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A rare case of ocular scarrings in a patient with Vogt-Koyanagi-Harada disease.

Abstract We here report the case of a 27-year old patient, followed-up in our Department for treatment of chronic Vogt-Koyanagi-Harada disease ( VKH disease). Fundus examination showed depigmentation of the retinal pigment epithelium and of the choroid, appearing as a pseudotumoral peripapillary lesion. Vogt-Koyanagi-Harada disease is a multisystem disorder, characterized by bilateral granulomatous panuveitis with serous exudative multifocal retinal detachment. Pathophysiology of this disease is unknown, but an immunological cellular reaction against melanocytes of the skin, the meninges, the retina, the uvea, the cochlea and the labyrinth is suspected. This disease mainly occurs in young subjects from the Far East as well as in pigmented subjects. Ocular involvement is often associated with neurological (meningeal stiffness, headache, sometimes associated with focal deficit and erebrospinal fluid (CSF) pleocytosis), auditory ( perceptive deafness) and cutaneous (vitiligo, poliosis, alopecia and canities) manifestations. It usually evolves in three phases: a prodromal phase mainly characterized by neurological signs, an acute uveitic phase, a chronic phase of convalescence characterized by choroidal and tegument depigmentation or a phase of recurrence during which subretinal neovessels and subretinal fibrosis may appear. Scarrings manifest during the chronic phase of VKH disease, which is dominated by diffuse depigmentation of the fundus of the eye, scars due to nummular chorioretinal atrophy, wheals due to diffuse depigmentation, macular scar remodeling. Pseudotumoral appearance is rare and atypical during the chronic phase of VKH disease. Treatment is based on intravenous corticosteroids followed by a cycle of oral therapy. Patient should be early treated with massive and prolonged therapy to improve prognosis.
PMID
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Authors

Mayor MeshTerms
Keywords

Ocular scarrings

Vogt-Koyanagi-Harada

multisystem disorder

Journal Title the pan african medical journal
Publication Year Start




PMID- 29721143
OWN - NLM
STAT- MEDLINE
DCOM- 20180510
LR  - 20180510
IS  - 1937-8688 (Electronic)
VI  - 28
DP  - 2017
TI  - [A rare case of ocular scarrings in a patient with Vogt-Koyanagi-Harada disease].
PG  - 313
LID - 10.11604/pamj.2017.28.313.4547 [doi]
AB  - We here report the case of a 27-year old patient, followed-up in our Department
      for treatment of chronic Vogt-Koyanagi-Harada disease ( VKH disease). Fundus
      examination showed depigmentation of the retinal pigment epithelium and of the
      choroid, appearing as a pseudotumoral peripapillary lesion. Vogt-Koyanagi-Harada 
      disease is a multisystem disorder, characterized by bilateral granulomatous
      panuveitis with serous exudative multifocal retinal detachment. Pathophysiology
      of this disease is unknown, but an immunological cellular reaction against
      melanocytes of the skin, the meninges, the retina, the uvea, the cochlea and the 
      labyrinth is suspected. This disease mainly occurs in young subjects from the Far
      East as well as in pigmented subjects. Ocular involvement is often associated
      with neurological (meningeal stiffness, headache, sometimes associated with focal
      deficit and erebrospinal fluid (CSF) pleocytosis), auditory ( perceptive
      deafness) and cutaneous (vitiligo, poliosis, alopecia and canities)
      manifestations. It usually evolves in three phases: a prodromal phase mainly
      characterized by neurological signs, an acute uveitic phase, a chronic phase of
      convalescence characterized by choroidal and tegument depigmentation or a phase
      of recurrence during which subretinal neovessels and subretinal fibrosis may
      appear. Scarrings manifest during the chronic phase of VKH disease, which is
      dominated by diffuse depigmentation of the fundus of the eye, scars due to
      nummular chorioretinal atrophy, wheals due to diffuse depigmentation, macular
      scar remodeling. Pseudotumoral appearance is rare and atypical during the chronic
      phase of VKH disease. Treatment is based on intravenous corticosteroids followed 
      by a cycle of oral therapy. Patient should be early treated with massive and
      prolonged therapy to improve prognosis.
FAU - Belmokhtar, Adil
AU  - Belmokhtar A
AD  - Universite Mohamed V Souissi, Service d'Ophtalmologie A, Hopital des Specialites,
      CHU IBN Sina, Rabat, Maroc.
FAU - Daoudi, Rajae
AU  - Daoudi R
AD  - Universite Mohamed V Souissi, Service d'Ophtalmologie A, Hopital des Specialites,
      CHU IBN Sina, Rabat, Maroc.
LA  - fre
PT  - Case Reports
PT  - Journal Article
TT  - Cas rare de lesions cicatricielles oculaires au cours du Vogt-Koyanagi-Harada.
DEP - 20171222
PL  - Uganda
TA  - Pan Afr Med J
JT  - The Pan African medical journal
JID - 101517926
SB  - IM
MH  - Adult
MH  - Cicatrix/*etiology/pathology
MH  - Female
MH  - Humans
MH  - Prognosis
MH  - Retinal Detachment/etiology
MH  - Retinal Pigment Epithelium/*pathology
MH  - Uveomeningoencephalitic Syndrome/*complications/physiopathology
PMC - PMC5927560
OTO - NOTNLM
OT  - Ocular scarrings
OT  - Vogt-Koyanagi-Harada
OT  - multisystem disorder
EDAT- 2018/05/04 06:00
MHDA- 2018/05/11 06:00
CRDT- 2018/05/04 06:00
PHST- 2014/05/12 00:00 [received]
PHST- 2015/03/12 00:00 [accepted]
PHST- 2018/05/04 06:00 [entrez]
PHST- 2018/05/04 06:00 [pubmed]
PHST- 2018/05/11 06:00 [medline]
AID - 10.11604/pamj.2017.28.313.4547 [doi]
AID - PAMJ-28-313 [pii]
PST - epublish
SO  - Pan Afr Med J. 2017 Dec 22;28:313. doi: 10.11604/pamj.2017.28.313.4547.
      eCollection 2017.