Total elbow arthroplasty as a reconstruction option for distal humerus osteosarcoma: A case report

IntroductionOsteosarcoma is the second most common malignant bone tumor. The humerus is the third most common anatomical location for osteosarcoma, however, osteosarcoma around the elbow joint is uncommon. The intricacy of the elbow joint, limited soft tissue coverage, and proximity to nerves and arteries make the surgical resection and reconstruction complicated.Case reportA 17-year-old boy came with a chief complaint of lump and pain on his left elbow. One month later, the patient felt there was a lump with a size of a marble on the left elbow, which getting bigger to a size of a tenis ball. Physical examination showed mass on the posterior aspect of the elbow with the size 14x12cm. Plain radiographs revealed osteolytic lesion and periosteal reaction of the distal humerus and the magnetic resonance imaging (MRI) showed a low intensity on T2-weighted imaging. Histopathological examination suggested osteosarcoma. The patient underwent neoadjuvant chemotherapy for 3 cycles. The patient was treated with limb salvage surgery by wide excision, cryosurgery followed by total elbow arthroplasty and ORIF with plate and screw. Postoperative plain radiographs showed the plate and screws are well-fixated. The patient can slowly regain his elbow motion without limitation one month postoperatively.ConclusionsDistal humerus in an unusual site for osteosarcoma. Total elbow arthroplasty and ORIF with plate and screw is a favorable reconstruction option for distal humerus osteosarcoma with excellent postoperative functional outcomes.     

Implantation metastasis in a 13-year-old girl: a case report

We report a case of a 13-year-old girl with an osteosarcoma of the right humerus, which had been diagnosed as an aneurysmal bone cyst at our institution. She underwent curettage and bone grafting of the lesion, which resulted in implantation metastasis of the tumour to the ilium. She died 15 months after presentation owing to pulmonary metastases. This report highlights the possibility of metastasis occurring by direct implantation to a graft donor site. We strongly recommend that a biopsy be performed in cases of presumed benign lesions before proceeding with the definitive surgery.     

A successful reconstruction using a frozen autograft and a pedicled latissimus dorsi flap after a S12345B shoulder girdle resection in a patient with osteosarcoma

We present a case of high-grade osteosarcoma in the proximal humerus in an 8-year-old boy. Massive tumor expansion required a S12345B shoulder girdle resection according to the system of the Musculoskeletal Tumor Society. After wide resection, only a small portion of the distal humerus and none of the rotator cuff muscles would be spared. Because the humeral portion would be too short to support the stem and the soft tissue would be insufficient to cover prosthetic components, we designed a composite reconstruction using a frozen autograft and a pedicled muscle flap. At 2 years postoperatively, our patient exhibited good adaptation and had acquired fine dexterity of the upper limb. To our knowledge, this is the first report regarding a successful composite reconstruction after a S12345B shoulder girdle resection.     

Primary parosteal osteosarcoma of the rib

Osteosarcoma is a common primary malignant tumour of bones that produces an osteoid matrix. Parosteal osteosarcoma is an uncommon neoplasm and principally affects the long bones, especially in the distal femur, proximal tibia and proximal humerus. Rarely, the tumour may arise in a flat bone, and rib involvement is very infrequent. An unusual case of primary parosteal osteosarcoma of the rib in an asymptomatic 28-year old female is presented here. A chest X-ray film suggested an intrapulmonary homogeneous mass, while a computed tomography scan revealed a broad-based calcified mass attached to the inner cortex of the right fourth anterior rib. The patient underwent a wide excision of the tumour together with adjacent intercostal muscles and chest wall reconstruction. Postoperative histopathological outcome was consistent with primary parosteal osteosarcoma.     

Management of the contralateral testicle in patients with unilateral testicular cancer

Patients with a prior history of a germ cell tumor of the testicle are known to have an increased risk of development of a second germ cell tumor in the contralateral testicle. It is believed that all patients who develop a germ cell tumor of the testicle have a precursor lesion know as carcinoma in situ (CIS) or intratubular germ cell neoplasia. Approximately 50% of these patients will subsequently go on to develop a germ cell tumor in the testicle. A biopsy of the contralateral testicle in a patient with a previous history of a germ cell tumor of the testicle has been advocated by some, while others recommend a biopsy only in patients with other risk factors as well. The arguments for biopsy are that intervention may be used to prevent the development of a second germ cell tumor when CIS is detected. In this review we present the arguments for and against a biopsy of the contralateral testicle, review the techniques of biopsy as well as its complications, and discuss the interventions employed to prevent CIS from progressing.     

Osteosarcoma developing after radiation and chemotherapy for primary mediastinal seminoma

This report presented an osteosarcoma arising from the same field after radiation and chemotherapy without surgery for primary mediastinal seminoma. A 45-year-old man had received from June 1979 to August 1981 irradiation and chemotherapy for the anterior-mediastinal seminoma. Since then he was been without evidence of disease for five years. In March 1987, he had suffered from an advanced tumor extending from the neck and the mediastinum to the right anterior chest wall. Further irradiation combined with thermotherapy were performed but without improvement. He was thereafter seen and admitted July 23 1987 to our hospital with complaint of back pain. Needle biopsy specimen revealed chondrosarcoma. Despite chemotherapy with CDDP, he died four months later after admission. Pathological finding of his autopsy revealed a widely invading osteosarcoma extending to extra- and intrathorax and the neck without evidence of germ cell component. To explain the pathogenesis of the secondarily developing osteosarcoma, two hypotheses are offered; 1. Malignancy of mesenchymal component in germ cell tumor. 2. Radiation-induced osteosarcoma. The latter hypothesis appears to be the most probable pathogenesis from his autopsy. These findings suggest that a long survival patient with malignant germ cell tumor after therapy should be never considered as being free from a potential risk of secondarily developing malignancy.     

Multiple myeloma and intramedullary nailing

We present the case of a 58 year old man who presented with a pathological fracture of his right humerus. Further investigations revealed he had IgA multiple myeloma. A skeletal survey showed lytic lesions in his contralateral humerus and both femora. He was treated with the insertion of intramedullary nails to both humeri and both femora, in addition to undergoing systemic therapy for his myeloma. This paper reports, for the first time, the use of four intramedullary nails in the same patient and examines the principles behind the management of pathological bone disease.     

血管重建在骨与软组织肉瘤保肢术中的应用

目的 探讨血管重建在骨与软组织肉瘤保肢术中的作用.方法 2004年8月至2009年6月累及肢体重要血管的骨与软组织肉瘤患者13例接受保肢手术,男8例,女5例;年龄14～63岁,平均38岁.恶性骨肿瘤4例,软组织肉瘤9例.1例位于肱骨上端,12例位于下肢.7例同时重建动脉和静脉,6例仅重建动脉.5例采用同侧或对侧大隐静脉移植,8例采用人工血管置换.结果 无一例出现重建血管感染.1例肱骨骨肉瘤行肱动脉人工血管置换的患者于术后第1天出现急性动脉栓塞,急诊取栓后保留了肢体.人工血管置换8例中出现肢体肿胀和伤口不愈合5例,大隐静脉移植5例伤口均一期愈合.全部患者随访7～45个月,平均19.4个月.1例肱骨骨肉瘤患者术后12个月出现局部复发,保肢成功率92.3%(12/13).1例术前肺转移患者于术后7.5个月死于肺转移,另4例患者于术后半年至1年出现肺转移.Kaplan-Meier生存曲线分析患者2年总生存率和无瘤生存率分别为90.9%和63.6%,重建动、静脉2年通畅率分别为100%和28.6%.12例下肢肿瘤患者末次随访时的1993年美国骨肿瘤学会评分系统评分平均为72%,肱骨骨肉瘤患者为33%.结论 肿瘤累及血管不是截肢的绝对适应证,肿瘤连同重要血管整块切除后进行血管重建的保肢手术可行,可获得良好的局部肿瘤控制和肢体功能.

Abstract：

Objective To determine the feasibility of limb salvage with major vascular reconstruction for sarcomas of extremities, focusing on the early complications, oncological and functional outcomes.Methods Between August 2004 and June 2009, 13 patients (mean age 38 years, range 14-63 years) underwent wide resection of upper and lower extremity sarcomas to include the involved arterial and venous segments. 4 patients had bone sarcomas and 9 soft tissue sarcomas, 6 patients only had underwent artery replacement and the other 7 had underwent both artery and vein. 8 resected vessels were reconstructed with vascular graft and 5 with ipsilateral or contralateral saphenous vein. Results No patient suffered from reperfusion injury and infection. The patient with osteosarcoma in proximal humerus had suffered acute arterial graft thrombosis at first day postoperatively; however, the limb was salvaged after successful thrombectomy. 5 of 8 patients with synthetic graft developed minor delayed wound healing and hematoma, but the other 5 patients with autologous saphenous vein replacement did not. The mean follow-up was 19.4 months (7-45months). One patient died of pulmonary metastasis; the patient with osteosarcoma in proximal humerus had developed local recurrence, but refused amputation and was alive with metastasis. No arterial occlusion was observed at final follow-up or at the time of death, but the patency of artery and vein was 100% and 28.6%respectively. One patient who has pulmonary metastases before operation died of metastases 7.5 months after operation. Four patients developed pulmonary metastases, therefore, Kaplan-Meier survival analysis showed that 2-year overall and metastasis-free survival rate was 90.9% and 63.6% respectively. Functional status were judged as good or excellent (mean MSTS score 72%) in 12 of 13 patients. Conclusion The study indicate that malignant involvement of major vessels is not a contraindication for limb-salvage. Vascular reconstruction is a feasible option in limb salvage surgery. Wide resection with vascular reconstruction provide acceptable oncological and functional outcome of limb salvage.     

Haemophilus aphrophilus osteomyelitis after dental prophylaxis. A case report.

A 36-year-old patient who was otherwise healthy had acute osteomyelitis of the humeral shaft develop after routine prophylactic dental cleaning and ultrasonic scaling. Haemophilus aphrophilus grew on cultures of material obtained during biopsy of the humerus, and pathologic examination confirmed the diagnosis of acute osteomyelitis. Haemophilus aphrophilus, a fastidious gram negative bacillus, is part of the normal oral flora and is a rare pathogen. Osteomyelitis caused by Haemophilus aphrophilus has not been reported to occur after routine dental prophylaxis. The patient was treated successfully with surgical debridement and appropriate antibiotics.     

右肱骨富于巨细胞性骨肉瘤1例

富于巨细胞性骨肉瘤是骨肉瘤的一种罕见类型,病理学上易误诊为巨细胞瘤.由于两者的治疗方法有差异,所以误诊往往影响术者选择正确的手术方法.本文治疗1例富于巨细胞性骨肉瘤的患者(女,16岁),术中行右肱骨近端肿瘤切除同时取不带血管的自体腓骨移植重建肱骨头及肱骨干,术后肩关节保留部分功能.     

Osteosarcoma of the skull base: a case report

Osteosarcoma, usually observed in long bones, is the second most frequent primitive malignant bone tumor after myeloma. The skull base is an exceptional localization. We report a case of skull base osteosarcoma managed in our department. A 23-year-old female was admitted for bilateral epistaxis, headache, decreasing visual acuity then blindness. Physical examination revealed bilateral blindness and exophthalmia. Cranial magnetic resonance imaging showed a voluminous mass in the skull base extending to the nasosinusal area. A rhinoseptal surgical approach was used but the tumor was so huge that excision was impossible. The biopsy identified at an osteoblastic osteosarcoma. Search for extension (chest computed tomography and abdominal ultrasonography) was negative. Chemotherapy was to be delivered before combination chemoradiotherapy but the patient died before any treatment could be started. Osteosarcoma of the skull base is very rare. The treatment is based on surgery which should be as complete as possible followed by chemoradiotherapy. Prognosis is poor. Median survival is around six months.     

Cystic fibrous dysplasia of the humerus

The patient was a 39-year-old woman with a 3-month history of worsening right distal humerus pain that was insidious in onset. At the time of the initial physical therapy visit, the patient complained of weakness and numbness throughout the entire right upper extremity, with an inability to perform daily activities. Magnetic resonance imaging revealed extensive bone marrow signal abnormalities extending from the proximal humeral epiphysis to the distal humeral diaphysis. Further evaluation with radiographs and computed tomography revealed multiple lytic lesions through the humerus. Tissue biopsy confirmed a diagnosis of cystic fibrous dysplasia.     

Fine needle aspiration biopsy of primary bone tumors

A review of 66 consecutive fine needle aspiration biopsies of primary bone tumors revealed that 48 (73%) were diagnostic. Twelve (18%) yielded inadequate specimens unsatisfactory for diagnosis, and five (8%) yielded specimens adequate for partial diagnosis. The only error, presumably attributable to sampling error, was an unappreciated dedifferentiated osteosarcoma arising in an otherwise typical giant cell tumor. Fine needle aspiration biopsy obviated the need for open biopsy in 24 patients and simplified surgery in an additional 24 patients by establishing the diagnosis before surgical intervention. A solitary soft tissue recurrence of a giant cell tumor has been the only local recurrence. A review of 26 consecutive patients with osteosarcoma revealed that seven tumors were diagnosed by primary open biopsy. Nineteen patients had fine needle aspiration biopsy, of which 15 were diagnostic and four required supplemental open biopsy. The elapsed time between the initial office visit and the diagnostic confirmation averaged 5 days for patients requiring open biopsy compared with 0 days for patients whose fine needle aspiration biopsy was diagnostic. The total estimated charge for fine needle aspiration biopsy of a distal femoral osteosarcoma was $1060.00 compared with $4312.25 for open biopsy. There have been no local recurrences in patients in either group. Fine needle aspiration biopsy provides an accurate, safe, efficient, well tolerated, and cost-effective method for diagnosing classic primary bone tumors, including osteosarcoma.     

上肢骨肿瘤切除后的自体骨移植重建

目的探讨应用自体骨移植对上肢骨肿瘤切除后的骨缺损修复重建的效果。方法1998年8月～2004年3月，收治上肢骨肿瘤切除后的骨缺损16例。男8例，女8例。年龄7～45岁。经病理确诊，肱骨近端尤文肉瘤和骨肉瘤各1例；肱骨远端尤文肉瘤2例；桡骨远端骨巨细胞瘤8例，高分化软骨肉瘤2例，恶性纤维组织细胞瘤和骨肉瘤各1例。2例肱骨近端肿瘤行自体锁骨代肱骨；2例肱骨远端肿瘤行自体腓骨代肱骨；12例桡骨远端肿瘤中，1例行自体髂骨移植，11例行自体腓骨代桡骨进行重建。采用MSTS系统进行术后功能评价。结果2例肱骨近端自体锁骨移植患者分别随访36个月和12个月，术后保持部分肩关节前屈和后伸功能，但外展功能丧失；MSTS评分分别为23分和22分。2例肱骨远端自体腓骨移植患者分别随访4个月和6个月，肘关节功能良好，移植骨连接处已经出现骨愈合；MSTS功能评分分别为24分和19分。12例桡骨远端自体骨移植患者中11例随访6～75个月，功能良好，无明显并发症；1例髂骨植骨的桡骨远端骨巨细胞瘤术后3个月移植骨完全愈合，至今随访75个月，肿瘤无复发。MSTS功能评分18~27分，平均22．6分。结论自体骨移植在上肢骨肿瘤切除后骨缺损的重建，尤其是儿童的骨缺损重建中，是一种较好的方法。     

Evaluation of a contralateral biopsy specimen in prostate cancer patients with unilateral suspicious lesions

INTRODUCTION: In patients with a clinically unilateral palpable and/or visible lesion confined to the prostate on digital rectal examination and transrectal ultrasonography, the findings of biopsy specimens of a clinically unsuspicious lobe do not reflect TNM staging results (2002 classification). In patients with such a unilateral lesion, we compared the biopsy results of a clinically unsuspicious lobe with the pathological assessment of the radical prostatectomy specimen and evaluated the importance of the results of biopsy specimens in an unsuspicious lobe. PATIENTS AND METHODS: Between April 2000 and August 2004, 97 prostatic cancer patients without neoadjuvant therapy underwent a radical retropubic prostatectomy. In the patients with a unilateral lesion on digital rectal examination and/or transrectal ultrasound, the preoperative prostate-specific antigen levels, the results of contralateral biopsy specimens, and contralateral cancer foci in radical prostatectomy specimens were examined. RESULTS: Of 39 patients with a unilateral palpable and/or visible lesion, 15 had contralateral positive biopsy findings, while 24 had contralateral negative biopsy findings. In a pathological analysis of prostatectomy specimens, a significantly higher rate of clinically significant cancer foci and a larger cancer volume in a clinically unsuspicious lobe have been observed in patients with contralateral positive biopsy findings than in patients with contralateral negative biopsy findings (p < 0.001). Moreover, contralateral cancer foci in patients with a contralateral positive biopsy specimen exhibited a more ominous state, such as seminal vesicle invasion, extraprostatic extension, and a positive surgical margin, than those in patients with a contralateral negative biopsy specimen (40.0 vs. 8.3%, p = 0.017). However, in a pathological analysis of both ipsilateral and contralateral cancer foci, the proportion of ominous pathological findings did not differ between the patients with a contralateral positive biopsy and those with a contralateral negative biopsy. CONCLUSIONS: In patients with clinically unilateral palpable and/or visible tumors confined to the prostate, the results of a bilateral biopsy need not be used to determine the clinical stage. However, in patients with positive biopsy results for an unsuspicious lobe, urologists should perform an extended surgical resection.     

A case of irreducible shoulder dislocation as an initial manifestation of osteosarcoma of the proximal humerus

We experienced a case of osteosarcoma of proximal humerus with an initial clinical manifestation of irreducible shoulder dislocation. An eleven-year-old girl was transferred due to shoulder dislocation after minor collision, which could not be reduced even under anesthesia. After failed closed reduction under anesthesia, open reduction through deltopectoral exposure was performed evacuating the organized fibrous tissue, even without the suspicion of primary bone malignancy. Histopathologic report was osteosarcoma. Retrospective review of the imaging studies revealed subperiosteal hemorrhage, organized hematoma underneath the deltoid musculature, and osseous parenchymal changes, which suggested bone tumor. Subsequently, lung metastases were detected, and the chemotherapy was accompanied by neutropenic fever and Stevens Johnson syndrome. She expired 1 month thereafter. High index of suspicion would have led to more early diagnosis and established more appropriate management plan. Osteosarcoma of proximal humerus should be included in the differential diagnosis of a shoulder dislocation that could not be readily reduced.     

Unreduced chronic dislocation of the humeral head with ipsilateral humeral shaft fracture: a case report

The purpose of reporting this case is to illustrate a treatment plan for a chronically anteriorly dislocated shoulder associated with an ipsilateral humerus fracture, a condition heretofore not addressed in the literature to our knowledge. An 18-year-old female, left hand dominant, injured her left upper extremity and liver in a motor vehicle accident. X-rays at time of injury revealed a diaphyseal facture of her left humerus. No x-rays of the shoulder were taken at time of injury. Treatment consisted of a plaster cast application and discharge at 1 week. The patient was seen again 4 weeks postinjury, at which time only humerus films were taken and the immobilization was continued. At 45 days postinjury, the patient complained of left shoulder pain, and shoulder x-rays at that time revealed an anterior subcoracoid dislocation of the left humeral head. At surgery 52 days postinjury, the humeral shaft fracture was found to be unstable and external fixation of both the fracture (2 pins above and below the fracture) and the reduced but still unstable humeral head was performed (a pin through the humeral head into the glenoid). The external fixator was removed at 3 weeks, and at a 3-year follow-up, the patient had acquired nearly full range of motion of her shoulder without pain and no significant limitations of her arm movements or activities. In conclusion, given a patient with a chronic anteriorly dislocated shoulder and a healing ipsilateral shaft fracture, an external fixation stabilization of both the fracture and the relocated repaired dislocation is a viable treatment option.     

Dedifferentiated chondrosarcoma arising in a solitary osteochondroma with leiomyosarcomatous component: a case report

BACKGROUND: A rare variant of dedifferentiated chondrosarcoma with leiomyosarcomatous component in a 63-year-old male is reported. CASE REPORT: He had a life-long osteochondroma adjacent to the left knee and recently developed swollen and pain. We initially suspected an intermediate grade chondrosarcoma arising in a solitary osteochondroma, but a biopsy specimen revealed a dedifferentiated component that histologically was classified as leiomyosarcoma. Multiple lung metastases were also present. The patient underwent amputation above knee and lung operation following adjuvant chemotherapy, but he died because of pulmonary metastasis 2 years after first admission. DISCUSSION: The differentiated component usually shows the histological features of osteosarcoma, malignant fibrous histiocytoma, and fibrosarcoma. Among the rare dedifferentiation with leiomyosarcomatous component, case arising in a solitary osteochondroma is not reported yet.     

The effect of biopsy on survival of patients with osteosarcoma.

A retrospective study of patients with osteosarcoma was undertaken to determine whether there was a relationship between biopsy and survival. Fifty-seven patients treated at the Karolinska Hospital, Stockholm, between 1938 and 1959 were included in this study, all of whom were less than thirty years old, had a metaphysial osteosarcoma in a long bone but had no pulmonary metastases at the time of diagnosis; all were treated by amputation. No clinical variants of osteosarcoma were included. Twenty-four of the fifty-seven patients had an amputation without a prior biopsy; the others had biopsies before amputation. These two groups were fairly closely matched in age, sex, site and size of tumour, and in the level of amputation; some patients in each group received radiation before operation. Evaluation of these two groups of patients revealed that the performance of a biopsy, with or without a delay of not more than thirty days between the biopsy and the definitive operation, had no adverse effect on survival.     

Long-term use of nerve block catheters in paediatric patients with cancer related pathologic fractures

We report three cases of children with osteosarcoma and pathologic fractures treated with long-term continuous nerve blocks for preoperative pain control. One patient with a left distal femoral diaphysis fracture had a femoral continuous nerve block catheter for 41 days without complications. Another with a fractured left proximal femoral shaft had three femoral continuous nerve block catheters for 33, 26 and 22 days respectively. The third patient, whose right proximal humerus was fractured, had a brachial plexus continuous nerve block catheter for 36 days without complication. In our experience, prolonged use of continuous nerve block is safe and effective in children with pathologic fractures for preoperative pain control.