Bone lesions of Langerhans cell histiocytosis triggered by trauma in children

Pediatr Int. 2022 Jan;64(1):e15199. doi: 10.1111/ped.15199.

Abstract

Background: Bone lesions of Langerhans cell histiocytosis (LCH) may be triggered by trauma.

Methods: The characteristics of pediatric patients in the JLSG-02 study cohort who developed a bone lesion at the trauma site at diagnosis of LCH were analyzed retrospectively.

Results: Of the 261 pediatric patients with LCH, 12 (4.6%), of median age 4.9 years, had trauma-triggered bone LCH lesions at diagnosis, making them significantly older than the remaining patients (P = 0.006). Trauma sites included the craniofacial regions in 10 patients and the lumbar spine and pelvis in one patient each. At the time of trauma, six patients had a bump at the site, whereas none had extradural hematomas or bone fractures. The median time from trauma to onset was 4 weeks. Of these 12 patients, three had isolated bone (IB) disease; four had multifocal bone (MFB) disease, including the bone lesion at the trauma site; and five had multisystem disease, including four with lesions in neighboring tissue and one with polyuria (posterior pituitary lesion) more than 1 year before the trauma-triggered bone lesion. Treatment responses were good in all 12 patients and none died, but relapses were observed in two patients, one each with IB and MFB disease.

Conclusions: About 5% of pediatric patients with LCH developed new trauma-triggered bone lesions at a relatively old age. These lesions can manifest as IB, or, in patients with underlying LCH diseases, as MFB or multisystem. Good clinical outcomes were observed in these patients.

Keywords: Langerhans cell histiocytosis; bone lesion; pediatric; trauma; triggered.

MeSH terms

  • Bone Diseases* / diagnosis
  • Bone Diseases* / etiology
  • Bone and Bones
  • Child
  • Child, Preschool
  • Cohort Studies
  • Histiocytosis, Langerhans-Cell* / complications
  • Histiocytosis, Langerhans-Cell* / diagnosis
  • Histiocytosis, Langerhans-Cell* / drug therapy
  • Humans
  • Retrospective Studies