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A case of generalized acanthosis nigricans with positive lupus erythematosus-related autoantibodies and antimicrosomal antibody: autoimmune acanthosis nigricans?

Kondo Y, Umegaki N, Terao M, Murota H, Kimura T, Katayama I - Case Rep Dermatol (2012)

Bottom Line: Neither internal malignancy nor other endocrinological disorders, including glucose intolerance, were detected during a 10-year clinical course with benign diffuse papillomatosis extending from the mucosa of the larynx to the esophagogastric junction.The case was complicated with chronic thyroiditis and interstitial pneumonia, which were not treated with any medication.AN skin lesions and mucosal papillomatosis regressed with oral cyclosporine A, accompanied by the lowering of autoantibody titers.

View Article: PubMed Central - PubMed

Affiliation: Department of Dermatology Integrated Medicine, Osaka University Graduate School of Medicine, Suita-City, Japan.

ABSTRACT
Acanthosis nigricans (AN) is a hyperpigmented keratotic skin lesion known to be associated with malignant disease and endocrinopathy. We report a very rare case of generalized AN with Sjögren's syndrome- and systemic lupus erythematosus-like features but without type B insulin resistance. Neither internal malignancy nor other endocrinological disorders, including glucose intolerance, were detected during a 10-year clinical course with benign diffuse papillomatosis extending from the mucosa of the larynx to the esophagogastric junction. The case was complicated with chronic thyroiditis and interstitial pneumonia, which were not treated with any medication. AN skin lesions and mucosal papillomatosis regressed with oral cyclosporine A, accompanied by the lowering of autoantibody titers. This is the first report of generalized AN involving an area from the mucosa of the larynx to the esophagogastric junction accompanied by autoimmune manifestations which responded to systemic immunosuppressive therapy.

No MeSH data available.


Related in: MedlinePlus

Histological findings. a, c Histological features of biopsy specimen from posterior neck (a, magnification ×40) and larynx (c, magnification ×200). Slight hyperkeratosis and acanthosis were seen undulating with dermal papillomatosis. b Upper endoscopy feature. A diffuse papillomatosis extending to the esophagus, where little normal mucosa was left. Carcinomatous changes such as ulcers or nodules were not observed.
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Figure 2: Histological findings. a, c Histological features of biopsy specimen from posterior neck (a, magnification ×40) and larynx (c, magnification ×200). Slight hyperkeratosis and acanthosis were seen undulating with dermal papillomatosis. b Upper endoscopy feature. A diffuse papillomatosis extending to the esophagus, where little normal mucosa was left. Carcinomatous changes such as ulcers or nodules were not observed.

Mentions: A 58-year-old Japanese male had noticed a diffuse pigmentation of his face and generalized hypotrichosis 10 years before his first visit to our clinic. He had been diagnosed with AN at the age of 55 years because of mucocutaneous manifestations such as diffuse papillomatosis. No internal malignancy was detected during repeated medical examinations over a 10-year clinical course. He was referred to our outpatient clinic in June 2006. His height was 168.5 cm and his weight 59 kg (body mass index 20.7). He presented with dirty-looking keratosis of the umbilicus, areola, and chest wall, diffuse papillomatosis of the palms and soles in addition to numerous acrochordons of the face, neck, and axilla. Hyperplastic and papillomatous changes of the lips and oral cavity accompanied by impaired taste sensation were also noted (fig. 1a–d). His past medical history showed interstitial pneumonia and chronic thyroiditis. He did not have any familial history of endocrine disorders, including AN. The biopsy specimens taken from skin of the posterior neck and mucosa of the pharynx and larynx revealed slight hyperkeratosis and acanthosis undulating with dermal papillomatosis (fig. 2a, c). Upper endoscopy showed a diffuse papillomatosis extending from the mucosa of the larynx to the esophagogastric junction, where little normal mucosa was left (fig. 2b). No epidermal inclusion bodies were observed and polymerase chain reaction analysis of human papilloma virus DNA in the mucosal region was negative. These findings were compatible with a diagnosis of AN [1, 13, 14, 15].


A case of generalized acanthosis nigricans with positive lupus erythematosus-related autoantibodies and antimicrosomal antibody: autoimmune acanthosis nigricans?

Kondo Y, Umegaki N, Terao M, Murota H, Kimura T, Katayama I - Case Rep Dermatol (2012)

Histological findings. a, c Histological features of biopsy specimen from posterior neck (a, magnification ×40) and larynx (c, magnification ×200). Slight hyperkeratosis and acanthosis were seen undulating with dermal papillomatosis. b Upper endoscopy feature. A diffuse papillomatosis extending to the esophagus, where little normal mucosa was left. Carcinomatous changes such as ulcers or nodules were not observed.
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC3362225&req=5

Figure 2: Histological findings. a, c Histological features of biopsy specimen from posterior neck (a, magnification ×40) and larynx (c, magnification ×200). Slight hyperkeratosis and acanthosis were seen undulating with dermal papillomatosis. b Upper endoscopy feature. A diffuse papillomatosis extending to the esophagus, where little normal mucosa was left. Carcinomatous changes such as ulcers or nodules were not observed.
Mentions: A 58-year-old Japanese male had noticed a diffuse pigmentation of his face and generalized hypotrichosis 10 years before his first visit to our clinic. He had been diagnosed with AN at the age of 55 years because of mucocutaneous manifestations such as diffuse papillomatosis. No internal malignancy was detected during repeated medical examinations over a 10-year clinical course. He was referred to our outpatient clinic in June 2006. His height was 168.5 cm and his weight 59 kg (body mass index 20.7). He presented with dirty-looking keratosis of the umbilicus, areola, and chest wall, diffuse papillomatosis of the palms and soles in addition to numerous acrochordons of the face, neck, and axilla. Hyperplastic and papillomatous changes of the lips and oral cavity accompanied by impaired taste sensation were also noted (fig. 1a–d). His past medical history showed interstitial pneumonia and chronic thyroiditis. He did not have any familial history of endocrine disorders, including AN. The biopsy specimens taken from skin of the posterior neck and mucosa of the pharynx and larynx revealed slight hyperkeratosis and acanthosis undulating with dermal papillomatosis (fig. 2a, c). Upper endoscopy showed a diffuse papillomatosis extending from the mucosa of the larynx to the esophagogastric junction, where little normal mucosa was left (fig. 2b). No epidermal inclusion bodies were observed and polymerase chain reaction analysis of human papilloma virus DNA in the mucosal region was negative. These findings were compatible with a diagnosis of AN [1, 13, 14, 15].

Bottom Line: Neither internal malignancy nor other endocrinological disorders, including glucose intolerance, were detected during a 10-year clinical course with benign diffuse papillomatosis extending from the mucosa of the larynx to the esophagogastric junction.The case was complicated with chronic thyroiditis and interstitial pneumonia, which were not treated with any medication.AN skin lesions and mucosal papillomatosis regressed with oral cyclosporine A, accompanied by the lowering of autoantibody titers.

View Article: PubMed Central - PubMed

Affiliation: Department of Dermatology Integrated Medicine, Osaka University Graduate School of Medicine, Suita-City, Japan.

ABSTRACT
Acanthosis nigricans (AN) is a hyperpigmented keratotic skin lesion known to be associated with malignant disease and endocrinopathy. We report a very rare case of generalized AN with Sjögren's syndrome- and systemic lupus erythematosus-like features but without type B insulin resistance. Neither internal malignancy nor other endocrinological disorders, including glucose intolerance, were detected during a 10-year clinical course with benign diffuse papillomatosis extending from the mucosa of the larynx to the esophagogastric junction. The case was complicated with chronic thyroiditis and interstitial pneumonia, which were not treated with any medication. AN skin lesions and mucosal papillomatosis regressed with oral cyclosporine A, accompanied by the lowering of autoantibody titers. This is the first report of generalized AN involving an area from the mucosa of the larynx to the esophagogastric junction accompanied by autoimmune manifestations which responded to systemic immunosuppressive therapy.

No MeSH data available.


Related in: MedlinePlus