Amyloidosis Cutis Dyschromica: A Case Treated with Acitretin

Özcan, Atilla; Şenol, Mustafa; Aydın, Nasuhi Engin; Karaca, Şemsettin

doi:10.1111/j.1346-8138.2005.tb00783.x

Cited by 34 publications

(36 citation statements)

References 15 publications

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“…However, only wide-spectrum cytokeratin antibodies or high molecular weight cytokeratin antibodies were used in these reports. Wide-spectrum anti-cytokeratin antibodies were positive in two of the three cases [12,16], and high molecular weight cytokeratin was positive in another patient [5]. In our series, 100% (6/6) of the cases showed immunohistochemical positivity for CK34βE12 and CK5/6 and negative or faint positive staining for pan-cytokeratin AE1/AE3.…”

Section: Discussionsupporting

confidence: 47%

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Amyloidosis cutis dyschromica

Qiao

Fang

Yao

2012

Orphanet J Rare Dis

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BackgroundAmyloidosis cutis dyschromica is a rarely documented variant of cutaneous amyloidosis. To date, only 26 cases have been reported.ObjectiveThe purpose of this study was to improve the clinical and histopathological data for this variant of amyloidosis and to highlight the immunohistochemical features of the disease. The published cases were also reviewed.MethodsWe performed a retrospective review of patients with amyloidosis cutis dyschromica in a single centre. The clinical, histopathological and immunohistochemical features were documented and analysed.ObservationsWe described 10 cases of amyloidosis cutis dyschromica. Six of them were female. Five patients were from the same family, and the other 5 were sporadic. The distinguishing features of the clinical presentation included generalised mottled hyper- and hypopigmented macules, which were asymptomatic or mild pruritic. The typical onset of the lesions occurred in childhood (n = 7) and occasionally after puberty (n = 3). No evidence of systemic amyloidosis deposition was observed in these cases of amyloidosis cutis dyschromica. Amyloid deposits were observed in the papillary dermis and were positive for the Congo red stain. An immunohistochemical study showed that the amyloid expresses cytokeratins CK34βE12 and CK5/6.ConclusionsWe described the largest series of amyloidosis cutis dyschromica to date and reviewed the published patients. This rare disease is featured by generalised mottled hyper- and hypopigmented lesions, and it is a rare variant of primary cutaneous amyloidosis without evidence of systemic amyloid deposition. Positive staining for the cytokeratins CK34βE12 and CK5/6 in amyloidosis cutis dyschromica suggests that the amyloid is derived from keratinocytes.

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Section: Discussionsupporting

confidence: 47%

“…The damage may then result in keratinocyte deposition and degenerate to amyloid at the dermo-epidermal junction [18]. Only 3 reports in the literature reported the keratin composition of the amyloid in ACD [5,12,16]. However, only wide-spectrum cytokeratin antibodies or high molecular weight cytokeratin antibodies were used in these reports.…”

Section: Discussionmentioning

confidence: 99%

Amyloidosis cutis dyschromica

Qiao

Fang

Yao

2012

Orphanet J Rare Dis

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“…Acitretin was given and seems to be effective because it may act by minimizing keratinization defect that causes keratin degeneration to amyloid as proposed by some to be the pathogenesis of this disorder. 10 Qiao et al in his series has convincingly shown 100% positivity to immunohistochemical staining for keratin, CK34 E12 and CK5/6, that the amyloid is thought to be of epidermal origin. 6 This finding seems to agree with the work of Moriwaki et al 9 that points at a disturbance of keratinocyte repairs after irradiation with ultraviolet.…”

Section: Discussionmentioning

confidence: 94%

Familial amyloidosis cutis dyschromica in three siblings: report from Indonesia

2014

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Amyloidosis cutis dyschromica (ACD) is an extremely rare type of primary cutaneous amyloidosis. To date there are fewer than 40 published cases worldwide; some were reported affecting several family members. Its resemblance to other common pigmentation disorders makes it rarely recognized at first sight. Our patient, the 12-year-old firstborn son of non-consanguineous parents presented with generalized mottled pigmentation starting from lower extremities. His siblings suffered from similar condition. The clue for diagnosis is the amyloid deposition in the papillary dermis. The etiology of ACD is still unknown, but genetic factors and ultraviolet radiation are implicated. It is proposed that disturbance of keratinocyte repair following ultraviolet radiation results in amyloid deposition. The treatment remains a challenge. Oral acitretin treatment, thought to repair keratinization defect, gave a slight improvement in our case. Our is the first case of ACD reported in Indonesia.

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“…topical corticosteroids, tretinoin, cryotherapy and carbon dioxide laser have varying effects. Systemic acitretin have been applied and suggest a good response [17]. …”

Section: Discussionmentioning

confidence: 99%

Amyloidosis cutis dyschromica in two female siblings: cases report

Yang

Lin

2011

BMC Dermatol

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BackgroundCutaneous amyloidosis has been classified into primary cutaneous amyloidosis (PCA, OMIM #105250), secondary cutaneous amyloidosis and systemic cutaneous amyloidosis. PCA is the deposition of amyloid in previously apparent normal skin without systemic involvement. Amyloidosis cutis dyschromica (ACD) is a rare distinct type of PCA. Here, the unique clinical and histological findings of two Chinese female siblings with ACD were described.Cases presentationsPatient 1 was a 34-year-old female, presented with mildly pruritic, diffuse mottled hyperpigmentation and hypopigmentation. The lesions involved all over the body since she was 10 years old. There were a few itchy blisters appearing on her arms, lower legs and truck, especially on the sun-exposed areas in summer. Some hypopigmented macules presented with slight atrophy. Patient 2 was 39-year-old, the elder sister of patient 1. She had similar skin lesions since the same age as the former. The atrophy and blisters on the skin of the patient with amyloidosis cutis dyschromica have not been described in previous literature. Histological examinations of the skin biopsies taken from both patients revealed amyloid deposits in the whole papillary dermis. Depending on the histological assessment, the two cases were diagnosed as amyloidosis cutis dyschromica.ConclusionThe two cases suggest that the atrophy and blisters may be the uncommon manifestations of amyloidosis cutis dyschromica. It alerts clinicians to consider the possibility of ACD when meeting patients with cutaneous dyschromia. Skin biopsy is essential and family consultation of genetic investigation is very important in such cases.

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Amyloidosis Cutis Dyschromica: A Case Treated with Acitretin

Cited by 34 publications

References 15 publications

Amyloidosis cutis dyschromica

Amyloidosis cutis dyschromica

Familial amyloidosis cutis dyschromica in three siblings: report from Indonesia

Amyloidosis cutis dyschromica in two female siblings: cases report

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