Langerhans cell histiocytosis: An exploratory epidemiologic study of 177 cases

Hamre, Merlin; Hedberg, J.; Buckley, Jonathan D.; Bhatia, Smita; Finlay, Jonathan L.; Meadows, Anna T.; Nesbit, Mark E.; Smithson, Anthony; Ll, Robison

doi:10.1002/(sici)1096-911x(199702)28:2<92::aid-mpo2>3.0.co;2-n

Cited by 62 publications

(51 citation statements)

References 16 publications

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“…[32][33][34] Little is known about the epidemiology of Langerhans histiocytosis. One study identified maternal urinary tract infections, feeding problems during infancy, and blood transfusions during infancy 35 as risk factors. Another study found increased risks associated with neonatal infections, solvent exposure, and family thyroid disease.…”

Section: Discussionmentioning

confidence: 99%

Cancer Risk in Children and Young Adults Conceived by In Vitro Fertilization

et al. 2010

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WHAT'S KNOWN ON THIS SUBJECT:No clear-cut increase in cancer risk after IVF has been found, but most studies were too small to answer the question. There are characteristics of children who are conceived by IVF that could increase cancer risk. WHAT THIS STUDY ADDS:This study is large enough to demonstrate that a slight but statistically significant cancer risk exists for children who are conceived by IVF. An interesting finding is the seeming increase in the risk for histiocytosis. abstract OBJECTIVES: Studies conducted so far have found no statistically significant increased risk for cancer among children who are born after in vitro fertilization (IVF). METHODS:We followed 26 692 children who were born after IVF during the years 1982-2005 by using the Swedish Cancer Register and compared the number of children who had cancer and were born after IVF with children who were not conceived by IVF. Adjustment was made for year of birth. RESULTS:Maternal age, parity, smoking, subfertility, previous miscarriages, BMI, and multiple births did not significantly affect cancer risk in offspring. High birth weight, premature delivery, and the presence of respiratory diagnoses and low Apgar score were risk factors for cancer. We identified 53 cases of cancer in children who were born after IVF against 38 expected cases: 18 of them with hematologic cancer (15 of them acute lymphoblastic leukemia), 17 with eye or central nervous system tumors, and 12 with other solid cancers. There were 6 cases of Langerhans histiocytosis against 1.0 expected. The total cancer risk estimate was 1.42 (95% confidence interval: 1.09 -1.87). CONCLUSIONS:We found a moderately increased risk for cancer in children who were conceived by IVF. Putative intermediary factors could be preterm birth and neonatal asphyxia.

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Section: Discussionmentioning

confidence: 99%

Cancer Risk in Children and Young Adults Conceived by In Vitro Fertilization

et al. 2010

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“…Most children with multisystem disease were aged less than 3 years at diagnosis, similarly to what has been described in most studies. 7,16 Classically, age lower than 2-3 years indicates poor prognosis. In the present study, probably because of the small number of patients, such influence did not have any statistical significance.…”

Section: Receiving Treatmentmentioning

confidence: 73%

“…7,[14][15][16][17] In most cases, diagnosis was presumptive. The definitive diagnosis was established in only 21.2% of children, which points out to the difficulty in establishing it in 10 In the present study, patients whose disease concentrated only on lymph nodes had a presumptive or probable diagnosis.…”

Section: Discussionmentioning

confidence: 99%

Histiocitose das células de Langerhans: experiência de 16 anos

Campos¹,

Viana²,

Oliveira³

et al. 2007

J. Pediatr. (Rio J.)

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Objectives: To describe the clinical course of Langerhans cell histiocytosis and to compare its outcome according to age, staging of the disease and treatment response.Methods: Retrospective analysis of data on 33 children with Langerhans cell histiocytosis followed at Hospital das Clínicas, Universidade Federal de Minas Gerais, Brazil, between 1988 and2004. Results: Age at diagnosis ranged from 2 months to 16 years (median: 2.5 years). Seventeen children were male.The follow-up period varied from 21 days to 16.2 years (median: 3.4 years). The most common clinical manifestations at diagnosis were osteolytic lesions, enlarged lymph nodes and skin lesions. The overall survival rate for the whole group was 86.1% at 16 years (95%CI 66.6-94.6%). Deaths occurred in patients with multisystem disease and organ dysfunction at diagnosis. Those patients who had a "better" response to treatment in the sixth week were likely to have a significantly higher overall survival rate than those who showed disease progression. Overall survival rate was significantly higher for patients with single-system disease. The disease-free survival rate for the whole group was 30.9% at 16 years (95%CI 15.6-47.5%), and was significantly higher for those with single-system disease. Age groups were not associated with different disease-free survival rates. Diabetes insipidus was the most common sequela. No cases of secondary neoplasms were observed. Conclusion:The clinical manifestations of Langerhans cell histiocytosis vary widely, with a high relapse rate and low mortality rate.J Pediatr (Rio J). 2007;83(1):79-86: Langerhans cell histiocytosis, diagnosis, therapy, complications.

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“…LCH is most commonly seen in boys between the age of 1-4 years. 8,9 Both patients in this study are girls which is rare presentation.…”

Section: Discussionmentioning

confidence: 99%

Langerhans Cell Histiocytosis of Ocular Region: A Report of Two Cases

Khatri¹

2018

RAPL

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Langerhans cell histiocytosis (LCH) is a rare monoclonal disorder of dendritic cells originating from bone marrow. We are reporting two cases of isolated involvement of ocular region both of which clinically appeared as soft tissue tumour. A young girl of two and half years presented with swelling of left eyelid. Microscopic examination of biopsy showed collections of round cells which were large having abundant cytoplasm and centrally placed nuclei. Plenty of eosinophils were seen in the background. The final diagnosis of LCH involving eyelid was given after histopathological examination. Second case report is of three-year-old girl presented to ophthalmology outpatient department with the complaints of painless progressive swelling of lateral aspect of left orbital wall for four months which were rapidly increasing in size for the past two months. Fine-needle aspiration (FNA) of both swellings was done and finding was suggestive of LCH. The final diagnosis of LCH was given on histopathological examination of biopsy tissue from both the swellings. Ocular region involvement by Langerhans cell histiocytosis is a very rare presentation.

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Langerhans cell histiocytosis: An exploratory epidemiologic study of 177 cases

Cited by 62 publications

References 16 publications

Cancer Risk in Children and Young Adults Conceived by In Vitro Fertilization

Cancer Risk in Children and Young Adults Conceived by In Vitro Fertilization

Histiocitose das células de Langerhans: experiência de 16 anos

Langerhans Cell Histiocytosis of Ocular Region: A Report of Two Cases

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