RESUMO
BACKGROUND: GAPO (growth retardation, alopecia, pseudoanodontia, and optic atrophy) as a rare genetic disorder includes growth retardation, alopecia, pseudoanodontia, and optic atrophy. It was reported to be associated with craniosynostosis and intracranial hypertension. CASE REPORT: A patient with such a rare disorder associated with multisuture craniosynostosis and headache is presented. Surgery has been done due to intracranial hypertension. CONCLUSIONS: Abnormal intraoperative findings including sever pericranium and dural adhesions and extraordinary bleeding related to this syndrome are described.
Assuntos
Alopecia/cirurgia , Anodontia/cirurgia , Craniossinostoses/cirurgia , Craniotomia/métodos , Transtornos do Crescimento/cirurgia , Hipertensão Intracraniana/cirurgia , Atrofias Ópticas Hereditárias/cirurgia , Alopecia/complicações , Anodontia/complicações , Pré-Escolar , Craniossinostoses/complicações , Feminino , Transtornos do Crescimento/complicações , Humanos , Hipertensão Intracraniana/complicações , Atrofias Ópticas Hereditárias/complicações , Resultado do TratamentoRESUMO
The goal of this study was to investigate whether poly (lactic-co-glycolic) acid (PLGA) nanoparticles could enhance sublingual immunotherapy (SLIT) efficacy. BALB/c mice sensitized to rChe a 3 were treated sublingually either with soluble rChe a 3 (100µg/dose) or PLGA-encapsulated rChe a 3 (5, 25, or 50µg/dose). SLIT with PLGA-encapsulated rChe a 3 (equivalent to 25 and 50µg rChe a 3 per dose) led to significantly increased antigen-specific IgG2a, along with no effect on allergen-specific IgE and IgG1 antibody levels. In addition, interleukin 4 (IL-4) levels in restimulated splenocytes were significantly less, while interferon-γ (IFN-γ), interleukin-10 (IL-10), and transforming growth factor-ß (TGF-ß) levels, as well as Foxp3 expression, were significantly greater than in the control groups. Our findings suggest that PLGA nanoparticle-based vaccination may help rational development of sublingual immunotherapy through reduction of the needed allergen doses and also significantly enhanced systemic T regulatory (Treg) and T helper 1 (Th1) immune responses.