TY - JOUR
T1 - Labyrinthine Sequestrum
T2 - A Case Report and Review of the Literature
AU - Guerin, Julie B.
AU - Vork, DIana L.
AU - Eguiguren, Lourdes
AU - Marston, Alexander P.
AU - Driscoll, Colin L.W.
AU - Carlson, Matthew L.
AU - Henry, Nancy K.
AU - Lane, John I.
N1 - Funding Information:
Address correspondence and reprint requests to Julie B. Guerin, M.D., Department of Diagnostic Radiology, Mayo Clinic, 200 First Street SW, Rochester, MN 55905; E-mail: [email protected] Financial support: This work was not funded by any agency or grant. The authors disclose no conflicts of interest.
Publisher Copyright:
© 2018, Otology & Neurotology, Inc.
PY - 2018/3/1
Y1 - 2018/3/1
N2 - Objective: To report the presentation, diagnosis, management, and convalescence of labyrinthine sequestrum (LS) and summarize all previously published cases. Patient(s): Eleven-year-old female with LS. Intervention(s): Multidisciplinary diagnostic evaluation and treatment. Main Outcome Measures: Imaging and laboratory findings, medical and surgical treatment. Results: We describe a case of LS secondary to medically recalcitrant suppurative otitis media in an 11-year-old female and review all eight previously reported cases. The index patient presented after 6 months of otitis media, profound unilateral hearing loss, with symptoms suggesting meningitis. Temporal bone CT demonstrated marked bony destruction of the left otic capsule. Gadolinium-enhanced MRI showed an enhancing process with evidence of meningitis and subdural empyema. The patient was treated with surgical debridement and culture directed antibiotic therapy. Posttreatment imaging showed resolution of intracranial infection with fibrous bony healing of the otic capsule resembling fibrous dysplasia. Conclusion: LS is a rare form of labyrinthitis characterized by centrifugal destruction of the otic capsule. The current index case highlights the importance of combined medical and surgical treatment and describes for the first time in the literature the fibrous ossification of the otic capsule following disease resolution.
AB - Objective: To report the presentation, diagnosis, management, and convalescence of labyrinthine sequestrum (LS) and summarize all previously published cases. Patient(s): Eleven-year-old female with LS. Intervention(s): Multidisciplinary diagnostic evaluation and treatment. Main Outcome Measures: Imaging and laboratory findings, medical and surgical treatment. Results: We describe a case of LS secondary to medically recalcitrant suppurative otitis media in an 11-year-old female and review all eight previously reported cases. The index patient presented after 6 months of otitis media, profound unilateral hearing loss, with symptoms suggesting meningitis. Temporal bone CT demonstrated marked bony destruction of the left otic capsule. Gadolinium-enhanced MRI showed an enhancing process with evidence of meningitis and subdural empyema. The patient was treated with surgical debridement and culture directed antibiotic therapy. Posttreatment imaging showed resolution of intracranial infection with fibrous bony healing of the otic capsule resembling fibrous dysplasia. Conclusion: LS is a rare form of labyrinthitis characterized by centrifugal destruction of the otic capsule. The current index case highlights the importance of combined medical and surgical treatment and describes for the first time in the literature the fibrous ossification of the otic capsule following disease resolution.
KW - Labyrinthitis
KW - Otitis media
KW - Pediatric
KW - Sequestrum
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U2 - 10.1097/MAO.0000000000001676
DO - 10.1097/MAO.0000000000001676
M3 - Review article
C2 - 29337715
AN - SCOPUS:85042487404
SN - 1531-7129
VL - 39
SP - 340
EP - 343
JO - Otology and Neurotology
JF - Otology and Neurotology
IS - 3
ER -