Acute unilateral maculopathy associated with adult onset of hand, foot and mouth disease: a case report

Abstract

Background To report the case of a 31-year-old patient with Hand, Foot and Mouth Disease (HFMD) and concurrent acute monocular maculopathy, and to describe multimodal imaging findings never before described including optical coherence tomography angiography (OCT-A). Case presentation Nine days after the onset of clinically highly probable but not laboratory-verified HFMD, a 31-year old male noticed a central scotoma, distorted lines and loss of visual acuity (Snellen visual acuity 20/400) in his right eye. Funduscopy revealed focal alterations in the retinal pigmented epithelium (RPE) and yellow retinal dots corresponding to focal dots of decreased fundus autofluorescence (FAF) surrounded by increased FAF. Spectral domain optical coherence tomography (SD-OCT) demonstrated irregularities in the ellipsoide zone, hyperreflective dots above the RPE and RPE thickening. Fundus fluorescein angiography (FAG) revealed central hypofluorescence in the macular area in the early phase, as well as increasing focal hyperfluorescence in the late phase corresponding with RPE defects observed in FAF. Indocyanine green angiography (ICGA) showed central hypofluorescence in the early and late phase, corresponding with areas of reduced flow in the choroidea and choriocapillaris as apparent in OCT-A. Visual acuity improved within 3 months without any systemic or local therapy. At his three-month follow-up, SD-OCT revealed subtle subretinal fluid that resolved spontaneously over time. No signs of choroidal neovascularization were observed. Twelve months following the onset of symptoms Snellen visual acuity was 400/400. Multimodal imaging revealed subtly changed, decreased FAF while the choroidal architecture recovered completely as demonstrated by OCT-A. Conclusions HFMD-associated maculopahty is an uncommon but important differential diagnosis of chorioretinitis with macular involvement. The prognosis can be good and the initially observed morphological pathologies such as impaired perfusion of the choroidal vessels can recover spontaneously over a period lasting 12 months. OCT-A can be employed as a non-invasive tool to detect the reduced perfusion of the choroidal vessels and for monitoring the disease course.

Is choriocapillaris the early target in acute maculopathy secondary to hand, foot, and mouth disease? A case report

“…European Journal of Ophthalmology 32 (2) manifestations confirm the hypothesis by Reich et al 10 In particular, we believe that HFMD maculopathy involves primarily choriocapillaris with a swelling of choroidal thickness, then RPE and ellipsoid layer in late stages, so we think that only an early diagnosis associated with a prompt treatment can improve the final outcome. In our opinion, a treatment should be started before the beginning of all retinal sequelae and not after appearance of clinical symptoms of the HFMD maculopathy.…”

“…8,9 Maculopathy can present many different manifestations, in particular unilateral alterations of RPE associated with irregularities in the ellipsoide zone, retinal vasculitis, unilateral hemorrhagic maculopathy, unilateral retinal exudates, or anterior segment deposits. 7,10,11 Maculopathy is usually self-limited, but scars of the outer layer of the retina with subfoveal ellipsoid zone disruption can occur, with a permanent visual impairment. In our case, in presence of ocular severe conditions, we decided to perform oral prednisolone therapy according to Agrawal et al 7 in order to reduce the inflammatory response.…”

Multimodal Imaging of Unilateral Acute Maculopathy Associated with Hand, Foot, and Mouth Disease: A Case Series

“…Various retinopathies associated with HFMD have been reported such as UAIM [ 2 , 3 , 4 , 5 , 6 , 7 , 8 , 9 ], unilateral multifocal retinitis [ 10 ], bilateral obstructive retinal vasculitis [ 11 ], and unilateral chorioretinitis [ 12 ]. UAIM associated with HFMD is most commonly described with more than a dozen cases.…”

“…It mainly affects children, but adults can also be infected. Several scientific publications have reported the association between HFMD and UAIM in young adults [ 2 , 3 , 4 , 5 , 6 , 7 , 8 ]. However, despite the increasing use of multimodal imaging in the management of retinal diseases, imaging findings remain limited in the scientific literature.…”

Immune responses of a CV-A16 live attenuated candidate strain and its protective effects in rhesus monkeys

“…As of June 2019, over 21 million HFMD cases were reported in China [ 6 ]. HFMD is a common contagious disease among children aged <5 years and occurs sporadically in adults [ 1 , 7 , 8 ]. Clinical manifestations of HFMD include fever, vesicles on hands, feet, mouth, anus, and central nervous system diseases such as aseptic meningitis, encephalitis, and neurogenic pulmonary oedema.…”