Childhood macrophagic myofasciitis: A series from the Indian subcontinent

Kakkar, Aanchal; Rajeshwari, Madhu; Nalwa, Aasma; Suri, Vaishali; Sarkar, Chitra; Chakrabarty, Biswaroop; Gulati, Sheffali; Sharma, Mehar Chand

doi:10.1002/mus.25467

Cited by 7 publications

(6 citation statements)

References 19 publications

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“…MMF lesions in our patients were found in vaccination sites, i.e., the quadriceps; however, these patients presented with a severe systemic illness, such as a delayed motor milestone or muscle weakness, congenital hypotonia, etc. (n = 1) ( Table 1), which is consistent with the reported clinical manifestations of MMF patients 2,5,7,8 . However, sensorineural hearing loss (n = 1) and tracheo-broncho-laryngomalacia (n = 1) may result from superimposed coincidental diseases and may not be associated with MMF.…”

Section: Discussionsupporting

confidence: 89%

“…After Gherardi et al first reported 14 French adult patients (median age: 43 years, range 30–70 years old, male: female = 6:8) with MMF in 1988, various adverse effects of aluminium hydroxide-containing vaccines have been reported (Supplementary Table 2 ) 6 , 18 . Several childhood cases have also been reported (median age: 20 months, range 4–70 months, male:female = 2:1) 2 , 4 , 5 , 7 , 8 .…”

Section: Discussionmentioning

confidence: 99%

“…Furthermore, myalgic encephalomyelitis, cognitive dysfunction, and aluminium neurotoxicity or neuropsychological symptoms, such as demyelinating CNS disorder, have been reported 20 – 23 . Hypotonia, delayed motor milestones, seizures, and irritability are common symptoms in children 5 , 7 , 8 . Autoimmune (autoinflammatory) syndrome induced by adjuvants (ASIA) 12 , 13 , 24 and Gulf War syndrome (GWS)-chronic fatigue syndrome, which can develop in soldiers subjected to multiple aluminium-containing vaccinations, are known to be associated with post-vaccinal disseminated encephalomyelitis 14 , 15 .…”

Section: Discussionmentioning

confidence: 99%

“…In adult cases, symptoms have been delayed from several months to years after vaccination, but some of the reported children began the symptoms immediately after vaccination and lasted for a long time. 5 …”

Section: Discussionmentioning

confidence: 99%

“…MMF is an unusual inflammatory myopathy found in patients with arthromyalgia and muscle weakness that develops several months to years after administering aluminium-containing vaccines. However, symptoms immediately developed after vaccination have also been reported 4 , 5 . Since Gherardi et al first described MMF in 1998, most reported patients have been adult, although several articles have documented child cases 2 , 4 – 8 .…”

Section: Introductionmentioning

confidence: 99%

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Macrophagic myofasciitis and subcutaneous pseudolymphoma caused by aluminium adjuvants

Kim

Lim

Kang

et al. 2020

Sci Rep

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Aluminium hydroxide is a well-known adjuvant used in vaccines. Although it can enhance an adaptive immune response to a co-administered antigen, it causes adverse effects, including macrophagic myofasciitis (MMf), subcutaneous pseudolymphoma, and drug hypersensitivity. the object of this study is to demonstrate pediatric cases of aluminium hydroxide-induced diseases focusing on its rarity, under-recognition, and distinctive pathology. Seven child patients with biopsy-proven MMf were retrieved from the Seoul national University Hospital (SnUH) pathology archives from 2015 to 2019. The medical records and immunisation history were reviewed, and a full pathological muscle examination was carried out. The mean age was 1.7 years (8.9-40 months), who had records of vaccination against hepatitis B, hepatitis A, and tetanus toxoid on the quadriceps muscle. The chief complaints were muscle weakness (n = 6), delayed motor milestones (n = 6), instability, dysarthria, and involuntary movement (n = 1), swallowing difficulty (n = 1), high myopia (n = 1), and palpable subcutaneous nodules with skin papules (n = 1). Muscle biopsy showed MMF (n = 6) and pseudolymphoma (n = 1) with pathognomic basophilic large macrophage infiltration, which had distinctive spiculated inclusions on electron microscopy. the intracytoplasmic aluminium was positive for pAS and Morin stains. Distinctive pathology and ultrastructure suggested an association with aluminium hydroxide-containing vaccines. to avoid misdiagnosis and mistreatment, we must further investigate this uncommon condition, and pharmaceutical companies should attempt to formulate better adjuvants that do not cause such adverse effects. Despite the availability of several vaccine adjuvants, these based on aluminium hydroxide (a crystalline compound) continues to be widely used worldwide 1. To date, the benefit of aluminium hydroxide-based adjuvants is not fully understood, in spite of its long-term usage. It has an enhancing effect on the adaptive immune response to a co-administered antigen through repository and pro-phagocytic effects and proinflammatory NOD-like receptor protein 3 (NLRP3) pathway activation 1. NLR proteins are involved in the immune response and help in activating and regulating responses to injury, toxins, or invasion by microorganisms. However, aluminium hydroxide has adverse effects, including macrophagic myofasciitis (MMF) and adverse drug reactions (ADRs) to aluminous adjuvants, which include aluminium neurotoxicity, autism spectrum disorder, and autoinflammatory syndrome 1,2. Currently, tetanus, hepatitis A, hepatitis B, human papillomavirus, Haemophilus influenzae type B, pneumococcal and meningococcal vaccines, and anthrax vaccines use aluminium hydroxide as an adjuvant 3. MMF is an unusual inflammatory myopathy found in patients with arthromyalgia and muscle weakness that develops several months to years after administering aluminium-containing vaccines. However, symptoms immediately developed after vaccination have also been reported 4, 5. Since Gherardi ...

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Section: Discussionsupporting

confidence: 89%