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 Table of Contents  
CASE REPORTS
Year : 2022  |  Volume : 37  |  Issue : 2  |  Page : 80-84

A rare case report: Giant cell tumor of ischial tuberosity and ischiopubic ramus


Department of Orthopedic Surgery, King George Medical College, Lucknow, Uttar Pradesh, India

Date of Submission06-Sep-2022
Date of Acceptance07-Sep-2022
Date of Web Publication19-Oct-2022

Correspondence Address:
Siddharth Deshwal
A1-26, Mansarovar Colony, Moradabad 244001, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jbjd.jbjd_22_22

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  Abstract 

The ischial tuberosity and ischiopubic ramus represent one of the rarest sites of giant cell tumors of bone. A rare case is presented here. The unusual site and characteristic features of giant cell tumors and current treatment options are reviewed.

Keywords: Giant cell tumor of bone, ischial tuberosity, ischiopubic ramus


How to cite this article:
Kumar D, Singh S, Deshwal S, Abbas Z, Kumar A. A rare case report: Giant cell tumor of ischial tuberosity and ischiopubic ramus. J Bone Joint Dis 2022;37:80-4

How to cite this URL:
Kumar D, Singh S, Deshwal S, Abbas Z, Kumar A. A rare case report: Giant cell tumor of ischial tuberosity and ischiopubic ramus. J Bone Joint Dis [serial online] 2022 [cited 2022 Nov 29];37:80-4. Available from: http://www.jbjd.in/text.asp?2022/37/2/80/358800




  Introduction Top


Giant cell tumor of bone is an uncommon neoplasm of uncertain origin that accounts for approximately 4%–5% of all bone tumors. The articular ends of long bones are characteristically involved. Pelvic involvement, excluding the sacrum, is quite unusual.[1] In particular, giant cell tumor of the ischium represents less than 0.5% of all giant cell tumors. The purpose of this presentation is to report a case in this location, to review the characteristic features of this unusual tumor, and to discuss current concepts of treatment of giant cell tumor of bone.


  Case Report Top


A 38-year-old man presented with pain in the left groin region for 2.5 years. Physical examination and laboratory data were not remarkable. A lesion involving the left ischial tuberosity and ipsilateral ischiopubic ramus was incidentally detected on radiographs of the pelvis region. X-ray [Figure 1] films of the pelvis revealed expansile lytic lesion of the left ischial tuberosity and ipsilateral ischiopubic ramus. Computed tomography scan films [Figure 2] were suggestive of a well-defined large osteoexpansile lesion localized ischial tuberosity and ipsilateral ischiopubic ramus. Magnetic resonance study [Figure 3] revealed a well-defined expansile lobulated, altered signal intensity lesion measuring approximately 43 × 58 × 82 mm involving the left ischium bone extending into the left ischial tuberosity with an involvement of the posterior column of acetabulum but not involving the hip joint. These findings were suggestive of neoplastic etiology and the most common differentials were giant cell tumor and aneurysmal bone cyst. Biopsy [Figure 4] report confirmed the diagnosis of the giant cell tumor. After the confirmation of diagnosis, the patient was given injection denosumab four cycles to decrease pain and may downstage surgery so that the tumor becomes operatable or it may avoid resections.
Figure 1: (a) Anteroposterior view of the pelvis demonstrates an expansile, lytic lesion of the left ischium. (b) Oblique view demonstrates the lack of reactive sclerosis, periosteal reaction, and matrix mineralization

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Figure 2: Computed tomography scan films were suggestive of a well-defined large osteoexpansile lesion localized ischial tuberosity and ipsilateral ischiopubic ramus

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Figure 3: Magnetic resonance study revealed a well-defined expansile lobulated, altered signal intensity lesion measuring approximately 43 × 58 × 82 mm involving the left ischium bone extending into the left ischial tuberosity with the involvement of posterior column of acetabulum, but not involving the hip joint

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Figure 4: Histopathological report suggestive of giant cell tumor of ischial tuberosity

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Surgical approach

Under spinal anesthesia, the patient was placed on the radiolucent operating table in the prone position. The intergluteal cleft was draped to avoid gastrointestinal contamination. The gluteal crease was marked before draping. Image-intensifier control was needed for the visualization of the pelvis. A pillow was kept below the leg to keep the knee in a position of flexion during the operation. After the disinfection of the skin, a single-use hip draping set was applied. The ischial tuberosity was palpated and localized. An incision was placed on the gluteal crease, centered at the vertical projection of the lateral facet of the ischial tuberosity. An approximately 8-cm-long incision was used for sufficient exposure. The subcutaneous fat is sharply dissected. The inferior border of the gluteus maximus was localized, and the fascia overlying the gluteus maximus was incised. The inferior border of the gluteus maximus was elevated with a gentle dissection to elevate the gluteus maximus muscle, which was the principal tissue preventing exposure. The lower limb was abducted to facilitate sciatic nerve dissection. The sciatic nerve was protected, and ischial tuberosity was exposed with the help of retractors. A cortical window was made in the tuberosity under C-arm guidance, and curettage was done [Figure 5]. A cavity was found empty with only a little amount of tumor tissue that was adhered to the cavity walls was curetted. The cavity was irrigated with distilled water, and finally the cavity was filled with methyl methacrylate (Palacos) [Figure 6], and the wound was closed in layers.
Figure 5: Intraoperative picture showing the extent of the lesion and cavity filling with bone cement

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Figure 6: Postoperative x-ray with bone cement in the cavity

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  Conclusion Top


Giant cell tumor of bone is an uncommon neoplasm. It typically occurs in the young and middle-aged adults after the growth plate closes. Its frequency gradually decreases in the later decades of life, and it is extremely rare in patients over 70 years of age. The distal femur, proximal tibia, and distal radius are the sites of approximately 60% of the giant cell tumors in most series. The sacrum is by far the most common site in the axial skeleton; tumors occur in this location almost as frequently as in the distal radius [Figure 7]. Giant cell tumor of bone is characterized radiographically as a lytic lesion with ill-defined margins. It may be expansile and extend beyond the cortex. Most tumors demonstrate the absence of a sclerotic border (unless the tumor has been present for a long time), a lack of periosteal reaction, and the absence of calcified or ossified matrix. These characteristic findings may be altered by pathologic fracture, secondary infection, or treatment. In our case scenario, an expansile lytic lesion of the left ischial tuberosity extending into the ipsilateral ischiopubic ramus is noted. The tumor’s subarticular location in the appendicular skeleton is the most useful guide to its correct radiographic diagnosis, especially when common long bones are involved in the young and middle-aged adults.[1],[2] Benign and malignant lesions have been described.[3],[4] Rare reported sites of the giant cell tumor include the calvaria, sphenoid, mandible, manubrium, scapula, ribs, proximal radius, carpal bones, distal fibula, calcaneus, metatarsals, cuneiforms, patella, vertebra, pubis, and ischium.[1],[2],[4],[5],[6],[7],[8],[9],[10],[11] Lesions not extending into the articular ends of the long bones are extremely rare and may be seen in the immature skeleton at the metaphysis. A review of the literature, however, reveals that the frequency of giant cell tumor at the ischium is slightly less than that seen at the pubis. The ilium, on the other hand, is the most frequent site of giant cell tumor of the pelvis (excluding the sacrum). Overall, approximately 4% of the giant cell tumors occur in the innominate bones. Sixty-four percent of these occur in the ilium, 22% in the pubis, and 14% in the ischium[1],[2],[4-7],[12] [Figure 8]. Our case of the giant cell tumor was large, osteoexpansile lesion localized ischial tuberosity and ipsilateral ischiopubic ramus. This case was subarticular in location, extending to the acetabulum. When possible, the treatment of giant cell tumors of bone is intralesional curettage and bone grafting, either autogenous or allogenous or can be bone with cement application.[5] There may be occasions where the instillation of methyl methacrylate is preferred to the bone graft. The use of methyl methacrylate has advantages because of heat and cytotoxic effect, which acts as an adjuvant treatment and has led to less recurrence. We have used four cycles of injection denosumab at days 1, 8, 15, and 28 days as the pain was not relieved much, and the patient was not willing for further injections because of financial constrain; we planned for curettage.
Figure 7: Sites of 1332 giant cell tumors of bone collected from the literature

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Figure 8: Sites of 57 giant cell tumors of the innominate bone collected from the literature

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Denosumab use in the treatment of giant cell tumor causes fast symptomatic and radiological recovery, is easy to use, and has an excellent adverse effect profile. No reconstruction was done as it was felt that this would not significantly improve the postoperative function and because reconstruction in this location can be complex and often adds to the morbidity of the surgical procedure. According to a study given by Mehmet Ali and Paydas,[13] curettage or resection material after the denosumab treatment was classified using a two-grade staging system developed according to the tumor response. Grade 1 was grouped as fibrous tissue, inflammation, and a small amount of woven bone formation; grade 2 was defined as woven bone, precancellous bone, and cancellous bone formation. We found no woven bone, and only fibrous tissue was harvested. So our study comes under stage 1. We found that the use of methyl methacrylate (Palacos) in filling the narrow cavity was very difficult because of the viscosity of the cement, and it was not possible to fill the entire cavity of the lesion keeping in mind that we could not extend the cavity because of the proximity of sciatic nerve, but this was our first experience. We will try to open the cavity next time with the standard approach, as with this approach, only limited area was visible. Further, we suggest the use of Simplex cement for easy administration. The patient is now planned for IV zoledronic acid therapy at 3-month interval for the first year and will be on a regular follow-up every 6 months from the second year onward.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Norman A Giant cell tumor. In: Radiology. Vol. V. 1st ed. Philadelphia: JB Lippincott; 1986.  Back to cited text no. 1
    
2.
Dahlin DC Giant cell tumor of bone: Highlights of 407 cases. AJR Am J Roentgenol 1985;144:955.  Back to cited text no. 2
    
3.
Jacobs TP, Michelsen J, Polay JS, D’Adamo AC, Canfield RE Giant cell tumor in Paget’s disease of bone: Familial and geographic clustering. Cancer 1979;44:742-7.  Back to cited text no. 3
    
4.
Upchurch KS, Simon LS, Schiller AL, Rosenthal DI, Campion EW, Krane SM Giant cell reparative granuloma of Paget’s disease of bone: A unique clinical entity. Ann Intern Med 1983;98:35-40.  Back to cited text no. 4
    
5.
Kattapuram SV, Phillips WC, Mankin HJ Giant cell tumor of bone: Radiographic changes following local excision and allograft replacement. Radiology 1986;161:493-8.  Back to cited text no. 5
    
6.
Dahlin DC, Cupps RE, Johnson EW Jr. Giant-cell tumor: A study of 195 cases. Cancer 1970;25:1061-70.  Back to cited text no. 6
    
7.
Goldenberg RR, Campbell CJ, Bonfiglio M Giant cell tumor of bone. J Bone Joint Surg [Am] 1970;52:619.  Back to cited text no. 7
    
8.
Hutter RV, Worcester JN Jr, Francis KC, Foote FW Jr, Stewart FW Benign and malignant giant cell tumors of bone. A clinicopathological analysis of the natural history of the disease. Cancer 1962;15:653-90.  Back to cited text no. 8
    
9.
Kuritzky AS, Joyce ST Giant cell tumor in the ischium. A therapeutic dilemma. JAMA 1977;238:2392-4.  Back to cited text no. 9
    
10.
McGrath PM Giant cell tumor of bone: An analysis of fifty-two cases. J Bone Joint Surg [Br] 1972;54:216.  Back to cited text no. 10
    
11.
Mnaymneh WA, Dudley HR, Mnaymneh LG Giant cell tumor of bone. J Bone Joint Surg [Am] 1964;46:63.  Back to cited text no. 11
    
12.
Mnaymneh W, Mnaymneh LG Giant cell tumor of the ischium: Unusual site and outcome. South Med J 1979;72:1012-4.  Back to cited text no. 12
    
13.
Ali M, Paydas S Clinical and pathological results of denosumab treatment for giant cell tumors of bone: Prospective study of 14 cases. AOTT 2017;51:1-6.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]



 

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