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Dang RR, Noonan V, Chigurupati R, Henry A. Treatment of tophaceous pseudogout in the temporomandibular joint with resection and alloplastic reconstruction: a single-staged approach. Oral Maxillofac Surg 2021; 26:505-509. [PMID: 34596805 DOI: 10.1007/s10006-021-01013-2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/18/2021] [Accepted: 09/23/2021] [Indexed: 11/26/2022]
Abstract
Tophaceous pseudogout is a rare variant of the calcium pyrophosphate dihydrate (CPPD) disorder, with predilection for the TMJ. It is characterized by calcific deposits composed of rhomboid- or rectangular-shaped crystals that exhibit birefringence when examined under polarized light. We hereby present a case of a 65-year-old man with left pre-auricular tenderness and malocclusion. CT imaging was notable for a left TMJ mineralized mass with erosion of the condylar head. Treatment involved excision of the mass with eminectomy, condylectomy, and a stock total TMJ reconstruction. In this report, important considerations for diagnosis, biopsy, and surgical treatment with emphasis on reconstruction for tophaceous pseudogout of the TMJ have been highlighted by the authors.
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Affiliation(s)
- Rushil R Dang
- Department of Oral and Maxillofacial Surgery, Boston Medical Center, Boston, MA, 02118, USA.
| | - Vikki Noonan
- Division of Oral and Maxillofacial Pathology, Boston University Henry M. Goldman School of Dental Medicine, 635 Albany Street, Boston, MA, 02118, USA
| | - Radhika Chigurupati
- Department of Oral and Maxillofacial Surgery, Boston Medical Center, Boston, MA, 02118, USA
| | - Andrew Henry
- Department of Oral and Maxillofacial Surgery, Boston Medical Center, Boston, MA, 02118, USA
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Tang T, Han FG. Calcium pyrophosphate deposition disease of the temporomandibular joint invading the middle cranial fossa: Two case reports. World J Clin Cases 2021; 9:2662-2670. [PMID: 33889634 PMCID: PMC8040178 DOI: 10.12998/wjcc.v9.i11.2662] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/15/2020] [Revised: 01/03/2021] [Accepted: 02/10/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Pseudogout is a benign joint lesion caused by the deposition of calcium pyro-phosphate dihydrate crystals, but it is invasive. Pseudogout of the temporo-mandibular joint (TMJ) is uncommon, and it rarely invades the skull base or penetrates into the middle cranial fossa. The disease has no characteristic clinical manifestations and is easily misdiagnosed.
CASE SUMMARY We present two cases of tophaceous pseudogout of the TMJ invading the middle cranial fossa. A 46-year-old woman with a history of diabetes for more than 10 years was admitted to the hospital due to swelling and pain in the right temporal region. Another patient, a 52-year-old man with a mass in the left TMJ for 6 years, was admitted to the hospital. Maxillofacial imaging showed a calcified mass and severe bone destruction of the skull base in the TMJ area. Both patients underwent excision of the lesion. The lesion was pathologically diagnosed as tophaceous pseudogout. The symptoms in these patients were relieved after surgery.
CONCLUSION Tophaceous pseudogout should be considered when there is a calcified mass in the TMJ with or without bone destruction. A pathological examination is the gold standard for diagnosing this disease. Surgical treatment is currently the recommended treatment, and the prognosis is good after surgery.
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Affiliation(s)
- Ting Tang
- Department of Radiology, The Affiliated Hospital of Southwest Medical University, Luzhou 646000, Sichuan Province, China
| | - Fu-Gang Han
- Department of Radiology, The Affiliated Hospital of Southwest Medical University, Luzhou 646000, Sichuan Province, China
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Loro LL, Bjørnland T. Calcium pyrophosphate deposition disease: A case report with bilateral involvement of the temporomandibular joints and concurrence of psoriatic arthritis. Clin Case Rep 2020; 8:640-643. [PMID: 32274026 PMCID: PMC7141746 DOI: 10.1002/ccr3.2715] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/19/2019] [Revised: 12/03/2019] [Accepted: 01/02/2020] [Indexed: 12/02/2022] Open
Abstract
Calcium pyrophosphate dehydrate deposition (CPDD) disease very rarely affects the temporomandibular joint (TMJ). It may resemble synovial chondromatosis, chondrosarcoma, chondroblastoma, or a parotid tumor. Clinical examination, CT, and MRI are important in making the correct diagnosis. Surgical removal of CPDD is necessary with or without excision of the TMJ.
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Affiliation(s)
- Lado Lako Loro
- Section of Maxillofacial SurgeryDepartment of Ophthalmology, Otolaryngology and Maxillofacial SurgeryMøre and Romsdal Hospital TrustÅlesund HospitalÅlesundNorway
| | - Tore Bjørnland
- Department of Oral Surgery and Oral MedicineFaculty of DentistryUniversity of OsloOsloNorway
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Hotokezaka Y, Hotokezaka H, Katayama I, Fujita S, Sasaki M, Eida S, Uetani M. A case of tophaceous pseudogout of the temporomandibular joint extending into the cranium. Oral Radiol 2019; 36:203-208. [PMID: 31559516 DOI: 10.1007/s11282-019-00410-4] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/01/2019] [Accepted: 09/11/2019] [Indexed: 02/07/2023]
Abstract
A case of tophaceous pseudogout (i.e., calcium pyrophosphate dihydrate crystal deposition disease) in the temporomandibular joint (TMJ) extending into the cranium is reported. A 59-year-old woman was referred to hospital with swelling and pain in the left cheek, and with trismus. Computed tomography imaging revealed a large, granular, calcified mass surrounding the left condylar head, partly destroying the cortex of the condylar head, and extending into the cranium by destroying the glenoid fossa. Magnetic resonance imaging revealed that the soft-tissue mass was of low-signal intensity on T1- and T2-weighted images, and was enhanced after intravenous injection of gadolinium. The mass was clinically and radiographically suspected to be a neoplastic lesion or a synovial osteochodromatosis. However, histological analysis demonstrated that the mass contained granulomatous lesion due to multiple nodular deposits of numerous rod-shaped and rhomboid crystals, which verified the diagnosis of tophaceous pseudogout. The lesion was excised surgically using a preauricular approach. Neither radiographic nor clinical examination demonstrated any signs of mass recurrence in the long-term 8- and 14-year postoperative recall examinations. Tophaceous pseudogout is a rare benign arthropathy that presents with clinical and radiographic features mimicking neoplastic conditions of the TMJ. Therefore, it is recommended that tophaceous pseudogout is considered in the differential diagnosis when a calcified mass lesion of the TMJ is encountered.
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Affiliation(s)
- Yuka Hotokezaka
- Department of Radiology and Cancer Biology, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki, 852-8588, Japan
| | - Hitoshi Hotokezaka
- Department of Orthodontics and Dentofacial Orthopedics, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki, 852-8588, Japan
| | - Ikuo Katayama
- Department of Radiology and Cancer Biology, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki, 852-8588, Japan.
| | - Shuichi Fujita
- Department of Oral Pathology, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki, 852-8588, Japan
| | - Miho Sasaki
- Department of Radiology and Cancer Biology, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki, 852-8588, Japan
| | - Sato Eida
- Department of Radiology and Cancer Biology, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki, 852-8588, Japan
| | - Masataka Uetani
- Department of Radiological Sciences, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki, 852-8588, Japan
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Fan J, Heimann A, Wu M. Temporal mandibular joint chondrocalcinosis (tophaceous pseudogout) diagnosed by ultrasound-guided fine-needle aspiration. Diagn Cytopathol 2019; 47:803-807. [PMID: 30908901 DOI: 10.1002/dc.24181] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/12/2018] [Revised: 02/20/2019] [Accepted: 03/12/2019] [Indexed: 12/11/2022]
Abstract
Tophaceous pseudogout is a calcium pyrophosphate dihydrate crystal (CPPD) deposition disease that frequently affects elderly patient in the temporomandibular joint (TMJ). A diagnosis of CPPD deposition disease in the TMJ is challenging due to its mimicking of other benign and malignant entities. Surgical exploration followed by histologic examination is by far the most frequently used diagnostic modality. We present a case of an 87-year-old female who presented with a right TMJ mass. A final diagnosis of tophaceous pseudogout was made on cellular material obtained by ultrasound-guided fine-needle aspiration (US-guided FNA). Based on our case and current available literature, ultrasound-guided FNA is a reliable tool for diagnosing tophaceous pseudogout of the TMJ.
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Affiliation(s)
- Jun Fan
- Department of Pathology, Stony Brook University Hospital, Stony Brook, New York
| | - Alan Heimann
- Department of Pathology, Stony Brook University Hospital, Stony Brook, New York
| | - Maoxin Wu
- Department of Pathology, Stony Brook University Hospital, Stony Brook, New York
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Laviv A, Sadow PM, Keith DA. Pseudogout in the Temporomandibular Joint With Imaging, Arthroscopic, Operative, and Pathologic Findings. Report of an Unusual Case. J Oral Maxillofac Surg 2015; 73:1106-12. [DOI: 10.1016/j.joms.2014.12.041] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/01/2014] [Revised: 12/28/2014] [Accepted: 12/30/2014] [Indexed: 12/11/2022]
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Tophaceous pseudogout of the temporomandibular joint: a series of 3 cases. Oral Surg Oral Med Oral Pathol Oral Radiol 2013; 117:369-75. [PMID: 24528794 DOI: 10.1016/j.oooo.2013.12.007] [Citation(s) in RCA: 27] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/24/2013] [Revised: 12/03/2013] [Accepted: 12/04/2013] [Indexed: 12/13/2022]
Abstract
OBJECTIVE We report a series of 3 cases of tophaceous pseudogout of the temporomandibular joint (TMJ). STUDY DESIGN Three patients, two men and one woman, ranging in age between 60 and 75 years, presented with unilateral painful swelling of the TMJ area associated with limitation of mouth opening. RESULTS Radiographic and computed tomographic images showed opaque masses in the supracondylar region of the TMJ. The preoperative clinical impression was a "neoplastic lesion" in the 3 cases. Microscopic examination revealed numerous deeply basophilic masses of calcified deposits, exhibiting birefringence under polarized light and morphologically consistent with calcium pyrophosphate dihydrate deposition, referred to in these cases as "tophaceous pseudogout." CONCLUSION Tophaceous pseudogout is a rare benign arthropathy that presents with clinical and radiographic features mimicking neoplastic conditions of the TMJ. Therefore, it is recommended that tophaceous pseudogout be considered in the differential diagnosis when a TMJ is involved with "neoplasm-like" lesions clinically and radiographically.
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Srinivasan V, Wensel A, Dutcher P, Newlands S, Johnson M, Vates GE. Calcium pyrophosphate deposition disease of the temporomandibular joint. J Neurol Surg Rep 2013; 73:6-8. [PMID: 23946918 PMCID: PMC3658656 DOI: 10.1055/s-0032-1329190] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/12/2022] Open
Abstract
Calcium pyrophosphate dihydrate deposition disease (CPDD, tophaceous pseudogout) is a rare crystal arthropathy characterized by calcium pyrophosphate crystal deposition in joint spaces, episodes of synovitis, and radiological features of chondrocalcinosis. We present a case of 61-year-old woman who presented with left temporomandibular joint (TMJ) pain, difficulty chewing, left facial numbness, left-sided hearing loss, and left TMJ swelling. Imaging of the temporal fossa revealed a large mass emanating from the temporal bone at the TMJ, extending into the greater wing of the sphenoid and involving the mastoid bone and air cells posteriorly. Fine needle aspiration demonstrated polarizable crystals with giant cells. Intraoperatively, the TMJ was completely eroded by the mass. Final pathology was consistent with tophaceous pseudogout. CPDD has rarely been reported involving the skull base. None of the cases originally described by McCarty had TMJ pseudogout. Symptoms are generally pain, swelling, and hearing loss. Management is nearly always surgical with many patients achieving symptomatic relief with resection. CPDD is associated with many medical problems (including renal failure, gout, and hyperparathyroidism), but our patient had none of these risk factors. This case demonstrates that CPDD can involve the skull base and is best treated with skull base surgical techniques.
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Affiliation(s)
- Vasisht Srinivasan
- Department of Neurosurgery, University of Rochester Medical Center, Rochester, New York
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Mikami T, Kumagai A, Aomura T, Javed F, Sugiyama Y, Mizuki H, Takeda Y. Cytopathologic diagnosis on joint lavage fluid for patients with temporomandibular joint disorders. Diagn Cytopathol 2013; 42:30-6. [DOI: 10.1002/dc.23023] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/19/2012] [Revised: 03/21/2013] [Accepted: 06/03/2013] [Indexed: 11/06/2022]
Affiliation(s)
- Toshinari Mikami
- Division of Anatomical and Cellular Pathology; Department of Pathology; Iwate Medical University; 2-1-1 Nishitokuta Yahaba Shiwa-gun Iwate 028-3694 Japan
| | - Akiko Kumagai
- Division of Maxillofacial Surgery; Department of Oral and Maxillofacial Surgery; School of Dentistry; Iwate Medical University; 19-1 Uchimaru Morioka Iwate 020-8505 Japan
| | - Tomoyuki Aomura
- Division of Maxillofacial Surgery; Department of Oral and Maxillofacial Surgery; School of Dentistry; Iwate Medical University; 19-1 Uchimaru Morioka Iwate 020-8505 Japan
| | - Fawad Javed
- Engineer Abdullah Bugshan Research Chair for Growth Factors and Bone Regeneration; 3D Imaging and Biomechanical Laboratory; College of Applied Medical Sciences; King Saud University; Riyadh 11545 Saudi Arabia
| | - Yoshiki Sugiyama
- Division of Maxillofacial Surgery; Department of Oral and Maxillofacial Surgery; School of Dentistry; Iwate Medical University; 19-1 Uchimaru Morioka Iwate 020-8505 Japan
| | - Harumi Mizuki
- Division of Maxillofacial Surgery; Department of Oral and Maxillofacial Surgery; School of Dentistry; Iwate Medical University; 19-1 Uchimaru Morioka Iwate 020-8505 Japan
| | - Yasunori Takeda
- Division of Anatomical and Cellular Pathology; Department of Pathology; Iwate Medical University; 2-1-1 Nishitokuta Yahaba Shiwa-gun Iwate 028-3694 Japan
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Meng J, Guo C, Luo H, Chen S, Ma X. A case of destructive calcium pyrophosphate dihydrate crystal deposition disease of the temporomandibular joint: a diagnostic challenge. Int J Oral Maxillofac Surg 2011; 40:1431-7. [DOI: 10.1016/j.ijom.2011.05.007] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/20/2010] [Revised: 04/25/2011] [Accepted: 05/10/2011] [Indexed: 01/16/2023]
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Zweifel D, Ettlin D, Schuknecht B, Obwegeser J. Tophaceuos calcium pyrophosphate dihydrate deposition disease of the temporomandibular joint: the preferential site? J Oral Maxillofac Surg 2011; 70:60-7. [PMID: 21855194 DOI: 10.1016/j.joms.2011.06.207] [Citation(s) in RCA: 24] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/16/2011] [Revised: 06/17/2011] [Accepted: 06/18/2011] [Indexed: 02/07/2023]
Affiliation(s)
- Daniel Zweifel
- Department of Craniomaxillofacial and Oral Surgery, University Hospital Zurich, Zurich, Switzerland.
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Sklenicka S, Dierks EJ, Jarmin J, Miles C. Pseudogout of the temporomandibular joint: an uncommon cause of temporomandibular joint pain and swelling. ACTA ACUST UNITED AC 2010; 111:709-14. [PMID: 21167760 DOI: 10.1016/j.tripleo.2010.07.018] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/12/2010] [Accepted: 07/17/2010] [Indexed: 02/07/2023]
Abstract
BACKGROUND Pseudogout, or calcium pyrophosphate deposition, is a rare cause of pain, swelling, and trismus of the temporomandibular joint (TMJ). Diagnosis and management of the lesion are discussed. CASE DESCRIPTION A 58-year-old female had a 2-month history of progressive swelling of right TMJ associated with trismus and facial pain. Imaging of the TMJ revealed a mixed radiolucent and radiopaque lesion associated with the right TMJ joint space. Surgical excision was performed successfully via preauricular approach. Pathology was consistent with calcium pyrophosphate deposition of the TMJ, also known as pseudogout. Surgical excision successfully treated her symptoms as expected. She is now disease free without recurrence. CLINICAL IMPLICATIONS Pseudogout is a rare cause of TMJ pain, swelling, and trismus that should be included in the differential of joint pain and dysfunction. It can be treated successfully with surgery.
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Affiliation(s)
- Scott Sklenicka
- North Florida Oral and Facial Surgery, Oral and Maxillofacial Surgery, Oregon Health and Science University, Jacksonville, FL, USA
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Takata Y, Koeda S, Inahara H, Kumamoto H, Kawamura H. Tophaceous pseudogout (tumoral calcium pyrophosphate dihydrate crystal deposition disease) of the temporomandibular joint: A case report. ACTA ACUST UNITED AC 2010. [DOI: 10.1016/j.ajoms.2009.11.006] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/23/2022]
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Kalish LH, Ng T, Kalnins I, Da Cruz MJ. Pseudogout mimicking an infratemporal fossa tumor. Head Neck 2009; 32:127-32. [PMID: 19283846 DOI: 10.1002/hed.21059] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/12/2022] Open
Abstract
BACKGROUND The infratemporal fossa is a well-concealed, complex anatomical space. Neoplasms arising in this area are heterogenous in nature and have an insidious onset and usually present late. METHODS AND RESULTS We present a case of a 71-year-old woman who presented with worsening facial pain, trismus, and a large mass in her infratemporal fossa with minimal associated temporomandibular joint destruction. She underwent a surgical excision of the mass, which revealed a pseudogout deposit. CONCLUSION The clinical and radiological features of patients with tophaceous pseudogout frequently mimic those of a benign or malignant neoplasm of the infratemporal fossa, often resulting in more radical surgery.
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Affiliation(s)
- Larry H Kalish
- Department of Otorhinolaryngology, Westmead Hospital, Sydney, Australia.
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Mikami T, Takeda Y, Ohira A, Hoshi H, Sugiyama Y, Yoshida Y, Ambo J. Tumoral calcium pyrophosphate dihydrate crystal deposition disease of the temporomandibular joint: Identification on crystallography. Pathol Int 2008; 58:723-9. [DOI: 10.1111/j.1440-1827.2008.02300.x] [Citation(s) in RCA: 14] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/20/2022]
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Abstract
PURPOSE OF REVIEW This review is an attempt to keep current in the sparse literature addressing the still underutilized area of crystal identification. RECENT FINDINGS The emphasis has been on the subtleties of the microscopic identification of common crystals and other less common potentially confusing crystals. Imaging is noted to provide increasing help, but microscopic crystal identification remains the gold standard. SUMMARY Quality control is still a concern as is the infrequency of attempted arthrocentesis for crystal identification.
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Affiliation(s)
- Lan X Chen
- VA Medical Center, Rheumatology Research 151K, Philadelphia, Pennsylvania 19104, USA
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