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CLINICAL NOTE

Anterior Neck Pain Due to Hyoid Malformation


Matthew M. Michalowicz, MD; Luis G. Izquierdo, MD; M. Peter Sorensen, MD

nterior neck pain often presents a challenging problem for the otolaryngologist because of the various causes. We report an unusual case of an asymmetric hyoid bone
impinging on the carotid sheath with resulting pain on head rotation. Although rare,
awareness of this condition is vital in successful treatment as surgical intervention
provides immediate and long-term resolution of the symptoms.
REPORT OF A CASE

A 21-year-old man with a several-year history of intermittent, severe right anterior


level II neck pain and swelling reported
that his symptoms worsened when he
turned his head to the right but slowly resolved over hours to days. The pain was
always on the right side and often caused
the patient to become nonfunctional for
days at a time. The patient became dependent on narcotic medication to control this
pain. Symptoms also included right torticollis and dysphagia. On physical examination, point tenderness was located in
proximity to the superior portion of the
right lateral hyoid bone.
Computed tomography showed a significantly asymmetric hyoid bone. The right
greater cornu of the hyoid extended laterally and was immediately adjacent to the
carotid sheath at the bifurcation of the carotid artery. In the Figure, the right lateral portion of the hyoid bone is almost seen
wrapping around the carotid sheath at this
level with definite asymmetry compared
with the left side. The left greater cornu of
the hyoid was shorter and not closely associated with the carotid sheath. The patient was brought to the operating room for
excision of the right greater cornu of his hyoid bone.
The right lateral portion of the hyoid
bone was excised without complication. After surgery, the patient had almost immediate relief of the pain. There was no need
Author Affiliations: Department of Otolaryngology, Walter Reed National Military
Medical Center, Bethesda, Maryland.

ARCH OTOLARYNGOL HEAD NECK SURG/ VOL 138 (NO. 3), MAR 2012
307

for further narcotic pain medication use. On


follow-up examination, full neck range of
motion was restored and his symptoms had
completely resolved, with no return of pain.
He is 7 years without incident.
COMMENT

Hyoid syndrome is a rare but important


cause of anterior cervical neck pain. Nir et
al1 experienced an incidence of 0.2% (2 of
1000) in their ear, nose, and throat clinic.
Due to the vague symptoms, the differential diagnosis is numerous, and these patients have often seen several primary care
physicians before they are referred to an otolaryngologist. Treatments during this time
typically range from reassurance to multiple antibiotic regimens without any relief
of symptoms. Many explanations have been
offered for the origin of the pain, but, to our
knowledge, there are few case reports that
involve an asymmetric hyoid bone impingement near the carotid artery bifurcation.
The hyoid syndrome, as defined by Lim2,
is a complex of chronic and recurrent focal lancinating or dull pains in the carotid
area at the level of the tip of the greater hyoid cornu. The syndrome is often characterized by point tenderness on the outside
of the neck that may be referred to the lateral part of the neck and radiating to the
ipsilateral ear. Palpation, using the method
first described by Brown3 in 1954, will demonstrate point tenderness at the posterior
end of the greater horn of the hyoid. The
hyoid bone is a central point of attachment for almost all muscles of the anterior
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Figure. Axial computed tomography. A and B, The close association of the right greater cornu of the hyoid
bone (arrows) and the right carotid sheath. The right hyoid bone is seen wrapping around the carotid sheath.

neck and it is involved in the action


of swallowing. As a result, this may
cause the patient to have a sensation
of a foreign body, dysphagia, or tightness or pressure on that side of the
neck. Recently, Colby and Del Gaudio4 described a stylohyoid complex
syndrome that causes neck and facial pain by an elongated hyoid, elongation of the styloid process, or ossified stylohyoid ligament (as in Eagle
syndrome) in an attempt to consolidate 3 separate pathologic syndromes that cause the same symptoms. Any of these structures can
irritate the carotid artery or cranial
nerves VII, IX, or X. Impingement of
the neurovascular bundle from an enlarged hyoid caused our patient pain.
Previously, standard radiographs
of the cervical spine, to include the
odontoid view, were useful in demonstrating variants or abnormalities
of the hyoid apparatus.5 Recent advances in computed tomographic
imaging have allowed physicians to
complete a diagnostic workup quickly
and accurately before rendering treatment. In an article by Sittel et al6 in
1998, spiral computed tomography
with 3-dimensional reconstruction
was used to diagnose an elongation
and malformation of the greater cornu
of the hyoid. With a confirmed diagnosis and exact pathological
mechanism revealed, surgery was
performed. The surgical approach can
be accurately determined based on
correlation between a physical examination and computed tomography. In our patient, a submandibu-

lar approach was used to expose the


hyoid bone.
Once a diagnosis is confirmed,
multiple treatment modalities have
been proposed. Karlan et al7 and Robinson et al8 described relief of point
tenderness using an injection of combination corticosteroid and lidocaine hydrochloride. In 1998, Nir et
al1 presented an article using nonsteroidal anti-inflammatory drugs as
treatment. Ninety-one percent of patients with symptoms of less than 6
weeks experienced symptom relief,
and the authors recommended this
anti-inflammatory drug regimen for
first-line treatment. It was believed
that the symptoms in most of these
cases were caused by inflammation at
a muscular insertion point on the hyoid as initially described by Ernest and
Salter.9 Surgical excision of the tip of
the greater cornu should be planned
for those patients failing medical
therapy or with known malformations, as in our patient. Kopstein,5
Bhide and Dehadray,10 and Lim2 described the surgical excision of the tip
of the greater cornu with their patients exhibiting full relief of symptoms immediately after surgery.
Although this specific clinical scenario is rare, it does serve as a reminder that there are many causes to
anterior cervical neck pain. An awareness that this syndrome exists is vital in successful treatment. Our patient responded well to surgical
excision of the right greater cornu of
the hyoid with immediate symptom
relief and without return of the pain.

ARCH OTOLARYNGOL HEAD NECK SURG/ VOL 138 (NO. 3), MAR 2012
308

Submitted for Publication: May 12,


2011; final revision received September 11, 2011; accepted November 20, 2011.
Correspondence: Matthew M. Michalowicz, MD, Department of Otolaryngology, Walter Reed National Military Medical Center, 8901 Rockville
Pike, Bethesda, MD 20889-5600
(matthew.michalowicz@med.navy
.mil).
Author Contributions: Drs Sorensen and Izquierdo had full access to all the data in the study and
take responsibility for the integrity
of the data and the accuracy of the
data analysis. Study concept and design: Michalowicz, Sorensen, and Izquierdo. Drafting of the manuscript:
Michalowicz. Critical revision of the
manuscript for important intellectual
content: Sorensen and Izquierdo. Administrative, technical, and material
support: Michalowicz and Izquierdo. Study supervision: Sorensen and
Izquierdo.
Financial Disclosure: None reported.
Disclaimer: The views expressed in
this article are those of the authors
and do not necessarily reflect the official policy or position of the US Departments of the Army, Navy, and
Defense, or the US government.
REFERENCES
1. Nir D, Hefer T, Joachims HZ. Hyoid bone syndrome and its treatment with nonsteroidal antiinflammatory drugs. Am J Otolaryngol. 1998;
19(5):296-300.
2. Lim RY. The hyoid bone syndrome. Otolaryngol
Head Neck Surg. 1982;90(2):198-200.
3. Brown LA. Hyoid bone syndrome. South Med J.
1954;47(11):1088-1091.
4. Colby CC, Del Gaudio JM. Stylohyoid complex
syndrome. Arch Otolaryngol Head Neck Surg. 2011;
137(3):248-252.
5. Kopstein E. Hyoid syndrome. Arch Otolaryngol.
1975;101(8):484-485.
6. Sittel C, Brochhagen HG, Eckel HE, Michel O.
Hyoid bone malformation confirmed by 3-dimensional computed tomography. Arch Otolaryngol
Head Neck Surg. 1998;124(7):799-801.
7. Karlan MS, Beroza L, Cassisi NJ. Anterior cervical pain syndromes. Otolaryngol Head Neck Surg.
1979;87(3):284-291.
8. Robinson PJ, Davis JP, Fraser JG. The hyoid syndrome: a pain in the neck. J Laryngol Otol. 1994;
108(10):855-858.
9. Ernest EA III, Salter EG. Hyoid bone syndrome: a
degenerative injury of the middle pharyngeal constrictor muscle with photomicroscopic evidence of
insertion tendinosis. J Prosthet Dent. 1991;66
(1):78-83.
10. Bhide AR, Dehadray AY. Excision of the greater
cornu of the hyoid in hyoid syndrome. Auris Nasus Larynx. 1980;7(1):1-6.

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