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CASE REPORT
Schwannoma Presenting with Anteromedial Knee Pain and Bone Defect K. Karthik, M.S. (Ortho), M.R.C.S.Ed,1,2 S. Aarthi, M.B.B.S., M.D., M.R.C.P.,1 and Ayeshah Gordon-Dixon, B.Sc., M.B.B.S.2
ABSTRACT
Tumors of the peripheral nerve sheath including schwannomas are known for their rarity and can present with neurological deficit. We report an interesting case of schwannoma of the infrapatellar branch of saphenous nerve presenting with anteromedial knee pain and proximal tibial metaphyseal defect. A 26-year-old man presented with pain and swelling (for the past 2 months) in the anteromedial aspect of knee. Investigations revealed scalloping lesion in the anteromedial aspect of proximal tibia with sclerosed margins; however, further studies ruled out the possibility of invasive tumor. Surgical exploration identified an encapsulated tumor lying over the defect in the proximal tibial metaphysis. Histopathological examination confirmed benign schwannoma and the patient became symptom-free after the surgery. Schwannoma of the infrapatellar branch of saphenous nerve can present with anteromedial knee pain. In patients with an eccentrically placed metaphyseal defect of the anteromedial tibia with sclerosed smooth margins, the possibility of nerve sheath tumors should always be ruled out. KEYWORDS: Schwannoma, infrapatellar branch of saphenous nerve, anteromedial knee pain, proximal tibial metaphyseal defect
S
Q1
chwannomas are benign neoplasms of the nerve sheath and are composed entirely of neoplastic Schwann cells.1,2 90% of schwannomas are sporadic, 3% occur with neurofibromatosis type 2 (NF2).1,2 They rarely occur as part of NF1 or after radiation therapy and commonly presents as a painless mass.1,2 The clinical course of nerve sheath tumor arising from infrapatellar branch of saphenous nerve has not been reported in the literature. However, pain in the anteromedial knee after injury to this nerve after trauma or surgery had been reported.3 We report an interesting case of schwannoma on the anteromedial part of proximal leg arising from the infrapatellar branch of the saphenous nerve presenting
with anteromedial knee pain and proximal tibial metaphyseal defect.
1 Department of Orthopaedic Surgery, James Cook University Hospital, Middlesbrough, United KingdomQ1; 2Queen Elizabeth Hospital, London, United Kingdom. Address for correspondence and reprint requests: K. Karthik, M.S. (Ortho), M.R.C.S.Ed., Clinical Fellow, Department of Orthopaedic Surgery, James Cook University Hospital, Marton Road, Middlesbrough, UK SE184QH (e-mail:
[email protected]).
J Knee Surg. Copyright # by Thieme Medical Publishers, Inc., 333 Seventh Avenue, New York, NY 10001, USA. Tel: +1 (212) 584-4662. Received: November 18, 2011. Accepted after revision: April 23, 2011. DOI: http://dx.doi.org/10.1055/s-0031-1280975. ISSN 1538-8506.
CASE REPORT A 26-year-old male athlete presented with pain and swelling (for the past 2 months) in the anteromedial aspect of left knee. There was no history of trauma and the patient had not noticed any change in size of the swelling. The knee pain was dull aching and continuous in nature. There was no radiation of the pain and the pain was aggravated with running, stretching exercises of the knee, and on applying direct pressure
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over the swelling. He had minimal relief of pain with analgesics. On examination the patient had a swelling over the anteromedial aspect of proximal tibia 2 cm below the lateral joint line. The swelling was around 3 3 cm in size, warm, acutely tender on pressure, and firm in consistency. Severity of pain associated with the swelling limited examination of this area. The skin over the swelling was normal and there were no superficial scars, sinuses, or dilated veins. There were no other swellings anywhere in the body. Tests for instability and meniscal injuries were negative except for a minimal increase in pain over the swelling while stressing medial collateral ligament. Radiograph of the left knee showed a scalloping lesion in proximal tibial metaphysis (Fig. 1A, B). The
Figure 1 (A) Anteroposterior view of the left knee showing a defect in the proximal tibial metaphysis with smooth margins. (B) Lateral view showing a flake of doubtful periosteal reaction anterior to the defect.
base and margin of the defect was well defined and smooth. Based on the clinical findings and radiographs a differential diagnosis of subacute osteomyelitis (eccentrically placed metaphyseal defect variant4), soft-tissue sarcoma, giant-cell tumor of the bone, or a benign soft-tissue tumor was made and the patient was further investigated. Blood investigations were normal and the ultrasound showed a nonvascular, well-defined echogenic mass confined to the defect. Computed tomography (CT) scan showed a well-demarcated mass lying on the smooth sclerosed defect in the proximal tibia (Fig. 2). Magnetic resonance imaging (MRI) demonstrated well-defined lesion with no extension into the soft tissues or bone. The tumor was explored through a midline incision (suggested by tumor surgeon) for excisional biopsy. Intraoperatively a rubbery, nonvascular, encapsulated tumor was identified lying in the bed of eroded proximal tibia. The tumor was covered and compressed into the bone by pes anserinus and part of medial collateral ligament. The tumor tissue was yellow in color and was attached to the infrapatellar branch of saphenous nerve. The tumor tissue had no connection with the bone or with the surrounding soft tissues. The tumor was enucleated out of the nerve without exposing the contents to the surrounding tissue. The nerve fascicles though atrophied, were found intact continuing distally. The specimen was sent for histopathological examination. The biopsy report came as benign schwannoma with Antoni A, Antoni B, and Verocay bodies (Fig. 3A–C). The patient had immediate relief of the characteristic pain after the surgery and remained symptom-free till the last follow-up at 1 year.
Figure 2 CT scan (axial view) showed a well-circumscribed mass on the smooth defect in the proximal tibial metaphysis. The defect is smooth with sclerosed margin.
SCHWANNOMA PRESENTING WITH ANTEROMEDIAL KNEE PAIN AND BONE DEFECT/KARTHIK ET AL
Figure 3 (A) Hematoxylin and eosin staining (original magnification 100) showing hypercellular areas of spindle-shaped cells demonstrating Antoni A pattern. (B) Hematoxylin and eosin staining (original magnification 100) showing hypocellular areas of spindle-shaped cells, loose, lacking arrangement in bundles and palisading demonstrating Antoni B pattern. (C) Hematoxylin and eosin staining (original magnification 100) showing Verocay bodies characterized by linear arrangements of elongated tumor nuclei (arrow).
DISCUSSION Schwannomas are solid, benign tumors that grow very slowly.1,2 This tumor has preponderance to occur in upper limbs including brachial plexus (69%) compared with 24% in the lower extremities.5,6 Schwannomas rarely can occur in association with NF1, NF2, or after radiation therapy.5,6 Sudden onset of pain or new symptoms is a concern for malignant transformation.5–7 Schwannoma has a true capsule composed of epineurium and can be excised with acceptable risk of injury to the nerve.5,8 A high incidence of transient neurological deficits has been reported after surgical excision of the tumor.8 However, careful and adequate excision of the tumor in solitary schwannomas are associated with good outcome.9,10 In large nerves the tumor is characteristically eccentric with respect
to the affected nerve.5–7 This finding is helpful when present and may be identified at imaging but is usually not seen in small nerves.5–7 Since the tumor affected a small infrapatellar branch of the saphenous nerve, none of the investigations identified schwannoma in our case. Schwannomas may grow for years without any symptoms if there are no structures anatomically that restrict their growth.5–8,11 Some schwannomas are symptomatic early, they usually cause neurological deficit by compressing the nerve by itself or over the bone.5–7 In our patient the tumor was surrounded by a tight osseomusculo-fascial compartment Q2that includes anterome- Q2 dial tibia, pes anserinus, and collateral ligament. This tight compartment led to the pressure erosion of the bone by the tumor. The patient was in constant pain as
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there was less space for the tumor to expand and it was compressed against the bone. Anteromedial knee pain had been reported due to injury of the infrapatellar branch of saphenous nerve or schwannoma of the saphenous nerve at the thigh, but no cases have been reported due to the tumor in the infrapatellar branch of saphenous nerve.11–13 Intraosseous schwannoma is a different entity where the tumor presents as an expansile lytic lesion with bone destruction. These tumors commonly occur in skull bones.14 In this case the tumor was extraosseous but eroded the bone due to the pressure from the surrounding structures. Despite an extensive literature search, the authors could not find a case similar to the one presented in this report.
CONCLUSION Schwannomas arising from the infrapatellar branch of saphenous nerve can present with knee pain, when the surrounding osseous, muscular, and fascial elements compress the tumor; the resulting pressure can also lead to bone erosion. In young athletes with nontraumatic anteromedial knee pain and bone defect the possibility of schwannoma should be ruled out. REFERENCES 1. Seddon HJ. Lesions of individual nerves. Lower limb. In: Seddon HJ, ed. Surgical Disorders of Peripheral Nerves. Baltimore: Williams and Wilkins; 1972:505–579 2. Sunderland S. Nerves and Nerve Lesions. 1st edition. Edinburgh: Churchill Livingstone; 1978
3. Tennent TD, Birch NC, Holmes MJ, Birch R, Goddard NJ. Knee pain and the infrapatellar branch of the saphenous nerve. J R Soc Med 1998;91(11):573–575 4. Roberts JM, Drummond DS, Breed AL, Chesney J. Subacute hematogenous osteomyelitis in children: a retrospective study. J Pediatr Orthop 1982;2(3):249–254 5. Kim DH, Murovic JA, Tiel RL, Moes G, Kline DG. A series of 397 peripheral neural sheath tumors: 30-year experience at Louisiana State University Health Sciences Center. J Neurosurg 2005;102(2):246–255 6. Campbell R. Tumours of peripheral and sympathetic nerves. In: Youmans JR, ed. Neurological Surgery. 3rd edition. Philadelphia: WB Saunders & Co.; 1990:3667–3670 7. Stuot AP. Tumours of peripheral nervous system. In: Atlas of Tumour Pathology, Section II, Fascicle 6. Washington DC: Q3 Armed forces institute of pathology; 1949Q3 8. Park MJ, Seo KN, Kang HJ. Neurological deficit after surgical enucleation of schwannomas of the upper limb. J Bone Joint Surg Br 2009;91(11):1482–1486 9. Knight DM, Birch R, Pringle J. Benign solitary schwannomas: a review of 234 cases. J Bone Joint Surg Br 2007;89(3): 382–387 10. Donner TR, Voorhies RM, Kline DG. Neural sheath tumors of major nerves. J Neurosurg 1994;81(3):362–373 11. Gazzeri R, Refice GM, Galarza M, Neroni M, Esposito S, Gazzeri G. Knee pain in saphenous nerve schwannoma: case report. Neurosurg Focus 2007;22(6):E11 12. Edwards JC, Green CT, Riefel E. Neurilemoma of the saphenous nerve presenting as pain in the knee. A case report. J Bone Joint Surg Am 1989;71(9):1410–1411 13. Tennent TD, Birch NC, Holmes MJ, Birch R, Goddard NJ. Knee pain and the infrapatellar branch of the saphenous nerve. J R Soc Med 1998;91(11):573–575 14. Gordon EJ. Solitary intraosseous neurilemmoma of the tibia: review of intraosseous neurilemmoma and neurofibroma. Clin Orthop Relat Res 1976;(117):271–282
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