Two cases of spontaneous regression of congenital cholesteatomas

Kodama, Kozue; Hara, Mariko; Hasegawa, Masayo; Matsuzawa, Shingo; Shinnabe, Masahiro; Kanazawa, Hiromi; Yoshida, Naohiro; Iino, Yukiko

doi:10.1016/j.ijporl.2011.07.008

Cited by 12 publications

(7 citation statements)

References 9 publications

(8 reference statements)

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“…Furthermore, authors tried to differentiate subtypes/phenotypes of C basing on index of severity such as age of onset, uni- or bi-lateral disease, signs of local aggressiveness, and relapsing, missing a possible link with different pathophysiological mechanisms underlying the clinical manifestations because endotypes are not yet known. For the same reason, it is difficult to predict the clinical behavior of C, which may vary from a slow-progressing benign diseases to more aggressive disease [ 13 ]. Consequently, determining an appropriate follow-up period duration for patients with C is critical, and good prognostic biomarkers are needed in selecting patients who will require immediate surgical intervention [ 8 ].…”

Section: Introductionmentioning

confidence: 99%

Endotyping of Cholesteatoma: Which Molecular Biomarkers? A Systematic Review

Cantone

Nola

Corso

et al. 2022

JPM

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Background: So far, no medical treatment is available for cholesteatoma (C) and the only effective therapy is complete surgical removal, but recurrence is common even after surgical treatment. While C is classically divided into two clinical phenotypes, congenital and acquired, only a few studies have focused on its potential biomarkers. This study aims to revise the literature to identify which biomarkers can define the endotype of C. Methods: We conducted a systematic review in accordance with the Preferred Reporting Items for Systematic Review and Meta-Analysis (PRISMA) process to identify published experimental articles about molecular biomarkers in C. Results: KGF and its receptor, MMP-9, KRT-1, KRT-10, and MIF might be considered biomarkers of recurrence, whereas Ki-67, TLR-4, RANKL, IL17, MMP-2, MMP-9, IL6, TNF-α, should be considered more specifically as biomarkers of bony erosion. Conclusions: These results are interesting especially from a prognostic point of view, nevertheless more studies are needed to search new biomarkers of C that could completely change not only the therapeutic standards of the disease, but also the clinical history of C itself in the era of precision medicine.

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Section: Introductionmentioning

confidence: 99%

Endotyping of Cholesteatoma: Which Molecular Biomarkers? A Systematic Review

Cantone

Nola

Corso

et al. 2022

JPM

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“…Koltai et al reported two cases in which the lesion had significantly extended beyond the tympanic membrane and into the external auditory canal [ 9 ]. In addition, there have been reports of cases in which CC disappeared spontaneously [ 25 ]; therefore, there is a possibility that latent patients may be cured without being diagnosed or treated. In any case, the calculated incidence of CC may be considered lower than the actual number, as the number of cases may be underestimated but not overestimated.…”

Section: Discussionmentioning

confidence: 99%

Epidemiology of Congenital Cholesteatoma: Surveys of the Last 17 Years in Japan

Kadowaki,

Ide,

Nakamura

et al. 2024

JCM

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(1) Background: The incidence of congenital cholesteatoma (CC) has rarely been discussed, particularly from a demographic viewpoint. Therefore, we conducted an epidemiological study of CC using local medical characteristics. (2) Methods: The participants were 100 patients (101 ears) who underwent initial surgical treatment at university hospitals in two rural prefectures between 2006 and 2022. A total of 68% of the patients were males and 32% were females, with a median age of 5 years. We reviewed the medical records for the date of birth, date of surgery, stage of disease, and first symptoms of the disease. (3) Results: The total incidence of CC was calculated to be 26.44 per 100,000 births and tended to increase. No significant difference was found between the incidences in the two prefectures. The number of surgeries performed was higher in the second half of the study period. No difference in the stage of progress was observed based on age. (4) Conclusions: The incidence of CC was estimated to be 26.44 per 100,000 newborn births. The number of patients with CC tended to increase; however, this can be attributed to an increase in the detection rate rather than the incidence.

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“…A limitation of this study is that some patients may not have been included; for example, patients who moved out before being diagnosed (since this study was conducted in a rural area, there must be more people moving out than moving in), patients who were referred to hospitals in other larger cities, and patients whose disease progressed undiagnosed and could no longer be distinguished from acquired cholesteatoma. In addition, there have been reports of cases in which CC disappeared spontaneously [12]; therefore, there is a possibility that latent patients may be cured without being diagnosed or treated. In any case, the calculated incidence of CC may be considered lower than the actual number, as the number of cases may be underestimated but not overestimated.…”

Section: Discussionmentioning

confidence: 99%

Epidemiology of Congenital Cholesteatoma

Kadowaki,

Ide,

Nakamura

et al. 2023

Preprint

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Background The incidence of congenital cholesteatoma (CC) has rarely been discussed, particularly from the demographic viewpoint. Therefore, we conducted an epidemiological study of CC using local medical characteristics. Materials and methods The participants were 100 patients (101 ears) who underwent initial surgical treatment at university hospitals in two rural prefectures between 2006 and 2022. Sixty-eight percent of the patients were males and 32% were females, with a median age of 5 years. We reviewed the medical records for the date of birth, date of surgery, stage of disease, and first symptom of the disease. Result The total incidence of CC was calculated to be 26.44 per 100,000 births and tended to increase. No significant difference was found between the incidences in the two prefectures. The number of surgeries performed was higher in the second half of the study period. No difference in the stage of progress was observed based on age. Conclusion The incidence of CC was estimated to be 26.44 per 100,000 new-born births. The number of patients with CC has tended to increase; however, this can be attributed to an increase in the detection rate rather than the incidence.

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Two cases of spontaneous regression of congenital cholesteatomas

Cited by 12 publications

References 9 publications

Endotyping of Cholesteatoma: Which Molecular Biomarkers? A Systematic Review

Endotyping of Cholesteatoma: Which Molecular Biomarkers? A Systematic Review

Epidemiology of Congenital Cholesteatoma: Surveys of the Last 17 Years in Japan

Epidemiology of Congenital Cholesteatoma

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