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Eur J Orthop Surg Traumatol

DOI 10.1007/s00590-017-1997-6

ORIGINAL ARTICLE • ELBOW - OSTEOBLASTOMA

Osteoblastoma of the elbow: analysis of 13 patients and literature


review
Andrea Angelini1 • Andrès Felipe Varela-Osorio2 • Giulia Trovarelli1 •

Antonio Berizzi1 • Gabriele Zanotti3 • Pietro Ruggieri1

Received: 8 April 2017 / Accepted: 16 June 2017


 Springer-Verlag France SAS 2017

Abstract 87% (range 50–100%), which corresponds to excellent


Purpose To analyze the clinical and radiographic charac- results.
teristics, treatment, and outcome of patients with elbow Conclusions Intralesional surgery is successful in tumor
osteoblastoma. control in most patients with osteoblastoma of the elbow.
Patients and methods We present 13 patients (7 males and Thermal ablation may be successful for smaller lesions.
6 females; mean age, 28 years) diagnosed and treated for Most of the patients had a good-to-excellent functional
an elbow osteoblastoma from 1975 to 2012. Mean follow- outcome even if they had tumor-related elbow stiffness at
up was 29 months (range 12–60 months). Clinical pre- diagnosis.
sentation, imaging, surgical treatment, complications, Level of evidence Therapeutic study, Level IV-1.
range of elbow motion, and functional outcome were
evaluated. The MSTS, DASH, and OXFORD scores were Keywords Osteoblastoma  Elbow  Elbow joint stiffness 
used. Curettage  Local recurrence
Results Main symptom was pain (all patients) accompa-
nied by stiffness (8 patients) and swelling or tumefaction (7
patients), with a median duration of symptoms of Introduction
32 months (range 6–96 months). Distal humerus was
affected in 10 patients, proximal ulna in 2 patients, and Tumors around the elbow may prove to be a diagnostic and
proximal radius in one patient. All patients underwent therapeutic challenge for the oncologist orthopedic. Elbow
surgical therapy that consisted of curettage of the lesion (7 joint (including distal humerus, proximal radius, and
patients), curettage and bone allografting (3 patients), wide proximal ulna) is an uncommon localization for primary
resection (2 patients; total distal humerus and resection of benign bone tumors. At Rizzoli Institute, only 2% of the
the radial head), and radiofrequency thermal ablation (1 total benign bone tumors are localized in the elbow, most
patient). One patient experienced a recurrence after surgi- commonly GCT and osteoid osteoma [1]. Osteoblastoma is
cal treatment. The mean MSTS score after treatment was a rare benign bone tumor, initially described by Jaffe in
1932 [2–4] that accounts for approximately 1% of all bone
neoplasms and 3% of all benign primary bone tumors. It
may occur in any area of the skeleton with a predilection
& Pietro Ruggieri for the spine and sacrum ([40% of the cases) [4, 5]. In the
pietro.ruggieri@unipd.it
long bones, osteoblastoma is generally observed in the
1
Department of Orthopedics and Orthopedic Oncology, meta-diaphysis, and more rarely in adult age it may extend
University of Padova, Via Giustiniani 3, 35128 Padua, Italy to the epiphysis. Cases of subperiosteal osteoblastoma have
2
Istituto Ortopedico Rizzoli, University of Bologna, Bologna, been reported [4]. In the current literature, there are few
Italy studies about osteoblastoma of the elbow, usually case
3
Department of Orthopedics, Civilian Hospital of Lugo, Lugo, reports that describe little information on the radiological
Ravenna, Italy and clinical aspects of this entity [3, 6–12]. Osteoblastoma

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Eur J Orthop Surg Traumatol

can be an aggressive benign bone tumor, but tragic con- collected consisted of hospital medical records, operative
sequences have been reported when mistaken for notes, and pathologic reports (Table 2). All patients or their
osteosarcoma [6, 13–15]. relatives gave written informed consent at the time of
We performed a search of the literature to identify admission for their data to be included in this study.
patients who had been treated for osteoblastoma of the Patients were studied for clinical and radiologic
elbow. English language and non-English language litera- findings; imaging studies included radiographs (all
ture were searched in PubMed. The search was carried out patients), bone scans (7 patients), computed tomography
using literature of the past 40 years (from 1975 to 2015) (CT) scans (9 patients), and/or magnetic resonance
[3, 6–12] (Table 1). imaging (MRI, 7 patients). All tumors were located in
This study aims to report the clinical and imaging the metaphysis of the distal humerus and proximal ulna
findings, incidence of local recurrence related with the type and radius. No tumor was located in the epiphysis of
of surgery, and clinical and functional outcome of a group these bones. The tumor involved the right elbow in 10
of patients diagnosed and treated for an osteoblastoma of cases (77%) and the left elbow in 3 cases (23%). Distal
the elbow. humerus was affected in 10 patients, proximal ulna in 2
patients, and proximal radius (Fig. 1) in one patient
(Table 2). Tumor volume was measured on coronal,
Patients and methods transverse, and sagittal MRI or CT scans of the lesion.
Four patients had previous elbow surgery, 3 patients had
We retrospectively studied all patients with histologically intralesional surgery (curettage in 2 patients, and curet-
verified osteoblastoma of the elbow, including distal tage and synovectomy in one patient), and 1 patient had
humerus, proximal radius, and proximal ulna, treated from previous elbow arthroscopy and ulnar nerve exploration
November 1975 to December 2015. There were 13 patients unsuspecting tumor pathology. These patients with pre-
with osteoblastoma of the elbow (7 males and 6 females), vious intralesional surgery experienced a local recur-
with a mean age of 28 years (range 7–62 years). The data rence and were referred to our institution for further

Table 1 Summary of the published studies reporting on osteoblastoma of the elbow


Study N. Age, Site Symptoms Stiffness Treatment LR Treatment of Function
pts gender relapse

McLoad et al. [8] 1 14,M D-H Pain - Curettage - - Good


Gil-Albarova et al. 1 21,M D-H Pain, ? Curettage - - Good
[17] stiffness
Lucas et al. [20] 2 n/a D-H n/a n/a n/a n/a n/a n/a
1 n/a P-R n/a n/a n/a n/a n/a n/a
2 n/a P-U n/a n/a n/a n/a n/a n/a
Kawaguchi et al. 1 12,M D-H Pain, ? Wide res. - - Normal
[18] swelling
Van Giffen et al. 1 21,F P-U Wrist pain - Curettage - - Normal
[22]
Louaste et al. [19] 1 40,M P-R Pain, ? Curettage - - Normal
swelling
Berry et al. [2] 2 n/a D-H n/a n/a n/a - n/a n/a
2 n/a P-R n/a n/a n/a 1 n/a n/a
2 n/a P-U n/a n/a n/a 2 n/a n/a
Spence et al. [21] 1 22,M D-H Pain ? Curettage 1 Thermoablation Good
Current study 13 Mean 28 D-H Pain (13) ? (8) Curettage (7) 1 Curettage Mean MSTS
(9) 87%
M7, F6 P-R Stiffness (8) Curettage ? graft
(2) (3)
P-U Swelling (7) Wide resection (2)
(2)
RFA (1)
Systematic review of the literature
Pts patients, LR local recurrence, D-H distal humerus, P-U proximal ulna, P-R proximal radius, RFA radiofrequency ablation, n/a not available

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Table 2 Demographic and clinical features of the 13 patients with elbow osteoblastoma included in this series
Patient no./ Duration of Stiffness Enneking Biopsy Site Tumor Previous Treatment Adjuvants LR MSTS
age/gender symptoms (mos) grade type volume treatment (%)
(cm3)

1/7/M 6 ? 3 Incis D-H 1.5 - Curettage - - 83


2/22/M 12 - 2 Incis D-H 6.6 - Curettage - - 79
3/30/M 24 - 2 Incis D-H 3.0 ? Curettage, bone graft and Phenol - 96
synovectomy
4/33/F 72 ? 2 Frozen P-U 1.9 - Curettage, bone graft - - 75
5/37/M 36 - 2 Trocar D-H 2.0 - RFA - - 95
6/9/F 18 ? 3 Frozen D-H 1.4 - Curettage/Graft Phenol - 100
7/30/F 12 - 3 Frozen D-H 8.5 - Curettage and synovectomy - - 85
8/62/F 12 ? 3 Incis D-H 4.8 - D-H res ? TEA - - 70
9/57/M 96 ? 2 Excis D-H 0.8 - Curettage Phenol - 100
10/9/F 12 - 2 Frozen P-R 2.5 - Curettage - 100
11/14/F 17 ? 2 Excis D-H 8.0 ? Curettage and synovectomy - ?(at 5 mos, repeat 90
curettage)
12/22/M 65 ? 2 Excis P-U 3.0 ? Curettage - - 86
13/40/M 16 ? 3 Excis D-H 7.5 - Curettage and radial - - 73
capitulum resection
Incis incisional, Excis excisional, D-H distal humerus, P-U proximal ulna, P-R proximal radius, RFA radiofrequency ablation, TEA total elbow arthroplasty, LR local recurrence

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excisional biopsy in 4 patients, each), or trocar biopsy (1


patient). Three patients had chronic synovitis and 2 of them
experienced elbow stiffness. One patient had histologic
suspicion of malignancy with epithelioid proliferation. The
final diagnosis by immunohistochemistry was osteoblas-
toma (patient no. 8). One patient was diagnosed as
osteoblastoma of the bone surface. Three patients were
initially diagnosed as osteoid osteoma (Table 2, patient no.
2, 4, and 5); re-review of the histological slides in these
patients confirmed the diagnosis of osteoblastoma (Fig. 2).
According to Enneking’s system for the Staging of Benign
Bone Tumors [16] and the clinical and radiographic fea-
tures, eight patients were stage 2 and five patients were
stage 3.
Routine follow-up examination was performed every
6 months for the first 2 years and then annually. Progres-
sion-free survival (PFS) was defined as the time elapsed
between date of treatment and tumor progression/local
recurrence or date of last follow-up for the patients who did
not have any such events. The time to local recurrence was
evaluated using the univariate Kaplan–Meier analysis [17].
Function was evaluated with the Musculoskeletal Tumor
Society (MSTS) score for the upper limb [18, 19], the
shortened disabilities of the arm, shoulder and hand ques-
tionnaire (QuickDASH) [20, 21], and the Elbow Oxford
score [22].

Results

Most common symptom at presentation was pain (all


patients) accompanied by elbow stiffness (8 patients) and
swelling or tumefaction (7 patients). Seven patients had
limited flexion–extension of their involved elbow joint, two
patients had limited pronation–supination, and 4 patients
had a full range of motion [23]. Of the eight patients with
elbow stiffness, five experienced swelling or palpable
tumefaction. Three patients had neurological symptoms,
specifically hand paresthesia at the distribution of the ulnar
nerve. The mean time from onset of symptoms to diagnosis
and treatment was 30 months (range 6–96 months).
Two patients had no evidence of tumor features at X-ray
(patients 7 and 9) (Fig. 3). Imaging findings included an
osteolytic round area with a central area of calcification
Fig. 1 a Antero-posterior and b lateral radiograph of the right elbow associated to the area of surrounding bone sclerosis (7
of a 9-year-old boy with nocturnal pain of several months’ duration
showed an osteoblastoma of the proximal radius. c Axial CT scan patients), marked osteolysis and/or bone destruction with-
shows a prevalent osteolytic lesion with internal matrix mineraliza- out a significant periosteal reaction (4 patients), periosteal
tion, cortical thinning in absence of cortical breakthrough reaction mimicking a Codmańs triangle (one patient), and a
nidus with surrounding sclerosis mimicking an osteoid
treatment. Two patients were administered previous osteoma (one patient).
intra-articular injections for epicondylitis. All patients underwent surgical treatment that consisted
Histologic diagnosis was obtained by open biopsy in 12 of curettage (7 patients), curettage and bone allografting (3
patients (incisional biopsy, frozen section biopsy and patients), wide resection (2 patients; a distal humerus and a

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Fig. 2 a Lateral radiograph of the right elbow of a 33-year-old shows a well-defined ostelytic lesion. c Axial CT scan showed the
female with a painful osteolytic lesion of the distal humerus exact extension of the lesion diagnosed as osteoblastoma at frozen
corresponding to the olecranon fossa. b Antero-posterior radiograph biopsy before curettage

humeral head resection), and radiofrequency thermal (Table 2; Patient no. 12). This patient was initially treated
ablation (1 patient). Phenol was used as local adjuvant to at another hospital and was referred to our institution for
curettage in 3 patients. Distal humeral resection and total further treatment due to inadequate tumor excision and
elbow arthroplasty were performed due to a clinical and local recurrences. He underwent repeat curettage at our
histological suspicion of malignancy, which, however, institution and presented local recurrence 10 months later
after histological and immunohistochemical examination that treated with another curettage without any evidence of
of the complete tumor specimen, proved to be an local recurrence at the last follow-up, 4 years after initial
osteoblastoma (Fig. 4). The patient with osteoblastoma of diagnosis and treatment.
the bone surface underwent radiofrequency thermal abla- Seven patients (Table 2; patient no. 1, 4, 6, 8, 9, 11, and
tion. Postoperative management included analgesics 13) with elbow stiffness at diagnosis had a tumor at the
administration and cast immobilization or progressive distal humerus. The mean MSTS score after treatment was
mobilization with a functional brace according to type of 87% (range 50–100%), which corresponds to excellent
surgery. results. The patient with distal humerus resection and total
At a mean follow-up of 29 months (range elbow arthroplasty had the worst results (MSTS score 70%)
12–60 months), overall local recurrence rate was 8% (case with limitation of elbow motion and lifting ability impaired
1/13). Twelve patients remained continuously local recur- that, however, did not interfere with daily activities. The
rence-free after treatment, and 1 patient experienced local mean Oxford Elbow Score improved from 28 points (range
recurrence after surgical treatment in our institution 23–35 points) to 42 points (range 36–50 points), whereas

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Fig. 3 a Lateral radiograph of the elbow in a 57 year-old male rugby suppression of the elbow revealed a nonspecific osteolytic lesion in
player with a persistent pain despite arthroscopic debridement and the medial condyle accompanied by severe articular inflammatory
ulnar nerve neurolysis shows marked degenerative features and volar changes. d, e, f CT scan confirmed an ostelytic lesion between the
calcification. b Coronal and c axial T2-weighted MRI with fat medial condyle and olecranon fossa with cortical breakthrough

Fig. 4 a Antero-posterior and b lateral radiograph of the elbow of a 62-year-old female with a large osteoblastoma of the distal humerus.
c Antero-posterior and d lateral radiograph show the result after distal humerus resection and reconstruction with total elbow arthroplasty

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the QuickDash score showed good-to-excellent results choice because it will detect the lesion and reveal marginal
(mean, 13.5 points; range 0–52 points). sclerosis, expansion, and internal matrix mineralization
[11]. On MRI, findings are relatively nonspecific with a
fairly well-defined expansive lesion [5, 11]. There is often
Discussion surrounding bone marrow edema and reactive synovitis in
intra-articular tumors [5, 11]. Main problems with MRI are
Elbow is an uncommon location for osteoblastoma that the edema is a nonspecific inflammatory response to
[2, 4, 12]. The present series aimed to address the clini- tumor and visualization of the margins between the osseous
copathological findings and outcome of a group of patients and soft tissue component of the tumor is less defined
with this entity and discuss the relationship with elbow resulting in inaccurate diagnosis of an aggressive or
joint stiffness. The main limitations of this study are the malignant tumor [5].
retrospective type of the analysis and the distribution of the Aggressive osteoblastoma can mimic osteosarcoma
treated patients over a 40-year period. However, consid- [4, 6, 15, 24]. There are several reports about aggressive
ering the relatively rare tumor location, the findings of our osteoblastoma in the mandible, proximal humerus, mas-
study would be useful for the related literature. toid process of temporal bone, and hand with marked
In a previous study [4], the mean age at presentation of bone destruction and periosteal reaction simulating the
patients with osteoblastoma of the elbow was 20.4 years, behavior of a malignant tumor [6, 25–27]. McLeod and
with a wide range of 6 months–75 years. The typical age at Dahlin [6] reported 3 cases of osteoblastoma with an
presentation is between 10 and 20 years of age with initial and erroneous diagnosis of osteogenic sarcoma due
approximately 90% being diagnosed before the age of to their size, shape, and histologic features with bizarre
30 years; males are slightly more commonly affected nuclei. Although the exact histopathologic differential
[2–5, 8, 10, 11]. The most common presenting symptoms diagnosis between osteosarcoma and osteoblastoma may
are pain and stiffness [2–4], while it is not uncommon the be difficult in some cases, current molecular genetic
presence of swelling and soft tissue mass [4, 6–8, 11]. In all examination may help solve this problem [4–6, 10]. In the
reported cases with articular involvement [3, 8–10], present study, three patients were initially diagnosed as
mobility is impaired, especially elbow extension. The bone sarcoma due to the clinical and radiographic fea-
present study confirmed these reports regarding age and tures; one of these patients was treated as a bone sarcoma;
gender, and clinical symptoms at presentation of the however, histological and immunohistochemical analysis
patients. Radiographs should be the initial imaging inves- of the final tumor specimen confirmed the diagnosis of
tigation at the staging process of a tumor patient; however, osteoblastoma.
the diagnosis of small osteoblastoma may be impaired on Treatment of osteoblastoma depends on the stage and
radiographs [11], as in 8 patients in the present study. site of the tumor [4, 5]. In stage 1 (latent) or stage 2 (ac-
Moreover, radiographic features may not be distinctive, tive), curettage is indicated, with or without local adjuvants
creating problems in the differential diagnosis from such as phenol. In stage 3 (aggressive) a marginal or wide
osteosarcoma, osteoid osteoma, aneurysmal bone cyst, resection is indicated depending on extension of bone
chondroblastoma, and chondrosarcoma. The dominant involvement. Wide resection, when indicated, should
finding is a single and round area of osteolysis, character- minimize the chance of local recurrence but at the cost of
ized by boundaries that are not always well defined, increase risk of surgical morbidity and articular function
sometimes marked by moderate reactive osteoesclerosis impairment [5, 28]. More recently, imaging-guided
(usually not as intense and extensive as in osteoid radiofrequency thermal ablation is being increasingly used
osteoma). Osteoblastoma may reach a considerable size, up for small osteoblastomas, less than 2 cm in maximum
10 cm in maximum diameter, and whether it is central or diameter [2–4, 7, 17, 28–31]. Rehnitz et al. [30] reported a
eccentric, it may attenuate the cortex and expand the bone, series of 77 patients with osteoblastoma and osteoid
at times with aspects that are similar to those of aneurysmal osteoma concluding that even large tumors and in those
bone cyst. Approximately, one-fourth of these tumors have tumors located in critical areas can safely be treated with
a radiographic appearance consistent with malignancy, radiofrequency thermal ablation, improving pain and
even if there is hardly any or only a fair amount of peri- quality of life and avoiding surgical complications.
osteal reaction [4, 6]. In the present study, 4 patients Osteoblastoma tend to be locally aggressive with
showed marked osteolysis or bone destruction on radio- recurrence rates ranging from 21 to 24% [3, 4, 6, 10]. The
graphs, but only one had intense periosteal reaction mim- local recurrence rates for osteoblastoma of the elbow after
icking a Codmańs triangle. Although bone scintigraphy curettage are not clear; some studies report a higher like-
will typically show marked tracer uptake, images are lihood of recurrence and a correlation between the inci-
nonspecific. CT scan is probably the imaging method of dence of recurrence and a previous inadequate treatment in

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non-referral centers [5, 32]. As in one patient in this series, Conflict of interest None of the authors have any financial and
Spense et al. [11] reported a case of distal humerus personal relationships with other people or organizations that could
inappropriately influence (bias) their work. Each author certifies that
osteoblastoma treated with curettage and then after recur- the manuscript has been read and approved and that the manuscript
rence with thermal ablation. Overall, in the present series, 4 represents honest work.
patients experienced a local recurrence; one patient after
initial treatment at our institution and 3 patients that were
referred to us for local recurrences. References
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