Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 10  |  Issue : 5  |  Page : 485-487  

Osteosarcoma of first metacarpal with bilateral lung metastasis: A rare case report


1 Department of Pathology, Government Medical College, Kota, Rajasthan, India
2 Department of Radiology, Government Medical College, Kota, Rajasthan, India
3 Department of Orthopedics, Government Medical College, Kota, Rajasthan, India

Date of Submission05-Oct-2016
Date of Acceptance05-Dec-2016
Date of Web Publication14-Nov-2017

Correspondence Address:
Mamta Jain
w/o Dr. Abhishek Jain, c/o Manish Book Depot, Rampura Bazar, Kota - 324 009, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0975-2870.218179

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  Abstract 


Osteosarcomas of hands are rare, (<1%) and these cases differ in presentation and behavior compared to those in usual locations. Histological grade was the only significant single prognostic variable for risk of death. High-grade osteosarcoma of the hand should be treated similar to those in conventional sites with ablative surgery and neoadjuvant chemotherapy. Here, we report a case of high-grade osteosarcoma of the first metacarpal. World literature shows occasionally reported case of this site. Rarity of the lesion and creating awareness among surgeons about existence of such a lesion in smaller bones is our motto. This would prompt them for an early effort for proper diagnostic procedure and subsequent early treatment so that prognosis of this grave disease can be improved. In the present case, diagnosis was delayed and pulmonary metastasis was present at time of diagnosis. The patient is presently undergoing chemotherapy and under regular follow-up.

Keywords: First metacarpal, osteosarcoma, pulmonary metastasis


How to cite this article:
Saxena R, Jain M, Singh H, Meena R P. Osteosarcoma of first metacarpal with bilateral lung metastasis: A rare case report. Med J DY Patil Univ 2017;10:485-7

How to cite this URL:
Saxena R, Jain M, Singh H, Meena R P. Osteosarcoma of first metacarpal with bilateral lung metastasis: A rare case report. Med J DY Patil Univ [serial online] 2017 [cited 2019 May 19];10:485-7. Available from: http://www.mjdrdypu.org/text.asp?2017/10/5/485/218179




  Introduction Top


Most of the hand tumors are benign, 1%–2% of hand tumors are malignant.[1] Osteosarcoma are malignant tumor characterized by direct formation of osteoid or immature bone by malignant cells. It usually develops in long bones and rare in short bones. Osteosarcoma of hand accounts for 0.18% of all osteosarcoma.[2] Hence, osteosarcoma of hand is clinically unexpected and diagnosis is often delayed or initially erroneous, leading to delayed or inappropriate treatment decisions. Although most of high grade osteosarcoma of hand has remarkably high survival rate.[3]

According to the American Cancer Society if osteosarcoma has spread only to lungs, the survival rate is closer to 40% but if it has already spread to other organs when it is first found, the 5-year survival rate is about 15%–30%.

The rarity of lesion with bilateral lung metastasis prompted us to report this case.


  Case Report Top


A 39-year-old female came to orthopedic outdoor due to enlarging mass with pain at base of thumb at dorsal aspect of the first metacarpal for 2 months. She was consulting a local practitioner for the same problem and got some relief after taking nonsteroidal anti-inflammatory drugs. She had no history of trauma, weight loss, evening rise of temperature, loss of appetite, no respiratory symptoms, or any other medical history.

On physical examination, diffuse swelling was palpable around the first metacarpal at dorsal aspect. It was irregular, firm, and tender. Swelling was tense and showed discoloration of overlying skin.

Imaging of thumb revealed multiple small osteolytic lesions with cortical break and larger soft tissue component, subcutaneous involvement, and causing displacement of adjacent muscular plane suggestive of osteosarcoma [Figure 1].
Figure 1: Magnetic resonance imaging (T2-weighted) of thumb showing multiple osteolytic lesions with cortical break

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Blood parameters such as serum lactate dehydrogenase and alkaline phosphatase were done and were found to be raised. Other blood tests such as complete blood count, liver function test, and renal function test were within normal limits.

Incisional biopsy was taken which shows atypical chondrocytes surrounded by pleomorphic richly cellular areas showing increased mitosis with fragments of osteoid by pleomorphic ovoid cells. Features were in favor of chondroblastic osteosarcoma [Figure 2].
Figure 2: H and E section revealing atypical spindle cells and formation of osteoid along with a quadripolar mitosis

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Before surgery, high-resolution computed tomography (CT) of lung was done which showed multiple diffusely distributed varying sized, well-defined soft tissue density space occupying lesion with internal punctate calcifications seen in bilateral lungs suggestive of bilateral pulmonary metastasis [Figure 3].
Figure 3: High-resolution computed tomography of lung showing multiple metastatic lesions

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The patient underwent below elbow amputation and receiving chemotherapy. Amputated specimen was sent for biopsy which confirmed our earlier diagnosis of chondroblastic osteosarcoma, and now, the patient is under regular follow-up.


  Discussion Top


A very few case of osteosarcoma of metacarpal bones has been published in literature, and together with bilateral lung metastasis is very rare.

Osteosarcoma occurs most commonly in 10–20 years age group and in seventh to eighth decade. Before 1970, prognosis for patient with high-grade osteosarcoma was poor with long-term survival <20%, but advances in adjuvant and neoadjuvant chemotherapy have improved 5-year disease-free survival to more than 60%.[4]

Kuniko et al. said that osteosarcoma of short tubular bone is extremely rare; but it should be included in differential diagnosis if radiological findings or clinical courses are not entirely typical of a more common benign process, to avoid incorrect clinicoradiological and pathological diagnosis.[5]

Lung is the most common site of metastases with 77%–92% of patient experiencing recurrence at this site.[6]

The patient with osteosarcoma of extremity who presents with lung metastasis has a poorer prognosis than those without metastasis. Nonetheless, with the combination of chemotherapy and complete surgical removal of primary and secondary lesions, the survival of patient with metastases has improved from <5% to >20%. However, when complete excision of all secondary lesions is impossible, the 5-year survival rate become extremely low and almost all of these patient die within 3 years from the time of diagnosis.[7]

Several epidemiologic risk factors have been related to the development of osteosarcoma, including a history of ionizing radiation exposure, fibrous dysplasia, retinoblastoma, or history of trauma. Risk factors were negative in our case.

No single feature on radiograph is diagnostic. Osteosarcoma lesions can be purely osteolytic, purely osteoblastic, or mixed. Imaging of primary lesion helps in delineating the location and extent of tumor and is critical for surgical planning. In our case, imaging demonstrated mainly osteolytic lesion with surrounding soft tissue involvement. CT of the chest is more sensitive than plain radiography for properly assessing pulmonary metastasis.[8]

On histologic examination of the tumor, two elements are most important; first, the type of tumor can be assessed by the biopsy specimen. Second, the response to treatment can be assessed only by evaluating the tissue resected after chemotherapy. The characteristic feature of osteosarcoma is the presence of osteoid in the lesion, even at the site distant from bone (e.g., lung). Various histologic subtypes have been described (osteogenic, chondroblastic, fibrogenic) although they are clinically indistinguishable.[9],[10]

The differential diagnosis of osteosarcoma of hand includes benign conditions such as osteoid osteoma and fibrous dysplasia or desmoplastic fibroma.[2] The clinical finding of bone pain and swelling in overlying soft tissue may result in mistaken diagnosis of osteoid osteoma.[11]

Metastatic disease at presentation or later, a pathologic fracture, larger tumor volume, and adequate margin at the time of surgery were associated with a significant lower survival. Hence, aggressive multiagent regimens are needed to improve survival.[12]


  Conclusion Top


Osteosarcomas of small hand bones are very rare condition and awareness among surgeons of this condition can significantly lower morbidity and mortality from this grave disease. A need for early diagnostic procedure and prompt ablative surgery can play a major role in providing a good prognosis to the patient.

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.
Sur YJ, Kang YK, Bahk WJ, Chang DK, Rhee SK. Metastatic malignant tumour in the hand. J Plast Surg Hand Surg 2011;45:90-5.  Back to cited text no. 1
    
2.
Okada K, Wold LE, Beabout JW, Shives TC. Osteosarcoma of the hand. A clinicopathologic study of 12 cases. Cancer 1993;72:719-25.  Back to cited text no. 2
    
3.
Daecke W, Bielack S, Martini AK, Ewerbeck V, Jürgens H, Kotz R, et al. Osteosarcoma of the hand and forearm: Experience of the Cooperative Osteosarcoma Study Group. Ann Surg Oncol 2005;12:322-31.  Back to cited text no. 3
    
4.
Bacci G, Picci P, Ferrari S, Ruggieri P, Casadei R, Tienghi A, et al. Primary chemotherapy and delayed surgery for nonmetastatic osteosarcoma of the extremities. Results in 164 patients preoperatively treated with high doses of methotrexate followed by cisplatin and doxorubicin. Cancer 1993;72:3227-38.  Back to cited text no. 4
    
5.
Abe K, Kumagai K, Hayashi T, Kinoshita N, Shindo H, Uetani M, et al. High-grade surface osteosarcoma of the hand. Skeletal Radiol 2007;36:869-73.  Back to cited text no. 5
    
6.
Strauss SJ, McTiernan A, Whelan JS. Late relapse of osteosarcoma: Implications for follow-up and screening. Pediatr Blood Cancer 2004;43:692-7.  Back to cited text no. 6
    
7.
Palmerini E, Staals EL, Ferrari S, Rinaldi R, Alberghini M, Mercuri M, et al. Nonresectable multiple lung metastases of high-grade osteosarcoma of the humerus: Stable after twelve years. A case report. J Bone Joint Surg Am 2008;90:2240-4.  Back to cited text no. 7
    
8.
Mirra JM, editor. Bone Tumors: Clinical, Radiological and Pathological Correlation. Philadelphia: Lea and Febiger; 1989. p. 250-2.  Back to cited text no. 8
    
9.
Bacci G, Ferrari S, Bertoni F, Ruggieri P, Picci P, Longhi A, et al. Long-term outcome for patients with nonmetastatic osteosarcoma of the extremity treated at the istituto ortopedico Rizzoli according to the istituto ortopedico Rizzoli/osteosarcoma-2 protocol: An updated report. J Clin Oncol 2000;18:4016-27.  Back to cited text no. 9
    
10.
Bacci G, Briccoli A, Longhi A, Ferrari S, Mercuri M, Faggioli F, et al. Treatment and outcome of recurrent osteosarcoma: Experience at Rizzoli in 235 patients initially treated with neoadjuvant chemotherapy. Acta Oncol 2005;44:748-55.  Back to cited text no. 10
    
11.
Ellman H, Gold RH, Mirra JM. Roentgenographically “benign” but rapidly lethal diaphyseal osteosarcoma; a case report. J Bone Joint Surg Am 1974;56:1267-9.  Back to cited text no. 11
    
12.
Carsi B, Rock MG. Primary osteosarcoma in adults older than 40 years. Clin Orthop Relat Res 2002;397:53-61.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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