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Electronic Physician (ISSN: 2008-5842) http://www.ephysician.ir
September 2017, Volume: 9, Issue: 9, Pages: 5310-5317, DOI: http://dx.doi.org/10.19082/5310
Corresponding author:
Assistant Professor Dr. Afshin Taheriazam, Tehran Medical Sciences Branch, Islamic Azad University, Khaghani
St, Shariati Ave, Tehran, Iran. Tel: +989121113486, Fa x: +982188033020, Email: afshintaheriazam@yahoo.com
Received: September 21, 2016, Accepted: May 12, 2017, Published: September 2017
iThenticate screening: January 03, 2017, English editing: August 06, 2017, Quality control: September 12, 2017
© 2017 The Authors. This is an open access article under the terms of the Creative Commons Attribution -NonCommercial-
NoDerivs License, which permits use and distribution in any medium, provided the original work is properly cited, the use is
non-commercial and no modifications or adaptations are made.
Page 5310
One-stage surgical excision of a huge bilateral multiple osteochondroma of the hip: a case report
Afshin Taheriazam1, Amin Saeidinia2
1M.D., Assistant Professor, Department of Orthopedics Surgery, Tehran Medical Sciences Branch, Islamic Azad
University, Tehran, Iran
2M.D., Assistant Researcher, Medical Faculty, Mashhad University of Medical Sciences, Mashhad, Iran
Type of article: Case report
Abstract
Osteochondroma or hereditary multiple exostoses is the most common benign bone tumor and is usually found in
young patients. Osteochondromata of the proximal femur or hip have been reported in 30% to 90% o f patients
with hereditary multiple exostoses. This article presents a 25 -year-old-male referred to our orthopedic clinic with
a complaint of pain in both groins for the last four years which has deteriorated in the past 6 months by limitation
of range of motion. Radiographic findings were consistent with huge hip osteochondroma and a histopathological
report confirmed the diagnosis. This was a case of a huge bilateral osteochondroma of the hip originating from
the posterior of the hips to the neck of the f emur and hip joints and part of proximal of femur, that underwent
one-stage surgical excision of the tumor with posterior approach and tumor resection. As we know, this case of
bilateral huge hip osteochondromas has rarely been presented previously by some one who has undergone one-
stage surgery treatment of a tumor this size. Bilateral hip osteochondroma is a rare condition and we showed that
one-stage excision can be performed successfully.
Keywords: Osteochondroma, One-stage bilateral excision, Hereditary multiple exostosis
1. Introduction
Hereditary multiple exostosis (HME), is a type of rare benign bone tumor and is an inherited autosomal dominant
disorder characterized by the presence of multiple exostoses or osteochondromas (1). Osteochondroma is a cartilage
capped bony projection arising on the external surface of the bone, containing a marrow cavity that is continuous
with that of the underlying bone (2). It is generally developed during the first decade of life and stopped growing
when the individual gained skeletal maturity (3). Although frequency and presence of osteochondroma can be
difficult to characterize because of its asymptomatic nature, it can be found as a palpable mass which may cause
pain due to fracture through a pedunculated stalk, o verlying bursal inflammation or impingement on an overlying
structure (4). The radiological pathognomonic characteristic of this tumor is the cortical and marrow continuity of
the lesions with the adjacent bone (5). The lesions may be solitary or multiple, the latter forming part of the
hereditary multiple exostoses syndrome (6). These lesions may also appear with complications such as bone
deformities, fractures (5, 7), neurological or vascular compromise (5, 8, 9), bursa formation and more rarely,
malignant transformation (10, 11). It is possible to diagnose multiple osteochondroma (MO) when at least two
osteochondromas of the juxta-epiphyseal region of long bones are observed through radiology (6). In the majority of
patients diagnosed with MO, a positive family history mutation in one of the EXT genes can be detected (6). With
an estimated prevalence of 1 per 50,000 individuals within the general population (12), it appears to be higher in
males than in females (male/female ratio 1.5:1) (13). In this case report we presented a bilateral hip multiple
osteochondroma with huge size in a 25-year-old male patient without family history of osteochondroma or HME,
who required surgical treatment.
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2. Case Presentation
A 25 year-old male with complaints of severe swelling and pain in both hips for the past 12 months was referred to
Erfan Hospital, Tehran, Iran in May 2015. He noted that the swelling of both hips had insidious onset and gradually
increased in size over a period of 4 years. Patient also mentioned t hat his aching was continuous pain, aggravated by
activities and relieved by rest and analgesics. His pain was radicular to the lower extremities by activity. His daily
activities were significantly restricted due to the mass effect of the tumor which rest ricted movement of the hip joint.
He had problems in walking and other motions related to the hip joints activities. In the recent 6 months before
admission, his chief complaint was in sitting. He suggested no manifestation of discharge or redness. There was no
history of trauma or fever. Skeletal survey showed other evidence of similar lesions in the patient; however, they
were smaller and did not cause problems for him. They were in the proximal of tibia, the distal of femur, the
proximal of humorous and in the scapula. He had no history of malignancy or known skeletal disorders in his
family. He was a smoker but he did not use opium or alcohol. The patient had no risk factor of tendency to the
malignancy. He had no history of previous similar mass in the past. He had no history of activity limitation in
childhood or adolescence. He did not use drugs and had no history of sensitivity to any substance or drug. On
examination, the patient had no evidence of any systemic illness. He had a limitation in range o f motion with 30°
flexion and 10° abduction in the hip joints. Local examination revealed two diffused, ill -defined, hard, globular
swellings of about 30×25 cm in the left hip and 40×30 cm in the right hip. They were found and aroused posterior of
the hips to neck of femur and hip joints and part of proximal of femur. Surface was irregular, hard in consistency
with ill-defined borders. Skin over the swelling was normal. It was not warm. There was no localized
lymphadenopathy and distal neurovascular deficit. There was no evidence of any laboratory abnormalities. Plain
radiographs (Figure 1), CT scan (Figure 2), three-dimensional CT scan (Figure 3) and MRI (Figure 4) were done,
and reports suggested it to be a huge multiple osteochondroma or hereditary multip le exostoses. As it is seen on 3D
CT and MRI, it had a sessile structure and extended outward broadly in the region of neck of femur, hip joint and
lesser and greater trochanter in both sides. So we decided to perform a one-stage complete excision of both hips, and
a frozen section evaluation of the tumor was made. The patient was located in supine position. We performed two
incisions simultaneously in both posterior of hips. A posterior trans-gluteal approach with splitting of gluteus
maximus was used and the sciatic nerve was explored in both sides (Figure 5). After complete dissection of the
tumor, it was removed en masse in both sides. There was no evidence of local invasion in either side. We performed
frozen section during the operation and it confirme d that there was no evidence of malignancy or tumoral cells. The
excised tumor was sent to pathology laboratory and i t was diagnosed osteochondroma. Then we closed and sutured
the tumor sites (Figure 6). General anesthesia was used for this procedure and t he operation lasted approximately
1:40 minutes. The patient maintained partial weight bearing for a period of four weeks. After 6 weeks, the patient
was able to return to his daily routine activities. The patient continued to receive follow -up treatment once every two
months for six months and then once every six months up to the present. At the last follow -up, the patient was
completely asymptomatic and the radiographs and MRI showed no evidence of local recurrence. He did not have
any dislocation, dysplasia or other signs in radiological follow-ups.
Figure1. Plain radiograph of pelvis showed bilateral cauliflower like mass attached to femoral neck and expands to
proximal of femur and posterolateral direction; a. AP view. b. Lateral view
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Figure 2. Multi Slice Computed Tomography of the hip demonstrates mass originating from posterior of the hips to
neck of femur and hip joints and part of proximal of femur without any soft tissue extension and malignant
degeneration.
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Figure 3. CT three-dimensional reconstruction of the pelvis showing multiple osteochondromata in both hips.
Figure 4. MRI of the hip with both sides mass originating from posterior of the hips without any soft tissue
extension and malignant degeneration but displacement of the adjac ent muscle and sciatic nerve.
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Figure 5. Showing surgical resection performed through posterolateral approach
Figure 6. Showing excision of tumor (a) and closure of the incision (b) and macroscopic view of the part of tumor
(c)
3. Discussion
The presented case has illustrated successful excision of hip osteochondromas and good outcomes in follow -up
results with one-stage surgical treatment with posterior trans-gluteal approach. As we know, this case of bilateral
huge hip osteochondromas has rarely been presented previously by someone who has undergone one -stage surgery
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treatment of a tumor this size.. Osteochondromas or exostoses, are the most common benign tumors of the bones,
with an estimated rate of 36% in all benign bone tumors and 8.5% in all bo ne tumors (14). In a descriptive study in
Iran, it was demonstrated that osteochondroma is the most common pathologic type of bone tumor by 35 percent
(15). Osteochondromas can be solitary and multiple, as a sporadic event or secondary to traumatic insult. Multiple
osteochondromas may be sporadic or familial such as HME, which is a relatively rare disease (12). The metaphyses
of long bones of the extremities, knee, scapulae, vertebrae, ilium and ribs can be involved in this disorder (16). The
majority of Osteochondroma masses discovered in younger patients are painless and present as slow growing mass.
However, due to complications such as bone deformity, fractures, mechanical joint pain, vascular and neurological
compromise and malignant transformation, significant symptoms may occur depending on the location of
osteochondroma (17, 18). Osteochondromata of the proximal femur have been reported to occur in 30% to 90% of
patients with hereditary multiple exostoses (19). Osteochondromas developing around knee and hip are usually
asymptomatic but can occasionally impinge surrounding vessels and nerves, and can cause various clinical
manifestations (20). Some of these clinical manifestations are femoral ante -version, impairment of hip flexion,
acetabular dysplasia resulting in sublaxation of the hip, valgus deformity of the knee, and angular limb deformities
(21). As in our case, the patient demonstrated symptoms including difficulty in sitting and walking. We showed that
the osteochondroma of the hip originated from proximal of both femurs and sessile were a cauliflower -like mass and
were huge in size. Makhdoon et al. reported three cases with osteochondroma of the hip (21). One of them was a 15 -
year-old male presented with left hip pain and prolonged walking and sporting activities. He underwent multiple
previous surgeries (left tibia, right distal femur, and upper extremities) for excision of osteochondromas. He had a
Trendelenburg gait, restricted range of motion in hip abduction and flexion on examination, and l eft hip dysplasia
and left femur neck osteochondroma causing left hip subluxation in radiography. He underwent left hip Bernese
periacetabular osteotomy and femoral neck osteoplasty with partial excision of the osteochondroma through a
modified Smith-Peterson approach (21). Another case was a four-year-old female with HME in the hip. She had no
complaints and her examination was unremarkable apart from palpable osteochondromas in the upper extremities
and distal femora. She was diagnosed by radiographs which revealed a left femoral neck osteochondroma with
bilateral coxa valga, and a left dysplastic hip, and left hip subluxation. She underwent left femoral varus derotational
osteotomy with partial excision of the osteochondroma through the lateral approach with application of a pediatric
dynamic hip screw. She had recurrence of the left hip osteochondroma with persistent left acetabular dysplasia after
4 years, and again operated by a proximal femoral varus osteotomy and extensive excision of the left femora l neck
osteochondroma through the lateral approach and a modified Dega osteotomy through a Smith -Petersen approach
(21). The last case that they reported was a thirteen -year-old female known for HME who had undergone multiple
previous surgeries in the lower extremities for excision of osteochondromas. She had limited flexion, abduction, and
internal/external rotation and the radiographs showed bilateral acetabular dysplasia with an increased left femoral
neck width secondary to osteochondromas. She underwent a left acetabular shelf procedure and femoral neck
osteoplasty through the anterior approach, but the acetabular osteochondroma was not excised (21). Muzaffar et al.
presented a young female with a history of hip pain for 3 years, with restriction of mov ements around the hip joint
and radicular pain which turned out to be a solitary osteochondroma of the femoral neck that was relieved by
surgical excision (22). Several surgical techniques have been reported in the previous studies and our study, which
contributed the main concerns for surgical resection of femoral neck and peritrochanteric osteochondromas to
exposure and femoral head vascularity. In our report, the senior author used the posterolateral approach. Rajaram
Manoharan et al. (4) reported a 24 year old male patient presented with a large swelling around the right hip.
Surgical excision with medial approach and lesser trochanter osteotomy was performed and at the latest follow up,
the patient was pain free and there was no evidence of any recurren ce. Ofiram and Porat have (23) reported a female
patient (16 years old) who had an osteochondroma at the femoral neck (circumferential) and floor of acetabulum in
association with acetabular dysplasia. They excised the lesion through the anterior approach with intraoperative hip
subluxation. No pelvic procedure was performed, and the patient remains asymptomatic at 3 years of follow -up.
Felix et al. (19) resected bilateral femoral neck osteochondromas in a 12-year-old female patient who also had
acetabular dysplasia. Bilateral staged resections, steel osteotomies, and proximal femoral varus osteotomies were
performed through the posterior approach. At 3 years of follow-up, no complications were reported. Shinozaki et al.
(24) resected a femoral neck osteocho ndroma in a 30-year-old male patient who had a dysplastic hip. The authors
resected the lesion through the anterior approach and posterior approach. A rotational osteotomy was also
performed. At 6 weeks of follow-up, recurrence of hip subluxation was obser ved and the greater trochanter was
transferred distally. Siebenrock et al. (25) have described the lateral approach to the hip with surgical hip dislocation
to allow access and adequate exposure of the femoral neck while preserving the vascular supply. The y presented
four adult patients with successful resection of femoral neck osteochondromas located in posterior, inferior, and
anterior regions of the femoral neck.
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4. Conclusions
It is concluded that the use of different approaches in hip osteochondroma s hould be determined by the status of the
patient, case by case, and that there is no specific approach for this condition. As we explained here, in the posterior
approach used in this case, short term follow-up had a good functional outcome. As it is mentioned, there were
several case reports of osteochondroma of hip joint, however they reported solitary osteochondroma of the hip and
bilateral huge hip involvement is not yet reported and also in regards to surgical technique, none have been used in
the one-stage surgical excision of tumor.
Acknowledgments:
We thank Erfan Hospital colleagues for their cooperation in management of the patient.
Conflict of Interest:
There is no conflict of interest to be declared.
Authors' contributions:
Both authors contributed to this project and article equally. Both authors read and approved the final manuscript.
References:
1) Albrecht S, Crutchfield JS, SeGall GK. On spinal osteochondromas. J Neurosurg. 1992; 77(2): 247 -52. doi:
10.3171/jns.1992.77.2.0247. PMID: 1625013.
2) de Andrea CE, Kroon HM, Wolterbeek R, Romeo S, Rosenberg AE, DeYoung BR, et al. Interobserver
Reliability in the Histopathological Diagnosis of Cartilaginous Tumors in Patients with Multiple
Osteochondromas. Mod Pathol. 2012; 25(9): 1275-83. doi: 10.1038/modpathol.2012.78. PMID: 22555180.
3) Yildirim C, Rodop O, Kuskucu M, Sahin O, Gamsizkan M. Giant solitary osteochondroma arising from the
fifth metatarsal bone: a case report. J Foot Ankle Surg. 2010; 49(3): 298.e9 -15. doi:
10.1053/j.jfas.2010.02.024. PMID: 20605564.
4) Rajaram Manoharan S, Gopinathan N, Vishwanathan V, Goni V, Tripathy S. Surgical Excision Of A Large
Sessile Osteochondroma Of The Lesser Trochanter Of Femur: A Case Report. Webmed Central. 2010;
1(11): WMC001237. doi: 10.9754/journal.wmc.2010.001 237.
5) Murphey MD, Choi JJ, Kransdorf MJ, Flemming DJ, Gannon FH. Imaging of osteochondroma: variants
and complications with radiologic-pathologic correlation. Radiographics. 2000; 20(5): 1407 -34. doi:
10.1148/radiographics.20.5.g00se171407. PMID: 10992031.
6) Bovee J. Multiple osteochondromas. Orphanet J Rare Dis. 2008; 3(1): 3. doi: 10.1186/1750 -1172-3-3.
PMID: 18271966.
7) Malhotra K, Nunn T, Chandramohan M, Shanker J. Metatarsal stress fractures secondary to soft -tissue
osteochondroma in the foot: case report and literature review. Foot Ankle Surg. 2011; 17(4): e51 -4. doi:
10.1016/j.fas.2011.05.003. PMID: 22017915.
8) Yu K, Meehan JP, Fritz A, Jamali AA. Osteochondroma of the femoral neck: a rare cause of sciatic nerve
compression. Orthopedics. 2010; 33(8). doi: 10.3928/01477447-20100625-26. PMID: 20704099.
9) Gruber-Szydlo K, Poreba R, Belowska-Bien K, Derkacz A, Badowski R, Andrzejak R, et al. Popliteal
artery thrombosis secondary to a tibial osteochondroma. Vasa. 2011; 40(3): 251 -5. doi: 10.1024/0301-
1526/a000101. PMID: 21638255.
10) Mahmoodi SM, Bahirwani RK, Abdull-Gaffar BA, Habib IF. Intrabursal vein abrasion and thrombosis. An
unusual complication of femoral osteochondroma. Saudi Med J. 2009; 30(12): 1604 -6. PMID: 19936429.
11) Martin C, Munk PL, O'Connell JX, Lee MJ, Masri B, Wambeek N. Malignant degeneration of an
osteochondroma with unusual intra-bursal invasion. Skeletal radiol. 1999; 28(9): 540 -3. doi:
10.1007/s002560050560. PMID: 10525800.
12) Schmale GA, Conrad EU, 3rd, Raskind WH. The natural history of hereditary m ultiple exostoses. J Bone
Joint Surg Am. 1994; 76(7): 986 -92. doi: 10.2106/00004623-199407000-00005. PMID: 8027127.
13) Legeai-Mallet L, Munnich A, Maroteaux P, Le Merrer M. Incomplete penetrance and expressivity skewing
in hereditary multiple exostoses. Clin genet. 1997; 52(1): 12-6. doi: 10.1111/j.1399-0004.1997.tb02508.x.
PMID: 9272707.
14) Kitsoulis P, Galani V, Stefanaki K, Paraskevas G, Karatzias G, Agnantis NJ, et al. Osteochondromas:
review of the clinical, radiological and pathological features. In Vivo. 2 008; 22(5): 633-46. PMID:
18853760.
Electronic physician
Page 5317
15) Solooki S, Vosoughi AR, Masoomi V. Epidemiology of musculoskeletal tumors in Shiraz, south of Iran.
Indian J Med Paediatr Oncol. 2011; 32(4): 187-91. doi: 10.4103/0971-5851.95138. PMID: 22563150,
PMCID: PMC3343243.
16) Liu W, Kong D, Tang J, Yu F. Giant costal osteochondroma in a man with multiple exostoses. Ann Thorac
Surg. 2013; 96(2): 675-7. doi: 10.1016/j.athoracsur.2012.12.007. PMID: 23910108.
17) Florez B, Monckeberg J, Castillo G, Beguiristain J. Solitary osteochondroma lo ng-term follow-up. J Pediatr
Orthop B. 2008; 17(2): 91-4. PMID: 18510166.
18) Paik NJ, Han TR, Lim SJ. Multiple peripheral nerve compressions related to malignantly transformed
hereditary multiple exostoses. Muscle Nerve. 2000; 23(8): 1290 -4. doi: 10.1002/1097-
4598(200008)23:8<1290::AID-MUS23>3.0.CO;2-M. PMID: 10918272.
19) Felix NA, Mazur JM, Loveless EA. Acetabular dysplasia associated with hereditary multiple exostoses. J
Bone Joint Surg Br. 2000; 82(4): 555-7. PMID: 10855881.
20) Tanigawa N, Kariya S, Kojima H, Komemushi A, Fujii H, Sawada S. Lower limb ischaemia caused by
fractured osteochondroma of the femur. Br J Radiol. 2007; 80(952): e78-80. doi: 10.1259/bjr/44678280.
21) Makhdom AM, Jiang F, Hamdy RC, Benaroch TE, Lavigne M, Saran N. Hip Joint Osteochondroma:
Systematic Review of the Literature and Report of Three Further Cases. Adv Orthop. 2014; 2014: 180254.
doi: 10.1155/2014/180254. PMID: 24963411.
22) Muzaffar N, Bashir N, Baba A, Ahmad A, Ahmad N. Isolated osteochondroma of the femoral neck
presenting as hip and leg pain. A case study. Ortop Traumatol Rehabil. 2012; 14(2): 183 -7. doi:
10.5604/15093492.992290. PMID: 22619103.
23) Ofiram E, Porat S. Progressive subluxation of the hip joint in a child with hereditary multiple exostosis. J
Pediatr Orthop B. 2004; 13(6): 371-3. doi: 10.1097/01202412-200411000-00004. PMID: 15599227.
24) Shinozaki T, Watanabe H, Inoue J, Ogiwara T. Rotational acetabular osteotomy in a dysplastic hip with
femoral neck osteochondromas. Orthopedics. 1998; 21(5): 588 -90. PMID: 9606700.
25) Siebenrock KA, Ganz R. Osteochondroma of the Femoral Neck. Clin Orthop Relat Res. 2002; 394: 211 -8.
doi: 10.1097/00003086-200201000-00025. PMID: 11795736.