Cholesteatoma Secondary to Temporal Bone Involvement by Langerhans Cell Histiocytosis

Roger, Gilles; Dupré, Marie; Leboulanger, Nicolas; Godey, Benoît; Thomas, Caroline; Gandemer, Virginie; Bordure, Philippe; Garabédian, Éréa-Noël; Donadieu, Jean

doi:10.1097/mao.0b013e318194f9b2

Cited by 12 publications

(4 citation statements)

References 10 publications

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“…“Vertebra plana” per se is not an indication for an orthopedic corset, and expert physiotherapy assessment should be considered; however, temporary immobilization may be required for symptomatic relief in the early phases of vertebral involvement. Patients with temporal bone lesions and recurrent otorrhea, may have a secondary cholesteatoma which may need specific treatment 21.…”

Section: Methodsmentioning

confidence: 99%

Langerhans cell histiocytosis (LCH): Guidelines for diagnosis, clinical work‐up, and treatment for patients till the age of 18 years

Haupt¹,

Minkov²,

Astigarraga³

et al. 2012

Pediatric Blood & Cancer

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567

693

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These guidelines for the management of patients up to 18 years with Langerhans cell histiocytosis (LCH) have been set up by a group of experts involved in the Euro Histio Net project who participated in national or international studies and in peer reviewed publications. Existing guidelines were reviewed and changed where new evidence was available in the literature up to 2012. Data and publications have been ranked according to evidence based medicine and when there was a lack of published data, consensus between experts was sought. Guidelines for diagnosis, initial clinical work-up, and treatment and long-term follow-up of LCH patients are presented.

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Section: Methodsmentioning

confidence: 99%

Langerhans cell histiocytosis (LCH): Guidelines for diagnosis, clinical work‐up, and treatment for patients till the age of 18 years

Haupt¹,

Minkov²,

Astigarraga³

et al. 2012

Pediatric Blood & Cancer

Self Cite

567

693

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“…In 2009, Roger et al 6 were the first to describe 3 cases of secondary acquired cholesteatoma in patients previously treated for temporal bone LCH. Their 3 cases had a bony defect in the posterior ear canal wall secondary to LCH involvement.…”

Section: Discussionmentioning

confidence: 99%

Cholesteatoma Masquerading as Recurrent Langerhans Cell Histiocytosis

Gendre¹,

Dréno²,

Boyer³

et al. 2023

Int Adv Otol

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Langerhans cell histiocytosis is a rare condition affecting the temporal bone in up to 60% of cases. Symptoms are non-specific and the differential diagnosis includes infection, benign lesions such as cholesteatoma, and malignant lesions of the skull base. Here, we report the case of a 14-year-old child referred with chronic ear discharge, and background of multifocal Langerhans cell histiocytosis 9 years prior. Recurrence of Langerhans cell histiocytosis was initially suspected and systemic treatment was considered. Further imaging workup and surgical exploration of the mastoid showed a secondary acquired cholesteatoma arising from a dehiscent posterior ear canal wall. Surgical removal of the cholesteatoma was performed with a canal wall down procedure. We review the presentation and management of temporal bone Langerhans cell histiocytosis. We recommend that cholesteatoma should be considered in case of recurrence of otological symptoms in patients with a background of Langerhans cell histiocytosis.

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“…Cholesteatoma formation in LCH patients is an uncommon occurrence. Cholesteatoma formation appears to occur at either the external auditory canal or mastoid in about 2-3 years after a patient is diagnosed with LCH [ 3 ]. Patients with LCH are no more likely to be diagnosed with cholesteatoma in comparison to the general population.…”

Section: Introductionmentioning

confidence: 99%

Secondary Acquired Cholesteatoma in Langerhans Cell Histiocytosis Patient

Alhelali¹,

Alsulami²,

Qasim³

et al. 2022

Cureus

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Langerhans cell histiocytosis (LCH) is a multi-faceted disease defined by the accumulation of dendritic cells in various organs with characteristics similar to the epidermal Langerhans cells and can affect any organ of the body. It is most commonly seen in young adults and children. Cholesteatoma is a congenital or acquired condition and is categorized into primary and secondary cholesteatomas. Only a few reported cases of primary or secondary cholesteatoma have been reported among patients treated for temporal bone LCH. We report a case of secondary acquired cholesteatoma in a six-year-old girl after five years of her LCH treatment. The patient initially presented with ear discharge and aural polyp which did not improve with medical management. A computed tomography scan of the mastoid showed a left middle ear cavity and temporal bone lesion with bony erosions and total obliteration of the left external auditory canal. The patient underwent multiple biopsies, and a histopathological evaluation confirmed the diagnosis of cholesteatoma. The diagnosis of LCH can be difficult due to variable clinical manifestations. Involvement of the ear as resemblance to other diseases such as mastoiditis and chronic otitis media are quite common. Computed tomography scan and biopsy are reliable tools for diagnosis. Cholesteatoma following LCH remains a rare entity, hence, critical examination at follow-up visits is needed. Surgery remains the treatment of choice for cholesteatoma patients.

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Cholesteatoma Secondary to Temporal Bone Involvement by Langerhans Cell Histiocytosis

Cited by 12 publications

References 10 publications

Langerhans cell histiocytosis (LCH): Guidelines for diagnosis, clinical work‐up, and treatment for patients till the age of 18 years

Langerhans cell histiocytosis (LCH): Guidelines for diagnosis, clinical work‐up, and treatment for patients till the age of 18 years

Cholesteatoma Masquerading as Recurrent Langerhans Cell Histiocytosis

Secondary Acquired Cholesteatoma in Langerhans Cell Histiocytosis Patient

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