Histiocytosis X: otologic presentations

DeMarino, David P.; Dutcher, Paul O.; Parkins, Charles W.; Hengerer, Arthur S.

doi:10.1016/s0165-5876(85)80062-8

Cited by 20 publications

(7 citation statements)

References 14 publications

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“…According to the histiocytosis study group, Langerhans cell histiocytosis was divided into three groups in 1987. LCH consists of three separate diseases including eosinophilic granuloma (EG), Hand-Schuller Christian disease and Letterer-Siwe disease [11,12]. Hand-Schuller Christian disease progresses with diabetes insipidus, exophthalmos, and lytic lesions and is observed in 15 -40% of patients.…”

Section: Discussionmentioning

confidence: 99%

Management of eosinophilic granuloma localized in the pelvis and extremities

Öztürk¹

2020

Ann Clin Anal Med

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Aim: Given the benign biology, and clinical course of eosinophilic granuloma and its higher incidence in pediatric patient group, a successful treatment plan with lower risk for complications is desired. In this study, we aimed to evaluate clinical characteristics, treatment, and outcomes of patients diagnosed with eosinophilic granuloma localized in the pelvis and extremities. Material and Method: Clinical findings, treatment methods, clinical outcomes and treatment failures were evaluated in a total of 19 patients (6 males, 13 females, mean age: 16.5 years, range: 1-37 years) diagnosed with eosinophilic granuloma localized in the pelvis and extremities between 2002 and 2016. Results: The most common mass localization was acetabulum (15%, n = 3). The mean tumor size was 3,7 cm (range: 1-8 cm). All patients presented with pain and had primary tumor. All patients in the study had solitary bone lesion. The mean duration of postoperative follow-up was 46 months (range: 24-95 months). Three patients underwent biopsy alone, and followed-up with adjuvant therapies. In 14 patients, curettage of the lesion was performed at single session during biopsy, and grafting was applied in six of these patients. Two patients underwent excision of the lesion. None of the patients developed recurrence, while a new lesion was detected in the cervical vertebra in one patient after treatment of acetabulum localized lesion. Discussion: The management of eosinophilic granuloma localized in the pelvis and extremities takes place in a wide spectrum varying from observation alone, to lesion curettage, chemotherapy and radiotherapy applications, and prophylactic administration, and treatment tailored for each patient should be planned with a multidisciplinary approach.

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Section: Discussionmentioning

confidence: 99%

Management of eosinophilic granuloma localized in the pelvis and extremities

Öztürk¹

2020

Ann Clin Anal Med

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“…Most LCH-related lytic lesions of the mastoid heal completely, with bone repair and complete reconstitution (4,10,11).…”

Section: Discussionmentioning

confidence: 99%

Cholesteatoma Secondary to Temporal Bone Involvement by Langerhans Cell Histiocytosis

Roger¹,

Dupré²,

Leboulanger³

et al. 2009

Otology &Amp; Neurotology

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“…Although the 2013 Histiocyte Society guidelines mention the use of topical steroids for skin lesions, some studies did note use of intralesional or topical steroids for localized otologic disease. 5,8,19,[22][23][24][25][26][27][28][29][30]65 In addition, 5 studies demonstrated the promising effectiveness of topical nitrogen mustard ear drops in 11 of 14 patients; however, larger studies need to be performed. 8,24,[27][28][29] At the present time, topical nitrogen mustard may be considered in treating otologic disease refractory to steroids.…”

Section: Managementmentioning

confidence: 99%

“…5,8,19,[22][23][24][25][26][27][28][29][30]65 In addition, 5 studies demonstrated the promising effectiveness of topical nitrogen mustard ear drops in 11 of 14 patients; however, larger studies need to be performed. 8,24,[27][28][29] At the present time, topical nitrogen mustard may be considered in treating otologic disease refractory to steroids. 8 Treatment can allow reossification of lytic lesions as early as 6 months and lead to improvement in hearing.…”

Section: Managementmentioning

confidence: 99%

Otologic Manifestations of Langerhans Cell Histiocytosis: A Systematic Review

Chen

Ashman

Bojrab

et al. 2021

Otolaryngol.--head neck surg.

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Objective To perform a systematic review to investigate common otologic manifestations of Langerhans cell histiocytosis, the incidence of these findings, methods for diagnosis, as well as medical and surgical management Data Sources PubMed/MEDLINE, Embase, and Cochrane Library. Review Methods A search of PubMed/MEDLINE, Embase, and Cochrane Library for all articles published between 1963 to 2020 was performed with variations and combinations of the following search terms: Langerhans cell histiocytosis, eosinophilic granuloma, Letterer-Siwe, Hand-Schüller-Christian, otitis, otologic, ear. A review of the references of all included articles was also conducted. Results Sixty-two articles encompassing 631 patients met inclusion criteria. Otologic symptoms at presentation were found in 246 (39%) patients in the reported studies with 48% reporting bilaterality. The mean age was 14.8 years with a male predominance (64%). The most common otologic presenting symptom was otorrhea (46%). A majority had the multisystem variant (52%). The most common treatment modalities were chemotherapy (52%), followed by surgery (50%), systemic steroids (45%), and radiotherapy (31%). Surgery was performed in 75.8% with unisystem involvement and in 50.6% with multisystem involvement. The most effective treatments included radiotherapy (56% success rate, 17% of treated patients), systemic steroids (44% success, 20% treated), chemotherapy (41% success, 21% treated), and surgical modalities (36% success, 19% treated). Conclusions Otologic manifestations that occur with the multisystem variant or are at high risk for central nervous system involvement necessitate systemic treatment. For unifocal lesions, surgery is recommended. Lastly, radiotherapy should be reserved for extensive lesions involving vital structures or presenting in older patients.

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Histiocytosis X: otologic presentations

Cited by 20 publications

References 14 publications

Management of eosinophilic granuloma localized in the pelvis and extremities

Management of eosinophilic granuloma localized in the pelvis and extremities

Cholesteatoma Secondary to Temporal Bone Involvement by Langerhans Cell Histiocytosis

Otologic Manifestations of Langerhans Cell Histiocytosis: A Systematic Review

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