Antireconverin antibodies in ocular chronic graft versus host disease: A new cause of nonparaneoplasic autoimmune retinopathy

Data de publicació: 01/09/2022 Data Ahead of Print: 01/09/2022

Autors de IIS La Fe

Marina Aguilar González

Autor
Emma Marin Paya

Autor
Romana Garcia Gil

Autor
Anselmo Feliciano Sanchez

Autor
Enrique España Gregori

Autor

Autors aliens a IIS La Fe

Gomez-Lechon Quiros, Luis

Grups d'Investigació

Investigación en Neuroinmunología y Esclerosis Múltiple

Leader

Bonaventura Casanova Estruch

Abstract

Introduction: A case of a 48-year-old male with a nonparaneoplasic autoinmune retinopathy (nPAIR) due to chronic graft versus host disease (GVHD) after an allogenic stem cell transplantation (ASCT) is described. Case report: The patient developed a bilateral rapidly progressive loss of visual acuity with bilateral optic disc edema and bilateral cystoid macular edema (CME) in the funduscopy, a ring scotoma in the visual field (VF) and photoreceptors dysfunction in the electroretinogram (ERG) 210 days after the ASCT. After ruling out other causes, the suspicion of autoimmune retinopathy (AIR) led to the study of antirecoverin antibodies which resulted positive. The exclusion of neoplasia discarded diagnosis of paraneoplasic autoinmune retinopathy (PAIR) and the temporal relationship with BMT led to the diagnosis of nonparaneoplasic autoinmune retinopathy (nPAIR) due to chronic graft versus host disease (GVHD). Oral corticosteroids led to resolution of the CME. Conclusions: Diagnosis of AIR requires a high index of suspicion based on the typical findings on visual field, optical coherence tomography (OCT) and ERG, which force requesting antirecoverin antibodies. However, diagnosis is often delayed because of the need to exclude other causes. Knowing typical symptoms and signs in for a quick action is important because an earlier diagnosis and treatment will improve visual prognosis since the loss of vision already established is irrecoverable. To our knowledge, this is the first reported case in the literature of nPAIR with CME and optic disc edema due to GVHS after ASCT.

Dades de la publicació

ISSN/ISSNe:: 1120-6721, 1724-6016
Tipus:: Article
Pàgines:: 105-110
DOI:: 10.1177/11206721221123779
PubMed:: 36052416
Factor d'Impacte:: 0,782 SCImago ℠
Quartil:: Q2 SCImago ℠

Documents

No hi ha documents

Mètriques

Filiacions

Aguilar Gonzalez, Marina:: Department of Ophthalmology, 16273Hospital Universitari i Politecnic La Fe, Valencia, Spain
Marin Paya, Emma:: Department of Ophthalmology, 16273Hospital Universitari i Politecnic La Fe, Valencia, Spain
Garcia Gil, Romana:: Department of Medical Retina, 16273Hospital Universitari i Politecnic La Fe, Valencia, Spain
Feliciano Sanchez, Anselmo:: Department of Medical Retina, 16273Hospital Universitari i Politecnic La Fe, Valencia, Spain
Gomez-Lechon Quiros, Luis:: Department of Rheumatology, Hospital Francesc de Borja, Gandía, Spain
Espana Gregori, Enrique:: Department of Oculoplastics and Neuroophthalmology, Hospital Universitario y Politécnico La Fe, Valencia, Spain

Keywords

Allogenic stem cell transplantation; antirecoverin antibodies; bone marrow transplant; cystoid macular edema; graft versus host disease; nonparaneoplasic autoimmune retinopathy

Projectes associats

PATRONES DE RM CEREBRAL Y MEDULAR EN RELACION CON LA PRESENCIA DE AUTOANTIGENOS NEURONALES RECONOCIDOS POR INMUNOGLOBULINAS M PRESENTES EN EL LIQUIDO CEFALORRAQUIDEO DE PACIENTES DE ESCLEROSIS MULTIPLE.

Investigador Principal: ISABEL BOSCÁ BLASCO

MERCK SERONO DR. CASANOVA . ALMIRALL S.A; FUNDACIÓN BIOGEN IDEC; MERCK S.L.U; FUNDACIÓN PARA LA INVESTIGACIÓN DEL HOSPITAL UNIVERSITARIO LA FE DE LA COMUNIDAD VALENCIANA . 2010