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845
Case
‘“f’
Acute Sinus
I.’(
Suppurative Thyroiditis Associated Fistula: Sonographic Findings
Hiroto Hatabu,1 Kanji and Junji Konishi
Kasagi,
Kazutaka
Yamamoto,
Yasuhiro
Acute suppurative thyroiditis associated with internal fistula from the piriform sinus is a rare clinical entity caused by bacterial infection via the remnant of the fourth branchial cleft
[1 -3].
The sonographic
sented.
Sonography
appearance
revealed
of the disease
the characteristic
is pre-
findings
of a
left-sided perithyroid hypoechoic area that involved the left lobe of the thyroid gland. Barium meal study confirmed that an internal
Case
fistula
was the route
of infection.
swelling
and drainages. The WBC count was 9.8 x 10/l, C-reactive protein was 9.9 mg/dl (0.10 g/l), erythrocyte sedimentation rate was 52 mm/ hr, serum level of T4 was 8.4 ,.g/dl (108 nmol/I; normal range, 5-11 zg/dl [64-1 42 nmol/l]), serum level of T3 was 68 ng/dl (1 .0 nmol/I; normal range, 90-1 70 ng/dl [1 .4-2.6 nmol/l]), and thyroid-stimulating hormone level was 0.96 U/ml (0.96 mU/I; normal range, 0.30-3.90 tU/ml [0.30-3.9OmU/l}). Sonography revealed a left-sided perithyroid area
involving
the
left
lobe
of the
thyroid
gland
(Figs.
1A
and 1 B), and a subsequent barium meal study revealed an internal fistula originating from the apex of the left side of the piriform sinus (Fig. 1 C). Complete surgical resection of the fistula resulted in a cure.
I
January
All authors:
AJR 155:845-847,
Akinari
Hidaka,
Keigo
Endo,
Discussion Acute suppurative thyroiditis associated with internal fistula from the piriform sinus can be characterized clinically as follows: (1) abrupt onset of painful swelling in the left side of the thyroid gland accompanied by fever and pain on swallowing,
occasionally
female
to male
with
ratio
upper
respiratory
tract
infection;
of 7 to 10; (3) age at onset
common
(2)
between
2
unless the fistula
pears at about the sixth week of development. woman was referred to our department because of of the left side of the neck with tenderness and
fever. She had had frequent episodes ofleft-sided neck swelling since she was 4 years old, which had been treated by repeated incisions
Received
Misaki,
is extirpated completely [1]. Embryologically, the branchial apparatus becomes evident during the third and fourth weeks of development and disap-
A 23-year-old
hypoechoic
lida, Takashi
with Piriform
and 12 years; and (4) recurrence
Report
anterior
Report
22, 1990;
Radiology October
accepted
and Nuclear
after revision Medicine,
pouches
may occur in the cervical region, but more than 95%
of them are formed from the second pouch and groove, which pass between the internal and external carotid arteries [2]. An internal fistula originating from the apex of the piriform sinus as a remnant of the fourth branchial cleft is rare; however, it has recently been recognized as the most common cause of acute suppurative thyroiditis [1 , 3]. Anatomically, the fistula runs anteroinferiorly from the apex of the piriform sinus into the semiclosed space around the thyroid gland (spatium perithyroideum). The latter is bordered by the
April 24, 1990.
Kyoto
1990 0361-803X/90/1554-0845
University
Hospital,
C American
Sakyo-ku, Roentgen
It is considered
that cervical cysts and fistulas are derived from remnants of branchial grooves and pouches with failure of fusion or burying of cell rests of the branchial grooves. Branchial cleft defects from any of the first to fourth branchial grooves and
Kyoto
606, Japan.
Ray Society
Address
reprint
requests
to H. Hatabu.
846
HATABU
ET AL.
AJR:155,
Downloaded from www.ajronline.org by Univ Of California-San Diego on 10/02/14 from IP address 132.239.1.231. Copyright ARRS. For personal use only; all rights reserved
.1
October
1990
}:
Fig. 1.-A, Real-time sonography (7.5 MHz) with a water-bath technique shows hypoechoic area surrounding anterolateral aspects of thyroid which continues to left lobe of thyroid gland (arrows). T = trachea; E = esophagus; A = left common carotid artery; V = left internal jugular vein. B, Longitudinal sonogram of left lobe of thyroid reveals poorly defined hypoechoic area. C, Barium meal study with oblique projection reveals a fistula originating from left side of piriform sinus (arrow).
Fig. 2.-14 year-old girl with recurrent episodes of sore throat, was refused by patient. Symptoms recurred 2 months later. A
fever, and swelling
of left side of neck. Surgical
intervention
A, Transverse sonogram shows pooriy defined perithyroid hypoechoic area that continues to left lobe of thyroid (arrows). left common carotid artery; V = left internal jugular vein. B, Barium meal study with anteroposterior view visualized a fistula originating from left side of piriform sinus (arrow). C, Barium meal study with right-left lateral view also showed fistula slightly curving anteriorly (arrow).
T
gland,
was recommended, =
trachea;
E
=
but
esophagus;
=
middle layer of cervical fascia covering the posterior surface of the sternothyroid muscle anteriorly, the prevertebral fascia posteriorly, the carotid sheath laterally, and the insertion of the sternothyroid muscle to the thyroid cartilage superiorly. After infection via the fistula, purulent exudate accumulates in this space [1]. On sonograms, the left-sided perithyroid hypoechoic area infiltrating the thyroid gland was, therefore, considered to be an abscess caused by bacterial infection. We have observed very similar findings in a 1 4-year-old girl with this disorder
(Fig. 2). Subacute thyroiditis, primary thyroid lymphoma, and undifferentiated carcinoma occasionally show poorly defined hypoechoic areas, which may simulate the sonographic findings in acute suppurative thyroiditis. However, the hypoechoic lesion
in these
three
conditions
arises
from
the
thyroid
gland
itself, unlike the predominantly perithyroid nature of the hypoechoic area in acute suppurative thyroiditis [4, 5]. When the inflammation is extensive, separation of thyroid from perithyroid
involvement
the right
lobe virtually
may
become
precludes
difficult.
the diagnosis
Involvement
of
of acute
sup-
Downloaded from www.ajronline.org by Univ Of California-San Diego on 10/02/14 from IP address 132.239.1.231. Copyright ARRS. For personal use only; all rights reserved
AJR:155,
October
1990
SONOGRAPHY
OF ACUTE
SUPPURATIVE
THYROIDITIS
purative thyroiditis [1 , 6]. Visualization of the fistula by barium meal study or fistulography is considered diagnostic for acute suppurative thyroiditis. We were able to show it in both of
tion.
our patients. Bacteremia or a persistent thyroglossal duct are other known causes of the disease. To our knowledge, only a few descriptions of sonographic features of acute suppurative thyroiditis have been published. Clair et al. [7] reported a case of acute suppurative thyroiditis as a complication of systemic lupus erythematosus and cor-
REFERENCES
ticosteroid enlargement atogenous
therapy, in which sonography revealed diffuse of the thyroid gland reflecting the probable heminfection, unlike that in our patients. Sonography is now widely used for morphologic examination of various thyroid diseases [8]. We recommend a careful
search of the hypopharynx for the fistula in patients with characteristic sonographic findings of a poorly defined inhomogeneous hypoechoic area surrounding and involving the left lobe ofthe thyroid, because piriform sinus fistula is virtually always pathogenic if the patient is young and is not immunosuppressed
or otherwise
at risk for primary
thyroid
infec-
In addition,
complete
sary to prevent
1 . Takai
extirpation
recurrence
S. Miyauchi
847
A, Matsuzuka
of the fistula
is neces-
of the disease.
F, Kuma
K, Kosaki
G. Internal
fistula
as
a route of infection in acute suppurative thyroiditis. Lancet 1979:1: 751-752 2. Proctor B, Proctor C. Congenital lesions of head and neck. Otolaryngol Clin North Am 19703:221-248 HM, Skolnick Ml. Fourth branched cleft (pharyngeal pouch) remnant. Trans Am Acad Ophthalmol Otolaryngol 1973;77:OAL368-370 4. Tokuda Y, Kasagi K, lida Y, et al. Sonography of subacute thyroiditis: changes in the findings during the course of the disease. JCU 1990;18:
3. Tucker
21-26 5. Takashima comparison
5, Morimoto
5, lkezoe
J, et al. Primary
of CT and US assessment.
Radiology
6. Miller M, Hill JL, Sun CC, O’Brien
thyroid
1989;171
lymphoma: :439-443
DS, Hailer JA. The diagnosis
and
management of pyriform sinus fistulae in infants and young children. J Pediatr Surg 1983;18:377-381 7. Clair MA, Mandelblatt 5, Baim AS, Perkes E, Goodman K. Sonographic features
8. Simeone sonography
of acute
JF,
suppurative
thyroiditis.
GH, Mueller of the thyroid. Radiology Daniels
JCU
1983;11
:222-224
PR, et al. High-resolution 1982;145:431-435
real-time
845
Case
‘“f’
Acute Sinus
I.’(
Suppurative Thyroiditis Associated Fistula: Sonographic Findings
Hiroto Hatabu,1 Kanji and Junji Konishi
Kasagi,
Kazutaka
Yamamoto,
Yasuhiro
Acute suppurative thyroiditis associated with internal fistula from the piriform sinus is a rare clinical entity caused by bacterial infection via the remnant of the fourth branchial cleft
[1 -3].
The sonographic
sented.
Sonography
appearance
revealed
of the disease
the characteristic
is pre-
findings
of a
left-sided perithyroid hypoechoic area that involved the left lobe of the thyroid gland. Barium meal study confirmed that an internal
Case
fistula
was the route
of infection.
swelling
and drainages. The WBC count was 9.8 x 10/l, C-reactive protein was 9.9 mg/dl (0.10 g/l), erythrocyte sedimentation rate was 52 mm/ hr, serum level of T4 was 8.4 ,.g/dl (108 nmol/I; normal range, 5-11 zg/dl [64-1 42 nmol/l]), serum level of T3 was 68 ng/dl (1 .0 nmol/I; normal range, 90-1 70 ng/dl [1 .4-2.6 nmol/l]), and thyroid-stimulating hormone level was 0.96 U/ml (0.96 mU/I; normal range, 0.30-3.90 tU/ml [0.30-3.9OmU/l}). Sonography revealed a left-sided perithyroid area
involving
the
left
lobe
of the
thyroid
gland
(Figs.
1A
and 1 B), and a subsequent barium meal study revealed an internal fistula originating from the apex of the left side of the piriform sinus (Fig. 1 C). Complete surgical resection of the fistula resulted in a cure.
I
January
All authors:
AJR 155:845-847,
Akinari
Hidaka,
Keigo
Endo,
Discussion Acute suppurative thyroiditis associated with internal fistula from the piriform sinus can be characterized clinically as follows: (1) abrupt onset of painful swelling in the left side of the thyroid gland accompanied by fever and pain on swallowing,
occasionally
female
to male
with
ratio
upper
respiratory
tract
infection;
of 7 to 10; (3) age at onset
common
(2)
between
2
unless the fistula
pears at about the sixth week of development. woman was referred to our department because of of the left side of the neck with tenderness and
fever. She had had frequent episodes ofleft-sided neck swelling since she was 4 years old, which had been treated by repeated incisions
Received
Misaki,
is extirpated completely [1]. Embryologically, the branchial apparatus becomes evident during the third and fourth weeks of development and disap-
A 23-year-old
hypoechoic
lida, Takashi
with Piriform
and 12 years; and (4) recurrence
Report
anterior
Report
22, 1990;
Radiology October
accepted
and Nuclear
after revision Medicine,
pouches
may occur in the cervical region, but more than 95%
of them are formed from the second pouch and groove, which pass between the internal and external carotid arteries [2]. An internal fistula originating from the apex of the piriform sinus as a remnant of the fourth branchial cleft is rare; however, it has recently been recognized as the most common cause of acute suppurative thyroiditis [1 , 3]. Anatomically, the fistula runs anteroinferiorly from the apex of the piriform sinus into the semiclosed space around the thyroid gland (spatium perithyroideum). The latter is bordered by the
April 24, 1990.
Kyoto
1990 0361-803X/90/1554-0845
University
Hospital,
C American
Sakyo-ku, Roentgen
It is considered
that cervical cysts and fistulas are derived from remnants of branchial grooves and pouches with failure of fusion or burying of cell rests of the branchial grooves. Branchial cleft defects from any of the first to fourth branchial grooves and
Kyoto
606, Japan.
Ray Society
Address
reprint
requests
to H. Hatabu.
846
HATABU
ET AL.
AJR:155,
Downloaded from www.ajronline.org by Univ Of California-San Diego on 10/02/14 from IP address 132.239.1.231. Copyright ARRS. For personal use only; all rights reserved
.1
October
1990
}:
Fig. 1.-A, Real-time sonography (7.5 MHz) with a water-bath technique shows hypoechoic area surrounding anterolateral aspects of thyroid which continues to left lobe of thyroid gland (arrows). T = trachea; E = esophagus; A = left common carotid artery; V = left internal jugular vein. B, Longitudinal sonogram of left lobe of thyroid reveals poorly defined hypoechoic area. C, Barium meal study with oblique projection reveals a fistula originating from left side of piriform sinus (arrow).
Fig. 2.-14 year-old girl with recurrent episodes of sore throat, was refused by patient. Symptoms recurred 2 months later. A
fever, and swelling
of left side of neck. Surgical
intervention
A, Transverse sonogram shows pooriy defined perithyroid hypoechoic area that continues to left lobe of thyroid (arrows). left common carotid artery; V = left internal jugular vein. B, Barium meal study with anteroposterior view visualized a fistula originating from left side of piriform sinus (arrow). C, Barium meal study with right-left lateral view also showed fistula slightly curving anteriorly (arrow).
T
gland,
was recommended, =
trachea;
E
=
but
esophagus;
=
middle layer of cervical fascia covering the posterior surface of the sternothyroid muscle anteriorly, the prevertebral fascia posteriorly, the carotid sheath laterally, and the insertion of the sternothyroid muscle to the thyroid cartilage superiorly. After infection via the fistula, purulent exudate accumulates in this space [1]. On sonograms, the left-sided perithyroid hypoechoic area infiltrating the thyroid gland was, therefore, considered to be an abscess caused by bacterial infection. We have observed very similar findings in a 1 4-year-old girl with this disorder
(Fig. 2). Subacute thyroiditis, primary thyroid lymphoma, and undifferentiated carcinoma occasionally show poorly defined hypoechoic areas, which may simulate the sonographic findings in acute suppurative thyroiditis. However, the hypoechoic lesion
in these
three
conditions
arises
from
the
thyroid
gland
itself, unlike the predominantly perithyroid nature of the hypoechoic area in acute suppurative thyroiditis [4, 5]. When the inflammation is extensive, separation of thyroid from perithyroid
involvement
the right
lobe virtually
may
become
precludes
difficult.
the diagnosis
Involvement
of
of acute
sup-
Downloaded from www.ajronline.org by Univ Of California-San Diego on 10/02/14 from IP address 132.239.1.231. Copyright ARRS. For personal use only; all rights reserved
AJR:155,
October
1990
SONOGRAPHY
OF ACUTE
SUPPURATIVE
THYROIDITIS
purative thyroiditis [1 , 6]. Visualization of the fistula by barium meal study or fistulography is considered diagnostic for acute suppurative thyroiditis. We were able to show it in both of
tion.
our patients. Bacteremia or a persistent thyroglossal duct are other known causes of the disease. To our knowledge, only a few descriptions of sonographic features of acute suppurative thyroiditis have been published. Clair et al. [7] reported a case of acute suppurative thyroiditis as a complication of systemic lupus erythematosus and cor-
REFERENCES
ticosteroid enlargement atogenous
therapy, in which sonography revealed diffuse of the thyroid gland reflecting the probable heminfection, unlike that in our patients. Sonography is now widely used for morphologic examination of various thyroid diseases [8]. We recommend a careful
search of the hypopharynx for the fistula in patients with characteristic sonographic findings of a poorly defined inhomogeneous hypoechoic area surrounding and involving the left lobe ofthe thyroid, because piriform sinus fistula is virtually always pathogenic if the patient is young and is not immunosuppressed
or otherwise
at risk for primary
thyroid
infec-
In addition,
complete
sary to prevent
1 . Takai
extirpation
recurrence
S. Miyauchi
847
A, Matsuzuka
of the fistula
is neces-
of the disease.
F, Kuma
K, Kosaki
G. Internal
fistula
as
a route of infection in acute suppurative thyroiditis. Lancet 1979:1: 751-752 2. Proctor B, Proctor C. Congenital lesions of head and neck. Otolaryngol Clin North Am 19703:221-248 HM, Skolnick Ml. Fourth branched cleft (pharyngeal pouch) remnant. Trans Am Acad Ophthalmol Otolaryngol 1973;77:OAL368-370 4. Tokuda Y, Kasagi K, lida Y, et al. Sonography of subacute thyroiditis: changes in the findings during the course of the disease. JCU 1990;18:
3. Tucker
21-26 5. Takashima comparison
5, Morimoto
5, lkezoe
J, et al. Primary
of CT and US assessment.
Radiology
6. Miller M, Hill JL, Sun CC, O’Brien
thyroid
1989;171
lymphoma: :439-443
DS, Hailer JA. The diagnosis
and
management of pyriform sinus fistulae in infants and young children. J Pediatr Surg 1983;18:377-381 7. Clair MA, Mandelblatt 5, Baim AS, Perkes E, Goodman K. Sonographic features
8. Simeone sonography
of acute
JF,
suppurative
thyroiditis.
GH, Mueller of the thyroid. Radiology Daniels
JCU
1983;11
:222-224
PR, et al. High-resolution 1982;145:431-435
real-time
