.elsevier.com/locate/anl
Pigmented villonodular synovitis occurring in the temporomandibular joint Fuminori Nomura a,*, Yosuke Ariizumi a, Yusuke Kiyokawa a, Akihisa Tasaki a, Yumiko Tateishi b, Nobuaki Koide a, Hiroaki Kawabe a, Takashi Sugawara c, Kentaro Tanaka d, Takahiro Asakage a a
Department of Head and Neck Surgry, Tokyo Medical and Dental University, Japan Department of Otorhinolaryngology, Tokyo Medical and Dental University, Japan c Department of Neurosurgery, Tokyo Medical and Dental University, Japan d Department of Plastic and Reconstructive Surgery, Tokyo Medical and Dental University, Japan b
A R T I C L E I N F O
A B S T R A C T
Article history: Received 20 August 2018 Accepted 30 October 2018 Available online xxx
Objective: Pigmented villonodular synovitis occurring in the region of the temporomandibular joint is a rare disease, requiring a review of the treatment method, follow-up period. Method: Refer to the past literature, along with a retrospective search. Results: An excision, including the skull base bone, was performed in all cases; however, recurrence was found in one case on which fractional excision was performed. Past reports have also indicated that en bloc resection was considered desirable. Conclusion: It is necessary to perform en bloc resection on patients with pigmented villonodular synovitis occurring in the region of the temporomandibular joint. Furthermore, due to reported cases of recurrence after a long period of time, follow-up observations of about 10 years are considered necessary. © 2018 Elsevier B.V. All rights reserved.
Keywords: Pigmented villonodular synovitis Tenosynovial giant cell tumor Temporomandibular joint Skul base sugery
1. Introduction Pigmented villonodular synovitis (PVNS) is also known as diffuse type tendon synovial giant cell tumor, most commonly occurring within the large joints of the extremities, with the highest frequency of occurrence in the knee joint (75%) followed by the hip joint and the foot joint. Temporomandibular joint development is rare. Clinical symptoms in the cases of temporomandibular joint development include painful or
* Corresponding author at: Department of Head and Neck Surgery, Tokyo Medical and Dental University, 1-5-45, Yushima, Bunkyo-ku, Tokyo, 1138519, Japan E-mail addresses: [email protected], [email protected] (F. Nomura).
indolent tumor formation, ear closure, and deafness, often accompanied by intracranial development. Using a CT, the erosion and destruction of bones, cyst formation, and soft tumor masses are revealed, occasionally exhibiting high signal reflecting the deposition of hemosiderin in the tissue. Because a gradual increase of an infiltrating local invasive nature may be exhibited, as well as many cases of recurrence, en bloc resection including an adequate safety area is recommended. We herein report, with pertinent bibliographical considerations, our experience in treating four cases of PVNS (Table 1). 2. Cases 1. A 40-year-old female. Her main complaints included pain in the lower right ear, a sensation of obstruction in the right ear,
https://doi.org/10.1016/j.anl.2018.10.021 0385-8146/© 2018 Elsevier B.V. All rights reserved.
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
2
Table 1 Clinical features of 4 cases of pigmented villonodular synovitis. Case
Age/gender
1 2 3 4
40/F 60/M 30/M 60/M
Tumor size(mm) 56 45 46 45 38 22 32 30 20 25 28 26
and autophony. At the age of 38, she found she was suffering from right transmission hearing loss, right temporomandibular joint pain, and a decline in feeling in her right cheek, which gradually exacerbated, leading to her paying a visit to an otolaryngology clinic at the age of 40 and resulting in a referral visit for further examination and treatment. A solid tumor was found at the front of her right ear, which was observed along with right ear canal blockage, right pharyngeal opening of eustachian tube obstruction, perceptual decline in the right cheek (V 2), and trismus (1.5 fingerbreadths). CT and MRI examinations found a tumorous lesion of 56 45 46 mm accompanied by bone destruction, from the right middle cranial base, sphenoid bone, and mastoid cells, to the masticatory muscle space, while the ossicular bone was deformed and retracted rearward.
Treatment Skul Skul Skul Skul
base base base base
Recurrence/follow-up surgery surgery surgery surgery
Yes/30m No/25m No/13m No/4m
The internal carotid artery and the tumor were in contact. Upon PET, the accumulation of SUVMAX 4.5 ! 5.1 was observed, in accordance with the tumor, and a malignant tumor was also suspected due to the degree of bone destruction, but no lymphnode metastasis was observed (Fig. 1). Hearing loss on the affected side was observed upon an audiometry examination (Fig. 2). Chondroma was suspected in the tissue biopsy, so total extirpation was performed. Following the right temporal craniotomy, an approach was made by cutting the zygomatic arch. The tumor had completely destroyed the bony part of the external auditory maetus, so the tumor was excised by split resection, separated from the pyramidal bone of the tympanic part of the temporal bone. The facial nerve was preserved and the defect was closed with the pericranial flap and temporal muscle flap. The postoperative pathological findings revealed that compact type circular cells with unevenly distributed
Fig. 1. (A) Contrast-enhanced CT axial image. Tumors with poor contrast effect are found from the temporomandibular joint to the parapharyngeal space. The tumor is squeezing the internal carotid artery backwards. (B) PET-CT SUV MAX5.1 axial image. (C) PET-CT coronal image. No obvious lymph node metastasis was observed. (D) MRI T2-weighted axial image. A tumor with a clear boundary of slightly high signals is found. (E) MRI T2-weigted coronal image. A tumor is progressing into the cranium.
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
Fig. 2. Pure tone hearing test: transmission hearing loss on the right side was observed.
nuclei and eosinophilic cytoplasm proliferated on their own, with the chondromyxoid matrix as background, and the nucleus was somewhat irregular shaped and constricted, revealing binuclear cells. The nuclear fission image was not noticeable, with multinucleated giant cells and several to a few dozen nuclei also mixed in. The formation of clear cartilage and calcification were observed. The tumor cells indicated Vimentin positive, S-100 positive. Clusterin positive, FGF23 negative, and others such as AE1/AE3, EMA, GFAP, p63, Desmin, a-SMA, PG-M1 were all negative. The Ki-67 positive rate was about 1.5% at the high areas. Local recurrence was suspected upon MRI 22 months later (Fig. 3), and therefore repeat surgery was performed. The findings on the removed specimens were similar to those of the previous surgical specimens. Eight months have passed with no obvious recurrence findings and no facial palsy.
3
2. A 60-year old male. His main complaint was a tumor at the front of his right ear. A tumor at the frontal part of his right ear was found 4 months earlier and a CT performed by a nearby clinic found a tumorous lesion in the right temporomandibular joint, which was in contact with the mid skull base, so he was referred to our clinic. Although the CT, which was taken due to a headache four years earlier, also revealed a tumor of 28 mm in size(Fig. 4A), there was no mention of the tumor at that time. A tumor in the right temporomandibular joint was felt, which was hard with poor mobility, no pulsation/redness, and mild tenderness. No neurological symptoms such as a decline in sensitivity were observed. In the image, a tumor with a calcification of 45 38 22 mm centered in the right lower jaw ostium was found, and although it was in contact with the middle cranial fossa, the boundary was clear and no obvious invasive findings were observed (Fig. 4B, C, and D). Although PVNS was primarily suspected in the tissue biopsy results, others such as a giant cell tumor and bone giant cell tumor were also suspected, leading to total extirpation surgery. The cheekbone was cut and a temporal craniotomy as well as resection of the skull base bone from the inner skull base were performed, preserving the third branch of the trigeminal nerve. An S-shaped incision was added from the front of the ear and the skin flap was lifted with a layer just under the platysma muscle. The superior branch of the facial nerve was temporarily cut upon identifying the trunk of the facial nerve. The mandibular bone was exposed and cut at the lower end of the tumor, such that the tumor was separated from the surrounding tissue and resected en bloc. The superior branch of the facial nerve was anastomosed, and filling the dead space with the temporal muscle flap (Fig. 5). The pathological findings suggested lesions in which monocytes proliferated invasively in and out of the joints as well as in the mandible. The mononuclear cells were small to medium in size and mixed with histiocyte-like cells which were
Fig. 3. (A) contrast-enhanced MRI TI-weighted axial image. Recurrent tumor with contrast effect was found in the pharyngeal space. (B) Contrast-enhanced MRI TIweighted axial image. Recurrent tumor is found in the parapharyngeal space and just under the dura.
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 4
F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
Fig. 4. (A) Simple CT axial image, taken 4 years prior to visiting our clinic. A tumor with calcification is found around the right temporomandibular joint. (B) Contrast-enhanced CT axial image. An increasing trend is found, compared with 4 years prior. (C) Contrast-enhanced CT coronal image. A tumor invading the skull base and the temporomandibular joint, accompanied by calcification, was found. (D) MRI T2-weighted axial image. A tumor with a clear boundary of slightly low signals is found.
constricted in the nucleus, with the cells having somewhat large and pale eosinophilic cytoplasm. Inflammatory cell invasion such as by osteoclastic multinucleated giant cells, hemosiderin phagocytic cells, foamy cells, lymphocytes, plasma cells, neutrophils were observed, at various ratios depending on the region on the solid growth of mononuclear cells (Fig. 6). Immunostaining revealed CD68: mononuclear cells, positive for multinucleated giant cells, CD 163: positive for mononuclear cells, negative for multinucleated giant cells, Desmin: very few positive, and a Ki-67 positive rate of 14.7% at the most. The surgical margin was negative. He developed facial nerve palsy (House–Blackmann 4/6), which gradually improved and eventually recovered to grade 2. No apparent recurrence was observed at 25 months following surgery. 3. A 30-year-old male. His main complaint was of pain in the right temporomandibular joint. The pain in his right jaw started one and a half years ago, upon which CT & MRI were performed at an oral surgery clinic, revealing a tumor in his right TMJ joint. A feeling of numbness on the right side of his face also appeared, leading to a referral visit. A hard tumor was found in his right temporomandibular joint and he suffered from poor mobility with a trismus of 2 fingerbreadth. Furthermore, a
decline in sensation in the second branch region of the right trigeminal nerve was observed. CT and MRI revealed a tumor of 32 30 20 mm in size, accompanied by calcification with a contrast effect in his right temporomandibular joint (Fig. 7). Although it was in contact with the middle cranial fossa and slight erosion of the bone was recognized, no progress was found in the cranium. A tissue biopsy was performed to find numerous giant cells, leading to diagnoses such as suspected PVNS, giant cell tumor, giant cell sarcoma, and chondroblast and giant cell tumor. Total extirpation was considered necessary and after the temporal craniotomy, resection of the mandibular region by the parotid gland method was performed in order to perform en bloc resection. The cheekbone branch of the facial nerve was temporarily transected and the anastomosis was reconstructed upon removing the tumor. The defects were filled with temporal muscle flap and pericranial flap. The pathologic findings indicated that oval to spindle-shaped mononuclear cells with egg-shaped nuclei grew densely accompanied by osteoclast-like multinucleated giant cells having dozens of large nuclei. Along with bleeding and deposition of high hemosiderin, mineralization and small bone formation nests were found. Furthermore, proliferation of fibroblast-like spindle-shaped cells were also partially observed, with no clear cartilage matrix found. Margin evaluation
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
5
Fig. 5. Case 2 Intraoperative photograph. (A) Temporal incision, cutting off the mandible by disconnecting the zygomatic arch (arrow). (B) After removing the tumor, filling the temporal muscle flap (arrow). Fixing the zygomatic arch with a titanium plate. (C) Resected specimen 50 43 42mm. No obvious tumor exposure is noticed.
was negative. He developed facial nerve palsy (HouseBrackmann 4/6) and eventually recovered to grade 2 after undergoing static facial nerve palsy reconstruction and upper eyelid descent surgery. There has been no recurrence 13 months following surgery. 4. A 60-year-old male. Because a temporomandibular joint tumor was suspected at a nearby clinic, due to a main complaint of a right ear anterior tumor from 7 months prior, he was referred to our clinic. At the first visit, CT and MRI revealed tumorous lesions, destroying and invading the mandible and the skull base, with a contrast effect of 25 28 26 mm in the right temporomandibular joint (Fig. 8). While cytodiagnosis was performed to find a suspected giant cell tumor, it was decided to perform surgery as malignant disease could also not be ruled out. An approach was made during the temporal craniotomy and en bloc resection, including mandibular joint prosthesis and the skull base bone, was performed. The upper arm branch of the facial nerve was temporarily transected to secure the operative field and anastomotic reconstruction was performed using the large auricular nerve upon removing the tumor. The pathological results indicated that multinuclear cells and mononuclear cells solidly grow and are tumorous lesions in which hemosiderin deposits were conspicuous around the lesion, involving the cartilage tissue of the temporomandibular joint, which partly invaded the mandible as well, resulting in a
diagnosis of tenosynovial giant cell tumor. The surgical margin was negative. He developed facial nerve palsy (House– Brackmann 4/6). Four months have passed following surgery and this patient is currently under observation. 3. Discussion PVNS is also known as a diffuse type tenosynovial giant cell tumor and was classified in addition to being localized in the WHO classification in 2013 [1]. Although both have many similarities in terms of their histopathology, diffuse type differs compared with the localized type, such that invasive development to the surrounding tissues is found. In this case, infiltration into the surrounding tissues was observed, leading to a diagnosis of diffuse type. It is a benign tumor that develops in the tendons, the tendon sheath, and the joint region, with the most common being cases of knee joint development [2], which often increases to local destruction along with bone destruction. The relapse rate is as high as 25–50% or higher when occurring in the extremities, so resection while ensuring a safety area is recommended for treatment [3–5]. Although the cause is still unknown and being discussed, with reports also existing on the cause being repeated trauma of the joint area and associated arthropathy [6], it was thought to
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 6
F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
Fig. 6. Case 2 Photo of hematoxylin eosin staining pathology. (A) Multinucleated giant cell (arrowhead) and hemosiderin phagocytosis cell (arrow) were found. (B) Multinucleated giant cell (arrowhead) and hemosiderin phagocytosis cell (arrow) were found. (C) Bone invasion of the tumor is observed (arrow).
be reactive inflammation resulting from chronic inflammation. However, because trisomy 7 was found in which organization only occasionally causes distant metastasis [7–9], it was determined to be a tumor. Temporomandibular joint PVNS was reported for the first time by Lapayowker et al. [10] in 1973 and the occurrence frequency is 1.8 in 1 million people, making it an extremely rare disease. Symptoms include painful or painless tumor formation, ear claudication, hearing loss, and temporomandibular joint symptoms (click sound or opening disorder), often accompanied by intracranial development. Depression on the trigeminal nerve may cause a decrease near the face or a numbness sensation in some cases. Even among these cases, Case 1 had progression into the skull, and Cases 2, 3 and 4 found destruction of the temporomandibular joint protrusions. Furthermore, while Case 1 had repeated otitis media with effusion, no lesions were observed in the nasopharynx. With regard to the presence or absence of the causative lesion in the diagnosis of exudative otitis media with pain and temporomandibular joint symptoms in adults, it is considered important to confirm the presence or absence of nasopharyngeal lesions, while also carefully examining the image findings to differentiate any neoplasmic gaps or tumorous lesions around the temporomandibular joint.
MRI is recommended for image examination, which reflects hemosiderin deposited in the tumor and becomes low signal at T2, and is useful for differentiating from other neoplastic lesions [11,12]. Although PET examinations are often performed, it is difficult to distinguish from malignant tumors. In Case 1, SUVMax combined with extensive bone destruction was as high as 5.1, resulting in a malignant tumor being suspected. Although cytology is the first method to be examined for diagnosis, no diagnosis is made by cytodiagnosis in PVNS, and tissue diagnosis is often required in many cases [13]. Even among these cases, diagnosis by cytological diagnosis was difficult, so a biopsy was performed in 3 cases. Even if a tissue examination were carried out, the differential diagnosis would still be quite diverse. Even these cases indicated pathological results including benign and malignant tumors such as chondroma, chondrosarcoma, giant cell sarcoma, chondroblast and giant cell tumor. If malignant or PVNS is suspected, total extirpation should be performed without hesitation, with the goal of en bloc resection Reports of recurrence were also observed in PVNS in the case of temporomandibular joint development [14–16], with a recurrence rate of 15–29% and treatment requiring resection with a sufficient safety area, similar to the case of the occurrence of orthopedic surgery [16,17]. Even among these cases, Case 1 underwent a fractional resection, with local
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
7
Fig. 7. (A) Contrast-enhanced CT axial image. A tumor with partial contrast effect is found, mainly in the temporomandibular joints accompanied by calcification. (B) Contrast-enhanced CT coronal image. Although the tumor is in contact with the skull base bone, no intracranial invasion is observed. (C) MRI T2-weighted axial image. A tumor with a clear boundary of low signals is found.
recurrence coming after 22 months. Cases 2, 3, and 4 underwent en bloc resections, in which the negative pathological margin was confirmed but clear recurrence has not been found as of yet. Early detection is important because en bloc resection becomes difficult once lesions grow large. Although there are reports of a possible decrease in the recurrence rate by performing radiotherapy via postoperative radiation (35–45 Gy), if en bloc resection was impossible [18], the effect is not clear [19]. An analysis of 16 cases reported after 2011 [12–14,16– 18,20–23] showed few reports on the preservation of a facial nerve during surgery. The facial nerve was preserved in five cases and all cases with paralysis improved during the follow-up observations. However, the tumor could only be completely resected in one case, 3 cases were described as macroscopic in total and 7 cases underwent partial resection, and two cases of relapse were reported.
Recurrence is considered to be caused by the fact that a split resection is often required in order to preserve the facial nerve. In case 1, the facial nerve was preserved without cutting during the surgery. Therefore, split resection was necessary and this led to recurrence. In the other 3 cases, the facial nerve was temporarily cut and then enblock resection was achieved. Depending on the size of the tumor, it may be better to perform a complete resection with a temporary cut of the nerve to reduce the risk of recurrence. Verspoor et al. [13] reported on 95 cases of temporomandibular joint PVNS, 11 cases of which recurred, with a median to recurrence of 24 months and a maximum of 240 months. Therefore, it is considered necessary to undergo follow-up observations for a long period of time, possibly for about 10 years after PVNS treatment. MRI is considered to be useful for image inspection.
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
8
Fig. 8. (A) Contrast-enhanced CT axial image. A tumor that invades the mandible with a nonuniform contrast effect is observed. (B) Contrast-enhanced CT coronal image. A tumor, infiltrating the temporomandibular joint into the skull base bone and progressing into the cranium, is found. (C) MRI T2-weighted axial image. Tumors mixed with low signals and high signals are found, mainly in the center of the temporomandibular joint.
4. Conclusion Although temporomandibular arthroplasty PVNS has low frequency and is a rare disease, early diagnosis is important due to the gradual increase thereof and progress into the cranium. It is important to conduct an image review at an early stage, in case of the transmission hearing loss on one side, or enlargement of ear front swelling, and prolonged temporomandibular joint symptoms. It is recommended to positively perform en bloc resection, because it is difficult to differentiate by tissue examination, with malignant tumors often incapable of being excluded, and PVNS is benign but the recurrence rate is high. Furthermore, because there are some cases of recurrence which occur after a long period of time, a sufficient period is also necessary for followup observation it therefore also required. Disclosures No financial interest is declared. References [1] Fletcher CDM, Bridge JA, Hogendoorn P, Mertens F. Tenosynovial giant cell tumour, diffuse type. In: Somerthausen N, Rijn M, editors. WHO
classification of tumors of soft tissue and bone. Lyon: IARC Press; 2013. p. 102–3. [2] de Visser E, Veth RP, Pruszczynski M, Wobbes T, Van de Putte LB. Diffuse and localized pigmented villonodular synovitis:evaluation of treatment of 38 patients. Arch Orthop Trauma Surg 1999;119:401–4. [3] Verspoor FG, Zee AA, Hannink G, van der Geest IC, Veth RP, Schreuder HW. Long-term follow-up results of primary and recurrent pigmented villonodular synovitis. Rheumatology 2014;53:2063–70. [4] Schwartz HS, Unni KK, Pritchard DJ. Pigmented villonodular synovitis: a retrospective review of affected large joints. Clin orthop 1989;247:243–55. [5] Byers PD, Cotton RE, Deacon OW, Lowy M, Newman PH, Sissons HA, et al. The diagnosis and treatment of pigmented villonodular synovitis. J Bone Joint Surg Br 1968;50:290–305. [6] Myers BW, Masi AT. Pigmented villonodular synovitis and tenosynovitis: a clinical epidemiologic study of 166 cases and literature review. Medicine 1980;59:223–38. [7] Ray RA, Morton CC, Lipinski KK, Corson JM, Fletcher JA. Cytogenetic evidence of clonality in a case of pigmented villonodular synovitis. Cancer 1991;67:121–5. [8] Sistla R, Jvs V, Afroz T. Malignant pigmented villonodular synovitis-a rare entity. J Orthop Case Rep 2014;4:9–11. [9] Righi A, Gambarotti M, Sbaraglia M, Frisoni T, Donati D, Vanel D, et al. Metastasizing tenosynovial giant cell tumour, diffuse type/pigmented villonodular synovitis. Clin Sarcoma Res 2015;6:15. [10] Lapayowker MS, Miller WT, Levy WM, Harwick RD. Pigmented villonodular synovitis of the temperomandibular joint. Radiology 1973;108:313–6. [11] Bemporad JA, Chaloupka JC, Putman CM, Roth TC, Tarro J, Mitra S, et al. Pigmented villonodular synovitis of the temporomandibular joint:
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
[12]
[13]
[14]
[15] [16]
[17]
diagnostic imaging and endovascular therapeutic embolization of a rare head and neck tumor. Am J Neuroradiol 1999;20:159–62. Wong JJ, Phal PM, Wiesenfeld D. Pigmented villonodular synovitis of the temporomandibular joint: a radiologic diagnosis and case report. J Oral Maxillofac Surg 2012;70:126–34. Verspoor FGM, Mastboom MJL, Weijs WLJ, Koetsveld AC, Schreuder HWB, Flucke U. Treatments of tenosynovial giant cell tumours of the temperomandibular joint: a report of three cases and a review of literature. Int J Oral Maxillofac Surg 2018;47:1288–94. Pina S, Fernandez M, Maya S, Garcia RA, Noor A, Pawha PS, et al. Recurrent temporal bone tenosynovial giant cell tumor with chondroid metaplasia: the use of imaging to assess recurrence. Neuroradiol J 2014;27:97–101. Allen A, Saran N. Recurrent pigmented villonodular synovitis of the temporomandibular joint. Radiol Case Rep 2018;13:499–502. Safaee M, Oh T, Sun MZ, Parsa AT, McDermott MW, El-Sayed IH, et al. Pigmented villonodular synovitis of the temporomandibular joint with intracranial extension: a case series and systematic review. Head Neck 2015;37:1213–24. Carlson ML, Osetinsky LM, Alon EE, Inwards CY, Lane JI, Moore EJ. Tenosynovial giant cell tumors of the temporomandibular joint and lateral skull base: review of 11 cases. Laryngoscope 2017;127:2340–6.
9
[18] Joshi K, Huang B, Scanga L, Buchman C, Chera BS. Postoperative radiotherapy for diffuse pigmented villonodular synovitis of the temporomandibular joint. Am J Otolaryngol 2015;36:106–13. [19] Verspoor FG, van der Geest IC, Vegt E, Veth RP, van der Graaf WT, Schreuder HW. Pigmented villonodular synovitis: current concepts about diagnosis and management. Future Oncol 2013;9:1515–31. [20] Bredell M, Schucknecht B, Bode-Lesniewska B. Tenosynovial, diffuse type giant cell tumor of the temporomandibular joint, diagnosis and management of a rare tumor. J Clin Med Res 2015;7:262–6. [21] Roman˜ach MJ, Brasileiro BF, Leo´n JE, Alves DB, de Almeida OP, Vargas PA. Pigmented villonodular synovitis of the temporomandibular joint: case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;111:e17–28. [22] Damodar D, Chan N, Kokot N. Pigmented villonodular synovitis of the temporomandibular joint: case report and review of the literature. Head Neck 2015;37(12):E194–9. [23] Vellutini EAS, Alonso N, Arap SS, Godoy LFS, Souza E, Souza RA, Mattedi RL, et al. Functional reconstruction of temporomandibular joint after resection of pigmented villonodular synovitis with extension to infratemporal fossa and skull base: a case report. Surg J (N Y) 2016;2:e78–82.
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
ANL-2534; No. of Pages 9 Auris Nasus Larynx xxx (2018) xxx–xxx Contents lists available at ScienceDirect
Auris Nasus Larynx journal homepage: www.elsevier.com/locate/anl
Pigmented villonodular synovitis occurring in the temporomandibular joint Fuminori Nomura a,*, Yosuke Ariizumi a, Yusuke Kiyokawa a, Akihisa Tasaki a, Yumiko Tateishi b, Nobuaki Koide a, Hiroaki Kawabe a, Takashi Sugawara c, Kentaro Tanaka d, Takahiro Asakage a a
Department of Head and Neck Surgry, Tokyo Medical and Dental University, Japan Department of Otorhinolaryngology, Tokyo Medical and Dental University, Japan c Department of Neurosurgery, Tokyo Medical and Dental University, Japan d Department of Plastic and Reconstructive Surgery, Tokyo Medical and Dental University, Japan b
A R T I C L E I N F O
A B S T R A C T
Article history: Received 20 August 2018 Accepted 30 October 2018 Available online xxx
Objective: Pigmented villonodular synovitis occurring in the region of the temporomandibular joint is a rare disease, requiring a review of the treatment method, follow-up period. Method: Refer to the past literature, along with a retrospective search. Results: An excision, including the skull base bone, was performed in all cases; however, recurrence was found in one case on which fractional excision was performed. Past reports have also indicated that en bloc resection was considered desirable. Conclusion: It is necessary to perform en bloc resection on patients with pigmented villonodular synovitis occurring in the region of the temporomandibular joint. Furthermore, due to reported cases of recurrence after a long period of time, follow-up observations of about 10 years are considered necessary. © 2018 Elsevier B.V. All rights reserved.
Keywords: Pigmented villonodular synovitis Tenosynovial giant cell tumor Temporomandibular joint Skul base sugery
1. Introduction Pigmented villonodular synovitis (PVNS) is also known as diffuse type tendon synovial giant cell tumor, most commonly occurring within the large joints of the extremities, with the highest frequency of occurrence in the knee joint (75%) followed by the hip joint and the foot joint. Temporomandibular joint development is rare. Clinical symptoms in the cases of temporomandibular joint development include painful or
* Corresponding author at: Department of Head and Neck Surgery, Tokyo Medical and Dental University, 1-5-45, Yushima, Bunkyo-ku, Tokyo, 1138519, Japan E-mail addresses: [email protected], [email protected] (F. Nomura).
indolent tumor formation, ear closure, and deafness, often accompanied by intracranial development. Using a CT, the erosion and destruction of bones, cyst formation, and soft tumor masses are revealed, occasionally exhibiting high signal reflecting the deposition of hemosiderin in the tissue. Because a gradual increase of an infiltrating local invasive nature may be exhibited, as well as many cases of recurrence, en bloc resection including an adequate safety area is recommended. We herein report, with pertinent bibliographical considerations, our experience in treating four cases of PVNS (Table 1). 2. Cases 1. A 40-year-old female. Her main complaints included pain in the lower right ear, a sensation of obstruction in the right ear,
https://doi.org/10.1016/j.anl.2018.10.021 0385-8146/© 2018 Elsevier B.V. All rights reserved.
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
2
Table 1 Clinical features of 4 cases of pigmented villonodular synovitis. Case
Age/gender
1 2 3 4
40/F 60/M 30/M 60/M
Tumor size(mm) 56 45 46 45 38 22 32 30 20 25 28 26
and autophony. At the age of 38, she found she was suffering from right transmission hearing loss, right temporomandibular joint pain, and a decline in feeling in her right cheek, which gradually exacerbated, leading to her paying a visit to an otolaryngology clinic at the age of 40 and resulting in a referral visit for further examination and treatment. A solid tumor was found at the front of her right ear, which was observed along with right ear canal blockage, right pharyngeal opening of eustachian tube obstruction, perceptual decline in the right cheek (V 2), and trismus (1.5 fingerbreadths). CT and MRI examinations found a tumorous lesion of 56 45 46 mm accompanied by bone destruction, from the right middle cranial base, sphenoid bone, and mastoid cells, to the masticatory muscle space, while the ossicular bone was deformed and retracted rearward.
Treatment Skul Skul Skul Skul
base base base base
Recurrence/follow-up surgery surgery surgery surgery
Yes/30m No/25m No/13m No/4m
The internal carotid artery and the tumor were in contact. Upon PET, the accumulation of SUVMAX 4.5 ! 5.1 was observed, in accordance with the tumor, and a malignant tumor was also suspected due to the degree of bone destruction, but no lymphnode metastasis was observed (Fig. 1). Hearing loss on the affected side was observed upon an audiometry examination (Fig. 2). Chondroma was suspected in the tissue biopsy, so total extirpation was performed. Following the right temporal craniotomy, an approach was made by cutting the zygomatic arch. The tumor had completely destroyed the bony part of the external auditory maetus, so the tumor was excised by split resection, separated from the pyramidal bone of the tympanic part of the temporal bone. The facial nerve was preserved and the defect was closed with the pericranial flap and temporal muscle flap. The postoperative pathological findings revealed that compact type circular cells with unevenly distributed
Fig. 1. (A) Contrast-enhanced CT axial image. Tumors with poor contrast effect are found from the temporomandibular joint to the parapharyngeal space. The tumor is squeezing the internal carotid artery backwards. (B) PET-CT SUV MAX5.1 axial image. (C) PET-CT coronal image. No obvious lymph node metastasis was observed. (D) MRI T2-weighted axial image. A tumor with a clear boundary of slightly high signals is found. (E) MRI T2-weigted coronal image. A tumor is progressing into the cranium.
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
Fig. 2. Pure tone hearing test: transmission hearing loss on the right side was observed.
nuclei and eosinophilic cytoplasm proliferated on their own, with the chondromyxoid matrix as background, and the nucleus was somewhat irregular shaped and constricted, revealing binuclear cells. The nuclear fission image was not noticeable, with multinucleated giant cells and several to a few dozen nuclei also mixed in. The formation of clear cartilage and calcification were observed. The tumor cells indicated Vimentin positive, S-100 positive. Clusterin positive, FGF23 negative, and others such as AE1/AE3, EMA, GFAP, p63, Desmin, a-SMA, PG-M1 were all negative. The Ki-67 positive rate was about 1.5% at the high areas. Local recurrence was suspected upon MRI 22 months later (Fig. 3), and therefore repeat surgery was performed. The findings on the removed specimens were similar to those of the previous surgical specimens. Eight months have passed with no obvious recurrence findings and no facial palsy.
3
2. A 60-year old male. His main complaint was a tumor at the front of his right ear. A tumor at the frontal part of his right ear was found 4 months earlier and a CT performed by a nearby clinic found a tumorous lesion in the right temporomandibular joint, which was in contact with the mid skull base, so he was referred to our clinic. Although the CT, which was taken due to a headache four years earlier, also revealed a tumor of 28 mm in size(Fig. 4A), there was no mention of the tumor at that time. A tumor in the right temporomandibular joint was felt, which was hard with poor mobility, no pulsation/redness, and mild tenderness. No neurological symptoms such as a decline in sensitivity were observed. In the image, a tumor with a calcification of 45 38 22 mm centered in the right lower jaw ostium was found, and although it was in contact with the middle cranial fossa, the boundary was clear and no obvious invasive findings were observed (Fig. 4B, C, and D). Although PVNS was primarily suspected in the tissue biopsy results, others such as a giant cell tumor and bone giant cell tumor were also suspected, leading to total extirpation surgery. The cheekbone was cut and a temporal craniotomy as well as resection of the skull base bone from the inner skull base were performed, preserving the third branch of the trigeminal nerve. An S-shaped incision was added from the front of the ear and the skin flap was lifted with a layer just under the platysma muscle. The superior branch of the facial nerve was temporarily cut upon identifying the trunk of the facial nerve. The mandibular bone was exposed and cut at the lower end of the tumor, such that the tumor was separated from the surrounding tissue and resected en bloc. The superior branch of the facial nerve was anastomosed, and filling the dead space with the temporal muscle flap (Fig. 5). The pathological findings suggested lesions in which monocytes proliferated invasively in and out of the joints as well as in the mandible. The mononuclear cells were small to medium in size and mixed with histiocyte-like cells which were
Fig. 3. (A) contrast-enhanced MRI TI-weighted axial image. Recurrent tumor with contrast effect was found in the pharyngeal space. (B) Contrast-enhanced MRI TIweighted axial image. Recurrent tumor is found in the parapharyngeal space and just under the dura.
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 4
F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
Fig. 4. (A) Simple CT axial image, taken 4 years prior to visiting our clinic. A tumor with calcification is found around the right temporomandibular joint. (B) Contrast-enhanced CT axial image. An increasing trend is found, compared with 4 years prior. (C) Contrast-enhanced CT coronal image. A tumor invading the skull base and the temporomandibular joint, accompanied by calcification, was found. (D) MRI T2-weighted axial image. A tumor with a clear boundary of slightly low signals is found.
constricted in the nucleus, with the cells having somewhat large and pale eosinophilic cytoplasm. Inflammatory cell invasion such as by osteoclastic multinucleated giant cells, hemosiderin phagocytic cells, foamy cells, lymphocytes, plasma cells, neutrophils were observed, at various ratios depending on the region on the solid growth of mononuclear cells (Fig. 6). Immunostaining revealed CD68: mononuclear cells, positive for multinucleated giant cells, CD 163: positive for mononuclear cells, negative for multinucleated giant cells, Desmin: very few positive, and a Ki-67 positive rate of 14.7% at the most. The surgical margin was negative. He developed facial nerve palsy (House–Blackmann 4/6), which gradually improved and eventually recovered to grade 2. No apparent recurrence was observed at 25 months following surgery. 3. A 30-year-old male. His main complaint was of pain in the right temporomandibular joint. The pain in his right jaw started one and a half years ago, upon which CT & MRI were performed at an oral surgery clinic, revealing a tumor in his right TMJ joint. A feeling of numbness on the right side of his face also appeared, leading to a referral visit. A hard tumor was found in his right temporomandibular joint and he suffered from poor mobility with a trismus of 2 fingerbreadth. Furthermore, a
decline in sensation in the second branch region of the right trigeminal nerve was observed. CT and MRI revealed a tumor of 32 30 20 mm in size, accompanied by calcification with a contrast effect in his right temporomandibular joint (Fig. 7). Although it was in contact with the middle cranial fossa and slight erosion of the bone was recognized, no progress was found in the cranium. A tissue biopsy was performed to find numerous giant cells, leading to diagnoses such as suspected PVNS, giant cell tumor, giant cell sarcoma, and chondroblast and giant cell tumor. Total extirpation was considered necessary and after the temporal craniotomy, resection of the mandibular region by the parotid gland method was performed in order to perform en bloc resection. The cheekbone branch of the facial nerve was temporarily transected and the anastomosis was reconstructed upon removing the tumor. The defects were filled with temporal muscle flap and pericranial flap. The pathologic findings indicated that oval to spindle-shaped mononuclear cells with egg-shaped nuclei grew densely accompanied by osteoclast-like multinucleated giant cells having dozens of large nuclei. Along with bleeding and deposition of high hemosiderin, mineralization and small bone formation nests were found. Furthermore, proliferation of fibroblast-like spindle-shaped cells were also partially observed, with no clear cartilage matrix found. Margin evaluation
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
5
Fig. 5. Case 2 Intraoperative photograph. (A) Temporal incision, cutting off the mandible by disconnecting the zygomatic arch (arrow). (B) After removing the tumor, filling the temporal muscle flap (arrow). Fixing the zygomatic arch with a titanium plate. (C) Resected specimen 50 43 42mm. No obvious tumor exposure is noticed.
was negative. He developed facial nerve palsy (HouseBrackmann 4/6) and eventually recovered to grade 2 after undergoing static facial nerve palsy reconstruction and upper eyelid descent surgery. There has been no recurrence 13 months following surgery. 4. A 60-year-old male. Because a temporomandibular joint tumor was suspected at a nearby clinic, due to a main complaint of a right ear anterior tumor from 7 months prior, he was referred to our clinic. At the first visit, CT and MRI revealed tumorous lesions, destroying and invading the mandible and the skull base, with a contrast effect of 25 28 26 mm in the right temporomandibular joint (Fig. 8). While cytodiagnosis was performed to find a suspected giant cell tumor, it was decided to perform surgery as malignant disease could also not be ruled out. An approach was made during the temporal craniotomy and en bloc resection, including mandibular joint prosthesis and the skull base bone, was performed. The upper arm branch of the facial nerve was temporarily transected to secure the operative field and anastomotic reconstruction was performed using the large auricular nerve upon removing the tumor. The pathological results indicated that multinuclear cells and mononuclear cells solidly grow and are tumorous lesions in which hemosiderin deposits were conspicuous around the lesion, involving the cartilage tissue of the temporomandibular joint, which partly invaded the mandible as well, resulting in a
diagnosis of tenosynovial giant cell tumor. The surgical margin was negative. He developed facial nerve palsy (House– Brackmann 4/6). Four months have passed following surgery and this patient is currently under observation. 3. Discussion PVNS is also known as a diffuse type tenosynovial giant cell tumor and was classified in addition to being localized in the WHO classification in 2013 [1]. Although both have many similarities in terms of their histopathology, diffuse type differs compared with the localized type, such that invasive development to the surrounding tissues is found. In this case, infiltration into the surrounding tissues was observed, leading to a diagnosis of diffuse type. It is a benign tumor that develops in the tendons, the tendon sheath, and the joint region, with the most common being cases of knee joint development [2], which often increases to local destruction along with bone destruction. The relapse rate is as high as 25–50% or higher when occurring in the extremities, so resection while ensuring a safety area is recommended for treatment [3–5]. Although the cause is still unknown and being discussed, with reports also existing on the cause being repeated trauma of the joint area and associated arthropathy [6], it was thought to
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 6
F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
Fig. 6. Case 2 Photo of hematoxylin eosin staining pathology. (A) Multinucleated giant cell (arrowhead) and hemosiderin phagocytosis cell (arrow) were found. (B) Multinucleated giant cell (arrowhead) and hemosiderin phagocytosis cell (arrow) were found. (C) Bone invasion of the tumor is observed (arrow).
be reactive inflammation resulting from chronic inflammation. However, because trisomy 7 was found in which organization only occasionally causes distant metastasis [7–9], it was determined to be a tumor. Temporomandibular joint PVNS was reported for the first time by Lapayowker et al. [10] in 1973 and the occurrence frequency is 1.8 in 1 million people, making it an extremely rare disease. Symptoms include painful or painless tumor formation, ear claudication, hearing loss, and temporomandibular joint symptoms (click sound or opening disorder), often accompanied by intracranial development. Depression on the trigeminal nerve may cause a decrease near the face or a numbness sensation in some cases. Even among these cases, Case 1 had progression into the skull, and Cases 2, 3 and 4 found destruction of the temporomandibular joint protrusions. Furthermore, while Case 1 had repeated otitis media with effusion, no lesions were observed in the nasopharynx. With regard to the presence or absence of the causative lesion in the diagnosis of exudative otitis media with pain and temporomandibular joint symptoms in adults, it is considered important to confirm the presence or absence of nasopharyngeal lesions, while also carefully examining the image findings to differentiate any neoplasmic gaps or tumorous lesions around the temporomandibular joint.
MRI is recommended for image examination, which reflects hemosiderin deposited in the tumor and becomes low signal at T2, and is useful for differentiating from other neoplastic lesions [11,12]. Although PET examinations are often performed, it is difficult to distinguish from malignant tumors. In Case 1, SUVMax combined with extensive bone destruction was as high as 5.1, resulting in a malignant tumor being suspected. Although cytology is the first method to be examined for diagnosis, no diagnosis is made by cytodiagnosis in PVNS, and tissue diagnosis is often required in many cases [13]. Even among these cases, diagnosis by cytological diagnosis was difficult, so a biopsy was performed in 3 cases. Even if a tissue examination were carried out, the differential diagnosis would still be quite diverse. Even these cases indicated pathological results including benign and malignant tumors such as chondroma, chondrosarcoma, giant cell sarcoma, chondroblast and giant cell tumor. If malignant or PVNS is suspected, total extirpation should be performed without hesitation, with the goal of en bloc resection Reports of recurrence were also observed in PVNS in the case of temporomandibular joint development [14–16], with a recurrence rate of 15–29% and treatment requiring resection with a sufficient safety area, similar to the case of the occurrence of orthopedic surgery [16,17]. Even among these cases, Case 1 underwent a fractional resection, with local
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
7
Fig. 7. (A) Contrast-enhanced CT axial image. A tumor with partial contrast effect is found, mainly in the temporomandibular joints accompanied by calcification. (B) Contrast-enhanced CT coronal image. Although the tumor is in contact with the skull base bone, no intracranial invasion is observed. (C) MRI T2-weighted axial image. A tumor with a clear boundary of low signals is found.
recurrence coming after 22 months. Cases 2, 3, and 4 underwent en bloc resections, in which the negative pathological margin was confirmed but clear recurrence has not been found as of yet. Early detection is important because en bloc resection becomes difficult once lesions grow large. Although there are reports of a possible decrease in the recurrence rate by performing radiotherapy via postoperative radiation (35–45 Gy), if en bloc resection was impossible [18], the effect is not clear [19]. An analysis of 16 cases reported after 2011 [12–14,16– 18,20–23] showed few reports on the preservation of a facial nerve during surgery. The facial nerve was preserved in five cases and all cases with paralysis improved during the follow-up observations. However, the tumor could only be completely resected in one case, 3 cases were described as macroscopic in total and 7 cases underwent partial resection, and two cases of relapse were reported.
Recurrence is considered to be caused by the fact that a split resection is often required in order to preserve the facial nerve. In case 1, the facial nerve was preserved without cutting during the surgery. Therefore, split resection was necessary and this led to recurrence. In the other 3 cases, the facial nerve was temporarily cut and then enblock resection was achieved. Depending on the size of the tumor, it may be better to perform a complete resection with a temporary cut of the nerve to reduce the risk of recurrence. Verspoor et al. [13] reported on 95 cases of temporomandibular joint PVNS, 11 cases of which recurred, with a median to recurrence of 24 months and a maximum of 240 months. Therefore, it is considered necessary to undergo follow-up observations for a long period of time, possibly for about 10 years after PVNS treatment. MRI is considered to be useful for image inspection.
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
8
Fig. 8. (A) Contrast-enhanced CT axial image. A tumor that invades the mandible with a nonuniform contrast effect is observed. (B) Contrast-enhanced CT coronal image. A tumor, infiltrating the temporomandibular joint into the skull base bone and progressing into the cranium, is found. (C) MRI T2-weighted axial image. Tumors mixed with low signals and high signals are found, mainly in the center of the temporomandibular joint.
4. Conclusion Although temporomandibular arthroplasty PVNS has low frequency and is a rare disease, early diagnosis is important due to the gradual increase thereof and progress into the cranium. It is important to conduct an image review at an early stage, in case of the transmission hearing loss on one side, or enlargement of ear front swelling, and prolonged temporomandibular joint symptoms. It is recommended to positively perform en bloc resection, because it is difficult to differentiate by tissue examination, with malignant tumors often incapable of being excluded, and PVNS is benign but the recurrence rate is high. Furthermore, because there are some cases of recurrence which occur after a long period of time, a sufficient period is also necessary for followup observation it therefore also required. Disclosures No financial interest is declared. References [1] Fletcher CDM, Bridge JA, Hogendoorn P, Mertens F. Tenosynovial giant cell tumour, diffuse type. In: Somerthausen N, Rijn M, editors. WHO
classification of tumors of soft tissue and bone. Lyon: IARC Press; 2013. p. 102–3. [2] de Visser E, Veth RP, Pruszczynski M, Wobbes T, Van de Putte LB. Diffuse and localized pigmented villonodular synovitis:evaluation of treatment of 38 patients. Arch Orthop Trauma Surg 1999;119:401–4. [3] Verspoor FG, Zee AA, Hannink G, van der Geest IC, Veth RP, Schreuder HW. Long-term follow-up results of primary and recurrent pigmented villonodular synovitis. Rheumatology 2014;53:2063–70. [4] Schwartz HS, Unni KK, Pritchard DJ. Pigmented villonodular synovitis: a retrospective review of affected large joints. Clin orthop 1989;247:243–55. [5] Byers PD, Cotton RE, Deacon OW, Lowy M, Newman PH, Sissons HA, et al. The diagnosis and treatment of pigmented villonodular synovitis. J Bone Joint Surg Br 1968;50:290–305. [6] Myers BW, Masi AT. Pigmented villonodular synovitis and tenosynovitis: a clinical epidemiologic study of 166 cases and literature review. Medicine 1980;59:223–38. [7] Ray RA, Morton CC, Lipinski KK, Corson JM, Fletcher JA. Cytogenetic evidence of clonality in a case of pigmented villonodular synovitis. Cancer 1991;67:121–5. [8] Sistla R, Jvs V, Afroz T. Malignant pigmented villonodular synovitis-a rare entity. J Orthop Case Rep 2014;4:9–11. [9] Righi A, Gambarotti M, Sbaraglia M, Frisoni T, Donati D, Vanel D, et al. Metastasizing tenosynovial giant cell tumour, diffuse type/pigmented villonodular synovitis. Clin Sarcoma Res 2015;6:15. [10] Lapayowker MS, Miller WT, Levy WM, Harwick RD. Pigmented villonodular synovitis of the temperomandibular joint. Radiology 1973;108:313–6. [11] Bemporad JA, Chaloupka JC, Putman CM, Roth TC, Tarro J, Mitra S, et al. Pigmented villonodular synovitis of the temporomandibular joint:
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021
G Model
ANL-2534; No. of Pages 9 F. Nomura et al. / Auris Nasus Larynx xxx (2018) xxx–xxx
[12]
[13]
[14]
[15] [16]
[17]
diagnostic imaging and endovascular therapeutic embolization of a rare head and neck tumor. Am J Neuroradiol 1999;20:159–62. Wong JJ, Phal PM, Wiesenfeld D. Pigmented villonodular synovitis of the temporomandibular joint: a radiologic diagnosis and case report. J Oral Maxillofac Surg 2012;70:126–34. Verspoor FGM, Mastboom MJL, Weijs WLJ, Koetsveld AC, Schreuder HWB, Flucke U. Treatments of tenosynovial giant cell tumours of the temperomandibular joint: a report of three cases and a review of literature. Int J Oral Maxillofac Surg 2018;47:1288–94. Pina S, Fernandez M, Maya S, Garcia RA, Noor A, Pawha PS, et al. Recurrent temporal bone tenosynovial giant cell tumor with chondroid metaplasia: the use of imaging to assess recurrence. Neuroradiol J 2014;27:97–101. Allen A, Saran N. Recurrent pigmented villonodular synovitis of the temporomandibular joint. Radiol Case Rep 2018;13:499–502. Safaee M, Oh T, Sun MZ, Parsa AT, McDermott MW, El-Sayed IH, et al. Pigmented villonodular synovitis of the temporomandibular joint with intracranial extension: a case series and systematic review. Head Neck 2015;37:1213–24. Carlson ML, Osetinsky LM, Alon EE, Inwards CY, Lane JI, Moore EJ. Tenosynovial giant cell tumors of the temporomandibular joint and lateral skull base: review of 11 cases. Laryngoscope 2017;127:2340–6.
9
[18] Joshi K, Huang B, Scanga L, Buchman C, Chera BS. Postoperative radiotherapy for diffuse pigmented villonodular synovitis of the temporomandibular joint. Am J Otolaryngol 2015;36:106–13. [19] Verspoor FG, van der Geest IC, Vegt E, Veth RP, van der Graaf WT, Schreuder HW. Pigmented villonodular synovitis: current concepts about diagnosis and management. Future Oncol 2013;9:1515–31. [20] Bredell M, Schucknecht B, Bode-Lesniewska B. Tenosynovial, diffuse type giant cell tumor of the temporomandibular joint, diagnosis and management of a rare tumor. J Clin Med Res 2015;7:262–6. [21] Roman˜ach MJ, Brasileiro BF, Leo´n JE, Alves DB, de Almeida OP, Vargas PA. Pigmented villonodular synovitis of the temporomandibular joint: case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;111:e17–28. [22] Damodar D, Chan N, Kokot N. Pigmented villonodular synovitis of the temporomandibular joint: case report and review of the literature. Head Neck 2015;37(12):E194–9. [23] Vellutini EAS, Alonso N, Arap SS, Godoy LFS, Souza E, Souza RA, Mattedi RL, et al. Functional reconstruction of temporomandibular joint after resection of pigmented villonodular synovitis with extension to infratemporal fossa and skull base: a case report. Surg J (N Y) 2016;2:e78–82.
Please cite this article in press as: Nomura F, et al. Pigmented villonodular synovitis occurring in the temporomandibular joint. Auris Nasus Larynx (2018), https://doi.org/10.1016/j.anl.2018.10.021