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Case Reports in Medicine 2019Vogt-Koyanagi-Harada (VKH) syndrome is a multisystemic autoimmune disease of uncertain pathogenesis. Infectious aetiology has been proposed which is suggested to lead to...
BACKGROUND
Vogt-Koyanagi-Harada (VKH) syndrome is a multisystemic autoimmune disease of uncertain pathogenesis. Infectious aetiology has been proposed which is suggested to lead to the loss of melanocytes in the skin, inner ear, meninges, and uvea in those who are genetically predisposed. Information regarding VKH syndrome is scanty among the African population.
CASE PRESENTATION
We report a 28-year-old HIV-uninfected Ugandan woman who had previously been well and presented with chronic bilateral panuveitis; symmetrical vitiligo patches on the head, trunk, and upper limbs; tinnitus; and poliosis of the scalp hair, eyelashes, and eyebrows. A flu-like syndrome preceded this. Several weeks of prednisolone and azathioprine therapy resulted in remarkable improvement of the ocular and inner ear symptoms.
CONCLUSION
A high index of suspicion is required in diagnosing VKH syndrome, even in sub-Saharan Africa where the disease is reported to be rare. Initiation of prompt and appropriate treatment prevents blindness and other complications.
PubMed: 31636673
DOI: 10.1155/2019/5192754 -
Actas Dermo-sifiliograficas 2015
Topics: Antihypertensive Agents; Cheek; Eyelashes; Female; Hair Diseases; Humans; Hypertrichosis; Hypopigmentation; Latanoprost; Middle Aged; Prostaglandins F, Synthetic
PubMed: 25065768
DOI: 10.1016/j.ad.2014.05.005 -
Skin Appendage Disorders Jan 2018
PubMed: 29457006
DOI: 10.1159/000477414 -
Pseudotumoral and Multiple Retinal Pigment Epithelium Proliferation in Vogt-Koyanagi-Harada Disease.Case Reports in Ophthalmological... 2015We report a case of pseudotumoral retinal pigment epithelium (RPE) proliferation in Vogt-Koyanagi-Harada (VKH) disease, in a 50-year-old female who presented with a...
We report a case of pseudotumoral retinal pigment epithelium (RPE) proliferation in Vogt-Koyanagi-Harada (VKH) disease, in a 50-year-old female who presented with a juxtapapillary and peripheral subretinal hyperpigmented lesions in the left eye and "sunset glow fundus," hyperpigmented striae, and multiple atrophic chorioretinal spots in the periphery. The darkly pigmented exuberant larger subretinal mass extended to the periphery with associated subretinal fibrosis. This patient demonstrated the entire clinical presentation of VKH disease, which tends to course with a chronic, bilateral, granulomatous panuveitis and exudative retinal detachment associated with poliosis, vitiligo, alopecia, and central nervous system and auditory signs. Our case is unique for the presence of exuberant, pseudotumoral RPE proliferation at the juxtapapillary region and peripheral area. Although this complication has rarely been reported, a high index of suspicion is warranted for early diagnosis and avoids unnecessary treatments of a pseudotumor.
PubMed: 26509089
DOI: 10.1155/2015/153831 -
The British Journal of Dermatology Jun 2017Cytotoxic T-lymphocyte-associated protein-4, programmed cell death protein and programmed cell death protein ligand 1 monoclonal antibodies (immune checkpoint...
Cytotoxic T-lymphocyte-associated protein-4, programmed cell death protein and programmed cell death protein ligand 1 monoclonal antibodies (immune checkpoint inhibitors), are used to treat various malignancies. Their mechanism of action involves the inhibition of negative regulators of immune activation, resulting in immune-related adverse events (irAEs) including endocrinopathies, pneumonitis, colitis, hepatitis and dermatological events. Dermatological irAEs include maculopapular rash, pruritus, vitiligo, blistering disorders, mucocutaneous lichenoid eruptions, rosacea and the exacerbation of psoriasis. Alopecia secondary to immune checkpoint inhibitors has been reported in 1·0-2·0% of treated patients. Our objective is to characterize for the first time the clinicopathology of patients with alopecia areata (AA) secondary to immune checkpoint inhibitors, including the first report of anti-PD-L1 therapy-induced AA, and review of the literature. Four cases of patients who developed partial or complete alopecia during treatment with immune checkpoint inhibitors for underlying cancer were identified from our clinics. Methods include the review of the history and clinicopathologic features. Three patients (75%) had AA and one had universalis. Two patients had a resolution after topical, oral or intralesional therapies and one had a resolution after immunotherapy was discontinued; all regrown hair exhibited poliosis. One of the four patients had coincident onychodystrophy. This report describes a series of four patients who developed partial or complete alopecia (i.e. areata and universalis) during treatment with immune checkpoint inhibitor therapies for cancer. The recognition and management of hair-related irAEs are important for pretherapy counselling and interventions that contribute to maintaining optimal health-related quality of life in patients.
Topics: Adult; Aged; Alopecia Areata; Antibodies, Monoclonal, Humanized; B7-H1 Antigen; CTLA-4 Antigen; Carcinoma, Renal Cell; Drug Therapy, Combination; Female; Humans; Immunotherapy; Kidney Neoplasms; Male; Melanoma; Middle Aged; Neoplasm Metastasis; Skin Neoplasms
PubMed: 27943234
DOI: 10.1111/bjd.15237 -
The British Journal of Dermatology Jan 2018Pembrolizumab is an immune checkpoint inhibitor that targets the programmed cell death (PD)-1 receptor. Common cutaneous adverse side-effects of PD-1 inhibitors include...
Pembrolizumab is an immune checkpoint inhibitor that targets the programmed cell death (PD)-1 receptor. Common cutaneous adverse side-effects of PD-1 inhibitors include maculopapular rash, pruritus, vitiligo and lichenoid skin and mucosal reactions. Here we describe a man in his sixties with metastatic melanoma treated with pembrolizumab who subsequently developed fading or disappearance of pigmented skin lesions, lightening of the skin, and poliosis of the eyebrows, eyelashes and scalp and body hair. Compared with baseline high-resolution three-dimensional total-body photography, we observed fading or disappearance of solar lentigines, seborrhoeic keratoses and melanocytic naevi, suggesting that PD-1 inhibitors may affect the evolution of these benign skin lesions. With dermatoscopic follow-up, altered lesions showed either blue-grey peppering/granularity or fading in colour without other identifiable features. No halo lesions or lesions with surrounding inflammation were identified. One changed pigmented lesion that showed blue-grey peppering/granularity on dermoscopy was biopsied and interpreted as a macular seborrhoeic keratosis with melanophages. Further studies are required to elucidate the effects of PD-1 inhibition on benign skin lesions.
Topics: Antibodies, Monoclonal, Humanized; Antineoplastic Agents, Immunological; Humans; Liver Neoplasms; Male; Melanoma; Middle Aged; Pigmentation Disorders; Skin Neoplasms
PubMed: 28132411
DOI: 10.1111/bjd.15354 -
The Pan African Medical Journal 2018Scleroatrophic lichen (SL) and vitiligo are two depigmenting disorders which may occur separately or, rarely, in combination. Their association may seem logical because...
Scleroatrophic lichen (SL) and vitiligo are two depigmenting disorders which may occur separately or, rarely, in combination. Their association may seem logical because both these disorders are characterized by the suspicion of an autoimmune pathogenesis. We here report the case of a 8-year old girl, with no notable medical history, presenting with achromic macules and papular nonpruritic lesions evolving over 6 months. Clinical examination showed two types of lesions (A): ovalaire achromic macules measuring 1-3 cm along their longer axis, located at the level of the front, of the neck, of the shoulders, as well as of the peri-mamelonar and of the genital region. These lesions exhibited slightly raised peripheral inflammatory border as well as a poliosis. The patient also had pearly white atrophic papular plaques at the level of the interscapular and abdominal regions as well as of the anterior face of the knees. Two biopsies were performed. Histological examination of an achromic macula showed vitiligo with inflammatory reaction while histological examination of an infiltrated lesion allowed the diagnosis of scleroatrophic lichen. Significant clinical improvement was obtained by local treatment with dermocorticoids. Therefore, this pathological association between scleroatrophic lichen and vitiligo in our patient, highlights the key role of epidermal lichenoid inflammatory process in the disappearance of melanocytes and, possibly, in the induction of a auto-immune process.
Topics: Biopsy; Child; Female; Humans; Inflammation; Lichen Sclerosus et Atrophicus; Melanocytes; Vitiligo
PubMed: 30344859
DOI: 10.11604/pamj.2018.30.75.13954 -
JAAD Case Reports Aug 2023
PubMed: 37521192
DOI: 10.1016/j.jdcr.2023.06.018 -
Case Reports in Oncology 2024Ipilimumab and nivolumab are checkpoint inhibitors that are known to cause a multitude of inflammatory ocular adverse events. Here we report a patient with poliosis and...
INTRODUCTION
Ipilimumab and nivolumab are checkpoint inhibitors that are known to cause a multitude of inflammatory ocular adverse events. Here we report a patient with poliosis and symptomatic depigmentation of the choroid and retinal pigment epithelium (RPE) associated with checkpoint inhibitor therapy for cutaneous melanoma.
CASE PRESENTATION
The patient presented with floaters in both eyes and concerns for intraocular metastases of metastatic cutaneous melanoma after 1 month of therapy with ipilimumab and nivolumab. External examination revealed poliosis of her eyebrows and eyelashes. Fundus photography demonstrated multiple 1-3 disc-diameter hypopigmented placoid flat areas in the RPE/choroid exposing underlying choroidal vessels in both eyes. At subsequent evaluation 7 months later (after an additional 6 months of checkpoint inhibitor therapy), the lesions appeared more blanched. Evaluation nearly 20 months after the initial presentation showed no significant changes from her prior visit despite cessation of checkpoint inhibitor therapy for 13 months.
CONCLUSION
Checkpoint inhibitor therapy for cutaneous melanoma metastases can cause depigmentation of the choroid and RPE that must be differentiated from progression of intraocular melanoma.
PubMed: 38264011
DOI: 10.1159/000535745 -
The Pan African Medical Journal 2017We 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...
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.
Topics: Adult; Cicatrix; Female; Humans; Prognosis; Retinal Detachment; Retinal Pigment Epithelium; Uveomeningoencephalitic Syndrome
PubMed: 29721143
DOI: 10.11604/pamj.2017.28.313.4547