title: Macrophagic myofasciitis in children is a localized reaction to vaccination.
abstract: Macrophagic myofasciitis is a novel, "inflammatory myopathy" described after a variety of vaccinations, almost exclusively in adults. We examined the relevance of histological findings of this myopathy to the clinical presentation in pediatric patients. Muscle biopsies from 8 children (7 months to 6 years old) with histological features of macrophagic myofasciitis were reviewed and correlated with the clinical manifestations. Patients underwent quadriceps muscle biopsy for suspected mitochondrial disease (4 patients), spinal muscular atrophy (2 patients), myoglobinuria (1 patient), and hypotonia with motor delay (1 patient). All biopsies showed identical granulomas composed of periodic acid-Schiff-positive and CD68-positive macrophages. Characteristic aluminum hydroxide crystals were identified by electron microscopy in 2 cases. The biopsy established diagnoses other than macrophagic myofasciitis in 5 patients: spinal muscular atrophy (2), Duchenne muscular dystrophy (1), phospho-glycerate kinase deficiency (1), and cytochrome c oxidase deficiency (1). Three children with manifestations and/or a family history of mitochondrial disease had otherwise morphologically normal muscle. All children had routine vaccinations between 2 months and 1 year before the biopsy, with up to 11 intramuscular injections, including the biopsy sites. There was no correlation between histological findings of macrophagic myofasciitis in biopsies and the clinical symptoms. We believe that macrophagic myofasciitis represents a localized histological hallmark of previous immunization with the aluminum hydroxide adjuvants contained in vaccines, rather than a primary or distinct inflammatory muscle disease.
authors.affiliation: Department of Pathology and Laboratory Medicine, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia. boleklach2@hotmail.com
authors.forename: Boleslaw
authors.initials: B
authors.lastname: Lach
authors.forename: Edward J
authors.initials: EJ
authors.lastname: Cupler
mesh_terms: D000536:Aluminum Hydroxide / Q000032:analysis; D015703:Antigens, CD / Q000032:analysis; D015214:Antigens, Differentiation, Myelomonocytic / Q000032:analysis; D001706:Biopsy; D002675:Child, Preschool; D003460:Crystallization; D003937:Diagnosis, Differential; D015721:Diphtheria-Tetanus-Pertussis Vaccine / Q000009:adverse effects*; D005208:Fasciitis / Q000473:pathology*; D005260:Female; D006099:Granuloma / Q000473:pathology; D018073:Haemophilus Vaccines / Q000009:adverse effects*; D017325:Hepatitis B Vaccines / Q000009:adverse effects*; D006801:Humans; D002479:Inclusion Bodies / Q000473:pathology; D007223:Infant; D008264:Macrophages / Q000473:pathology*; D008297:Male; D022542:Measles-Mumps-Rubella Vaccine / Q000009:adverse effects*; D008854:Microscopy, Electron; D018482:Muscle, Skeletal / Q000473:pathology*; D009134:Muscular Atrophy, Spinal / Q000473:pathology; D009220:Myositis / Q000473:pathology*; D010735:Phosphoglycerate Kinase / Q000172:deficiency; D000097382:CD68 Molecule
chemical_list: D000536:Aluminum Hydroxide; D015703:Antigens, CD; D015214:Antigens, Differentiation, Myelomonocytic; D015721:Diphtheria-Tetanus-Pertussis Vaccine; D018073:Haemophilus Vaccines; D017325:Hepatitis B Vaccines; D022542:Measles-Mumps-Rubella Vaccine; D010735:Phosphoglycerate Kinase; C067980:CD68 antigen, human; D000097382:CD68 Molecule