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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 52  |  Issue : 5  |  Page : 183-188

Gigantic giant cell tumor of the anterolateral rib treated with wide resection


1 Department of Orthopaedic and Traumatology, Dr.Cipto Mangunkusumo General Hospital, Faculty of Medicine Universitas Indonesia, Jakarta, Indonesia
2 Department of Surgery, Division of Cardiovascular and Thorasic Surgery, Dr.Cipto Mangunkusumo General Hospital, Faculty of Medicine Universitas Indonesia, Jakarta, Indonesia

Date of Submission20-Mar-2019
Date of Decision13-Jun-2019
Date of Acceptance23-Jul-2019
Date of Web Publication25-Oct-2019

Correspondence Address:
Dr. Achmad Fauzi Kamal
Department of Orthopaedic and Traumatology, Dr.Cipto Mangunkusumo General Hospital, Faculty of Medicine Universitas Indonesia, Jl. Diponegoro 7, Jakarta 10530
Indonesia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/fjs.fjs_28_19

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  Abstract 


Giant cell tumors usually arise in the epiphyseal region of the limbs, and their occurrence in the ribs is unusual. We presented a case of a 26-year-old male with a 2 years progressively growing lump on his right anterolateral chest. There was no pain and cardiopulmonary symptoms. Chest radiograph revealed an osteolytic lesion over the right 10th rib with chondroid matrix and a wide transition zone that grew expansively accompanied with the destruction of the 10th rib. Abdominal computed tomography scan confirmed a soft-tissue mass with 15 cm × 11 cm × 10 cm in dimension with the destruction of anterolateral 10th rib. The patient underwent en bloc-wide excision, including the 9th–11th ribs, followed by diaphragm repair and chest wall reconstruction.

Keywords: Chest wall reconstruction, giant cell tumor, wide excision


How to cite this article:
Kamal AF, Setiawan E, Wardoyo S. Gigantic giant cell tumor of the anterolateral rib treated with wide resection. Formos J Surg 2019;52:183-8

How to cite this URL:
Kamal AF, Setiawan E, Wardoyo S. Gigantic giant cell tumor of the anterolateral rib treated with wide resection. Formos J Surg [serial online] 2019 [cited 2019 Nov 12];52:183-8. Available from: http://www.e-fjs.org/text.asp?2019/52/5/183/269925




  Introduction Top


Giant cell tumor (GCT) is a benign neoplasm and characterized histologically by multinucleated giant cells with a background of mononuclear stromal cells. The multinucleated giant cells appear similar to osteoclasts, which led to the older term osteoclastoma.[1],[2] Despite being categorized as a benign lesion, GCT may be locally aggressive and recur after surgical resection.[2],[3]

GCTs usually arise in the epiphyseal region of the limbs, and their occurrence in the ribs is unusual.[4] These are usually found posteriorly in the ribs (epiphysis of head and tubercle), and their presentation anteriorly is very rare. Many large series have reported an incidence of around 1% in the ribs. Gupta and Mittal showed that most of these involved the posterior aspect of the rib after reviewing 15 cases.[5]

In this report, we present a rare case of gigantic GCT involving the anterolateral side of 9th–11th ribs treated with wide excision of the tumor, diaphragm repair, and reconstruction of the chest wall.


  Case Report Top


A 26-year-old male presented with a 2 years progressively growing lump on his right anterolateral chest. In the beginning, the lump was about marble-sized (2 cm diameter), and it grew progressively. Injust in 3 months, the lump had become about 6 cm in diameter. There was no pain related to the growing lump, and the patient still did his daily activity normally. He did not complain about any cardiopulmonary symptoms related to the mass. He underwent some investigation to confirm the diagnosis, including plain X-ray, computed tomography (CT) scans, and core biopsy. The histopathological result from core biopsy was suitable for GCT of the bone. The patient was medically advised to have the mass excised, but he refused it and opted for traditional medicine. He consumed traditional herbs, but the history of massage was denied. Thirty months after the first symptom, the patient came back to the doctor with a bigger mass size. On physical examination, the mass was around 14 cm in diameter with the normal overlying skin, without any ulcer and venectation [Figure 1]. The patient underwent some examinations again to enforce the diagnosis. The alkaline phosphatase level was 136 U/L, slightly increased erythrocyte sedimentation rate level; other laboratory findings were within normal limit. Chest and lumbosacral radiographs revealed an osteolytic lesion over the right 10th rib with chondroid matrix and wide transition zone, that grew expansively which resulted in thinning cortex and endosteal scalloping accompanied with the destruction of the rib [Figure 2].
Figure 1: Clinical presentation showed a lump on the right anterolateral side of the thoracoabdominal region

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Figure 2: (a) Chest and lumbosacral radiographs revealed an osteolytic lesion over the right 10th rib with chondroid matrix and wide transition zone, that grew expansively which resulted in thinning cortex and endosteal scalloping accompanied with the destruction of the rib. (b) Preoperative lumbosacral anteroposterior radiograph demonstrates increased mass compared to radiographic findings on the first admission

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Initial abdominal CT scans showed a soft-tissue mass with 15 cm × 11 cm × 10 cm in dimension with the destruction of the anterior side of the 10th rib [Figure 3]b. The tumor size was increased compared with the previous abdominal CT scan examination showing 5.9 cm × 8.6 cm × 10.3 cm in dimension [Figure 3]a. The mass compressed the right liver lobe medially and right kidney posteriorly.
Figure 3: (a) Initial abdominal computed tomography scan showed a soft-tissue mass with 15 cm × 11 cm × 10 cm in dimension with the destruction of the anterior side of the tenth rib. (b) The tumor size was increased compared with the previous abdominal computed tomography scan examination showing 5.9 cm × 8.6 cm × 10.3 cm in dimension

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The patient underwent wide resection of the tumor and reconstruction of the chest wall. He was placed on the right lateral decubitus position on the operating table under general anesthesia. The skin incision was conducted above the tumor mass (20 cm in length). The cutis, subcutis, and muscle were placed superiorly and inferiorly until the margin of the tumor was clearly palpable. The thoracic wall was exposed and 9th–11th ribs were identified and osteotomized proximally and distally around 2 cm from the mass. The mass was attached to the diaphragm and extended to the abdominal cavity. The mass was excised including partially muscle, diaphragm, and peritoneum attached to the tumor mass (wide excision). The peritoneum was primarily sutured and the defect of the diaphragm was repaired. The chest wall defect was reconstructed by prolene mesh application [Figure 4]. The final histopathology revealed GCT of bone accompanied by secondary aneurysmal bone cyst (ABC) with tumor-free surgical margins [Figure 5].
Figure 4: Intraoperative procedure. (a) He was placed on the right lateral decubitus position on the operating table under general anesthesia. (b) The incision was conducted above the tumor mass until the margin of the tumor were clearly palpable. (c). The thoracic wall was exposed and 9th–11th ribs were identified and osteotomized. (d) The mass was excised, including partially diaphragm and peritoneum attached to the tumor mass. (e) The peritoneum was primarily sutured and the defect of the diaphragm was repaired. Chest wall defect was reconstructed by prolene mesh

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Figure 5: (a) Gross pathology of the tumor. (b) Histopathology revealed giant cell tumor of bone accompanied by secondary aneurysmal bone cyst with tumor-free surgical margins

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A day postoperatively, the patient was instable condition using water-sealed drainage with re-expansion lung. The patient was discharged from our hospital on postoperative day 7. The outpatient follow-up evaluation showed that the patient was in very good general health. The patient showed no recurrence and asymptomatic 12 months after the surgical procedure.


  Discussion Top


Ninety percent of GCT exhibits the typical epiphyseal location.[6],[7] The sites most commonly affected are the distal femoral epiphysis (36%) and proximal tibial epiphysis (17%), followed by the distal radius (15%). Although rarely occurring in the axial skeleton, among the 2% of GCTs that affect that region such as sacrum and rib.[8] Involvement of the anterior arc of a rib is a very rare occurrence with only a few cases recorded previously in the literature.[9],[10],[11] The most GCTs have been located in the posterior arc of the rib (the head or the tubercle of the rib).[12] Furthermore, because of its rarity, GCT arising from the ribs are difficult to diagnose, especially when the tumor is located in the anterior arc of the ribs,[11],[12] inline with our case.

GCTs are aggressive tumors and present with the signs and symptoms of various pains, swelling or mass.[4],[10],[13] Limitation of motion about a joint when the GCT is located in the extremity.[4] Some patients present with pathological fracture resulting from the weakening of the cortical bone.[4],[8] Twenty percent have an associated soft-tissue mass with a cortical breakthrough on radiographic images. The patient typically has a benign course. Less than 5% of patients will show evidence of metastatic involvement, usually to the lung.[14] A study mentioned that patients experienced symptoms for an average of 10 months prior to the first treatment of benign GCT.[4],[10] Sharma and Armstrong[4] reported published cases of GCTs arising from the arc of the rib on the surgical specimen size. Eight of 13 cases, 4 of 13 cases, and 1 of 13 cases had <10 cm, >10–15 cm, and more than 15 cm in diameter, respectively.[4]

Our patient noted a painless growing mass over a period of 2 years, and it began more progressively near his time of surgery. This delayed surgery was due to the painless lesion, and he was scared of dealing with the surgery. Therefore, the extremely large tumor size (>15 cm specimen surgical size) was found at the time of surgical resection. We assumed that it is” a gigantic” case.

Normally, the radiograph shows a purely lytic expansive lesion, which is eccentric from the axis of the rib and does not contain calcifications. It has distinct outlines and no peripheral sclerotic bone changes. GCT can also show cortical thinning, cortical bone destruction, and soft-tissue mass formation.[2],[6],[15],[16] Unless a fracture is present, no periosteal reaction is found. CT scan confirms that calcification is not present and can be used to assess the cortical damage and to examine the extension of the lesion into the soft tissues.[2],[16],[17]

Secondary ABC formation occurs in up to 14% of cases of GCT of bone. These entities can mimic each other at radiologic evaluation. GCT can be an underlying condition of secondary ABC.[18] The development of hemorrhage within cystic spaces (ABC) of a GCT leads to the fluid levels identified on cross-sectional imaging.[2],[16]

A range of differential diagnoses of GCT may need to be considered, and biopsy is warranted to provide histological confirmation of the lesion.[4],[8] Microscopically, the two basic components of benign GCTs are stroma and multinucleated giant cells, the stromal cells are mononuclear and may be spindle-shaped, ovoid, or round, while the multinucleated giant cells may be so large that the numerous nuclei are almost uncountable.[4]

The clinical, radiological, and histopathological characteristics of GCTs of the ribs are similar to those of various tumors and pseudotumors, such as Brown tumors.[7] Other lesions, such as an ABC, telangiectatic osteosarcoma, malignant fibrous histiocytoma, and chondroblastoma, are considered in the differential diagnosis.[8],[18] In our case, the initial impression of the musculoskeletal radiologist included chondrosarcoma, metastatic tumor, and malignant fibrous histiocytoma. GCT was not included in the differential diagnosis.

No standard treatment for GCT of the rib has been established.[10] Current methods available to treat GCT include curettage with or without the use of alcohol, liquid nitrogen, phenol or methyl acrylate or bone graft, and complete surgical resection of the affected segment of bone.[4],[7],[11] Although GCT is described as benign, it has a high rate of local recurrence and can metastasize to the lungs.[11] Thus, wide surgical excision and curettage with regular follow-up remain the mainstay of management.[4],[11]

Some authors have reported that a wide excision with chest wall reconstruction had been successful for treating GCT arising from the rib.[7],[10],[18],[19],[20] Aggressive surgical resection with wide margins must be the first choice of treatment for this tumor.[14] Various studies suggest that a wide resection is associated with a decreased risk of local recurrence when compared with intralesional curettage and may increase the recurrence-free survival rate from 84% to 100%.[7] Meanwhile, Su et al.[19] reported the recurrence rate after wide resection (3.2%) was much lower than after curettage (17.9%). In one series, 81% of recurrences occurred within 2 years, although it is generally agreed that the recurrence rate can be markedly reduced with an appropriate technique.[11] Su et al.[19] mentioned that the only significant factor was the completeness of surgical removal of the tumor. In our case, the patient underwent en bloc-wide excision and chest wall reconstruction to prevent the recurrence of GCT. Moreover, a margin of 1–2 cm of healthy tissue (bony and muscle chest wall) is achieved, like a recommendation from Collado Otero et al.[20]

Radiation therapy is not recommended because most of the malignant transformations in GCT are associated with previous radiation therapy.[9] Even in cases with pulmonary metastasis, aggressive surgical extirpation is recommended.[4],[14],[18] Dahlin[21] reported that radiotherapy was not recommended because most of the malignant transformations in GCT were associated with the previous radiotherapy. However, in patients with GCT who cannot undergo surgery due to medical comorbidity, the extent of the tumor, or predicted unacceptable functional outcome, radiotherapy has been recommended.[10]

Reconstruction of the bony chest wall has various options and techniques available, each with its own advantages and disadvantages. The reconstruction is not required if the defect is <5 cm in any greatest diameter and even defects up to 10 cm in the posterior wall are well covered by the scapula.[22] There is a wide choice of prosthetic material available for reconstruction for the bony chest wall. Goretex and prolene mesh tend to be the regularly used prosthesis vicryl mesh used for temporary stabilization.[22],[23] In this case, wide excision and reconstruction of the chest wall had been carried out. Although the prolene mesh does not achieve a watertight seal and is difficult to stretch and suture, it is far cheaper and available any time. Therefore, we chose it to reconstruct our case. Goretex mesh is watertight but has to be a thicker 2 mm mesh to hold the sutures at the tension required for stabilization of the chest wall.[22] Another option of material for reconstruction of the defect in the chest wall is a double-layer polypropylene (Marlex) mesh sandwiched. A mild degree of paradoxical motion was well tolerated because the area of instability was relatively small.[20]

We also resected the tumor which was directly extended to the diaphragm creating a defect and repaired it. Regarding the literature, reconstruction of the diaphragm involving tumor should be done by an experienced surgeon because the lack of appropriate repair can lead to life-threatening complications. A poorly reconstructed diaphragm can result in symptomatic atelectasis or recurrent pneumonia if the diaphragm is repaired with a patch that is too large. Alternatively, if too much tension is placed on a repair, it can ultimately break down and lead to a diaphragmatic hernia with potential for serious complications. This can be of rapid or insidious onset, and it is wise to suggest long-term radiographic follow-up for any patient who has had diaphragm resection.[22]

The tumor is locally aggressive, but distant metastases are rare. Pulmonary metastases may be solitary or multiple are seen in 2% of patients with GCTs, an average of 3–4 years after primary diagnosis. Some of these metastases may slowly grow (benign pulmonary implants) and some may regress spontaneously. They rarely prove fatal and hence the term benign metastasis is appropriate. Few factors are responsible for metastasis, such as local recurrence, surgical manipulation, and location in the distal radius.[9] Even in cases with pulmonary metastasis, aggressive surgical extirpation is recommended.[4],[14],[18] In other words, it is generally agreed that wide (complete) resection may reduce potential metastases.


  Conclusion Top


We reported a case of gigantic GCT originating from the anterior arc of the 10th rib. Although GCT of the rib is rare, the differential diagnosis of an anterior chest wall mass should include GCT of the rib. In a year follow-up, wide excision with chest wall reconstruction has proven to be a successful treatment of a “gigantic” GCT originating from the rib.

Ethical statement

A case report was exempt from our ethical committee.

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

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Sharma A, Armstrong AE. Giant cell tumor of rib arising anteriorly as a large inframammary mass: A case report and review of the literature. Case Rep Med 2012;2012:850509.  Back to cited text no. 4
    
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Gupta V, Mittal R. Giant cell tumor of rib – Rare location on the anterior aspect. Arch Orthop Trauma Surg 2000;120:231-2.  Back to cited text no. 5
    
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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