A Demeyere

BB Koch

Hiroaki Fushiki

Hirohiko Tachino

HL Evans

ID Bough Jr

J Aoki

JA Greer Jr

K Itoh

Masayuki Ishida

R Hediger

Y Umekita

Yukio Watanabe

Journal of Medical Case Reports

English

PubMed

Springer - Publisher Connector

A low-grade chondrosarcoma presenting as an unusual cervical mass in the hyoid bone: a case report

CASE REPORT Open Access
A low-grade chondrosarcoma presenting
as an unusual cervical mass in the hyoid bone:
a case report
Hirohiko Tachino, Hiroaki Fushiki*, Masayuki Ishida and Yukio Watanabe
Abstract
Introduction: A chondrosarcoma originating from the hyoid bone is very rare. Here, we describe a case of low-
grade chondrosarcoma of hyoid origin and discuss its preoperative imaging features, including those on positron
emission tomography-computed tomography, and its recurrence rate.
Case presentation: A 42-year-old Japanese man noticed a mass in the right submandibular region of his neck. A
hard 3.0 × 2.8 cm tumor was noted on the right side of his hyoid bone. The mass was immobile and moved with
deglutition.
Conclusion: Even though radiographic studies, including positron emission tomography-computed tomography,
were inconclusive, the cartilaginous tumor was surgically removed en bloc, and the tumor was diagnosed based on
the results of pathological investigations. Close follow-up is recommended in such cases due to the potential for
recurrences, because local recurrence occurred in 50% of the reported cases of grade one chondrosarcomas.
Introduction
Chondrosarcoma, a malignant tumor characterized by
the production of a cartilage matrix, accounts for about
11% cases of primary malignant bone tumors [1]. The
parts of the body where it develops at relatively high
incidences include the long bones, the pelvis and the
ribs [1]. The head and neck area are seldom affected;
chondrosarcomas in this area reportedly account for 1%
to 12% of all reported cases of the disease [1]. Chondro-
sarcoma of the hyoid bone is extremely rare, with only
15 cases being reported in international literature. Here,
we describe a case of low-grade chondrosarcoma of
hyoid origin. We also discuss its preoperative imaging
features and its recurrence rate.
Case presentation
A 42-year-old Japanese man noticed a mass in the right
submandibular region of his neck in June 2010. He vis-
ited the department of otolaryngology at a nearby general
hospital the next month and was referred for medical
treatment. More than two weeks later, he visited our
University Hospital. A hard 3.0 × 2.8 cm tumor was
noted on the right side of his hyoid bone. The mass was
immobile and moved with deglutition. No spontaneous
or tender pain was noted.
Laryngeal endoscopy revealed no contributory abnor-
mal findings. Computed tomography (CT) demonstrated
a gourd-shaped distension of bone in the area extending
from the body of the hyoid bone to the right greater
horn. A multilocular cystic area of low density was
observed inside the distension, and central ossification
was evident in the anterior lesion (Figure 1a). Magnetic
resonance imaging (MRI) of his neck revealed a gourd-
shaped nodular lesion of 2.5 × 2.2 cm (upper part) and
1.6 × 1.0 cm (lower part) on the right side of his hyoid
bone. The signal intensity was low in T1-weighted
images and high in T2-weighted images, with a mosaic
pattern being observed in the high signals (Figure 1b, c).
Based on these findings, a cartilaginous tumor originat-
ing from the right side of his hyoid bone was diagnosed.
No abnormal accumulation was detected by bone scinti-
graphy or fluorodeoxyglucose positron emission tomo-
graphy (FDG-PET) (Figure 2). Therefore, high-grade
malignant chondrosarcoma was diagnosed by exclusion.* Correspondence: hfushiki@med.u-toyama.ac.jp
Department of Otolaryngology, Head and Neck Surgery, University of
Toyama, Toyama, Japan
Tachino et al. Journal of Medical Case Reports 2012, 6:21
http://www.jmedicalcasereports.com/content/6/1/21 JOURNAL OF MEDICAL
CASE REPORTS
© 2012 Tachino et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons
Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in
any medium, provided the original work is properly cited.
a.
b.  c.
Figure 1 Preoperative imaging by CT and MRI. (a) Computed tomography of his neck. A gourd-shaped distension of bone was present on
the right side of his hyoid bone. A multilocular cystic area of low density was observed in the distention. Central ossification was evident in the
anterior lesion. (b) Magnetic resonance imaging T1-weighted image with low signal intensity. (c) MRI T2-weighted image high signal intensity. A
mosaic pattern was visible in the high signal area.
Tachino et al. Journal of Medical Case Reports 2012, 6:21
http://www.jmedicalcasereports.com/content/6/1/21
Page 2 of 5
An analysis of his blood samples revealed no contribu-
tory abnormalities.
Surgery was performed to extirpate the tumor. His
geniohyoid, mylohyoid, sternohyoid, and thyrohyoid
muscles were detached from his hyoid bone. The tumor
mass and surgical margins were resected bilaterally on
the left side of the body of his hyoid bone and the right
greater horn. There were almost no adhesions between
the tumor and the tissue around it. Following a favor-
able postoperative course, our patient was discharged
from hospital.
A macroscopic examination identified the cartilaginous
tumor as a semi-lobular mass covered by a thin fibrous
coat. Histopathological examination revealed that it was
relatively well differentiated, but with a somewhat high
cell density and many binuclear cells. An open chromatin
pattern was evident and intranuclear structures were
observed in many cells. The condition was diagnosed as
chondrosarcoma, grade one (Figure 3a-c). No additional
surgical treatment was required. Our patient has been
periodically followed up at our outpatient clinic. Twelve
months postoperatively, the clinical course has been
favorable and no local recurrences or remote metastases
have been detected.
Discussion
Generally, the most useful diagnostic imaging examina-
tion for chondrosarcoma is CT, which detects irregular
bone destruction. Intratumoral calcification is noted in
about 75% of patients using this tool [2,3]. MRI is useful
for surgical planning and for evaluating the degree of
invasion of the tumor into the surrounding soft tissues.
Signal intensity is low in T1-weighted images and high
in T2-weighted images; the calcified portion is signal-
Figure 2 Positron emission tomography-computed tomography. No abnormal accumulation in the hyoid tumor or systemic abnormalities
were evident.
Tachino et al. Journal of Medical Case Reports 2012, 6:21
http://www.jmedicalcasereports.com/content/6/1/21
Page 3 of 5
free. A characteristic finding is a mosaic pattern in high
signal areas in T2-weighted images [4]. In our case, a
CT examination demonstrated no evidence of bone
destruction, but revealed intratumoral calcification. On
MRI examination, the signal intensity was low in T1-
weighted images and high in T2-weighted images, with
a.
b. c.
Figure 3 Macroscopic and histopathological examination. (a) The extirpated specimen was revealed to be a semi-lobular cartilaginous tumor
mass covered by a thin fibrous coat. (b,c) Histopathological findings: (b) hematoxylin-eosin (HE)-stained, ×2; (c) HE-stained, ×40. The cartilaginous
tumor mass is relatively well differentiated, the cell density is somewhat high and many binuclear cells were seen; an open chromatin pattern is
evident with intranuclear structures observable in many cells.
Tachino et al. Journal of Medical Case Reports 2012, 6:21
http://www.jmedicalcasereports.com/content/6/1/21
Page 4 of 5
a mosaic pattern; however, no invasion into the tissue
around the lesion was seen.
In most cases, bone scintigraphy examination reveals
poor uptake in the chondrosarcoma itself and morbid
uptake in the destroyed area of the affected bone [5].
FDG-PET is useful for grading tumors and for evaluating
local recurrences and metastatic diseases [6]. A standar-
dized uptake value of 1.3 has been reported as a border
value that distinguishes between benign and malignant
tumors [7]. However, bone scintigraphy and FDG-PET
did not rule out the presence of chondrosarcoma in our
case. The negative test results may have been obtained
partly because the hyoid bone itself is small, and partly
because the reactive bone formation was inconspicuous
on histology. Therefore, it may be impossible to distin-
guish chondroma from low-grade chondrosarcoma of
hyoid origin by means of preoperative imaging tests,
including positron emission tomography-computed
tomography (PET-CT).
Generally, the occurrence of metastases becomes more
probable as the degree of malignancy increases, resulting
in poor prognoses [8]. The five- year survival rate for all
cases of chondrosarcoma is reportedly 90% for grade one,
81% for grade two and 43% for grade three [9]. However,
in the few grade one cases of hyoid origin with available
outcome records, local recurrences occurred in five of
the 10 reported cases. Therefore, close follow-up is
necessary.
Conclusion
The present case did not involve union of the lesion to
the tissue around it. The tumor and surrounding intact
hyoid bone tissue were completely resected. Pathological
results led to the diagnosis of a grade one tumor of low
malignancy. Even though our radiographic studies,
including PET-CT, were inconclusive, we surgically
removed the cartilaginous tumor en bloc and the tumor
was diagnosed based on the results of pathological
investigations. Chondrosarcomas of hyoid origin have a
high incidence of local recurrence and are sometimes
metastatic. Therefore, it seems necessary to closely fol-
low-up the patient for a prolonged period of time.
Consent
Written informed consent was obtained from the patient
for publication of this case report and any accompany-
ing images. A copy of the written consent is available
for review by the Editor-in-Chief of this journal.
Authors’ contributions
HT was involved in drafting the manuscript and reviewing the literature. HF
was a major contributor in revising the manuscript and getting informed
consent from our patient. MI was involved in reviewing the literature. YW
reviewed the manuscript. All authors were responsible for the diagnosis,
treatment and follow-up of the patient described in this case report. All
authors read and approved the final manuscript.
Competing interests
The authors declare that they have no competing interests.
Received: 25 September 2011 Accepted: 18 January 2012
Published: 18 January 2012
References
1. Koch BB, Karnell LH, Hoffman HT, Apostolakis LW, Robinson RA, Zhen W,
Menck HR: National cancer database report on chondrosarcoma of the
head and neck. Head Neck 2000, 22(4):408-425.
2. Demeyere A, De Somer F, Perdieus D, Van Den Hauwe L, Lemmens L,
Schillebeeckx J: Chondrosarcoma of the hyoid bone. Eur Radiol 2000,
10(2):308-309.
3. Hediger R, McEniff N, Karmody C, Eustace S: Recurrent chondrosarcoma of
the hyoid bone. Clin Imaging 1997, 21(1):69-72.
4. Itoh K, Nobori T, Fukuda K, Furuta S, Ohyama M: Chondrosarcoma of the
hyoid bone. J Laryngol Otol 1993, 107(7):642-646.
5. Greer JA Jr, Devine KD, Dahlin DC: Gardner’s syndrome and
chondrosarcoma of the hyoid bone. Arch Otolaryngol 1977,
103(7):425-427.
6. Umekita Y, Yoshida A, Yoshida H: Parosteal chondrosarcoma attached to
the hyoid bone: A case report. Int J Oral Maxillofac Surg 1998,
27(3):215-216.
7. Aoki J, Watanabe H, Shinozaki T, Tokunaga M, Inoue T, Endo K: FDG-PET in
differential diagnosis and grading of chondrosarcomas. J Comput Assist
Tomogr 1999, 23(4):603-608.
8. Bough ID Jr, Chiles PJ, Fratalli MA, Vernose G: Laryngeal chondrosarcoma:
two unusual cases. Am J Otolaryngol 1995, 16(2):126-131.
9. Evans HL, Ayala AG, Romsdahl MM: Prognostic factors in chondrosarcoma
of bone: a clinicopathologic analysis with emphasis on histologic
grading. . Cancer 1977, 40(2):818-831.
doi:10.1186/1752-1947-6-21
Cite this article as: Tachino et al.: A low-grade chondrosarcoma
presenting as an unusual cervical mass in the hyoid bone: a case
report. Journal of Medical Case Reports 2012 6:21.
Submit your next manuscript to BioMed Central
and take full advantage of: 
• Convenient online submission
• Thorough peer review
• No space constraints or color figure charges
• Immediate publication on acceptance
• Inclusion in PubMed, CAS, Scopus and Google Scholar
• Research which is freely available for redistribution
Submit your manuscript at 
www.biomedcentral.com/submit
Tachino et al. Journal of Medical Case Reports 2012, 6:21
http://www.jmedicalcasereports.com/content/6/1/21
Page 5 of 5


Abstract
          
            Introduction
            A chondrosarcoma originating from the hyoid bone is very rare. Here, we describe a case of low-grade chondrosarcoma of hyoid origin and discuss its preoperative imaging features, including those on positron emission tomography-computed tomography, and its recurrence rate.
          
          
            Case presentation
            A 42-year-old Japanese man noticed a mass in the right submandibular region of his neck. A hard 3.0 × 2.8 cm tumor was noted on the right side of his hyoid bone. The mass was immobile and moved with deglutition.
          
          
            Conclusion
            Even though radiographic studies, including positron emission tomography-computed tomography, were inconclusive, the cartilaginous tumor was surgically removed en bloc, and the tumor was diagnosed based on the results of pathological investigations. Close follow-up is recommended in such cases due to the potential for recurrences, because local recurrence occurred in 50% of the reported cases of grade one chondrosarcomas.
          </jats:sec

Crossref

Chondrosarcoma of the hyoid bone.

Chondrosarcoma of the hyoid bone. Eur Radiol

Endo K: FDG-PET in differential diagnosis and grading of chondrosarcomas.

Gardner’s syndrome and chondrosarcoma of the hyoid bone. Arch Otolaryngol

Menck HR: National cancer database report on chondrosarcoma of the head and neck. Head Neck

Parosteal chondrosarcoma attached to the hyoid bone: A case report.

Prognostic factors in chondrosarcoma of bone: a clinicopathologic analysis with emphasis on histologic grading. . Cancer

Recurrent chondrosarcoma of the hyoid bone. Clin Imaging

Vernose G: Laryngeal chondrosarcoma: two unusual cases.

file:///data/core-remote/dit/data/Springer-OA/pdf/3e8/aHR0cDovL2xpbmsuc3ByaW5nZXIuY29tLzEwLjExODYvMTc1Mi0xOTQ3LTYtMjEucGRm.pdf

A low-grade chondrosarcoma presenting as an unusual cervical mass in the hyoid bone: a case report

Abstract

Similar works

Full text

Available Versions

Springer - Publisher Connector

Springer - Publisher Connector

Crossref