A rare disorder mimics otitis media: Langerhans cell histiocytosis of the temporal bone in a child with interstitial pulmonary fibrosis

Sm, Xie; W, Liu; Yy, Xiang; Za, Xiao; Hm, Ren; Aq, Peng; Wu, Wei; Xm, Yang; Dh, Xie; Tf, Yin; Jh, Ren

doi:10.1016/j.amjoto.2014.06.018

Cited by 5 publications

(4 citation statements)

References 28 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Exclusion criteria included articles that were non‐English or not relevant, reported on nonhuman populations, and did not provide access to full text or extraction of relevant data. All studies were assessed for grade of evidence ( Table 1) 1,3–63 and queried for demographics, otologic manifestations, diagnosis, clinical and radiologic findings, as well as medical and surgical management.…”

Section: Methodsmentioning

confidence: 99%

“…The most common otologic symptom was otorrhea, in 46% of patients in this review, which is consistent with previous studies reporting 45% to 55%. 4,11 Conductive hearing is more typical when 45 1 1 1 Yavas et al 62 1 1 1 1 1 1 S Jeunon et al 27 1 1 1 U Meyer et al 46 1 1 Alexander et al 50 1 1 1 1 1 U Arsovic, Tomanovic, and Bukurov 53 1 Bozdemir et al 26 1 1 1 1 1 1 1C Coleman et al 13 1 1 1 1C Gungadeen, Kullar, and Yates 16 1 1 1 Nishal, Modi, and Balar 41 1 1 1 Robison et al 48 1 1 1 1 Abdel-Aziz et al 7 12 10 4 8 10 C, 2 M Skoulakis et al 22 1 1 1 1 1 1 Xie et al 43 1 1 1 1 1 U Ehrhardt et al 44 1 1 Henderson et al 19 1 1 1 1 S Baser et al 18 1 45 1 1 1 V I 1 Yavas et al 62 1 1 1 V I 1 M A Jeunon et al 27 1 1 1 1 VI 1 Meyer et al 46 1 1 Alexander et al 50 1 1 CU Arsovic, Tomanovic, and Bukurov 53 1 1 MA 1 Bozdemir et al 26 1 1 1 1 1M A Coleman et al 13 1 1 1 V I 1 Gungadeen, Kullar, and Yates 16 1 1 AZ 1 1 CU Nishal, Modi, and Balar 41 Robison et al 48 1 1 1 1 M A 1 MT 1 CU Abdel-Aziz et al 7 12 12 MA 12 Skoulakis et al 22 1 1 1 1 1M T Xie et al 43 1 1 1 V I 1 C U Ehrhardt et al 44 1 19 1 1 1 1U S Baser et al 18 1 hearing loss is present, since otic capsule involvement is rare and the disease usually manifests in the middle ear or external auditory canal. 1,4 Occurrence of sensorineural hearing loss may represent otic capsule involvement.…”

Section: Presentationmentioning

confidence: 99%

See 1 more Smart Citation

Otologic Manifestations of Langerhans Cell Histiocytosis: A Systematic Review

Chen

Ashman

Bojrab

et al. 2021

Otolaryngol.--head neck surg.

View full text Add to dashboard Cite

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.

show abstract

Section: Methodsmentioning

confidence: 99%

Section: Presentationmentioning

confidence: 99%

Otologic Manifestations of Langerhans Cell Histiocytosis: A Systematic Review

Chen

Ashman

Bojrab

et al. 2021

Otolaryngol.--head neck surg.

View full text Add to dashboard Cite

show abstract

“…A study was done on the quality of life of parents caring for children with rare diseases. The results of a study on the quality of life of parents caring for children with rare diseases is severely reduced compared to parents of healthy children [5,6,2].There are studies that prove that correct and early diagnosis of rare childhood diseases is very important , as it often has fatal consequences for young families. Even with a known disease, in many countries, the there are intolerably long delays in diagnosis.…”

Section: Diagnosis Of Rare Diseasesmentioning

confidence: 99%

Current Status of Research on Rare Diseases in Children in China

Zhang

Sun²,

Ma³

et al. 2022

IJTDH

View full text Add to dashboard Cite

Objective: In order to understand and study the diagnosis and treatment status of rare diseases in children across the country. Methods: The data information retrieved by keywords of rare diseases in children was sorted and analyzed through the Internet, the Internet, official data and other databases. Results: Rare diseases are very critical, and have high disability and mortality rates, and 70% of them occur in children, rare diseases are receiving more attention. Conclusion: Strengthen the public and medical staff's attention to rare diseases, and improve the diagnosis and treatment technology level of medicine.

show abstract

“…LCH typically presents with a skin rash and/or bone lesion and can affect single or multiple organ systems including marrow, liver, and lungs. Common presentations of a lytic bone lesion secondary to LCH include cervical lymphadenopathy (40.9%) and otitis media (22.7%) [ 3 ]. It can also affect the lungs and the central nervous system (CNS).…”

Section: Introductionmentioning

confidence: 99%

Langerhans Cell Histiocytosis Causing Central Nervous System Vasculitis Leading to Stroke in a Two-year-old Boy: A Case Report

Froelich

Rizk

2019

Cureus

View full text Add to dashboard Cite

Here, the authors present the first documented case of a patient developing central nervous system (CNS) vasculitis secondary to Langerhans cell histiocytosis (LCH) ultimately leading to stroke. LCH is a rare histiocytic disorder affecting males and females equally and typically presents in pediatric patients with a median age of 30 months. Presentation of the disease can be single-site or multisystem; and, classification of treatment is further demarcated by high risk and low risk depending on the organ systems involved. Treatment of LCH typically involves vinblastine and prednisone, as well as salvage treatment as needed.

show abstract

A rare disorder mimics otitis media: Langerhans cell histiocytosis of the temporal bone in a child with interstitial pulmonary fibrosis

Cited by 5 publications

References 28 publications

Otologic Manifestations of Langerhans Cell Histiocytosis: A Systematic Review

Otologic Manifestations of Langerhans Cell Histiocytosis: A Systematic Review

Current Status of Research on Rare Diseases in Children in China

Langerhans Cell Histiocytosis Causing Central Nervous System Vasculitis Leading to Stroke in a Two-year-old Boy: A Case Report

Contact Info

Product

Resources

About