The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
FIGURE 4. The intraoperative view of the bony projection.
FIGURE 5. Histopathologic findings (hematoxylin-eosin staining; original magnification, 40).
deformity seemed irreversible after a 5-month follow-up. So a surgical revision was performed. More than a century ago, Wolff14 formulated his law of bone remodeling stating that bone issue adapts to functional stress by changes in structure and mass. From then on, a great number of researches, in vivo and in vitro, have been carried out to investigate the response of the bone to mechanical stimuli and the underlying mechanism.15–17 The bone remodeling secondary to tissue expansion may be considered as a special situation following the Wolff law. Compression loaded on the bone is the definitive factor determining the changes of the bone. However, there seemed to be many other factors contributing to the final result of bone remodeling, including age and sex of the patient, the duration of expansion, the intraexpander pressure and so forth. Stuehmer et al18 and von See et al19 described bone resorption on the interface of hydrogel expander and underlying bone. They believed separating the surface of expander from the underlying bone, with polydioxanone foil and titanium plates, respectively, could prevent the resorption of bone. That is to say, the expander itself contributed to the absorption of the bone. Menick20 advocated that the cheek should be repaired with a cheek flap, although the composite defect encompasses the nose and adjacent cheek. In this case, the 2 expanded flaps met at the nasal-face junction. After the removal of expanders, no changes of the maxilla were detected. In this patient, the forehead expander was inserted into a pocket under the frontalis muscle(ie, supraperiosteal plane) and contacted with the underlying frontal bone. The cheek expander, on the contrary, was inserted into a subcutaneous pocket where the expander did not touch the surface of the mandible or its periosteum. This may partially explain the differences of bone change between the frontal bone and the maxilla after tissue expansion according to Stuehmer et al18 and von See et al.19 As Calobrace and Downey21 pointed out, this case demonstrated not only the profound bony deformity that can result from tissue expansion but also the striking ability of the pediatric skull to remold. Hence, complications of bone deformity should be noticed in children undergoing skin expansion.
REFERENCES 1. Radovan C. Tissue expansion in soft-tissue reconstruction. Plast Reconstr Surg 1984;74:482–492 2. Gibstein LA, Abramson DL, Bartlett RA, et al. Tissue expansion in children: a retrospective study of complications. Ann Plast Surg 1997;38:358–364 3. Manders EK, Schenden MJ, Furrey JA, et al. Soft-tissue expansion: concepts and complications. Plast Reconstr Surg 1984;74:493–507
Brief Clinical Studies
4. Hemmer KM, Marsh JL, Picker S. Calvarial erosion after scalp expansion. Ann Plast Surg 1987;19:454–459 5. Fudem GM, Orgel MG. Full-thickness erosion of the skull secondary to tissue expansion for scalp reconstruction. Plast Reconstr Surg 1988;82:368–369 6. Paletta CE, Bass J, Shehadi SI. Outer table skull erosion causing rupture of scalp expander. Ann Plast Surg 1989;23:538–542 7. McKinney P, Edelson R, Terrasse A, et al. Chest-wall deformity following soft-tissue expansion for breast reconstruction. Plast Reconstr Surg 1987;80:442–444 8. Tirone L, La Rusca I, Ciccarelli F. Rib fracture as a complication of tissue expansion in breast reconstruction. Plast Reconstr Surg 2010;126:2290–2291 9. Tseng J, Huang AH, Wong MS, et al. Rib fractures: a complication of radiation therapy and tissue expansion for breast reconstruction. Plast Reconstr Surg 2010;125:65e–66e 10. Wallis KL, Gurusinghe AD, Anwar MU, et al. Osteophyte-induced rupture of a scalp tissue expander. Ann Plast Surg 2011;67:606–607 11. Colonna M, Cavallini M, De Angelis A, et al. The effects of scalp expansion on the cranial bone: a clinical, histological, and instrumental study. Ann Plast Surg 1996;36:255–260 12. Schmelzeisen R, Schimming R, Schwipper V, et al. Influence of tissue expanders on the growing craniofacial skeleton. J Craniomaxillofac Surg 1999;27:153–159 13. El-Saadi MM, Nasr MA. The effect of tissue expansion on skull bones in the paediatric age group from 2 to 7 years. J Plast Reconstr Aesthet Surg 2008;61:413–418 14. Wolff J. Das gesetz der transformation der knochen, Berlin, Germany: Verlag von August Hirschwald; 1892 15. Klein-Nulend J, van der Plas A, Semeins CM, et al. Sensitivity of osteocytes to biomechanical stress in vitro. FASEB J 1995;9:441–445 16. Rubin CT, Lanyon LE. Kappa Delta Award paper. Osteoregulatory nature of mechanical stimuli: function as a determinant for adaptive remodeling in bone. J Orthop Res 1987;5:300–310 17. Burger EH, Klein-Nulen J. Responses of bone cells to biomechanical forces in vitro. Adv Dent Res 1999;13:93–98 18. Stuehmer C, Ruker M, Schumann P, et al. Osseous alterations at the interface of hydrogel expanders and underlying bone. J Craniomaxillofac Surg 2009;37:258–262 19. von See C, Rucker M, Schumann P, et al. Micro-computed tomography and histologic evaluation of the interface of hydrogel expander and underlying bone: influence of pressure distributors on bone resorption. J Oral Maxillofac Surg 2010;68:2179–2184 20. Menick FJ. Nasal reconstruction. Plast Reconstr Surg 2010;125:138e–150e 21. Calobrace MB, Downey SE. Calvarial deformity and remodeling following prolonged scalp expansion in a child. Ann Plast Surg 1997;39:186–189
Pigmented Villonodular Synovitis of the Temporomandibular Joint With Intracranial Extension Ying Chen, BS, Xie-Yi Cai, MD, Chi Yang, MD, Min-Jie Chen, MD, Ya-Ting Qiu, MD, Ziang Zhuo, MS Abstract: Pigmented villonodular synovitis is an uncommon benign tumor-like proliferative lesion with an undetermined origin. Involvement of the temporomandibular joint is uncommon. Although pigmented villonodular synovitis is a benign lesion, it can grow with an aggressive pattern, and it extends extra-articularly in most of the reported cases, about one-third of them exhibiting intracranial involvement. The authors reported an additional case of a 47-year-old
© 2015 Mutaz B. Habal, MD
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woman with intracranial extension, who had a history of joint pain and trismus. The preoperative diagnosis was made with arthroscopy. The lesion was completely excised via preauricular approach and condylotomy. The bone defect was covered by the pedicled temporalis myofascial fat flap. The patient has been symptom-free for 40 months postoperatively. Key Words: Temporomandibular joint, pigmented villonodular synovitis, skull base, treatment
P
igmented villonodular synovitis (PVNS) is an uncommon benign tumor-like proliferative lesion with an undetermined origin arising from the synovial membranes of joints, bursas, and tendon sheaths, which was first fully described by Jaffe et al1 in 1941. Its etiology remains elusive. Possible etiologies include disturbances of lipid metabolism, neoplasm, inflammation, trauma, and hemorrhage.2,3 It has an annual incidence of 1.8 cases per million population.4 The knee is commonly involved in 80% of cases, but any joint may be affected.5 Most cases are monoarticular and do not metastasize, although they may be locally destructive. Involvement of the temporomandibular joint (TMJ) is uncommon, and about 60 cases have been published in the English-language literature to our knowledge. The predominant symptoms in the TMJ are preauricular mass or swelling, pain, and limitation of mandibular movement. Although PVNS is a benign lesion, it can grow with an aggressive pattern, and it extends extra-articularly in most of the reported cases, about one-third of them exhibiting intracranial involvement.6–13 It is commonly accepted that surgery is the only curative treatment for PVNS, with partial or complete excision of the synovium and involved bones. Despite these maneuvers, a relatively high rate of recurrence of PVNS in other joints has been reported, in the range of 33% to 50%.14 Therefore, some authors proposed that radiotherapy could be used in the management of recurrent cases with moderate radiation doses.5 All reported PVNSs in TMJ have been treated by surgical excision, and we reported 4 cases managed with arthroscopy.15,16 However, it is difficult to predict the recurrence rate in TMJ because of the lack of long-term follow-up information. Herein, we report an additional case of PVNS in TMJ with intracranial extension.
CLINICAL REPORT In November 2010, a 47 year-old woman was referred to the Department of Oral and Maxillofacial Surgery, Ninth People’s Hospital, Shanghai Jiao Tong University School of Medicine, with a chief From the Department of Oral and Maxillofacial Surgery, Ninth People’s Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai Key Laboratory of Stomatology, Shanghai, China. Received June 12, 2014. Accepted for publication September 13, 2014. Address correspondence and reprint requests to Chi Yang, MD, and Xie-Yi Cai, MD, Department of Oral & Maxillofacial Surgery, Shanghai Ninth People’s Hospital, Shanghai Jiaotong University, 639 Zhi Zao Ju Rd, Shanghai 200011, People’s Republic of China; E-mail: [email protected]; [email protected] This project was supported by the National Natural Science Foundation of China (grant 81200766) and Science and Technology Commission of Shanghai (13140902702). The authors report no conflicts of interest. Copyright © 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001341
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FIGURE 1. Magnetic resonance imaging scan of the TMJ of the 47-year-old female patient showing joint effusion with capsular expansion and a mass with mixed signal intensity. A, In the parasagittal plane. B, In the coronal plane.
complaint of pain in the left TMJ region that had developed 1 year ago and progressively increased over the past 2 months. She had no hearing loss and facial nerve palsy. Physical examination revealed a symmetric face with no swelling or mass over the left preauricular region, a maximal mouth opening of 28 mm, slight deviation of the mandibular midline, and a normal occlusion. The patient had no history of trauma or rheumatoid arthrosis. A magnetic resonance imaging (MRI) scan of the TMJ revealed joint effusion with capsular expansion and a mass with mixed signal intensity in the anterior recess in T1-weighted images on the left side (Figs. 1A, B). Clinical and radiological findings prompted a diagnosis of synovial chondromatosis or synovitis on the left TMJ. To clarify the diagnosis, a diagnostic arthroscopy of the left TMJ was performed under local anesthesia in November 2010. Under arthroscope, the articular disc was seen to be in normal position, and a widespread orange synovium in the anterior recess was noticed (Fig. 2). A large brown hyperplastic nodule occupied the medial part of anterior recess and medial groove, which was fragile and prone to bleed. It extended to the glenoid fossa, and the cartilage surface was involved. According to the arthroscopic features, a provisional diagnosis of PVNS was given. To investigate the extent of the lesion, an enhanced computed tomographic (CT) scan was applied after arthroscopy, which showed that the medial joint space was obviously widened, and the corresponding TMJ fossa and skull base were thinned and perforated. However, no condylar resorption or destruction was revealed on CT scans (Fig. 3). Three days later, the patient underwent surgical excision of the lesion via a modified preauricular approach under general anesthesia. The lesion was located in the anteromedial side of the TMJ and extended superiorly and medially into the middle cranial fossa, with erosion of the glenoid fossa. It was brown, with a soft and friable texture(Fig. 4). For better exposure of the lesion, condylar osteotomy was performed at the level of the condylar neck. Frozen biopsy was obtained, and giant cell granuloma was diagnosed during the operation. The lesion was completely excised, and the involved retrodiscal tissue was also removed. On closer examination, the lesion appeared to be protruding onto the dura mater but had not perforated it. Because of the erosion and perforation of the skull base, the temporalis myofascial fat flap with the pedicle of middle temporal vessels was used to cover the cranial bone defect. Finally, the condyle was repositioned and fixed with 2 mini–titanium plates. Patient's occlusion did not change after surgery. Histologic examination disclosed that the lesion was composed of predominantly plump histiocytes with intermixed, variably distributed multinucleated giant cells, and the mass was heavily pigmented with hemosiderin (Fig. 5). So a final diagnosis of PVNS was given.
FIGURE 2. Arthroscopic vision showing a widespread orange synovium.
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
FIGURE 3. Preoperative CT showing the widened medial joint space and the thinned and perforated skull base.
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FIGURE 5. Histological examination showed that the lesion was composed of predominantly plump histiocytes with intermixed multinucleated giant cells and deposits of hemosiderin (hematoxylin-eosin staining. original magnification 200).
The postoperative course was uneventful. A 40-month postoperative follow-up revealed good symmetry, normal occlusion, and no restriction of jaw movement. Panoramic radiograph, CT scan, and MRI showed no evidence of recurrence. The condyle grew well. The cranial bone defect was getting smaller, although it has not completely healed (Figs. 6A, B).
DISCUSSION Pigmented villonodular synovitis is a benign, yet locally aggressive proliferative lesion, which preferentially affects the synovial tissue of the large joints, such as the knee and hip of adult patients in their third to fifth decades of life.1–3 It can involve any articular site, including the ankle, shoulder, and elbow and rarely involves the joints of the hands, feet, spine, and TMJ. To the best our knowledge, about 60 patients with PVNS affecting the TMJ region have been reported in English-written literatures and about one-third of the cases present intracranial extension.6–13 The most common clinical manifestations for the patients with intracranial extension are painful/painless preauricular swelling/mass, trismus, altered hearing, and tinnitus. Advanced imaging methods, including contrast-enhanced CT and MRI, are helpful to the correct diagnosis. The glenoid fossa erosions and intracranial extension, associated with peri-articular soft tissue swelling with or without calcification, can be demonstrated on CT.17 The characteristic feature of the lesion on MRI is an intermediate- to low-intensity signal on T1W images and a low intensity on T2-weighted depending on the hemosiderin concentration.18 The present case reported a history of arthralgia and limitation of mouth opening, without preauricular swelling or mass. Joint effusion with capsular expansion and a mass with mixed signal intensity was found on MRI. Therefore, a preoperative diagnosis of synovial chondromatosis was given, and the patient underwent a diagnostic arthroscopy. Then, a provisional diagnosis of PVNS was made because of its typical manifestation under arthroscope. Similar with our reported cases with intra-articular PVNS,15,16 it is difficult to make an early diagnosis for the present case because of atypical symptoms and unspecific imaging information. It has been reported that fine-needle aspiration biopsy is a relatively simple method to establish the preoperative diagnosis for PVNS. In our patient, it is almost impossible to perform fine-needle aspiration biopsy, but diagnostic arthroscopy is very helpful for early diagnosis of PVNS. Furthermore, contrast-enhanced CT scan is necessary for investigating the extent of the lesion and bony erosion. Intracranial extension has been finally found with CT scan for the present patient. It is commonly accepted that a complete excision with synovectomy and/or capsulectomy is the treatment choice for PVNS.
FIGURE 4. Transoperative view showing the lesion was located in the anteromedial side of the TMJ and extended superiorly and medially into the middle cranial fossa.
FIGURE 6. The follow-up radiological images showing no evidence of recurrence at 40 months after operation: A, Computed tomography scan. B, Magnetic resonance imaging scan.
Conversely, an incomplete resection will result in a high rate of recurrence. Some authors proposed that radiotherapy could be used in the management of recurrent cases with moderate radiation doses.5 For the patients with intracranial involvement, a coronal or hemicoronal incision with preauricular and frontal extension is often used to expose the lesion. Besides complete excision of the lesion, craniectomy and/or condylectomy may be performed when the condyle is also involved. Because malignancy or malignant transformation of the lesion has not been reported, dura mater was not excised except for 1 patient.13 Radiotherapy has been used after excision in 3 patients. Even so, recurrence of PVNS was found in 1 of the 3 patients 3 years later.13 For the present case, the lesion was located in the medial part of the joint, and no condylar bone destruction was shown. Therefore, a modified preauricular approach was used, and condylar osteotomy was performed for better exposure. The dura mater was intact, and it remained untreated. We thought that craniectomy was not necessary, because PVNS was a benign and proliferative lesion. The cranial bone defect was covered by a pedicled temporalis myofascial fat flap rotated inferiorly. Compared with other surgical approaches for intracranial extension, our surgery was less invasive, and a good exposure of the surgical field could also be obtained. No recurrence has been found until now, but a longer follow-up is still needed.
CONCLUSIONS In summary, PVNS is an uncommon clinical entity that rarely affects the TMJ. One case of PVNS of the TMJ with extension into the middle cranial fossa has been reviewed. Arthroscopy was a useful diagnostic method for the patients with atypical clinical manifestation and imaging findings. The lesion could be completely excised via preauricular approach and condylotomy.
REFERENCES 1. Jaffe JL, Lichtenstein L, Sutro CJ. Pigmented villonodular synovitis, bursitis, and tenosynovitis: discussion of synovial and bursal equivalents of tenosynovial lesions commonly denoted as xanthoma, giant-cell tumor, or myeloplaxoma of tendon sheath lesion itself. Arch Pathol 1941;31:731–765 2. Goldman AB, DiCarlo EF. Pigmented villonodular synovitis. Diagnosis and differential diagnosis. Radiol Clin North Am 1988;26:1327 3. Oda y, Izumi T, Harimaya K, et al. Pigmented villonodular synovitis with chondroid metaplasia, resembling chondroblastoma of the bone: a report of three cases. Mod Pathol 2007;20:545–551 4. Cascone P, Rinna C, Ungari C, et al. Pigmented villonodular synovitis of the temporomandibular joint. J Craniofac Surg 2005;16:712–716
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5. Lee M, Mahroof S, Pringle J, et al. Diffuse pigmented villonodular synovitis of the foot and ankle treated with surgery and radiotherapy. Int Orthop 2005;29:403–405 6. Bemporad JA, Chaloupka JC, Putman CM, et al. Pigmented villonodular synovitis of the temporomandibular joint: diagnostic imaging and endovascular therapeutic embolization of a rare head and neck tumor. AJNR Am J Neuroradiol 1999;20:159–162 7. Day JD, Yoo A, Muckle R. Pigmented villonodular synovitis of the temporomandibular joint: a rare tumor of the temporal skull base. J Neurosurg 2008;109:140–143 8. Eisig S, Dorfman HD, Cusamano RJ, et al. Pigmented villonodular synovitis of the temporomandibular joint. Case report and review of the literature. Oral Surg Oral Med Oral Pathol 1992;73:328–333 9. Herman CR, Swift JQ, Schiffman EL. Pigmented villonodular synovitis of the temporomandibular joint with intracranial extension: a case and literature review. Int J Oral Maxillofac Surg 2009;38:795–801 10. Tosun F, Carrau RL, Weissman J. Pigmented villonodular synovitis of the temporomandibular joint: an extensive case with skull-base involvement. Am J Otolaryngol 2004;25:204–207 11. Cai J, Cai Z, Gao Y. Pigmented villonodular synovitis of the temporomandibular joint: a case report and the literature review. Int J Oral Maxillofac Surg 2011;25:1314–1322 12. Liu YK, Chan JY, Chang CJ, et al. Pigmented villonodular synovitis of the temporomandibular joint presenting as a middle cranial fossa tumor. J Oral Maxillofac Surg 2012;70:367 13. Romañach MJ, Brasileiro BF, León JE, et al. 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–e28 14. Reñaga RI, Salavert GA, Vasquez RA, et al. Pigmented villonodular synovitis of the temporomandibular joint. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:459–460 15. Cai XY, Yang C, Chen MJ, et al. Simultaneous pigmented villonodular synovitis and synovial chondromatosis of the temporomandibular joint: case report. Int J Oral Maxillofac Surg 2009;38:1215–1218 16. Cai XY, Yang C, Chen MJ, et al. Arthroscopic management of intra-articular pigmented villonodular synovitis oftemporomandibular joint. Int J Oral Maxillofac Surg 2011;40:150–154 17. Le WJ, Li MH, Yu Q, et al. Pigmented villonodular synovitis of the temporomandibular joint: CT imaging findings. Clin Imaging 2014;38:6–10 18. Wong JJ, Phal PM, Wiesenfeld D. Pigmented villonodular synovitis of the temporomandibular joint: a radiologic diagnosis and case report. J Oral Maxillofac Surg 2012;70:126–134
Coexistence of Spontaneous Spinal and Undiagnosed Cranial Subdual Hematomas Zhenwen Cui, MD,* Zhihong Zhong, MD,* Baofeng Wang, MD,* Qingsun Sun, MD, PhD,* Chunlong Zhong, MD, PhD,† Liuguan Bian, MD, PhD*
rarer. We report a unique case of spinal SDH combined with bilateral intracranial SDH, in which the cranial lesion was detected after the evacuation of spinal SDH. The undiagnosed chronic SDH developed acute-on-chronic SDH after the evacuation of spinal SDH. The patient had an uneventful clinical course, and a satisfactory outcome was achieved. The reason for reporting this case is to draw attention to the possibility of concurrent cranial SDH in patients with unexplained spinal SDH. The removal of the spinal SDH may exacerbate intracranial hemorrhage and consequently lead to the potential occurrence of tentorial herniation in patients with accompanied cranial SDH. Key Words: Spinal subdual hematoma, intracranial subdual hematoma, surgery, undiagnosed, spontaneous
S
pinal subdual hematomas (SDHs) are uncommon in general clinical practice with a reported incidence of 6.5% in all spinal hematomas, most of which are usually associated with trauma, lumbar puncture, anticoagulation, cranial surgery, and vascular malformations.1 In rare instances, spontaneous evolution of an SDH may occur.2,3 In this article, we describe a case of spinal SDH combined with bilateral intracranial SDH, in which the cranial lesion was detected after the evacuation of spinal SDH. The patient had an uneventful clinical course, but some lessons still should be learned.
CLINICAL REPORT A Chinese 45-year-old man was admitted to the hospital with 1-week history of a progressive saddle pain and dysuresia. He denied any history of trauma of the back or head. The specific hematologic investigations, liver function tests, and coagulation profile were all within reference range. Furthermore, the patient has no family history of bleeding diathesis; he was also not under any antiplatelet or anticoagulant agents. The physical examination demonstrated sensory disturbance in saddle region, lower-limb paresis (grade 4/5), and no other abnormalities. Magnetic resonance imaging (MRI) disclosed an occupying lesion extending from L4 to S3 vertebral levels with a hyperintense signal on T1-weighted images, and a hypointense signal on T2-weighted images (Fig. 1). The patient underwent surgery because of the severe saddle pain. Intraoperatively, no vascular malformations or tumors were detected, and the diagnosis of spinal SDH was made. The saddle pain and dysuresia were significantly improved after the surgery. On postoperative day 2, the patient complained headache and nausea. We performed a computed tomography (CT) scan of the head, which revealed bilateral chronic SDH with a little rebleeding
Abstract: Spinal subdual hematoma (SDH) is an uncommon pathology, and its simultaneous occurrence with cranial SDH is even From the *Department of Neurosurgery, Ruijin Hospital, and †Department of Neurosurgery, Renji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China. Received June 21, 2014. Accepted for publication September 13, 2014. Address correspondence and reprint requests to Liuguan Bian, MD, PhD, 197 Rui Jin Er Rd, Shanghai 200025, China; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001343
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FIGURE 1. Lumbar sagittal MRI scan demonstrating an occupying lesion extending from L4 to S3 vertebral levels with hypointense signal on T2-weighted images (A) and slightly hyperintense signal on T1-weighted images (B). C and D, Axial T2-weighted MRI scan reveals that the cauda equina nerve was tightly surrounded and compressed.
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
FIGURE 4. The intraoperative view of the bony projection.
FIGURE 5. Histopathologic findings (hematoxylin-eosin staining; original magnification, 40).
deformity seemed irreversible after a 5-month follow-up. So a surgical revision was performed. More than a century ago, Wolff14 formulated his law of bone remodeling stating that bone issue adapts to functional stress by changes in structure and mass. From then on, a great number of researches, in vivo and in vitro, have been carried out to investigate the response of the bone to mechanical stimuli and the underlying mechanism.15–17 The bone remodeling secondary to tissue expansion may be considered as a special situation following the Wolff law. Compression loaded on the bone is the definitive factor determining the changes of the bone. However, there seemed to be many other factors contributing to the final result of bone remodeling, including age and sex of the patient, the duration of expansion, the intraexpander pressure and so forth. Stuehmer et al18 and von See et al19 described bone resorption on the interface of hydrogel expander and underlying bone. They believed separating the surface of expander from the underlying bone, with polydioxanone foil and titanium plates, respectively, could prevent the resorption of bone. That is to say, the expander itself contributed to the absorption of the bone. Menick20 advocated that the cheek should be repaired with a cheek flap, although the composite defect encompasses the nose and adjacent cheek. In this case, the 2 expanded flaps met at the nasal-face junction. After the removal of expanders, no changes of the maxilla were detected. In this patient, the forehead expander was inserted into a pocket under the frontalis muscle(ie, supraperiosteal plane) and contacted with the underlying frontal bone. The cheek expander, on the contrary, was inserted into a subcutaneous pocket where the expander did not touch the surface of the mandible or its periosteum. This may partially explain the differences of bone change between the frontal bone and the maxilla after tissue expansion according to Stuehmer et al18 and von See et al.19 As Calobrace and Downey21 pointed out, this case demonstrated not only the profound bony deformity that can result from tissue expansion but also the striking ability of the pediatric skull to remold. Hence, complications of bone deformity should be noticed in children undergoing skin expansion.
REFERENCES 1. Radovan C. Tissue expansion in soft-tissue reconstruction. Plast Reconstr Surg 1984;74:482–492 2. Gibstein LA, Abramson DL, Bartlett RA, et al. Tissue expansion in children: a retrospective study of complications. Ann Plast Surg 1997;38:358–364 3. Manders EK, Schenden MJ, Furrey JA, et al. Soft-tissue expansion: concepts and complications. Plast Reconstr Surg 1984;74:493–507
Brief Clinical Studies
4. Hemmer KM, Marsh JL, Picker S. Calvarial erosion after scalp expansion. Ann Plast Surg 1987;19:454–459 5. Fudem GM, Orgel MG. Full-thickness erosion of the skull secondary to tissue expansion for scalp reconstruction. Plast Reconstr Surg 1988;82:368–369 6. Paletta CE, Bass J, Shehadi SI. Outer table skull erosion causing rupture of scalp expander. Ann Plast Surg 1989;23:538–542 7. McKinney P, Edelson R, Terrasse A, et al. Chest-wall deformity following soft-tissue expansion for breast reconstruction. Plast Reconstr Surg 1987;80:442–444 8. Tirone L, La Rusca I, Ciccarelli F. Rib fracture as a complication of tissue expansion in breast reconstruction. Plast Reconstr Surg 2010;126:2290–2291 9. Tseng J, Huang AH, Wong MS, et al. Rib fractures: a complication of radiation therapy and tissue expansion for breast reconstruction. Plast Reconstr Surg 2010;125:65e–66e 10. Wallis KL, Gurusinghe AD, Anwar MU, et al. Osteophyte-induced rupture of a scalp tissue expander. Ann Plast Surg 2011;67:606–607 11. Colonna M, Cavallini M, De Angelis A, et al. The effects of scalp expansion on the cranial bone: a clinical, histological, and instrumental study. Ann Plast Surg 1996;36:255–260 12. Schmelzeisen R, Schimming R, Schwipper V, et al. Influence of tissue expanders on the growing craniofacial skeleton. J Craniomaxillofac Surg 1999;27:153–159 13. El-Saadi MM, Nasr MA. The effect of tissue expansion on skull bones in the paediatric age group from 2 to 7 years. J Plast Reconstr Aesthet Surg 2008;61:413–418 14. Wolff J. Das gesetz der transformation der knochen, Berlin, Germany: Verlag von August Hirschwald; 1892 15. Klein-Nulend J, van der Plas A, Semeins CM, et al. Sensitivity of osteocytes to biomechanical stress in vitro. FASEB J 1995;9:441–445 16. Rubin CT, Lanyon LE. Kappa Delta Award paper. Osteoregulatory nature of mechanical stimuli: function as a determinant for adaptive remodeling in bone. J Orthop Res 1987;5:300–310 17. Burger EH, Klein-Nulen J. Responses of bone cells to biomechanical forces in vitro. Adv Dent Res 1999;13:93–98 18. Stuehmer C, Ruker M, Schumann P, et al. Osseous alterations at the interface of hydrogel expanders and underlying bone. J Craniomaxillofac Surg 2009;37:258–262 19. von See C, Rucker M, Schumann P, et al. Micro-computed tomography and histologic evaluation of the interface of hydrogel expander and underlying bone: influence of pressure distributors on bone resorption. J Oral Maxillofac Surg 2010;68:2179–2184 20. Menick FJ. Nasal reconstruction. Plast Reconstr Surg 2010;125:138e–150e 21. Calobrace MB, Downey SE. Calvarial deformity and remodeling following prolonged scalp expansion in a child. Ann Plast Surg 1997;39:186–189
Pigmented Villonodular Synovitis of the Temporomandibular Joint With Intracranial Extension Ying Chen, BS, Xie-Yi Cai, MD, Chi Yang, MD, Min-Jie Chen, MD, Ya-Ting Qiu, MD, Ziang Zhuo, MS Abstract: Pigmented villonodular synovitis is an uncommon benign tumor-like proliferative lesion with an undetermined origin. Involvement of the temporomandibular joint is uncommon. Although pigmented villonodular synovitis is a benign lesion, it can grow with an aggressive pattern, and it extends extra-articularly in most of the reported cases, about one-third of them exhibiting intracranial involvement. The authors reported an additional case of a 47-year-old
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e115
The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
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woman with intracranial extension, who had a history of joint pain and trismus. The preoperative diagnosis was made with arthroscopy. The lesion was completely excised via preauricular approach and condylotomy. The bone defect was covered by the pedicled temporalis myofascial fat flap. The patient has been symptom-free for 40 months postoperatively. Key Words: Temporomandibular joint, pigmented villonodular synovitis, skull base, treatment
P
igmented villonodular synovitis (PVNS) is an uncommon benign tumor-like proliferative lesion with an undetermined origin arising from the synovial membranes of joints, bursas, and tendon sheaths, which was first fully described by Jaffe et al1 in 1941. Its etiology remains elusive. Possible etiologies include disturbances of lipid metabolism, neoplasm, inflammation, trauma, and hemorrhage.2,3 It has an annual incidence of 1.8 cases per million population.4 The knee is commonly involved in 80% of cases, but any joint may be affected.5 Most cases are monoarticular and do not metastasize, although they may be locally destructive. Involvement of the temporomandibular joint (TMJ) is uncommon, and about 60 cases have been published in the English-language literature to our knowledge. The predominant symptoms in the TMJ are preauricular mass or swelling, pain, and limitation of mandibular movement. Although PVNS is a benign lesion, it can grow with an aggressive pattern, and it extends extra-articularly in most of the reported cases, about one-third of them exhibiting intracranial involvement.6–13 It is commonly accepted that surgery is the only curative treatment for PVNS, with partial or complete excision of the synovium and involved bones. Despite these maneuvers, a relatively high rate of recurrence of PVNS in other joints has been reported, in the range of 33% to 50%.14 Therefore, some authors proposed that radiotherapy could be used in the management of recurrent cases with moderate radiation doses.5 All reported PVNSs in TMJ have been treated by surgical excision, and we reported 4 cases managed with arthroscopy.15,16 However, it is difficult to predict the recurrence rate in TMJ because of the lack of long-term follow-up information. Herein, we report an additional case of PVNS in TMJ with intracranial extension.
CLINICAL REPORT In November 2010, a 47 year-old woman was referred to the Department of Oral and Maxillofacial Surgery, Ninth People’s Hospital, Shanghai Jiao Tong University School of Medicine, with a chief From the Department of Oral and Maxillofacial Surgery, Ninth People’s Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai Key Laboratory of Stomatology, Shanghai, China. Received June 12, 2014. Accepted for publication September 13, 2014. Address correspondence and reprint requests to Chi Yang, MD, and Xie-Yi Cai, MD, Department of Oral & Maxillofacial Surgery, Shanghai Ninth People’s Hospital, Shanghai Jiaotong University, 639 Zhi Zao Ju Rd, Shanghai 200011, People’s Republic of China; E-mail: [email protected]; [email protected] This project was supported by the National Natural Science Foundation of China (grant 81200766) and Science and Technology Commission of Shanghai (13140902702). The authors report no conflicts of interest. Copyright © 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001341
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FIGURE 1. Magnetic resonance imaging scan of the TMJ of the 47-year-old female patient showing joint effusion with capsular expansion and a mass with mixed signal intensity. A, In the parasagittal plane. B, In the coronal plane.
complaint of pain in the left TMJ region that had developed 1 year ago and progressively increased over the past 2 months. She had no hearing loss and facial nerve palsy. Physical examination revealed a symmetric face with no swelling or mass over the left preauricular region, a maximal mouth opening of 28 mm, slight deviation of the mandibular midline, and a normal occlusion. The patient had no history of trauma or rheumatoid arthrosis. A magnetic resonance imaging (MRI) scan of the TMJ revealed joint effusion with capsular expansion and a mass with mixed signal intensity in the anterior recess in T1-weighted images on the left side (Figs. 1A, B). Clinical and radiological findings prompted a diagnosis of synovial chondromatosis or synovitis on the left TMJ. To clarify the diagnosis, a diagnostic arthroscopy of the left TMJ was performed under local anesthesia in November 2010. Under arthroscope, the articular disc was seen to be in normal position, and a widespread orange synovium in the anterior recess was noticed (Fig. 2). A large brown hyperplastic nodule occupied the medial part of anterior recess and medial groove, which was fragile and prone to bleed. It extended to the glenoid fossa, and the cartilage surface was involved. According to the arthroscopic features, a provisional diagnosis of PVNS was given. To investigate the extent of the lesion, an enhanced computed tomographic (CT) scan was applied after arthroscopy, which showed that the medial joint space was obviously widened, and the corresponding TMJ fossa and skull base were thinned and perforated. However, no condylar resorption or destruction was revealed on CT scans (Fig. 3). Three days later, the patient underwent surgical excision of the lesion via a modified preauricular approach under general anesthesia. The lesion was located in the anteromedial side of the TMJ and extended superiorly and medially into the middle cranial fossa, with erosion of the glenoid fossa. It was brown, with a soft and friable texture(Fig. 4). For better exposure of the lesion, condylar osteotomy was performed at the level of the condylar neck. Frozen biopsy was obtained, and giant cell granuloma was diagnosed during the operation. The lesion was completely excised, and the involved retrodiscal tissue was also removed. On closer examination, the lesion appeared to be protruding onto the dura mater but had not perforated it. Because of the erosion and perforation of the skull base, the temporalis myofascial fat flap with the pedicle of middle temporal vessels was used to cover the cranial bone defect. Finally, the condyle was repositioned and fixed with 2 mini–titanium plates. Patient's occlusion did not change after surgery. Histologic examination disclosed that the lesion was composed of predominantly plump histiocytes with intermixed, variably distributed multinucleated giant cells, and the mass was heavily pigmented with hemosiderin (Fig. 5). So a final diagnosis of PVNS was given.
FIGURE 2. Arthroscopic vision showing a widespread orange synovium.
© 2015 Mutaz B. Habal, MD
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The Journal of Craniofacial Surgery • Volume 26, Number 2, March 2015
FIGURE 3. Preoperative CT showing the widened medial joint space and the thinned and perforated skull base.
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FIGURE 5. Histological examination showed that the lesion was composed of predominantly plump histiocytes with intermixed multinucleated giant cells and deposits of hemosiderin (hematoxylin-eosin staining. original magnification 200).
The postoperative course was uneventful. A 40-month postoperative follow-up revealed good symmetry, normal occlusion, and no restriction of jaw movement. Panoramic radiograph, CT scan, and MRI showed no evidence of recurrence. The condyle grew well. The cranial bone defect was getting smaller, although it has not completely healed (Figs. 6A, B).
DISCUSSION Pigmented villonodular synovitis is a benign, yet locally aggressive proliferative lesion, which preferentially affects the synovial tissue of the large joints, such as the knee and hip of adult patients in their third to fifth decades of life.1–3 It can involve any articular site, including the ankle, shoulder, and elbow and rarely involves the joints of the hands, feet, spine, and TMJ. To the best our knowledge, about 60 patients with PVNS affecting the TMJ region have been reported in English-written literatures and about one-third of the cases present intracranial extension.6–13 The most common clinical manifestations for the patients with intracranial extension are painful/painless preauricular swelling/mass, trismus, altered hearing, and tinnitus. Advanced imaging methods, including contrast-enhanced CT and MRI, are helpful to the correct diagnosis. The glenoid fossa erosions and intracranial extension, associated with peri-articular soft tissue swelling with or without calcification, can be demonstrated on CT.17 The characteristic feature of the lesion on MRI is an intermediate- to low-intensity signal on T1W images and a low intensity on T2-weighted depending on the hemosiderin concentration.18 The present case reported a history of arthralgia and limitation of mouth opening, without preauricular swelling or mass. Joint effusion with capsular expansion and a mass with mixed signal intensity was found on MRI. Therefore, a preoperative diagnosis of synovial chondromatosis was given, and the patient underwent a diagnostic arthroscopy. Then, a provisional diagnosis of PVNS was made because of its typical manifestation under arthroscope. Similar with our reported cases with intra-articular PVNS,15,16 it is difficult to make an early diagnosis for the present case because of atypical symptoms and unspecific imaging information. It has been reported that fine-needle aspiration biopsy is a relatively simple method to establish the preoperative diagnosis for PVNS. In our patient, it is almost impossible to perform fine-needle aspiration biopsy, but diagnostic arthroscopy is very helpful for early diagnosis of PVNS. Furthermore, contrast-enhanced CT scan is necessary for investigating the extent of the lesion and bony erosion. Intracranial extension has been finally found with CT scan for the present patient. It is commonly accepted that a complete excision with synovectomy and/or capsulectomy is the treatment choice for PVNS.
FIGURE 4. Transoperative view showing the lesion was located in the anteromedial side of the TMJ and extended superiorly and medially into the middle cranial fossa.
FIGURE 6. The follow-up radiological images showing no evidence of recurrence at 40 months after operation: A, Computed tomography scan. B, Magnetic resonance imaging scan.
Conversely, an incomplete resection will result in a high rate of recurrence. Some authors proposed that radiotherapy could be used in the management of recurrent cases with moderate radiation doses.5 For the patients with intracranial involvement, a coronal or hemicoronal incision with preauricular and frontal extension is often used to expose the lesion. Besides complete excision of the lesion, craniectomy and/or condylectomy may be performed when the condyle is also involved. Because malignancy or malignant transformation of the lesion has not been reported, dura mater was not excised except for 1 patient.13 Radiotherapy has been used after excision in 3 patients. Even so, recurrence of PVNS was found in 1 of the 3 patients 3 years later.13 For the present case, the lesion was located in the medial part of the joint, and no condylar bone destruction was shown. Therefore, a modified preauricular approach was used, and condylar osteotomy was performed for better exposure. The dura mater was intact, and it remained untreated. We thought that craniectomy was not necessary, because PVNS was a benign and proliferative lesion. The cranial bone defect was covered by a pedicled temporalis myofascial fat flap rotated inferiorly. Compared with other surgical approaches for intracranial extension, our surgery was less invasive, and a good exposure of the surgical field could also be obtained. No recurrence has been found until now, but a longer follow-up is still needed.
CONCLUSIONS In summary, PVNS is an uncommon clinical entity that rarely affects the TMJ. One case of PVNS of the TMJ with extension into the middle cranial fossa has been reviewed. Arthroscopy was a useful diagnostic method for the patients with atypical clinical manifestation and imaging findings. The lesion could be completely excised via preauricular approach and condylotomy.
REFERENCES 1. Jaffe JL, Lichtenstein L, Sutro CJ. Pigmented villonodular synovitis, bursitis, and tenosynovitis: discussion of synovial and bursal equivalents of tenosynovial lesions commonly denoted as xanthoma, giant-cell tumor, or myeloplaxoma of tendon sheath lesion itself. Arch Pathol 1941;31:731–765 2. Goldman AB, DiCarlo EF. Pigmented villonodular synovitis. Diagnosis and differential diagnosis. Radiol Clin North Am 1988;26:1327 3. Oda y, Izumi T, Harimaya K, et al. Pigmented villonodular synovitis with chondroid metaplasia, resembling chondroblastoma of the bone: a report of three cases. Mod Pathol 2007;20:545–551 4. Cascone P, Rinna C, Ungari C, et al. Pigmented villonodular synovitis of the temporomandibular joint. J Craniofac Surg 2005;16:712–716
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5. Lee M, Mahroof S, Pringle J, et al. Diffuse pigmented villonodular synovitis of the foot and ankle treated with surgery and radiotherapy. Int Orthop 2005;29:403–405 6. Bemporad JA, Chaloupka JC, Putman CM, et al. Pigmented villonodular synovitis of the temporomandibular joint: diagnostic imaging and endovascular therapeutic embolization of a rare head and neck tumor. AJNR Am J Neuroradiol 1999;20:159–162 7. Day JD, Yoo A, Muckle R. Pigmented villonodular synovitis of the temporomandibular joint: a rare tumor of the temporal skull base. J Neurosurg 2008;109:140–143 8. Eisig S, Dorfman HD, Cusamano RJ, et al. Pigmented villonodular synovitis of the temporomandibular joint. Case report and review of the literature. Oral Surg Oral Med Oral Pathol 1992;73:328–333 9. Herman CR, Swift JQ, Schiffman EL. Pigmented villonodular synovitis of the temporomandibular joint with intracranial extension: a case and literature review. Int J Oral Maxillofac Surg 2009;38:795–801 10. Tosun F, Carrau RL, Weissman J. Pigmented villonodular synovitis of the temporomandibular joint: an extensive case with skull-base involvement. Am J Otolaryngol 2004;25:204–207 11. Cai J, Cai Z, Gao Y. Pigmented villonodular synovitis of the temporomandibular joint: a case report and the literature review. Int J Oral Maxillofac Surg 2011;25:1314–1322 12. Liu YK, Chan JY, Chang CJ, et al. Pigmented villonodular synovitis of the temporomandibular joint presenting as a middle cranial fossa tumor. J Oral Maxillofac Surg 2012;70:367 13. Romañach MJ, Brasileiro BF, León JE, et al. 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–e28 14. Reñaga RI, Salavert GA, Vasquez RA, et al. Pigmented villonodular synovitis of the temporomandibular joint. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:459–460 15. Cai XY, Yang C, Chen MJ, et al. Simultaneous pigmented villonodular synovitis and synovial chondromatosis of the temporomandibular joint: case report. Int J Oral Maxillofac Surg 2009;38:1215–1218 16. Cai XY, Yang C, Chen MJ, et al. Arthroscopic management of intra-articular pigmented villonodular synovitis oftemporomandibular joint. Int J Oral Maxillofac Surg 2011;40:150–154 17. Le WJ, Li MH, Yu Q, et al. Pigmented villonodular synovitis of the temporomandibular joint: CT imaging findings. Clin Imaging 2014;38:6–10 18. Wong JJ, Phal PM, Wiesenfeld D. Pigmented villonodular synovitis of the temporomandibular joint: a radiologic diagnosis and case report. J Oral Maxillofac Surg 2012;70:126–134
Coexistence of Spontaneous Spinal and Undiagnosed Cranial Subdual Hematomas Zhenwen Cui, MD,* Zhihong Zhong, MD,* Baofeng Wang, MD,* Qingsun Sun, MD, PhD,* Chunlong Zhong, MD, PhD,† Liuguan Bian, MD, PhD*
rarer. We report a unique case of spinal SDH combined with bilateral intracranial SDH, in which the cranial lesion was detected after the evacuation of spinal SDH. The undiagnosed chronic SDH developed acute-on-chronic SDH after the evacuation of spinal SDH. The patient had an uneventful clinical course, and a satisfactory outcome was achieved. The reason for reporting this case is to draw attention to the possibility of concurrent cranial SDH in patients with unexplained spinal SDH. The removal of the spinal SDH may exacerbate intracranial hemorrhage and consequently lead to the potential occurrence of tentorial herniation in patients with accompanied cranial SDH. Key Words: Spinal subdual hematoma, intracranial subdual hematoma, surgery, undiagnosed, spontaneous
S
pinal subdual hematomas (SDHs) are uncommon in general clinical practice with a reported incidence of 6.5% in all spinal hematomas, most of which are usually associated with trauma, lumbar puncture, anticoagulation, cranial surgery, and vascular malformations.1 In rare instances, spontaneous evolution of an SDH may occur.2,3 In this article, we describe a case of spinal SDH combined with bilateral intracranial SDH, in which the cranial lesion was detected after the evacuation of spinal SDH. The patient had an uneventful clinical course, but some lessons still should be learned.
CLINICAL REPORT A Chinese 45-year-old man was admitted to the hospital with 1-week history of a progressive saddle pain and dysuresia. He denied any history of trauma of the back or head. The specific hematologic investigations, liver function tests, and coagulation profile were all within reference range. Furthermore, the patient has no family history of bleeding diathesis; he was also not under any antiplatelet or anticoagulant agents. The physical examination demonstrated sensory disturbance in saddle region, lower-limb paresis (grade 4/5), and no other abnormalities. Magnetic resonance imaging (MRI) disclosed an occupying lesion extending from L4 to S3 vertebral levels with a hyperintense signal on T1-weighted images, and a hypointense signal on T2-weighted images (Fig. 1). The patient underwent surgery because of the severe saddle pain. Intraoperatively, no vascular malformations or tumors were detected, and the diagnosis of spinal SDH was made. The saddle pain and dysuresia were significantly improved after the surgery. On postoperative day 2, the patient complained headache and nausea. We performed a computed tomography (CT) scan of the head, which revealed bilateral chronic SDH with a little rebleeding
Abstract: Spinal subdual hematoma (SDH) is an uncommon pathology, and its simultaneous occurrence with cranial SDH is even From the *Department of Neurosurgery, Ruijin Hospital, and †Department of Neurosurgery, Renji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China. Received June 21, 2014. Accepted for publication September 13, 2014. Address correspondence and reprint requests to Liuguan Bian, MD, PhD, 197 Rui Jin Er Rd, Shanghai 200025, China; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001343
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FIGURE 1. Lumbar sagittal MRI scan demonstrating an occupying lesion extending from L4 to S3 vertebral levels with hypointense signal on T2-weighted images (A) and slightly hyperintense signal on T1-weighted images (B). C and D, Axial T2-weighted MRI scan reveals that the cauda equina nerve was tightly surrounded and compressed.
© 2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
