Intramembranous and Mesotympanic Cholesteatomas Associated with an Intact Tympanic Membrane in Children

Sobol, Steven M.; Stroud, Malcolm H.; Reichert, Timothy J.; Spector, Gershon J.; Faw, Kenneth D.; Ogura, Joseph H.

doi:10.1177/000348948008900404

Cited by 30 publications

(23 citation statements)

References 11 publications

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“…Several variations of the invagination theory have been proposed, 12,13 most based on Ruedi's 14 suggestion that otitis media causes an inflammatory injury of the tympanic membrane that invaginates into the middle ear to form a CC. A review of the literature fails to reveal any buttressing histologic or clinical evidence for this postulate.…”

Section: Invaginationmentioning

confidence: 99%

The Natural History of Congenital Cholesteatoma

Koltai¹,

Nelson²,

Castellon³

et al. 2002

Arch Otolaryngol Head Neck Surg

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Congenital cholesteatoma appears to have a predictable trajectory of growth, starting as a small pearl in the middle ear, eventually growing to involve the ossicles and mastoid, and causing varying degrees of destruction and functional impairment. The clinical picture of a young child with otorrhea, conductive hearing loss, tympanic membrane perforation in a nontraditional location, and a mastoid filled with cholesteatoma may represent the end point in the natural history of CC, despite the fact that this type of lesion is outside the accepted definition of CC.

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Section: Invaginationmentioning

confidence: 99%

The Natural History of Congenital Cholesteatoma

Koltai¹,

Nelson²,

Castellon³

et al. 2002

Arch Otolaryngol Head Neck Surg

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“…Table 2 summarizes the data found in the English literature [5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22][23][24] .…”

Section: Discussionmentioning

confidence: 99%

“…According to the papillary proliferation theory [27,28] , cholesteatoma of TM may develop by epithelial proliferation in response to an inflammatory process, as in the case of previous otitis. This theory is supported by some authors based on the evidence of recurrent otitis media in the children they presented [7,8] , but according to Casale's definition [23] , the evidence of a previous inflammatory process makes doubtful the congenital nature of the mass.When cholesteatoma develops in subjects without any history of a previous inflammatory process of the external or middle ear, like in the case reported here, an embryologic origin is deeply suspected. This could be explained by the persistence of an epidermoid formation (derivative of the first branchial groove) that normally regresses after the 33 rd week of gestation [3,[29][30][31] .…”

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confidence: 93%

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confidence: 93%

“…RCT: randomized controlled trial • a white mass inserted into a normal pars tensa, without connection with the pars flaccida and/or promontorium; • normal pars flaccida; • no prior history of otorrhea or TM perforation; • no previous otologic procedures or trauma. Five studies [6,7,11,13,20] described a total of 14 cases of 36 pediatric patients with a history of recurrent otitis media; however, they were classified as "congenital" because they did not show anamnestic records of otorrhea, TM perforation, previous otologic procedures, or ear trauma. In all the living adult case's ITMCs, we did not find any otologic history (or previous otitis media episodes) and no inflammatory processes were found intraoperatively.…”

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confidence: 99%

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Congenital Intratympanic Cholesteatoma in an Adult Patient: A Case Report and Review of the Literature

Pedruzzi

Mion²,

Comacchio³

2016

Int Adv Otol

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Congenital cholesteatoma within the tympanic membrane is an uncommon entity, with only few cases being documented. The aetiopathogenesis of this lesion is still unknown; however, when cholesteatoma develops in subjects without any history of previous ear inflammation, as in the case we report here, an embryologic origin is deeply suspected. An acquired origin is hypothesized in patients with a previous history of an inflammatory process of the external or middle ear because of the proliferation of the basal cell layer of the tympanic membrane epithelium. We report a rare case of congenital cholesteatoma of the tympanic membrane in an adult patient and review the literature KEYWORDS: Congenital intratympanic cholesteatoma, intratympanic cholesteatoma, congenital cholesteatoma of the tympanic membrane, cholesteatoma of the tympanic membrane Barbara Pedruzzi, Marta Mion, Francesco ComacchioDepartment of Otolaryngology, Azienda Ospedaliera di Padova, Padova, Italy Case ReportIn 1936, Teed [3] cited the first five cases of ITMC to support the theory of a congenital origin of cholesteatomas. From the English literature, he cited the first case reported by Hinton in 1863 [4] and one other case.We identified a list of potential citations for inclusion in this review, following which the titles and abstracts on this list were screened by two independent reviewers.All the retrieved full-texts were included in the review by consensus of all the authors ( Figure 5).As a quality assessment strategy, the included studies were methodologically appraised according to the National Institute for Health and Clinical Excellence's levels of evidence (Table 1): all the studies could be categorized as evidence level 3.We analyzed the data from these cases, including our own case, to study their demographic details, and clinical features and management, for a total of 42 patients and 20 articles, including our own research (Table 2). Table 2 summarizes the data found in the English literature [5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22][23][24] . 120J Int Adv Otol 2016; 12(1): 119-24 The age range was 5 months to 57 years, with 27 (64.3%) cases involving patients less than 4 years of age, out of which seven (16.7%) were 1-year old or less. There was a history of one or more episodes of otitis media (n=14) when ITMC was noticed on otoscopy. The lesion was first noted by pediatricians (n=14), by general practitioners (n=4), or by otolaryngologists (n=16). In three cases, the analyses were conducted post-mortem during autoptic exams [9] . This highlights the need for routine otoscopy by primary care physicians to detect abnormalities and make an early referral to otolaryngologists. ITMC should always be considered in the differential diagnosis of white lesions in TM. Otomicroscopy can help differentiate ITMC from tympanosclerosis. Only in one case had the lesion occupied the whole TM, while the umbo was involved in nine patients; this region is the most depressed part of TM, where the manubrium of the malleus is fir...

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