Acta Med. Okayama, 2024
Vol. 78, No. 4, pp. 349‑355
CopyrightⒸ 2024 by Okayama University Medical School.

Case Report

http://escholarship.lib.okayama-u.ac.jp/amo/

Middle-Ear Salivary Gland Choristoma with Congenital,
Single-Sided Hearing Loss
Yuichiro Tominagaa, Akiko Sugayab,c＊, Shin Kariyad, Aiko Shimizub,
Yuko Kataokab,c, and Mizuo Andoba

Department of Otolaryngology, Head and Neck Surgery, Hiroshima City, Hiroshima Citizens Hospital, Hiroshima 730-8518, Japan,b
Department of Otolaryngology, Head and Neck Surgery, Okayama University Graduate School of Medicine,
Dentistry and Pharmaceutical Sciences, Okayama 700-8558, Japan,c
Okayama Kanariya Gakuen, Okayama 700-0927, Japan,dDepartment of Otolaryngology, Head and Neck Surgery, Kawasaki Medical School Hospital, Kurashiki, Okayama 701-0192, Japan

Middle-ear salivary gland choristoma (SGCh) is a rare, benign tumor that causes conductive hearing loss owing
to middle-ear morphological abnormalities. Early diagnosis is challenging, and surgical resection is indispensable for a definitive diagnosis. We report the case of a 3-year-old boy diagnosed with middle-ear SGCh during
the follow-up period for left-sided hearing loss discovered at newborn hearing screening (NHS). Long-term
follow-up after the NHS result, subsequent computed tomography/magnetic resonance imaging, and surgicalresection led to its relatively early diagnosis and treatment.
Key words: middle-ear salivary gland choristoma, middle-ear morphological abnormalities, newborn hearing
screening, unilateral hearing loss, surgical resection

Salivary gland choristoma (SGCh) is a rare, benign
tumor most commonly situated in the posterior
pituitary gland, parotid lymph nodes, and middle ear.
Fewer than 50 cases [1-43] of middle-ear SGCh havebeen reported. This tumor is typically accompanied bymorphological abnormalities in the middle ear, such as
malformations of the ossicular chain, particularly theincus and stapes, and dehiscence of the facial nervefrom the fallopian canal. Although such malformationsof the middle ear may cause congenital unilateral hearing loss, many cases are not diagnosed early in life. Thismay be because of physicians’ lack of awareness of thistumor or the risk of facial nerve palsy associated with its
diagnosis, as invasive surgery is required for a definitive
diagnosis.
Here, we report a case of middle-ear SGCh in an
Received January 5, 2024 ; accepted April 26, 2024.
Corresponding author. Phone : +81-86-235-7307; Fax : +81-86-235-7308
E-mail : sugaya-a@cc.okayama-u.ac.jp (A. Sugaya)＊

infant who was followed up for unilateral hearing loss
detected during newborn hearing screening (NHS), a
test of the automated auditory brainstem response(ABR) universally performed a few days after birth.
Although reports of this tumor and its early diagnosis
are rare, clinicians should be aware of SGCh in the differential diagnosis of congenital unilateral hearing loss.

Case
A 3-year-old boy with left-sided hearing loss that
had been diagnosed during NHS was brought to the
outpatient clinic of our hospital owing to a mass in his
left middle ear. He was born at 38 weeks’ gestationalage, weighing 2,450 g. His right ear had passed the
NHS test, but he was referred to the maternity hospitalfor his left ear. Conventional auditory brainstem response
Conflict of Interest Disclosures: No potential conflict of interest relevantto this article was reported.

350

Tominaga et al.

(ABR) testing performed at the child development center Okayama Kanariya Gakuen (OKG) when the patientwas 2 months old showed V-waves at 30 dB in the right
ear and 70 dB in the left ear. He was subsequently followed up for left-sided hearing loss at OKG. His ABRs
slightly improved, yielding V-waves at 20 dB in theright ear and 50 dB in the left ear at the follow-ups at
7 and 13 months. The patient’s left tympanic membrane was cloudy upon otoscopic observation at both
follow-up visits, and also at 18 months (Fig. 1), twomonths after a mass in the boy’s left anterior ear had
been resected (at a different institution) and diagnosed
as a lipoma.
When the boy was 2 years old, temporal-bone computed tomography (CT) (Fig. 1) revealed a shadow inthe left middle ear. Although the scutum was distinct,
the long process of the incus and the stapes were
unclear. Diffusion-weighted magnetic resonance imaging (MRI) of the brain (Fig. 2) revealed no signs of cholesteatoma, and the patient continued follow-up withaudiology.
A

Acta Med. Okayama Vol. 78, No. 4

Two months later, the decision was finally made toperform a biopsy of the mass under general anesthesia
for the differential diagnosis of a possible tumor or secondary acquired cholesteatoma. A soft white mass inthe middle ear with no epithelium—consistent with acholesteatoma—was observed using a postauricularapproach. Small tissue samples were submitted forpathological examination; we preserved the chorda
tympani but did not visualize the facial nerve. The frozensection and histopathological examinations revealed
glandular tissue. As these results revealed no malignancy, no additional treatment was performed.
The patient’s left middle ear shadow remained
unchanged upon follow-up CT when he was 3 years
old; however, a mastoid shadow had newly appeared(Fig. 3). Exploratory tympanotomy and mass removal
were performed when the patient was 3 years 7 months
old. The tumor was removed, and a tympanostomy
tube was placed because of the presence of otitis media
with effusion. Although intraoperative pathology indicated the presence of glandular tissue, almost all of thevisible tumors were resected for a definitive diagnosis.
After resection, the facial nerve was detected inferior towhere the tumors were located, and the malleus wasA

B

Fig. 1(A) The left tympanic cavity was cloudy. (B) Computed
tomography (CT) at 2 years of age, coronal view. A soft-tissue
mass was observed in the tympanic cavity, contiguous with thefacial nerve canal (△).

B

Fig. 2Magnetic resonance imaging (MRI) at 2 years of age,
axial view. (A) A T2-weighted image revealed the tumor as a strong
signal (○). (B) Diﬀusion-weighted image revealed no high-signal
area where the tumor was located.

Rare Case of Middle-Ear Choristoma

August 2024

intact, but the long process of the incus and the stapes
could not be seen. The stapes footplate could not be
detected owing to the presence of granulation tissue;

351

reconstruction of the ossicles was not performed.
Histopathological examination revealed that the tumor
was an SGCh of the middle ear (Fig. 4).
Follow-up CT was performed when the patient was
4 years 5 months old using a new photon-counting
detector that had been made available at our institution,
the NAEOTOM Alpha (Siemens Healthineers,
Forchheim, Germany) (Fig. 5). It revealed that themass remnants had not increased in size. Pure-toneaudiometry (Fig. 6) when the patient was 5 years
2 months old revealed conductive hearing loss in theleft ear.

A
Fig. 3CT at 3 years of age. The soft-tissue shadow extended tothe tympanic cavity and the mastoid cells (○).
A

tumor
facialnerve

B
B

Fig. 4(A) A schema of intraoperative ﬁndings of the tumor andthe picture after resection. The facial nerve (△) was shifted
inferiorly. (B) Histology of salivary gland choristoma (SGCh) of the
middle ear. Hematoxylin-eosin staining was performed. Mature
salivary gland tissue with a mixture of serous (○) and mucous (◇)
glands with little atypia was observed, covered by a multilineage
simple columnar epithelium (△).

Fig. 5(A) CT and (B) three-dimensional CT at 4 years 5 monthsof age. Soft-tissue shadows had decreased, but the long processof the incus and the stapes were deformed.

Tominaga et al.

352

Acta Med. Okayama Vol. 78, No. 4

(dB)
-20
-10
0
10
20
hearing level

30
40
50
60
70
80
90
100
110
120
130

125

250

500

1,000 2,000 4,000 8,000 (Hz)frequency

Fig. 6
Standard pure-tone audiometry at 5 years 2 months of age
revealed conductive hearing loss in the left ear. ○, right ear air
conduction; ×, left ear air conduction; ], left ear bone conduction;↓, scale out.

Discussion
Middle-ear SGCh is a rare diagnosis, and prior to
our case, 41 patients have been reported, with only oneof those having bilateral lesions; the total number ofcases comes to to 43, as shown in Table 1 [1-43]. Theage at diagnosis ranged from 9 months to 52 years, andthe majority of patients were female. In 39/40 cases,
this benign tumor was unilateral (97.5%), with the left
side being the most commonly affected (26 cases,
65.0%). SGChs are most common in the posterior pituitary, periparotid lymph nodes, and middle ear, butthey have also been reported in the external auditory
canal, thyroid gland, mediastinum, prostate, vulva,
rectum, buccal muscle, larynx, optic nerve, and internal auditory canal [44-46]. Although the pathogenesis
of SGCh remains controversial, many reports have suggested that abnormal differentiation of the first andsecond branchial arches during the fetal period may
cause this condition [9]. Moreover, middle-ear malformations and inferior shifts of the facial nerve due toaberrant deviations of the facial nerve canal may becaused by this tumor [27]. There are also cases accom-

panied by inner ear malformation [43], though this was
not observed in the present case.
Important clinical findings include an abnormalroute of the facial nerve in the tympanic chamber andossicular anomalies. Deficiencies or deformities of theincus have been reported in 29 cases (82.9%) and thoseof the stapes in 28 cases (84.8%). Deformities of themalleus were present in 7 cases (29.2%). Typical symptoms of this tumor include conductive hearing loss,
otorrhea, and tinnitus [47]. Otoscopic examination inthis case suggested a mass behind the intact, dull,
opaque tympanic membrane. CT revealed the tumor asa soft-tissue shadow with low absorption. The middle-ear mass must be differentiated from congenitalcholesteatoma as well as from a dermoid cyst, teratoma,
glomus tympanicum, granuloma, neuroma, and glioma, all of which manifest as unilateral hearing loss
without perforation of the tympanic membrane [11].
Despite its rarity, physicians should be aware of thiscondition to enable an early diagnosis and informed
decision-making in conjunction with parents.
A definitive diagnosis of middle-ear SGCh is possible only upon histopathological examination, and surgical resection is the only treatment. No malignant
transformation of this tumor was observed among the
43 reported cases (Table 1), and some authors have suggested that complete tumor excision is not an absolute
requirement [29 , 48]; however, others have proposedthat a small chance of malignant transformation exists,
which warrants complete excision of the tumor [25].
Reports of diagnoses of middle-ear SGCh are rare
because of the tumor’s rarity and the high risk of complications owing to its unique location behind the tympanum and close to the facial nerve, which causes adownward shift of the horizontal facial nerve. The useof a nerve stimulator during surgery is strongly recommended to mitigate the risk of facial nerve palsy[48 , 49].
In the present case, we performed surgery for a
middle-ear SGCh that was diagnosed via CT and MRI
during follow-ups after NHS-based referral. Its removal
was necessary to rule out the possibility of cholesteatoma or malignancy. Long-term follow-up of patientswith hearing loss discovered via NHS, subsequent CT
and MRI, and surgical resection may be useful for early
diagnosis and treatment. In addition, facial nerve
damage was successfully avoided in this case by using anerve stimulator, even though the facial nerve was dis-Rare Case of Middle-Ear Choristoma

August 2024
Table 1
ReferenceNo

353

Reported cases of middle-ear salivary gland choristoma
Author

Sex

Side

Age

Malleus

[1]
[2]
[3]

Chen S et al.
Jancikova J et al.
Toros SZ et al.

F
F
F

L
R
R

6Y
10 Y
7Y

Normal

[4]
[5]
[6]
[7]

Supiyaphun P et al.
Ziari K et al.
Ha SL et al.
Morimoto N et al.

F
M
F
M

L
L
R
L

10 Y
39 Y
3Y
13 Y

Malformed
NormalNormalNormal[8]

Bottrill ID et al.

M

R

5Y

[9]
[10]
[11]

Yamanobe Y et al.
Hinni ML et al.
Mohd Saiful AI et al.

F
M
F

L
R

8Y
10 Y
1Y

[12]

Enoz M et al.

F

L

14 Y

[13]

Young A et al.

F

L

14 Y

Normal

[14]
[15]
[16]
[17]

Yazici D et al.
Boleas-Aguirre MS et al.
Raghavan V et al.
Purnell PR et al.

F
M
M
M

L
L
L
L

32 Y
12 Y
6Y
6Y

NormalNormalNormal
Malformed[18]

Su QY et al.

F

L

8Y

[19]
[20]
[21]

Steﬀen TN et al.
Fois P et al.
Buckmiller LM et al.

F
F
M

R
R

52 Y
22 Y
9M

Partialdeﬁciencyof head[22]

Lee DK et al.

M

R

11 M

[23]
[24]
[25]

Amrhein P et al.
DvoryanchikoVV et al.
Ookouchi Y et al.

F
F
F

R
L
R

[26]

Namdar I et al.

F

L

5Y

[27]

Kartush JM et al.

F

R

19 Y

[28]
[29]
[30]
[31]

Moore PJ et al.
Cannon CR
Mischke RE et al.
Abadir WF et al.

F
F
F
F

L
R
L
L

5Y
24 Y
9Y
10 Y

Normal
Malformed
Normal[32]

Perry BP et al.

M

L

3Y

Normal[33]

Hociota D et al.

M

L

50 Y

[34]
[35]
[36]

Wine CJ et al.
Cejas Mendez DL et al.
Quaranta A et al.

M
F
M

L
L
L

3Y

[37]
[38]
[39]
[40]

Bruner RC
Anderhuber W et al.
Grachev NS et al.
Peron DL et al.

F
M
M

6Y
4Y
8Y
20 Y

[41]
[42]

Simoni P et al.
Nigauri et al.

F
M

R
L
L
Bilateral
L
R

[43]

Yoshimura et al.

F

L

Malformed

Malformed
NormalNormalNormalIncus

Malformed
Partial deﬁciency oflong process andlenticular process
Malformed
NormalNormalPartial deﬁciency oflong process
Malformed

Malformed
Partial deﬁciency oflong process

Partial deﬁciency oflong process
Normal
Deﬁciency
Normal
Malformed
Partial deﬁciency oflong process
Malformed
Malformed

Malformed longprocess
Mlalformed
Absence oflenticular process
Absence oflenticular process
Absence oflenticular process
Deﬁciency
Malformed
Malformed
Deﬁciency
Floating loosely intympanic cavity
Partial deﬁciency oflong process
NormalPartial deﬁciency oflong process

Stapes

Deﬁciency

Malformed
Erosion of tumor
NormalAbsence ofsuperstructure
Absence ofsuperstructure
Malformed
Deﬁciency
Absence of thesuperstructure
Absence ofsuperstructure
Partial deﬁciency
Partial deﬁciency
NormalAbsence ofsuperstructure
Normal

Malformed
Malformed
Absence ofsuperstructure
Deformity ofsuperstructure
Malformed
Absence ofsuperstructure
Absence ofsuperstructure

IS joint
Dislocation
Dislocation

Dislocation
Dislocation

Malformed

10M
5Y

NormalNormal10 Y

NormalNormalPartial deﬁciency oflong process
Partial deﬁciency oflong process

NormalAbsence ofsuperstructure
Not observed

Hearing
augmentation
repairOCR

Y
N

Dislocation

None

Y

Dislocation

None

N

Dislocation

None

N

Dislocation

None

N

None
None

N

None

N

Dislocation
Dislocation

BAHA

N
N

Dislocation

OCR

OCR

OCR, BAHA
Dislocation

None

Dislocation

Y
Y

None

None

N
Y
Y

Dislocation

Y

Dislocation

Y

Normal

Malformed

None
None

None

Dislocation

Malformed

Y
NNY

Dislocation

Absence ofsuperstructure
Absence ofsuperstructure
Deﬁciency

Malformed
Malformed

None

None

Dislocation

Malformed

Facialnerve
involvedYN
N

N

Deﬁciency
Malformed

Not observed

Recurrenceor tumor
growth
None
None
None

None
Dislocation

None
None

Dislocation
Dislocation

None
None

OCR

Y
N

Y
N

OCRmischNYYNo, number; M, male; F, female; L, left; R, right; Y, year; M, month; IS joint, incus stapes joint; OCR, ossicular chain reconstruction; BAHA, bone-anchoredhearing aids; empty cell, no description

354

Tominaga et al.

Acta Med. Okayama Vol. 78, No. 4

covered in the vicinity of the tumor. During surgery,
we identified a deficiency in the long process of theincus and the stapes and an inferior shift of the facial
nerve, as reported in numerous previous cases. Only asmall shadow in the middle ear was observed upon CT
ten months after removal of the mass.
Since the view of the stapes footplate was occluded,
the risk of facial nerve palsy after second-look operationis considered to be relatively high in this case. At present, we are carefully monitoring the progress of thetumor and considering future reconstruction of theossicles via tympanoplasty to mitigate the patient’s conductive hearing loss.
Acknowledgments. We would like to thank Editage (www. editage.
com) and KN International for English language editing. This work was
supported by AMED (grant number:23dk0310122h0001).

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