| Cholesteatoma secondary to temporal bone involvement by Langerhans cell histiocytosis: a complication amenable to curative surgery. | |
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MedLine Citation:
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PMID: 19169133 Owner: NLM Status: MEDLINE |
Abstract/OtherAbstract:
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OBJECTIVES: To describe secondary acquired cholesteatoma in patients previously treated for Langerhans cell histiocytosis (LCH). To focus on misleading symptoms suggesting LCH relapse. PATIENTS: This study involved 3 patients aged 12, 20, and 58 months whose conditions were diagnosed with LCH. All 3 had involvement of the mastoid and of the skin of the external ear canal (EAC) at diagnosis. They were treated with steroids and vinblastine. INTERVENTIONS: Serial computed tomographic (CT) scans and clinical follow-up. MAIN OUTCOME MEASURE: Exploratory surgery of the mastoid. RESULTS: Otorrhea recurred in all 3 patients at 24, 17, and 26 months, respectively, with difficulties to clinically identify a hole in the posterior part of the EAC. The otorrhea was considered a sign of a new occurrence of LCH, leading to systemic chemotherapy in 2 cases. A CT scan showed a defect in the posterior wall of the EAC and suggested cholesteatomatous invasion of the mastoid (2 true cholesteatomas and 1 precholesteatomatous case).Surgery (canal wall up mastoidectomies) successfully removed the cholesteatoma (bilateral in 2 cases) and reconstructed the bony defect using cartilage. Biopsies ruled out LCH recurrence. CONCLUSION: Secondary acquired cholesteatoma (through a bony defect of the EAC) may occur in patients previously treated for LCH. Recurrence of symptoms and bone destruction on CT may wrongly suggest LCH recurrence. Surgery allows removal of the cholesteatoma as well as EAC reconstruction and control biopsy. |
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Authors:
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Gilles Roger; Marie Dupré; Nicolas Leboulanger; Benoit Godey; Caroline Thomas; Virginie Gandemer; Philippe Bordure; Erea-Noel Garabedian; Jean Donadieu |
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Publication Detail:
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Type: Case Reports; Journal Article; Research Support, Non-U.S. Gov't |
Journal Detail:
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Title: Otology & neurotology : official publication of the American Otological Society, American Neurotology Society [and] European Academy of Otology and Neurotology Volume: 30 ISSN: 1537-4505 ISO Abbreviation: Otol. Neurotol. Publication Date: 2009 Feb |
Date Detail:
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Created Date: 2009-01-26 Completed Date: 2009-04-16 Revised Date: - |
Medline Journal Info:
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Nlm Unique ID: 100961504 Medline TA: Otol Neurotol Country: United States |
Other Details:
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Languages: eng Pagination: 190-3 Citation Subset: IM |
Affiliation:
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ENT Department, Armand Trousseau Children's Hospital, University Paris VI, AP-HP, Paris, France. gilles.roger@trs.aphp.fr |
Export Citation:
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| MeSH Terms | |
Descriptor/Qualifier:
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Antineoplastic Agents, Phytogenic
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therapeutic use Cerebrospinal Fluid Otorrhea / etiology Child, Preschool Cholesteatoma, Middle Ear / etiology*, surgery* Ear Canal / pathology Female Histiocytosis, Langerhans-Cell / complications*, drug therapy, pathology* Humans Infant Male Mastoid / pathology Otologic Surgical Procedures* Steroids / therapeutic use Temporal Bone / pathology* Tomography, X-Ray Computed Treatment Outcome Vinblastine / therapeutic use |
| Chemical | |
Reg. No./Substance:
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0/Antineoplastic Agents, Phytogenic; 0/Steroids; 865-21-4/Vinblastine |
From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine
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