RESUMEN
A 56-year-old-man presented a 2-month history of chalazion in the eyelids without response to treatment and with an inconclusive biopsy. Laboratory results confirmed the presence of Enterobacter cloacae and Streptococcus gordonii infection. Despite appropriate intravenous antibiotic treatment, clinical worsening was observed. Radical surgical excision and total tarsorrhaphy were performed. Following treatment, the patient was asymptomatic for 6 weeks until he developed acute renal failure, generalised arthralgia, acute hypertensive anterior uveitis, and dacryoadenitis. Lacrimal gland biopsy revealed a perivascular granulomatous inflammation with necrotic foci. Renal biopsy showed a necrotizing extracapillary glomerulonephritis. Blood tests showed elevated levels of C-reactive protein, positive rheumatoid factor, and proteinase 3-anti-neutrophil cytoplasmic antibody. The patients was therefore diagnosed with granulomatosis with polyangiitis and treated with cyclophosphamide and corticosteroids, with good systemic and orbital response. Surgical reconstruction of the eyelid was subsequently performed. This case describes, for the first time in the literature, an eyelid granuloma as the presenting sign in GPA, highlighting the importance of differential diagnosis of eyelid lesions with unusual characteristics.
Asunto(s)
Enfermedades de los Párpados/diagnóstico , Granuloma/diagnóstico , Granulomatosis con Poliangitis/diagnóstico , Úlcera/diagnóstico , Anticuerpos Anticitoplasma de Neutrófilos/sangre , Proteína C-Reactiva/metabolismo , Ciclofosfamida/uso terapéutico , Diagnóstico Diferencial , Quimioterapia Combinada , Enfermedades de los Párpados/tratamiento farmacológico , Glucocorticoides/uso terapéutico , Granuloma/tratamiento farmacológico , Granulomatosis con Poliangitis/tratamiento farmacológico , Humanos , Inmunosupresores/uso terapéutico , Masculino , Metilprednisolona/uso terapéutico , Persona de Mediana Edad , Mieloblastina/sangre , Tomografía Computarizada por Rayos X , Úlcera/tratamiento farmacológicoRESUMEN
CONTEXT: Inhibition of the neonatal fragment crystallizable receptor (FcRn) reduces pathogenic thyrotropin receptor antibodies (TSH-R-Ab) that drive pathology in thyroid eye disease (TED). OBJECTIVE: We report the first clinical studies of an FcRn inhibitor, batoclimab, in TED. DESIGN: Proof-of-concept (POC) and randomized, double-blind placebo-controlled trials. SETTING: Multicenter. PARTICIPANTS: Patients with moderate-to-severe, active TED. INTERVENTION: In the POC trial, patients received weekly subcutaneous injections of batoclimab 680 mg for 2 weeks, followed by 340 mg for 4 weeks. In the double-blind trial, patients were randomized 2:2:1:2 to weekly batoclimab (680 mg, 340 mg, 255 mg) or placebo for 12 weeks. MAIN OUTCOME: Change from baseline in serum anti-TSH-R-Ab and total IgG (POC); 12-week proptosis response (randomized trial). RESULTS: The randomized trial was terminated because of an unanticipated increase in serum cholesterol; therefore, data from 65 of the planned 77 patients were analyzed. Both trials showed marked decreases in pathogenic anti-TSH-R-Ab and total IgG serum levels (P < .001) with batoclimab. In the randomized trial, there was no statistically significant difference with batoclimab vs placebo in proptosis response at 12 weeks, although significant differences were observed at several earlier timepoints. In addition, orbital muscle volume decreased (P < .03) at 12 weeks, whereas quality of life (appearance subscale) improved (P < .03) at 19 weeks in the 680-mg group. Batoclimab was generally well tolerated, with albumin reductions and increases in lipids that reversed upon discontinuation. CONCLUSIONS: These results provide insight into the efficacy and safety of batoclimab and support its further investigation as a potential therapy for TED.
Asunto(s)
Exoftalmia , Oftalmopatía de Graves , Recién Nacido , Humanos , Oftalmopatía de Graves/tratamiento farmacológico , Calidad de Vida , Anticuerpos Monoclonales/uso terapéutico , Inmunoglobulina G/uso terapéutico , Método Doble Ciego , Resultado del TratamientoRESUMEN
OBJECTIVE: The aim of this article is to highlight our experience with autologous pericranium graft in wide exposures (≥3 mm). The pericranium graft was taken from the parietal region of the scalp in six consecutive clinical cases of orbital implant exposure in anophthalmic sockets. MATERIAL AND METHODS: This is a prospective, descriptive case series study of patients who had orbital implant exposures and were treated with autologous pericranium graft. RESULTS: The average postoperative follow-up period was 10 months, and the mean time for conjunctivalization of the graft was 3 months. In all cases, complete conjunctivalization was achieved, and no re-exposure of the implant was observed. CONCLUSIONS: The pericranium graft from the parietal region is an effective technique to treat both small and large orbital implant exposures with no comorbidity. Time to complete conjunctivalization is similar to that of other autologous grafts. It is a simple and convenient method for the oculoplastic surgeon that constitutes a good alternative for covering anophthalmic cavities. Further studies with more cases and longer follow-up are required to confirm the effectiveness of this technique.
Asunto(s)
Autoinjertos/trasplante , Implantes Orbitales , Periostio/trasplante , Dehiscencia de la Herida Operatoria/cirugía , Adulto , Anciano , Anciano de 80 o más Años , Anoftalmos/cirugía , Femenino , Estudios de Seguimiento , Humanos , Masculino , Persona de Mediana Edad , Hueso Parietal/cirugía , Estudios Prospectivos , Sitio Donante de Trasplante/cirugíaAsunto(s)
Linfoma/radioterapia , Neoplasias Orbitales/radioterapia , Traumatismos por Radiación/etiología , Anciano , Femenino , Estudios de Seguimiento , Humanos , Linfoma/diagnóstico , Linfoma/mortalidad , Linfoma/patología , Masculino , Persona de Mediana Edad , Neoplasias Orbitales/diagnóstico , Neoplasias Orbitales/mortalidad , Neoplasias Orbitales/patología , Traumatismos por Radiación/diagnóstico , Traumatismos por Radiación/epidemiología , Estudios Retrospectivos , España , Resultado del TratamientoRESUMEN
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