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Rare case of salivary gland choristoma originating in inner ear in

Rare case of salivary gland choristoma originating in inner ear in

Salivary gland choristoma (SGCh) is a rare benign tumor reported
in several unusual sites, such as the gastrointestinal tract, the optic nerve,
and the internal auditory canal, but never reported in the inner ear. It is a rare benign tumor characterized by a
histopathological finding of normal salivary tissue, mucous and mixed
predilection, and occasionally, fibrosis . SGCh is distinguished from ectopic
salivary glands which are present in abnormal locations without fibrous
capsules .

In a recent development, a team of doctors
under Yoshiharu Yamanobe, have reported an extremely rare case of a large inner
ear SGCh extending into the middle ear and the internal auditory canal (IAC),
with congenital unilateral profound hearing loss and ipsilateral progressive
facial nerve palsy.The case has been reported in Journal of Otolaryngology – Head & Neck Surgery.

The primary sites of SGCh have been reported in
several organs, such as the gastrointestinal tract, the optic nerve, and the
internal auditory canal . SGCh in the middle ear can sometimes be accompanied
by an aberrant facial nerve and middle ear malformations. “We had
reasons to consider that the inner ear was the primary site in this case.
First, the patient had a congenital profound hearing loss. Second, no previous
abnormalities in the tympanic membrane had been detected. Third, imaging
studies suggested that the tumor was mainly located in the inner ear.”the team
elaborated.

An 8-year-old girl with a history of left profound congenital
hearing loss presented to the department of Otolaryngology-Head and Neck
Surgery, Keio University School of Medicine, Japan, with ipsilateral progressive severe facial nerve palsy
(House-Brackmann Grade VI).

Clinical examination revealed the left tympanic membrane was
swollen with a pulsatile tumor. Radiological investigations revealed a
multilocular tumor in the inner ear extending into the middle ear and internal
auditory canal (IAC).

Suspecting a diagnosis of
salivary gland choristoma, the team performed a partial resection of the tumor
by transmastoid approach to preserve the anatomical structure of the facial
nerve. The tumor was pathologically diagnosed as SGCh. Anatomical preservation of the facial nerve
was considered significant to retain any chances for recovery of the facial
palsy after surger. No intraoperative or postoperative complication such as
spinal fluid leakage was observed.

Two years after surgery,
her facial function recovered to House-Brackmann Grade II and the residual
tumor did not show regrowth on MRI.

“Although the natural course of this rare tumor is unknown, a
partial resection is an acceptable treatment procedure when functional recovery
of the facial nerve is anticipated.”the team of physicians concluded. The
residual tumor needs to be carefully monitored in future using MRI, and total
removal of the tumor through transotic approach may be necessary if there is an
increase in the size of the residual tumor.”the team concluded.

For full article follow the link: https://doi.org/10.1186/s40463-021-00511-3

Source:
Journal
of Otolaryngology – Head & Neck Surgery

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